preservation of the congruence of the hip in perthes ...€¦ · evaluate msa outcome based on gait...
TRANSCRIPT
"PRESERVATION OF THE CONGRUENCE OF THE HIP IN PERTHES DISEASE USING SHELF ACETABULOPLASTY AND THE RELATIONSHIP TO THE KINEMATICS AND KINETICS OF THE HIP DURING GAIT, TOGETHER WITH STANDING BALANCE" MASTER’S THESIS
Florian Stul Emmeline Deloddere Student number: 01307899 Student number: 01308609
Supervisors: Prof. dr. Frank Plasschaert (promotor), Prof. dr. Malcolm Forward (copromotor) A dissertation submitted to Ghent University in partial fulfilment of the requirements for the degree
of Master of Medicine in Medicine
Academic year: 2017-2018
“The author and the promotor give the permission to use this thesis for consultation and to copy
parts of it for personal use. Every other use is subject to the copyright laws, more specifically the
source must be extensively specified when using results from this thesis.”
Date
(handtekening)
Name (student) (promotor)
Acknowledgments This thesis is part of the master of science in medicine of the UGent and is organised in
association with the university hospital of Ghent (UZ Gent). Enabling students to get a taste of
the possibilities and joys of research. We would like to express our sincere gratitude to the
people who supported us in the process of establishing our thesis.
First of all, we want to thank our promotor, Prof. Dr. F. Plasschaert (MD, Phd), to submit such an
interesting and promising study. Hereby creating the possibility to work on a study leading to a
clinically relevant result. Even though our promotor had an extremely busy agenda, he made it
possible to find time to evaluate the course of the thesis and give us usefull directions.
Secondly, we want to thank Prof. Dr. M. Forward, copromotor of the thesis. Without his help and
extensive gait analysis expertise, the research wouldn’t have been possible. He taught us how to
operate the gait lab and helped us through the elaborate process of collecting, processing and
interpreting data. At any day, any time, he was there to help us solve problems and supported us
to create the study to what it has become.
Thirdly, we want to thank Dr. Lauwagie (additional supervisor), Mrs. De Dobbelaere (gait lab
assistant) and Mrs. Fruyt (secretary pediatric orthopaedics) for the logistic support and help
when needed and Dr. Mullie and Dr. Willemot for the support on the processing of the X-rays of
the patients.
Special thanks also to our parents for always encouraging and supporting us. They made it
possible for us to study medicine and created the environment needed to succeed through the
years of the education.
We also want to thank our thesis-partner, although we didn’t know each other at the start, we
both feel that we became a good team. We shared, for a year and a half, progress and
frustrations related to the thesis, we solved problems and also created a few, but in the end we
finished the thesis.
Last, but most definitely not least, we want thank the control persons and patients (and their
parents) who participated in our study. None of this would have been possible without their
selfless participation.
Table of Contents ABSTRACT (English) .................................................................................................................. 1
Introduction ............................................................................................................................. 1
Methodology ............................................................................................................................ 1
Results .................................................................................................................................... 1
Conclusion .............................................................................................................................. 1
ABSTRACT (Nederlands) ........................................................................................................... 2
Introductie ............................................................................................................................... 2
Methodologie ........................................................................................................................... 2
Resultaten ............................................................................................................................... 2
Conclusie ................................................................................................................................ 3
PREFACE ................................................................................................................................... 4
Research question .................................................................................................................. 4
Aim of the study ....................................................................................................................... 4
Relevance ............................................................................................................................... 4
1 INTRODUCTION ................................................................................................................. 6
Legg-Calvé-Perthes Disease: What is LCP? ................................................................. 6 1.1
Clinical presentation ..................................................................................................... 6 1.2
Pathophysiology ........................................................................................................... 7 1.3
1.4 Radiological techniques and classifications ....................................................................... 8
Therapy ........................................................................................................................ 9 1.5
1.5.1 Containment therapy ........................................................................................... 10
1.5.2 Remedial therapy: ................................................................................................ 12
1.5.3 Salvage therapy: .................................................................................................. 12
Gait Analysis ............................................................................................................... 12 1.6
1.6.1 Kinematics ........................................................................................................... 13
1.6.2 Kinetics ................................................................................................................ 13
1.6.3 Construction of the 3D-model .............................................................................. 13
IOWA Hip Score ......................................................................................................... 14 1.7
2 Methodology ...................................................................................................................... 15
LCPD patients ............................................................................................................ 15 2.1
Controls ...................................................................................................................... 16 2.2
Experimental set-up .................................................................................................... 16 2.3
Radiologic evaluation of the LCPD patients ................................................................ 17 2.4
2.4.1 Pre-processing .................................................................................................... 17
2.4.2 Processing ........................................................................................................... 18
Gait Analysis ............................................................................................................... 20 2.5
2.5.1 Technical specifications of the lab for motion analysis of the UZ Ghent ............... 20
2.5.2 Positioning of the markers: .................................................................................. 20
2.5.3 Sequence of the gait analysis .............................................................................. 22
2.5.4 Data processing – gait analysis ........................................................................... 23
IOWA Hip Score ......................................................................................................... 23 2.6
Data Analysis .............................................................................................................. 23 2.7
2.7.1 Kinematics ........................................................................................................... 23
2.7.2 Kinetics ................................................................................................................ 24
Statistical Analysis ...................................................................................................... 26 2.8
2.8.1 Comparison of the PIG and the functional (PiG-Matlab) model ............................ 26
2.8.2 Group comparison ............................................................................................... 26
2.8.3 Side comparison .................................................................................................. 27
2.8.4 Correlation IOWA – gait variables ........................................................................ 28
3 Results .............................................................................................................................. 29
General variables of the groups .................................................................................. 29 3.1
Gait and balance variables comparison ...................................................................... 29 3.2
3.2.1 Group comparison PiG-Matlab Model .................................................................. 30
3.2.2 Side comparison PiG-Matlab Model ..................................................................... 32
3.2.3 Differences between both gait analysis models ................................................... 34
IOWA Hip Score ......................................................................................................... 35 3.3
3.3.1 Comparison of the patient groups ........................................................................ 35
3.3.2 Correlations IOWA – gait variables ...................................................................... 35
4 Discussion ......................................................................................................................... 37
The research question ................................................................................................ 37 4.1
Discussion of the results ............................................................................................. 37 4.2
4.2.1 Gait model ........................................................................................................... 37
4.2.2 Overall group comparison .................................................................................... 39
4.2.3 Side by side comparison ...................................................................................... 43
The study design choices ........................................................................................... 44 4.3
Study limitations .......................................................................................................... 45 4.4
Future research .......................................................................................................... 46 4.5
Conclusion .................................................................................................................. 46 4.6
5 References ........................................................................................................................ 48
6 Appendix ............................................................................................................................... I
Schematic illustration of the gait lab ............................................................................... I 6.1
Radiographic parameters .............................................................................................. II 6.2
6.2.1 Coverage and subluxation ..................................................................................... II
6.2.2 Congruence: SDS ................................................................................................. VI
IOWA hip score (chart) .............................................................................................. VIII 6.3
Results PiG Model ....................................................................................................... IX 6.4
6.4.1 Group comparison PiG Model ............................................................................... IX
6.4.2 Side comparison ................................................................................................... IX
Tables results ............................................................................................................... X 6.5
1
ABSTRACT (English)
Introduction Legg-Calvé-Perthes disease (LCPD) is a pediatric orthopedic disease, affecting the mobility of
the hip and hip joint loading, leading to premature osteoarthritis. Modified Shelf Acetabuloplasty
(mSA) is a recent developed containment therapy for severe LCPD patients. This study aims to
evaluate mSA outcome based on gait analysis. Kinematics and kinetics of the hip and pelvis are
used to obtain objective data to evaluate hip mobility, hip joint loading and gait patterns.
Methodology Twenty-eight patients are divided into four quartiles based on Principal Component Analysis
(PCA) processed X-ray measurements. Only the best and worst quartile are selected in further
analysis, creating a group of patients with good radiographic outcome and a group of patients
with bad radiographic outcome. Subsequently, in order to compare both patient groups and the
seven healthy controls, computerized gait analysis is performed based on Plug-in-Gait (PiG) and
PiG-Matlab Models. Kinematic and kinetic parameters are evaluated, for the three planes
(frontal, sagittal and transverse), in order to obtain a complete evaluation. The analysis is based
on both a group comparison and an affected/unaffected side comparison within the groups.
Results In the group comparison, a reduced Hip Extension, Hip Range of Motion (ROM) and increased
Pelvic Tilt is observed for the affected side. A trend of reduction is detected for Hip Abduction in
the group of patients with bad outcome, but no significance is detected for Pelvic Obliquity,
Balance Standing or Trendelenburg gait. The bad outcome patient group shows a significant
reduction for Hip Flexion, Hip ROM and Hip External Rotation Moment for the affected side.
Conclusion In conclusion the patient group with good outcome shows no compromised gait pattern. The
abnormalities observed in the patient group with bad outcome are mostly due to aberrant hip
extension, other affected parameters are compensation mechanisms for the affected hip
extension. The major clinically important variables: Hip Abduction, Trendelenburg gait and Hip
Internal Rotation are unaffected in both patient groups. This is an important result, especially
concerning the Hip Abduction and the Hip Abduction Moment, because of its consequences with
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respect to the long-term complications, such as premature osteoarthritis.
ABSTRACT (Nederlands)
Introductie De ziekte van Legg-Calvé-Perthes (LCPD) is een orthopedische ziekte, die voorkomt op
kinderleeftijd. Bij LCPD is de mobiliteit en belasting van de heup aangetast. Dit leidt tot
vroegtijdige artrose. Modified Shelf Acetabuloplasty (mSA) is een recent ontwikkelde
containment behandeling voor patiënten met een ernstige vorm van LCPD. Deze studie heeft als
doel het resultaat van mSA te evalueren a.d.h.v. ganganalyse. Er wordt gebruik gemaakt van
kinematica en kinetica van de heup en het bekken om objectieve data te verzamelen. Op basis
van deze data wordt een evaluatie gemaakt van de heupmobiliteit, heupbelasting en
gangpatronen.
Methodologie Achtentwintig patiënten worden onderverdeeld in vier kwartielen, gebaseerd op RX variabelen,
die a.d.h.v. Principal Component Analysis (PCA) werden verwerkt. Enkel het beste en slechtste
kwartiel wordt hierbij weerhouden voor verdere analyse. Op die manier worden twee
patiëntengroepen gecreëerd: een patiëntengroep met een goed radiografisch resultaat en een
patiëntengroep met een slecht radiografisch resultaat. Daarna wordt er een ganganalyse
afgenomen, om beide patiëntengroepen met elkaar en met de zeven controles te vergelijken. De
ganganalyse werd afgenomen gebruikmakende van het Plug-In-Gait (PiG) en PiG-Matlab
Model. Kinematische en kinetische data wordt geëvalueerd in drie vlakken (sagittaal, frontaal,
transversaal) om een complete evaluatie te verkrijgen. De analyse is gebaseerd op een
groepsvergelijking en op een vergelijking binnen de groep van het gezonde en het aangetaste
been.
Resultaten Bij het vergelijken van de drie groepen wordt voor het aangetaste been een afname van de
heupextensie en heupbewegingsbereik gezien in combinatie met een toename van de
anterieure bekkenkanteling. In de groep met slecht resultaat wordt een trend tot reductie gezien
voor heup abductie, maar er wordt geen significant verschil t.o.v. de andere groepen gezien voor
bekkenuitzakking en balans in stand. Ook een Trendelenburg gangpatroon wordt niet
waargenomen. Voor de groep met slecht resultaat wordt bij het vergelijken van beide zijden een
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significant reductie waargenomen voor heupflexie, heupbewegingsbereik en heup exorotatie
moment.
Conclusie Voor de patiënten met goed resultaat wordt geen afwijking van het gangpatroon gezien. De
abnormaliteiten die geobserveerd worden voor de groep met slecht resultaat kunnen
grotendeels verklaard worden door aberrante heupextensie. De andere afwijkende parameters
behoren namelijk tot de compensatiemechanismen van de aangetaste heupextensie. De klinisch
relevante variabelen: heupabductie, Trendelenburg gangpatroon en endorotatie van de heup,
zijn niet afwijkend in beide patiëntengroepen. Zeker voor heupabductie en heupabductie
moment, zijn dit belangrijke resultaten, aangezien beide parameters een belangrijke impact
hebben op langetermijnscomplicaties, zoals bijvoorbeeld vroegtijdige artrose.
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PREFACE
Research question What is the relationship between modified Shelf Acetabuloplasty (mSA) and the kinematics and
kinetics of the hip during gait, together with standing balance? And as an additional question, is
Plug-in-Gait (PiG) or PiG-Matlab the most valid model in patients with Legg-Calvé-Perthes
(LCPD)?
Aim of the study The aim of this thesis is to evaluate the effect of a modified Shelf Acetabuloplasty procedure on
the kinematics and kinetics of the hip as well as the standing balance, when treating children
with Legg-Calvé-Perthes (LCP). The purpose of modified Shelf Acetabuloplasty is to increase
the femoral head coverage in order to prevent malformations and incongruence of the hip joint
as they can occur, during the healing process. We therefore evaluate routine postoperative
follow-up X-rays of LCP patients treated by Prof. dr. Frank Plasschaert (UZ Gent), using his
modified Shelf Acetabuloplasty. The patients are scored for parameters representing coverage
and congruence. Based on these parameters, patients were divided into two groups: (a) better
radiographic results and (b) worse radiographic results. Instrumented Gait Assessments were
used to compare these patient groups for hip mobility and hip power, by analysing the
kinematics and kinetics.
The hypothesis states that in both groups of LCP (better and worse radiographic results) good
functional results will be obtained. This is based on the presumption that the shelf will only
influence the ultimate shape of the hip during the healing process of LCP. If the shelf is resorbed
into the acetabulum or migrates after the healing process, this will cause a bad radiographic
result. The hypothesis however states that the shelf will have already fulfilled its effect and the
resorption and/or migration has no further impact on the function of the hip. The focus of this
research is the hip function. Concerning the possible development of early onset osteoarthritis,
no predictions are made.
Relevance LCPD is a self-limiting disease in the longer-term, however deformation of the femoral head that
might develop during the process of LCP disease predisposes patients to premature secondary
degenerative arthritis. Depending on its severity, arthritis can compromise the quality of life of
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patients and entail high costs for the health care system (e.g. when total hip replacement is
needed.)
Defining a treatment for LCP that not only improves short-term outcome in maintaining the
function of the hip, but which also addresses the longer-term outcome with respect to arthritis
would offer an important break through in the management of LCP.
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1 INTRODUCTION
Legg-Calvé-Perthes Disease: What is LCP? 1.1Arthur Thornton Legg first describes Legg-Calvé-Perthes Disease (LCPD) in 1909. In the same
period, a few years after the discovery of the X-ray technology, Jacques Calvé and Georg
Perthes report on similar observations (1).
Nowadays, the reported incidence of LCPD varies from 8 to 32 per 100 000, depending on the
literature (1). LCPD affects boys 4-5 times more than girls and bilateral LCP is more often in
girls. The impact of the development of LCPD and gender on the prognosis do remain unclear
but might be related to a lower remodelling potention in girls of similar chronological age than
boys (1,2).
Multiple causal factors of LCPD have been investigated but, to the best of our knowledge, no
distinct causal factor has been identified thus far (3,4). Hypercoagulation has more recently been
put forward as a possible cause for the femoral head necrosis. A significant relationship between
LCP and a factor V Leiden mutation was shown by a meta-analysis of Woratanarat et al.(3).
Till today, general consensus reports LCP to be a multifactorial disease with genetic
predisposition influenced by environmental causal factors. LCP could be caused by multiple
etiological factors with a common pathological and clinical presentation. For instance acetabular
retroversion and obesity are conditions frequently reported to be associated with LCPD (4).
Clinical presentation 1.2The age of onset of LCP is between 2 and 12 years of age (5), with a peak incidence in children
between 4 and 8 (1,3,5). The importance of the age of onset within the disease process of LCP
will be focussed on a bit more in the sections about pathophysiology (cfr 1.3 Pathophysiology)
and therapy (cfr. 1.5 Therapy).
The clinical presentation of LCP is highly variable and has different characteristics throughout
the phases of the disease (5). First, in the early stages, LCP is characterised by a limping gait,
followed by an ill-defined pain along the anterior side of the hip and medial thigh (3). Sometimes,
only isolated knee pain does exist (5). The range of motion of the hip joint is often affected by
LCP. Usually a loss in abduction and internal rotation are found. A progression towards a
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Trendelenburg gait pattern might develop (1,3,5). Normally the maximum flexion and extension
of the hip are not affected by LCPD. Although less specific, the children affected by LCP show a
relatively small stature and the bone age can be delayed when compared to unaffected peers
(1). Experienced pain (due to synovitis in the early stages), range of motion, abduction, internal
rotation, flexion and extension are among the variables evaluated in this study since they seem
to be the clinical variables affected by LCP.
Pathophysiology 1.3Disruption of the blood supply in the femoral head, due to the accumulation of multiple infarcts,
is generally accepted to be the cause of LCPD (4,6,7).
The pathophysiological process of LCPD can be divided into four stages: the avascular phase
(disruption of the blood supply and necrosis of bone and cartilage), continued by the
fragmentation phase (collapse of the trabecular bone), followed by the reossification phase
(reformation of bone) and finally the healing phase (maturation of the bone) (7). The weakening
and deformation of the bone vary throughout the different healing stages. The deformation of the
femoral head, occurring in LCPD, can be approached from a mechanical point of view. The
ischaemia during the avascular phase reduces the load-carrying capacity of the femoral head
(necrotic bone) and induces an inability to resist deformation. Fractures and deformity of the
femoral head can develop within this phase due to a loading force, exceeding the capacity of the
femoral head (8).
During the avascular phase a disruption of the blood supply leads to necrosis and
apoptosis of both bone marrow and articular cartilage in the hip joint and has secondary effects
on the soft tissue. The combination of soft tissue changes and muscle spasms can cause an
extrusion of the femoral head out of the acetabulum, resulting in an increased lever arm putting
the vulnerable femoral head under pressure. Extrusion is an important factor in the development
of femoral head deformation. The abnormal loading of the hip in combination with a weakened
bone structure challenges the hip joint (9). Within this phase the femoral head is extremely
vulnerable to deformation, since fractures cannot repair themselves in avascular bone.
Following the ischaemic damage, the fragmentation phase is initiated by the
neovascularisation, inducing a first step within the healing process. The higher concentration of
osteoclasts leads to the resorption of the bone necrosis. During this phase radiolucent areas can
be determined (4,10). The resorbed bone is replaced by fibrovascular tissue. This phase is still
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characterised by a weak bone structure and an even higher potential to develop fractures and
deformation of the femoral head. Research has shown that inhibition of bone resorption, using
bisphosphonates or RANKL inhibitors, should counter the weakening of the bone structure and
conserve both trabecular bone and sphericity of the femoral head (8).
During the reossification phase , ensuing the fragmentation phase, the fibrovascular
tissue is replaced by new-formed bone. The process ends with the healing phase : the bone
reobtains its trabecular structure and a normal radiodensity can be observed, as the maturation
of the bone in the femoral head is complete (7).
Age has an important impact on the earlier described risk for deformation of the femoral head
within the disease process. Depending on the age of the subject, a different response to
hypoxemia and angiogenesis can be observed. The neovascularisation is much more activated
in children younger than 6 years in comparison to children older than 8 years. Therefore,
younger children with LCPD are expected to experience less deformation. Furthermore, young
children have a relatively higher proportion of cartilage covering the bony core of the femoral
head. This makes younger patients less vulnerable to fractures in the underlying weakened bone
(8).
1.4 Radiological techniques and classifications
Throughout the years several radiologic classifications for LCP, using X-rays, have been
developed. The most relevant are the Waldenström, Catterall, Salter-Thompson, Herring Lateral
pillar, Stulberg and Mose classification. These classifications differ w.r.t. the phase of the
disease at which they can be applied, the measurements on which they are based and the
purpose of its use. Below we briefly summarize the main ideas of these classification methods.
To specify the stage of the disease the Waldenström classification can be used. The
classification is applied during the active phase of the disease. The Catterall, Salter-Thompson
and Herring lateral pillar classifications focus on the prognostic short-term outcome based on the
extent of necrosis, subchondral fracture or loss of height of the lateral pillar of the femoral
epiphysis. The Stulberg classification is based on deformity that develops in the later stages of
LCPD. The higher classes (II-IV) have a high risk at premature osteoarthritis (8).
The Mose classification uses a template of concentric circles, of which the radii differ 1
millimetre, to define the sphericity of the femoral head (11).
Next to the above classifications, descriptive coverage and congruence parameters can be
9
measured to classify subjects. One study reported the measurement of angles to have higher
interobserver reliability than radiographic classifications, but the importance of clearly defined
landmarks is fundamental (12,13). Coverage can be measured by the Sharp angle (SA), the
center-edge angle (CEA) of Wilberg, subluxation ratio (SR), femoral head size ratio (FHSR) and
femoral head coverage ratio (FHCR). The congruence is measured using the Sphericity
Deviation Score. The Sharp angle is a measure of acetabular dysplasia for which a good inter-
observer reliability as well as good agreement could be found in skeletally immature subjects
(14–16). The center-edge angle of Wilberg and the subluxation ratio, also referred to as “Medial
joint space ratio” are measures for subluxation (17,18). The femoral head shape is a very
important determinant of long-term outcome in LCPD (3). Significant correlations were found
between clinical outcome and femoral head size ratio (19). Wiig et al. found femoral head
coverage to be a reliable measurement for quantifying the acetabular covering of the femoral
head, characterised by relatively small inter-observer differences (13). The congruence of the
femoral head can be quantified using the, recently developed, Sphericity Deviation Score (SDS).
SDS is a continuous variable, which is an important advantage over the traditional classifications
described above. Especially because sample sizes in LCPD research are often limited (20). In
addition, it is applicable for unilateral and bilateral cases and validated for application during both
the healing stage and at skeletal maturity. In recent literature the intraobserver and interobserver
reproducibility were found to be satisfactory (21). An overview of the formulas of the variables
can be found in the appendix (6.2 Radiographic parameters (Appendix)).
Therapy 1.5LCPD is a self-limited disorder, leading to the occlusion of the femoral blood supply, which
results in bone necrosis (8). Usually a spontaneous revascularization is seen over a period of 2
to 4 years. The course of disease can occur with or without sequellae. In a significant number of
patients, influenced by age, femoral deformation can develop during the natural course of LCP.
The goal of treatment of LCP does focus on an avoidance of deformation of the femoral head
within the disease process in order to minimise physical impairments and the development of
early onset secondary degenerative osteoarthritis (2,22).
Deformation of the femoral head is commonly seen in the late stage of the fragmentation phase
or the early stage of the reossification phase. Deformation can be used as a marker point to
divide the course of the disorder into two parts: an early or pre-deformation part and a later or
post-deformation part. Following this division, preventive treatment (containment) should be
initiated during the pre-deformation part of the disorder. Any treatment started after the onset of
10
the deformation should be regarded a remedial or salvage treatment (22).
Based on the previous discussion, treatment in LCPD can be categorised into the following
categories: containment therapy (pre-deformation), remedial therapy (post-deformation) and
salvage therapy (treatment of possible sequellae). An overview of the treatment options is
depicted below (cfr. Figure 1).
1.5.1 Containment therapy
The goal of containment therapy is to stop increased loading on the antero-lateral side of the
epiphysis of the hip, by avoiding extrusion of the femoral head and preventing deformation and
the consequent incongruence (6). This goal can be reached by using a brace (to fixate the hip in
abduction and internal rotation) or by surgery. One of the options for surgery is Shelf
Acetabuloplasty (9). Whatever technique is chosen, the common goal is to protect the
anterolateral part of the femoral head against (abnormal) loading, potentially leading to
deformation of the femoral head (before it can repair itself) (7,9,23).
The decision of which treatment strategy should be applied, is based on the severity (extent of
the necrosis) and stage of disease. Age and remodeling potential do influence the prognosis of
Figure 1: overview LCPD treatment options
11
the disorder and therefore the choice of treatment. The choice of what to do (or not to do) when
is a difficult task to the clinician involved in the treatment of LCPD. Therefore, an estimation of
the prognosis and need for treatment is made based on the head at risk signs, decreased
mobility, patients age and radiological classification. At present, the radiological classification
being frequently used, is the Herring’s lateral pillar classification (24). Today, the lateral pillar
classification together with age is the dominant factor when deciding upon treatment (25,26).
Based on these principles four treatment groups have been defined:
1) group A Herring lateral pillar classification
2) group B and group B/C Herring lateral pillar classification under the age of 8
3) group B and group B/C Herring lateral pillar classification over the age of 8
4) group C Herring lateral pillar classification.
In general, a good prognosis is to be expected in patients of the first two groups. In these groups
a surgical intervention is not necessary. It is however important to advise a four monthly X-ray
follow-up in patients from the age of seven. In order to detect extrusion and proceed with
containment therapy if needed (9). For the last two groups previous studies have shown surgery
to produce significantly better results than conservative treatment (26). The goal of surgical
containment procedures is the maintenance of the sphericity (varus osteotomy, innominate
osteotomy, pelvic osteotomy) and/or centralisation of the femoral head (innominate osteotomy,
triple osteotomy). Despite the surgical options mentioned above, the prognosis in subjects older
than eight is often poor. The success rate of surgery decreases with increasing age.
Shelf Acetabuloplasty, developed to overcome the problems associated with femoral and Salter
innominate osteotomy, is probably not only effective in older subjects but also in other subjects,
older than five presenting with a severe case of LCPD (27–30). Shelf Acetabuloplasty has been
successfully applied in severe late-onset LCPD but there were concerns regarding the damage
at the lateral acetabular epiphysis, graft resorption and graft migration. However, only for the
graft migration, evidence was found (14,28,31). By stabilizing the shelf, using the reflected head
of the rectus femoris tendon, graft migration can be prevented (27,31). Modified Shelf
Acetabuloplasty, the technique studied in this thesis, is a further improvement of the Shelf
Acetabuloplasty, and combines the Albee Graft and Staheli Shelf technique. The technique
meets the principles of containment therapy. The advantages of this new technique are: the
intrinsic stability (which makes a hip spica cast obsolete and thus shortens the required hospital
admission), the in-built compression, the not-affected growth of the acetabulum, no tendency for
graft resorption and a major pain relieving effect immediately after the operation. After
performing the modified Shelf Acetabuloplasty the following effects have been observed:
12
increase of the femoral head coverage ratio, decrease of the subluxation ratio and good shelf
height ratio (32).
1.5.2 Remedial therapy:
When the disorder already proceeded to the late fragmentation phase or early reconstitution
phase, remedial therapy is indicated. The goal is to minimise the commencing deformation
effects. Results increase when the containment therapy is applied in earlier stages of the
disorder (9).
1.5.3 Salvage therapy:
After the healing process has ended, abnormalities can occur under the form of sequellae.
These abnormalities are cam and pincer impingement, functional retroversion and enlarged and
diminished trochanteric impingement. Recent attempts were made to reshape the deformed
femoral head. These treatments result in pain reduction, increased mobility and improved hip
abductor strength (9). Shelf Acetabuloplasty can be used as a salvage therapy, but the success
rate decreases compared to when it is used as containment therapy (30). Overall, the long-term
prognosis for salvage therapy is poor and often a hip prosthesis is required after a few years. As
reported by Joseph B., 39% of the patients has undergone a total hip replacement within eight
years after the salvage surgery (9). In addition, Lehmann et al. reported that 30% of the
adolescents undergoing total hip replacement for degenerative joint disease, secondary to
LCPD, had previously been treated with joint preserving salvage therapy (33). These findings
stress the importance of treatments early in the course of the disease.
Gait Analysis 1.6The purpose of using gait analysis in this study is to collect kinetic and kinematic data, derive
relevant quantitative summary parameters and obtain an objective assessment of the gait
pattern. Using gait analysis enables a more accurate assessment of the gait pattern than visual
gait assessment. It is broadly acknowledged as a useful research tool (34). The kinematics,
being the joint motions of the subject, are measured using a set of digital infrared cameras
positioned 360 ° around the gait platform (Cfr. Appendix: Figure 3: schematic illustration of the
gait lab). Simultaneously, data from force plates mounted within the walkway floor is collected,
enabling calculation of joint force and moments during gait (35).
13
1.6.1 Kinematics
Kinematic data consists of the position and orientation of body segments, angles of joints, linear
and angular velocities and accelerations. It thus describes the movements of the subject in a
(three dimensional) geometrical way over time (referred to as gait cycle). The movement of the
subject through space is monitored by the optico-electronic video cameras. The cameras
register markers attached to the subject’s skin, positioned in such a way they are seen by at
least two cameras. Photogrammetric (or stereometric) reconstruction techniques are used
(together with calibration data) to derive three dimensional coordinates of markers in space over
time. The markers placed on the subject’s skin act as repère points to digitise the subject.
Groups of three or more markers are used on each body segment of the subject to be able to
determine position and orientation in 3D space. The markers are placed onto the skin, often on
anatomical bony landmarks (36).
The correct marker placement on anatomical landmarks is extremely important to reduce data
noise and collect useful data (depending on the model used) (37–39). The goal is to place the
markers1 in a repeatable, consistent and clinically valid manner. Skin movement and muscle
contractions, interfere with the steady position of the marker and cause noise (39). To this day,
despite the development of new techniques and models, correct and repeatable marker
placement remains a major challenge in gait analysis (38).
1.6.2 Kinetics
The kinetics describe the joint forces and moments during movement. Kinetic data is most often
derived using kinematic data and data derived from force plates. The ground reaction forces,
starting at heeling strike and ending at toe offset, are measured using the force plates, built into
the walkway of the gait lab. Since the ground reaction forces result from the movement and force
generation of the entire body, they can be used to estimate kinetic data of the whole body,
based inverse dynamic calculations. When the kinetic data is combined with the kinematic data,
the joint forces and moments of separate body segments can be estimated (36).
1.6.3 Construction of the 3D-model
In 3D kinematic modelling, body segments are most commonly modelled as simplified rigid
1 The margin of error in locating the anatomical landmarks can be reduced using image techniques to determine the patient specific, individual geomitry of the hip joint (39). These techniques are not used in this study because of the exposure to radiation and the required labour-intensive processing of the data.
14
structures joined by joints.
Two methods exist to determine and digitalize the joint axes: the predictive and the functional
method.
1) Predictive method: markers are located on anatomically defined landmarks. The
landmarks are chosen to be repeatedly palpable, and not to give the best definition of the
joint axis. The joint centre and axis is estimated based on anthropometric data, derived
from empirical cadaveric data. This use of palpable bony landmarks makes this
technique more susceptible to the experience of the researcher than the functional
method (36,39).
2) Functional method: the functional joint centre and axes are defined based on movements
of the body segments relative to each other. The data of these movements is acquired
during a series of dynamic trials. Using this technique on the hip is challenging because
the hip joint can move in multiple planes. An important advantage is that it is less
susceptible to variation in marker placement (36,39). The functional method is
recommended by the International Society of Biomechanics (ISB) for subjects with
sufficient range of motion. It is especially preferred if an aberrant morphology of the joint
is expected (40). If the subject is not capable of carrying out the required movement
protocol, the functional method cannot be used (39,41).
IOWA Hip Score 1.7
The scoring system developed in 1963 by Carroll Larson, is still wildly used as a measurement
of clinical outcome. It separately scores aspects of function, gait, freedom from pain, absence of
deformity and range of motion. Disadvantages are the inter-observer variability and the fact that
the subject’s perception might be that the examined functional aspects are irrelevant (42).
15
2 Methodology
LCPD patients 2.1
The study population consists of 38 patients, treated with modified Shelf Acetabuloplasty in the
hospitals UZ Ghent and St.-Jan of Bruges. All patients were operated by the surgeon and
promoter Professor Dr. Plasschaert. Six patients are excluded based on co-existing pathology,
more precise a traumatic tetraplegia, osteomyelitis and other operations such as osteotomies,
leaving 32 patients. The exclusions are made to exclude factors, not related with Perthes
disease, but possibly interfering with the gait pattern. These patients were all diagnosed with
LCPD and operated between 2004 and 2014. Bilateral Perthes patients are not excluded in this
study, but are excluded from certain parts of the statistical analysis. Finally, four of the remaining
32 patients are excluded because the required X-rays are not available.
The residual 28 patients are divided in four quartiles, based on radiologic parameters for
coverage and congruence (cfr. 2.4 Radiologic evaluation of the LCPD patients). The most recent
follow-up X-ray photographs are used. No additional X-rays were made for this study (range in
time after operation: 1 – 102 months; mean: 36,73). All patients were observed on an
anteroposterior standing X-ray and a frog-leg Lauenstein X-ray. Based on the radiological
classification (cfr methodology: 2.4 Radiologic evaluation of the LCPD patients), the patients in
the best and worst outcome quartiles were included in the study2 (the mid 50% is excluded to
reduce confounding factors/noise). The best quartile will be referred to as the patients with good
outcome and the worst quartile will be referred to as the patients with bad outcome. Four
patients, two patients of the seven patients with good outcome and two patients of the seven
patients with bad outcome, choose not to participate in the study. Therefore, the two next
available patients with the best outcome out of the middle group are included to complete the
best quartile. The same is done with the two worse patients out of the middle group to complete
the worst quartile. The general characteristics of the both patient groups (i.e. age, height, weight
and leg length discrepancy) can be found in Table 2 (cfr Results: 3.1 General variables of the
groups). The final 14 patients are all inhabitants of Flanders.
2 One patient is included based on coverage and clinical evaluation. It is the worst patient out of 38 based on gait pattern and therefore this result does not affect the final result. It makes the result less reproducible but more realistic and the statistical tests more sensitive to differences between groups.
16
All patients went through both a clinical examination3 and a gait analysis. The Medical Ethical
committee of UZ Ghent approved this study and all patients signed an informed consent before
testing. Both examinations took place on the same day in the Lab of Gait analysis of the
University Hospital of Ghent; executed by the authors of this thesis, i.e. two students, second
master of medicine.
Controls 2.2In this study seven healthy controls participated. They are matched with the LCPD patients
based on age (mean: 14,7 years (SD: 3,93)). The general characteristics of the control group
(i.e. age, height, weight and leg length discrepancy) can be found in Table 2 (cfr Results: 3.1
General variables of the groups). All healthy controls are inhabitants of East- and West-
Flanders. The exclusion criteria are neurologic disorders affecting gait, pathology or operations
of the lower limbs and pelvis, inability to walk unaided and any disorder interfering with a normal
gait pattern. In the context of gait analysis, the healthy controls are recruited as reference group
to define the normal kinematics and kinetics in the frontal, sagittal and transverse plane. The
control group went through an identical clinical and gait examination as the patient group3. The
Medical Ethical Committee of UZ Ghent approved this study and the healthy controls signed an
informed consent before enrolment.
Experimental set-up 2.3
The modified Shelf Acetabuloplasty (mSA), as containment treatment for LCPD, is superior to
the classic Shelf Acetabuloplasty, regarding post-operative advantages and stability of the graft
(cfr.1.5.1 Containment therapy). The goal is to evaluate the functional outcome of the operation.
Therefore, a gait analysis is used (43). The patient group is divided in four quartiles based on
radiological parameters as will be explained below (cfr 2.4 Radiologic evaluation of the LCPD
patients). The outer quartiles define the patients with best and worst outcome. Both quartiles are
compared based on kinematics and kinetics in dynamic trials and standing balance in a gait
assessment. The control group defines the normal hip mobility, gait and balance. The hypothesis
of the study is that the effect of the mSA lays in the influence during the healing stage and not
the final result (measured on X-ray). Therefore, patients with the best and worst X-ray outcome
3 The anthropometric data required to construct the subject-specific kinematic model with the Vicon software, is gathered during the clinical exam prior to the gait analysis.
17
are compared. If the hypothesis is correct, then both patient groups should have acceptable gait4
and balance (based on the control group) after the mSA.
Radiologic evaluation of the LCPD patients 2.4
None of the classifications, described in the introduction (cfr 0 1.4 Radiological techniques and
classifications), are found to be applicable in the context of our study and for our study
population, as they need to be applied during the fragmentation phase of the disease or at
skeletal maturity (8). The follow-up time differed largely amongst the patients in this study and
largely exceeded the stage in which these classifications do apply.
The Mose classification is not used because of the limited discriminative possibilities for the
patients in this study. Therefore, another approach is chosen, which is explained below.
In order to determine the best and worst 25% of the patients, a ranking variable is created based
on two sets of quantitative variables. The first set of variables: the CEA (left/right), SA(left/right),
SR, FHSR and the FHCR (left/right) determines the coverage. The second set determines the
congruence of the hip. Only one variable, has proven to be sensitive to our patient group, the
SDS. Therefore, only the SDS is used to score congruence. The measurement protocol of each
parameter can be found in the appendix (cfr Appendix: 6.2 Radiographic parameters). The
coverage variables are measured using Autodesk® Graphic version 3.0.1. The SDS
measurements are made using GeoGebra 3.0.4.0.
Both coverage and congruence variables are equally important in the overall ranking system. All
variables are measured by two independent observers and compared based on the intraclass
correlation coefficient (ICC) (44). The ICC shows significance (P<0,001) for all variables. The
statistic calculations are performed using SPSS statistics version 23. Primary, the 5 coverage
variables are combined into one coverage variable. Subsequently a ranking variable is created
by combining the coverage variable and congruence variable.
2.4.1 Pre-processing
The coverage variable is based on the combination of five variables (cfr. 2.4 Radiologic
evaluation of the LCPD patients). The only patient with bilateral Perthes, was bilaterally
operated, and only the worst side is included in further analysis. All variables are continuous, but
differ in variance. In order to use the PCA (cfr. 2.4.2 Processing) it is preferable to normalise the 4 As defined by the control group.
18
variance (45). For this reason, the variables of the coverage and congruence are both
standardized by creating the z-scores (� � ����� ��� ��������
�����������������) (46), and rescaled to a
range between 0 and 1 (� ������ �!"#$#"�"�%
"�&#"�"�%.
2.4.2 Processing
Left with five coverage variables, there was a further dimension reduction based on the PCA.
Due to lack of an outcome variable or a variable describing each patient’s condition it is not
possible to use regression techniques to determine the weight for each variable. For this reason,
the PCA is used for both a dimension reduction and the determination of the weight of the PCA
components (45). The 5 variables can be reduced to 3 principle components with a 7% loss of
information by observer 1 and 11% by observer 2. Applying the K-means clustering technique it
is possible to subdivide the patients in quartiles (47). However, the scatterplot (Graph 1) shows
an important spread of the data. Consequently, K-means clustering will most likely not be able to
identify clearly separated clusters. Therefore, the components are combined to one coverage
variable as follows:
First, the three continuous components are categorized into 10 groups in order to enlarge the
differences. To each component a weight is ascribed based on the importance of the PCA
calculation5 assigned to the components (cfr. Table 1) (45)
('�()*+��,-�.�.+ ���/#�$0 �%%�2�3�4��5������
��/#�$0 �%%�2�3��������5������4��5������).
The weighted sum of the components constitute the coverage variable, i.e. coverage variable =
W1*C1 + W2*C2 + W3*C3 (Sum of the weighted Principal Components)6. Finally, the coverage
variable and the congruence variable (only consisting of the SDS) are add up to form the final
ranking variable.
5 The % of Variance is used as measure to define the importance given to each component by PCA. 6 Weight component 1 : 2,864; Weight component 2: 1,446; Weight component 3: 1 (set as the reference weight).
19
The patients are also evaluated by Professor Dr. Plasschaert based on his clinical expertise.
Eleven of the fifteen patients are classified in the same quartile, which is quite acceptable as, in
previous studies the interobserver reliability of the on-sight X-ray evaluation was found to be
poor (20).
Table 1 : Principal Component Analysis (PCA) Output: weigth Component 1: 2,864; weigth Component 2: 1,446; weigth Component 3: 1.
Graph 1: Scatterplot of the PCA components, based on the three used components
20
Gait Analysis 2.5
2.5.1 Technical specifications of the lab for motion analysis of the UZ Ghent
The Cerebral Palsy Reference Centre UZG houses a 24 x 16 megapixel camera motion capture
system (VICON Motion SystemsTM, Oxford, United Kingdom).
3D data is collected at 100 frames per second in this study, using the infrared motion capture
combined with force plate data derived from five force platforms (KistlerTM Instrument
Corporation, Amherst, New York, U.S.) located in the walkway. Conventional digital video
(movie) data, for the transverse, sagittal and coronal plane, is synchronously collected for each
trial.
Kinematic, kinetic and video data are collected and processed using Nexus® 2.6.1. (VICONTM
Motion Systems, Oxford, UK) and data presented using Polygon® 4.3.3 (VICONTM Motion
Systems, Oxford, UK)
The dimensions of the walkway in the gait lab are specified in the appendix (cfr. Appendix:
Figure 3).
2.5.2 Positioning of the markers:
Two versions of the Plug-in-Gait® model (VICON Motion Systems) are used in this study, i.e. for
the predictive method (PiG) and for the functional method (PiG-Matlab). The models and 3D
retroreflective markersets are based on the model described by Davis et al. (35). The standard
Plug-in-Gait® model (PiG) has been in clinical use in many labs for some years. A recent
revision written in Matlab (PiG-Matlab) takes advantage of new methods of analysis of specific
dynamic/functional trials to calculate the knee flexion/extension axes and hip joint centre
locations, using these in place of empirically derived hip joint centre and knee axis locations. The
methods for utilising the function joint and joint axis locations are implemented as a series of
Nexus plug-in routines connected as an “Advanced Gait Workflow” (AGW)
Markers are placed at the seventh cervical vertebra, tenth dorsal vertebra, right scapula,
posterior superior iliac spine, incisura jugularis, transition sternum-xyphoid, anterior superior iliac
spine, lateral collateral ligamentum of the knee, dorsum of the foot in line with the second
metatarsal bone, lateral malleolus and on the Achilles tendon at the level of the longitudinal axis
of the foot.
Markers are also positioned at the lateral side of the upper leg and lower leg (approximately
halfway) and on the ventral side of the upper leg, above and below the knee. In order to
21
minimise the influence of muscle contractions during gait, careful attention is payed during the
positioning of the markers (positioned as little as possible on the muscle belly). The marker
below the knee is positioned on the margo anterior of the tibia. Marker placement is illustrated
below in Figure 2. The subjects are asked to wear clothes not covering any of the markers at
any moment of the gait analysis. During the gait analysis subjects are barefoot.
Figure 2: Positioning of the markers conform the PiG-Matlab Model.
CLAV: clavicular (incisura jugularis); STERN: sternum; C7: cervical vertebral 7; D10: thoracal vertebral 10; RBAK: back of the right scapula; RASI/LASI: right and left anterior superior iliac spine; RPSI/LPSI: right and left posterior superior iliac spine; RTHI/LTHI: right and left tigh; RTHIA/LTHIA: right and left tigh additional marker; RKNEE/LKNEE: right and left knee; RTIB/LTIB: right and left tibia; RTIBA/LTIBA: right and left tibia additional marker; RANK/LANK: right and left ankle; RTOE/LTOE: right and left toe; RHE/LHE: right and left heel.
22
2.5.3 Sequence of the gait analysis
2.5.3.1 Calibration
Calibration is carried out at the start of each gait analysis.
2.5.3.2 Static analysis
Knee Alignment Devices (KAD’s)(© Motion Lab Systems, Baton Rouge, USA) (48) are used
during a standing static trial in PiG. When a KAD is used, the flexion-axis of the knee is
calculated using the VICON Clinical Manager Software application of Oxford Metrics Ltd. (VCM),
applying the empirical predictive method (cfr 1.5.3 Construction of the 3D-model). Once the axes
are calculated, the KAD is removed and medial knee markers are placed.
2.5.3.3 Advanced Gait Workflow (AGW) lower body
Following the PiG Static Trial, the subjects go through the AGW lower body protocol. This
protocol is used to recalculate the hip and knee axes, using the functional method (cfr. 1.6.3
Construction of the 3D-model). During the protocol the subject is asked to perform a combined
movement of the hip: pure anteflexion, anteflexion-abduction, pure abduction, abduction-
retroflexion and pure retroflexion. To determine the knee axis, the subject is asked to perform
about seven flexions of the knee. For the hip movement as well as for the knee flexions, the
subjects are asked to return to the anatomical position after each movement, before going
through with the subsequent movement. The combined movements of the hip and the flexions of
the knee are carried out once for each leg and the subjects are asked to carry them out in a way
that is comfortable (not forcing themselves to exceed their usual range of motion.)
2.5.3.4 Dynamic analysis
After estimating the joint axes for the lower limbs by means of the static analysis and performing
the AGW lower body protocol, the data required to construct the kinematic model is collected
and the dynamic analysis is executed. The subject is asked to walk on the walkway, from one
end to the other (following the longitudinal axis; cfr. Figure 3), at a self-selected comfortable
walking speed, in a straight line. For each subject at least 15 trials are collected to make sure at
least five runs can be withheld, that include a correct foot-force plate contact for both the left and
right foot. A correct foot-force plate contact is defined as an isolated foot contact on the force
plate, within the borders of the force plate, and during a natural, comfortable walk (a hesitant
contact should be avoided). Attention is payed to the fatigue level of the subject.
23
2.5.3.5 Balance analysis
The balance capabilities of the subject are assessed during unipodal stance. The subject is
asked to hold the unipodal position for 30 seconds, eyes closed and standing on a single force
plate. The test is performed whilst standing on each leg in turn.
2.5.4 Data processing – gait analysis
Standard reconstruction and trajectory labelling techniques are used to process the 3D data
before running the two modelling procedures for PiG and PiG-Matlab.
IOWA Hip Score 2.6In this study, the decision is made to reduce the IOWA hip score to the parameters 'pain free'
and 'function' (cfr. Appendix: 6.3 IOWA Hip Score (chart)) . The score is based on the subjective
experience of the patient and given by the researcher after questioning the patient. The score is
given by the researcher to exclude the risk of incorrect interpretation of the scale by the patient
(e.g. one of the patients wanted to give himself a score of 35, the maximum, for 'pain free', but
after further questioning by the researcher, the patient declared knee pain was present after
physical exercise. Knee pain can originate from the hip and the patient was therefore not given
the maximum score by the researcher (49).
Data Analysis 2.7During the gait assessment 21 outcome parameters of the hip are measured for both affected
and unaffected side in the two patient groups and the control group. The 21 parameters consist
of 18 dynamic parameters (11 kinematic and seven kinetic parameters), step time and two
standing balance variables. All parameters are derived from either the PiG model or the PiG-
Matlab model.
2.7.1 Kinematics
2.7.1.1 Sagittal Plane
(1) Hip Maximum Flexion(°): is calculated as the maximum of the hip flexion during gait at
comfortable speed.
(2) Hip Maximum Extension(°): is calculated as the maximum of the hip extension during gait
at comfortable speed.
(3) Hip Range of Motion(°): the hip range of motion is calculated as the difference between
24
maximal hip flexion and maximal hip extension. It describes the range of motion in the
sagittal plane during a comfortable gait speed.
(4) Anterior Pelvic tilt(°): is calculated as the maximum of the pelvic tilt during gait at
comfortable speed.
2.7.1.2 Frontal Plane
(5) Hip Maximum Abduction(°): is calculated as the maximum hip abduction during gait at
comfortable speed.
(6) Knee Maximum Varus(°): is calculated as the maximum knee abduction during gait at
comfortable speed.
(7) Knee Maximum Valgus(°): is calculated as the minimum knee abduction during gait at
comfortable speed.
(8) Knee Mean Varus(°): is calculated as the mean knee abduction during gait at comfortable
speed.
(9) Pelvic Minimum Obliquity(°): is calculated as the minimum pelvic obliquity during gait at
comfortable speed.
2.7.1.3 Transverse Plane
(10) Hip Maximum Internal Rotation(°): is the maximum internal rotation during gait at
comfortable speed.
(11) Hip Maximum External Rotation(°): is the maximum external rotation during gait at
comfortable speed.
2.7.2 Kinetics
2.7.2.1 Sagittal Plane
(12) Hip Extension Moment First Peak(Nm/kg): Is calculated as the first extension moment
peak during a cycle, meaning the maximum in the first 60% of a cycle, during gait at
comfortable speed.
(13) Hip Maximum Flexion Moment (Nm/kg): is the maximum of the flexion moment during
gait at comfortable speed.
2.7.2.2 Frontal Plane
(14) Hip Maximum Abduction Moment (Nm/kg): is calculated as the maximum of the hip
25
abduction moment during gait at comfortable speed.
(15) Hip Mean Abduction Moment (Nm/kg): the mean abduction moment is the mean of all
frames in a cycle during gait at comfortable speed.
(16) Knee varus moment (Nm/kg): is the maximal varus moment during gait at comfortable
speed.
2.7.2.3 Transverse Plane
(17) Hip Maximum Internal Rotation Moment (Nm/kg): is the maximum internal rotation
moment during gait at comfortable speed.
(18) Hip Maximum External Rotation Moment (Nm/kg): is the maximum external rotation
moment during gait at comfortable speed.
2.7.2.4 General
(19) Step Time (s): is defined as the amount of time spent during single step. It is the time
between heel strike of one side and heel strike of the contra-lateral side.
(20) Balance Path Length (mm): is the total distance (sum of all separate paths) covered
during a balance test of 30s.
(21) Balance Path Velocity (mm/s): is defined as the ratio of Path Length to Balance time.
Balance time is defined as the time a subject could hold the demanded balance position.
The maximum demanded time was 30 seconds.
26
Statistical Analysis 2.8The statistical analysis consists of five parts:
1) The possible presence of confounding factors is checked.
2) A comparison is made between the results obtained from the Plug-in-Gait (PiG) and
Matlab (PiG-Matlab) models, in order to determine which model should be used.
3) A comparison is made between the gait and balance data from the healthy controls, the
good outcome patients and the bad outcome patients using a group comparison.
4) The unaffected side and affected sides are compared within each group.
5) The correlation between IOWA function score and IOWA pain score on the one hand and
the dynamic variables on the other hand is tested.
The significance level is set on α < 0,05. The SPSS Software package version 23 is used.
2.8.1 Comparison of the PIG and the functional (PiG-Matlab) model
In the data collection process the data for both gait models (PiG and PiG-Matlab) are
simultaneously measured. Therefore, a perfect pairwise comparison of the exact same trial,
cycle and subject is possible, excluding the interference of any confounding factor. The only
difference in the outcome is the difference in the processing of the data and the identification of
the correct joint axis. In the comparison of the models a pairwise analysis is performed for all
variables and the affected and unaffected side are tested separately. The comparison is done by
performing the Wilcoxon test or Sign test (asympt. Sign. (2-tailed)), depending on the
symmetricality of the data distribution.
2.8.2 Group comparison
The comparison between the control group, the patient group with good outcome and the patient
group with bad outcome, is both carried out using the data of the separated trials7 as well as for
the means8 of the trials per subject.
The normality of the distribution of each variable is analysed by means of the Shapiro-Wilkinson
test. The Shapiro-Wilkinson test is used because the test is considered to be more robust than
the Kolmogorov-Smirnov test for small sample sizes, which applies here. The normality is tested
7 The separate trials are only tested for the PiG Model. The reason is explained later in 3.2 Gait and balance variables comparison. The means are tested in both models and compared. 8 The means are calculated based on at least seven trials for each patient.
27
for the population as a whole and for the groups separately. The null hypothesis states that the
data has a normal distribution, if a not-significant result is found, the null hypothesis is not
rejected. Further testing is done by performing the Levene’s test. The Levene’s test analyses the
homogeneity of variances. This protocol is also used for the side comparison and the correlation
testing.
The group comparison between all three groups is tested by means of the non-parametric
Kruskall-Wallis test (asympt. Sign. (2-tailed)) for continuous variables. For each variable, all four
assumptions9 of the Kruskal-Wallis test are fit. The null hypothesis states that all samples are
originate from the same population. The Mann-Whitney U-test is performed to compare the
scores of the two patient groups for IOWA function score. The Fisher’s exact test is used for the
categorical variable IOWA pain score (the conditions of the chi-square test are not met (50)). If
needed, the Mann-Whitney U-test is used as the post-hoc test for the continuous variables, to
determine between which groups the significant difference is located (α < 0,015 after Bonferroni
Adjustment for three samples10. A P-value between 0,015 and 0,05 is accepted as a possible
trend.) The comparison of the affected and unaffected side is done separately. For the healthy
control group both sides are unaffected. The ‘unaffected’ side of the bilateral affected patient,
classified in the bad outcome patient group, is excluded from the group comparison of the
unaffected sides. Both sides are excluded from the side comparison, since the 'unaffected' side
cannot act as a healthy control.
To check for possible confounding variables a comparison of the groups is performed for the
general variables: Age, Length, Weight, Leg length discrepancy, Follow-up time since shelf and
Age at time of operation.
2.8.3 Side comparison
The side difference of the affected and unaffected side of the patients (the affected and
unaffected side are considered to be paired) is assessed by the Wilcoxon test or Sign test
(asympt. Sign. (2-tailed)), depending on the symmetricality of the data distribution. The
symmetricality is assessed using a boxplot plotting the distribution of the differences between
9 The assumptions for the Kruskal-Wallis test are: 1) k > 2 (k: independent samples), 2) continuous or ordinal variable, 3) not parametric continuous variable; 4) number of each sample is at least 5. 10 Bonferroni adjustment is the significance level (α) divided by the number of possible paired tests (N) between samples, N is based on the number of sample means (n).
6 � 789:� ; <�� � �
= > �
$�$�?%
@
28
the affected and unaffected side. Only the means of the trials for each subject are used in further
analysis. The means are based on the average of 7 to 10 trials for each patient.
2.8.4 Correlation IOWA – gait variables
In order to explore correlations between the IOWA function score and IOWA pain score on one
hand and the gait variables (kinematics, kinetics, Step Time and balance variables) the
Spearman rank coefficient is used. The conditions for the Pearson correlation coefficient11 are
not met for the continuous variable IOWA function score and the ordinal variable, IOWA pain
score.
11 Conditions for the Pearson correlation coefficient: 1) the variables need to be continuous and 2) have a normal distribution (50).
29
3 Results
General variables of the groups 3.1When comparing the general variables (Age, Length, Weight, Leg length discrepancy, Follow-up
time since mSA and Age at time of operation) of the healthy controls, the good outcome patients
and the bad outcome patients, no significant differences are found. Consequently, no
confounding variables are identified (cfr. Table 2).
Gait and balance variables comparison 3.2In the dynamic trials eleven kinematic and seven kinetic variables are statistically analysed
based on the averages of the trials (cfr. 2.8.2 Group comparison). Primary the three groups are
overall tested for both kinematic and kinetic variables. Followed by a comparison between the
affected and unaffected side within each patient. Both the PiG-Matlab model and the PiG model
are statistically analysed using the same tests, therefore a comparison between both models is
possible. In the last phase of the analysis, differences between both models are tested.
When the separate trials (not the averages) are compared between the sides almost every
variable seems to be significantly different for the patient groups and even in the control group
six variables are significant (Hip Maximum Extension, Hip Range of Motion, Hip Maximum
Abduction, Hip Maximum Flexion Moment, Hip Maximum Abduction Moment, Hip Mean
Abduction Moment). In order to reduce confounding factors and compensate for the variance
between both sides within the same trial, the averages of the separate trials of each patient are
used. After this analysis a more clinical relevant result is obtained.
In order not to overload the results section, only the PiG-Matlab analysis is presented, which
Table 2: overview general variables study population (SD = standard deviation) (* Follow-up time is the time elapsed since mSA operation)
30
seems after further analysis the most superior model for this study. The reasoning for this
superiority is presented and explained in the discussion (cfr. 3.3 Differences between both gait
analysis models). This form of presenting the results is in no way to reduce the quality of the
study, but to bring brevity and structure to the results. The interested reader is referred to the
Appendix, in which all PiG data and analysis is presented (cfr. Appendix: 6.4 Results PiG Model)
3.2.1 Group comparison PiG-Matlab Model
3.2.1.1 Comparison of the unaffected sides
The comparison of the unaffected sides, between the three groups in the PiG-Matlab model,
based on the Kruskal-Wallis test, shows no significance for 15 variables (0,063 < P-value <
0,961). For Hip Maximum Extension, Hip Maximum Abduction and Anterior Pelvic Tilt a
significant difference between the groups is detected (0,015 < P-value < 0,038).
The significant variables indicate a difference between the three groups. Therefore the
unaffected side of the patients can only be used for the not-significant variables as the ‘perfectly
matched healthy control’ in the side by side comparison (cfr 3.2.2 Side comparison PiG-Matlab
Model). After performing the post hoc Mann-Whitney U-test in the PiG-Matlab model, no
significant differences are found for Hip Maximum Abduction and Anterior Pelvic Tilt. The
variable Hip Maximum Extension shows a significant difference in the comparison of the control
group and the bad outcome patients (P-value: 0,014) and a trend is detected after post hoc
comparison of the good and bad outcome patients (P-value: 0,022) (Graph 2). A trend is
detected for Hip Maximum Abduction between the controls and the bad outcome patients (P-
value: 0,022) (Graph 3). The variable Anterior pelvic Tilt shows a trend between the controls and
the bad outcome patients (P-value: 0,022) (Graph 4).
No significant differences are detected between the control group and the good outcome
patients (0,073 < P-value < 0,318).
31
3.2.1.2 Comparison of the affected sides
In the PiG-Matlab model, the variables Hip Maximum Extension (Graph 2), Anterior Pelvic Tilt
(Graph 4) and Hip Range of Motion (Graph 5) are shown to be significantly different when
Graph 3: Box plot of Hip Maximum Abduction (º); Grey : affected side, Black : unaffected side; separately the controls (n=7), good outcome patients (n=7) and the bad outcome patients (n=6). The dot represents an outlier (number = patient number). M = median.
M: 8,2
M: 12,1
M: 10,7
M: 8,0
M: 10,7
M: 5,3
Graph 2: Box plot of Hip Maximum Extension (º); Grey : affected side, Black : unaffected side; separately the controls (n=7), good outcome patients (n=7) and bad outcome patients (n=6). The dot represents an outlier (number = patient number). M = median.
M: -9,5 M: -9,3
M: -3,6
M: -6,3
M: 10,9
M: -1,3
Graph 4: Box plot of Anterior Pelvic Tilt (º); Grey : affected side, Black : unaffected side; separately the controls (n=7), good outcome patients (n=7) and bad outcome patients (n=6). The dots represent outliers (numbers = patient numbers). M = median.
M: 11,6 M: 11,2
M: 15,1 M: 15,2
M: 25, 4 M: 25,7
32
comparing the three groups (0,004 < P-value < 0,025). For the other variables no significant
differences are found (0,198 < P-value < 0,872). Further exploration of group differences,
applying Mann-Whitney U-tests, reveals the following results: significant differences between the
controls and the bad outcome patients for the variables Hip Maximum Extension, Hip Range of
Motion and Anterior Pelvic Tilt (0,001 < P-value ≤ 0,011). No significant result is detected when
comparing the healthy controls and the good outcome patients, however a trend is found for Hip
Range of Motion (P-value: 0,038). When comparing both patient groups, a trend is found for Hip
Maximum Extension (P-value: 0,026).
3.2.1.3 Step Time
No significant difference between the groups is found for this variable, both for the unaffected (p-
value: 0,581) and for the affected side (p-value: 0,71).
3.2.1.4 Balance standing
The group comparison reveals that the groups do not differ statistically when comparing the
unaffected and affected sides for the variables Balance Path Velocity and Balance Path Length
(P-values resp. 0,838 (unaff. side), 0,233 (aff. side) and 0,078 (unaff. side), 0,306 (aff. side)).
3.2.2 Side comparison PiG-Matlab Model
The affected and unaffected side are pairwise compared within the groups, using the average
values.
Graph 5: Box plot of Hip Range of Motion (º); Grey : affected side, Black : unaffected side; separately the controls (n=7), good outcome patients (n=7) and bad outcome patients (n=6). The dot represents an outlier (number = patient number). M = median.
M: 47,6 M: 46,6
M: 40,8
M: 46,8
M: 37,9
M: 43,0
33
3.2.2.1 Control group
Within the control group, no significant difference is shown based on the PiG-Matlab model
(0,125 < P-value < 1,000). Step Time and the Balance Variables are also not significant (0,453 <
P-value < 1,000).
The null hypothesis indicates no differences between the left and right sides of the control group.
3.2.2.2 Good outcome patients
The PiG-Matlab model shows no significant results for all variables (0,125 < P-value < 1,000).
No significant difference is found for Step Time (P-value 0,219) and the balance variables (0,375
< P-values < 1,000).
3.2.2.3 Bad outcome patients
Three variables are significant as indicated by the PiG-Matlab model: Hip Maximum Flexion
(Graph 6), Hip Range of Motion (Graph 5) and Hip Maximum External Rotation Moment (P-
values: 0,031) (Graph 7).
As in the control group and the good outcome patients no significant difference is found for Step
Time (P-value: 1,000) and the balance variables (P-values: 0,687).
Graph 7: Box plot of Hip Maximum External Rotation Moment (Nm/kg); Grey : affected side, Black : unaffected side; separately the controls (n=7), good outcome patients (n=7) and bad outcome patients (n=6). The dots represent outliers (numbers = patient numbers). M = median.
M: -0,10 M: -0,10 M: -0,08
M: -0,11 M: -0,06
M: -0,12
Graph 6: Box plot of Hip Maximum Flexion (º); Grey : affected side, Black : unaffected side; separately the controls (n=7), good outcome patients (n=7) and bad outcome patients (n=6). The dot represents an outlier (number = patient number). M = median.
M: 39,1 M: 38,7
M: 39,5 M: 40,2
M: 44,2
M: 49,2
34
3.2.3 Differences between both gait analysis models
For the hip variables significant results are shown for Hip Maximum Flexion (aff. and unaff. side)
Graph 10: Box plot of Hip Maximum External Rotation(º) ; Grey : affected side, Black : unaffected side; separately the controls (n=7), good outcome patients (n=7) and bad outcome patients (n=6). The dot represents an outlier (number = patient number).M = median.
M: -12,8 M: -9,0
M: -7,3 M: -7,9
M: -13,8 M: -17,9
Graph 9: Box plot of Hip Mean Abduction Moment (Nm/kg); Grey : affected side, Black : unaffected side; separately the controls (n=7), good outcome patients (n=7) and bad outcome patients (n=6). The dot represents an outlier (number = patient number). M = median.
M: 0,30
M: 0,42
M: 0,34 M: 0,38 M: 0,39
M: 0, 43
M: 0,59
M: 0,77
M: 0,70 M: 0,71 M: 0,69
M: 0,78
Graph 8: Box plot of Hip Maximum Abduction Moment (Nm/kg); Grey : affected side, Black : unaffected side; separately the controls (n=7), good outcome patients (n=7) and bad outcome patients (n=6). The dot represents an outlier (number = patient number). M = median.
35
(P-value: 0,006), Hip Maximum Abduction (aff. and unaff. side) (P-values < 0,001), Hip
Maximum Internal Rotation (aff. and unaff. side) (P-value < 0,001). All knee kinematic variables
in the frontal plane show a highly significant result (0,001 < P-value < 0,005).
IOWA Hip Score 3.3
3.3.1 Comparison of the patient groups
No significant difference between the patients with good and bad outcome is found for IOWA
function score (P-value: 0,784) and IOWA pain score (P-value: 1,000) (Graph 11).
Note that none of the patients were given a point for ability to drive, because none of them had
acquired a driver’s licence yet. The data for one of the patients within the group with good
outcome, is missing.
3.3.2 Correlations IOWA – gait variables
If the correlation is calculated including all patients, a significant correlation is found between
IOWA function score and Balance Path Velocity of the unaffected side (r =0,678) and between
the Balance Path Length of the affected side (r = -0,588).
When the correlation is calculated, including only the patients with good outcome, a significant
correlation is found for IOWA function score and Hip Maximum Flexion of the affected side (r
=0,788).
Graph 11: bar chart IOWA pain score (/35pt); Grey: patients with good outcome (n=6), Black: patients with bad outcome (n= 7).
36
When considering the patients with bad outcome, significant correlations are found for IOWA
function score and Hip Maximum Flexion of the affected side (r =0,805), IOWA function score
and Hip Maximum Internal Rotation of the affected side (r =-0,823), IOWA function score and
Maximum and Mean Varus of the knee of the affected side (r for both = -0,842), IOWA pain
score and Pelvic drop at the affected side (r = -0,917), IOWA function score and Hip Extension
Moment First Peak at the affected side (r =0,805), IOWA function score and Hip Maximum
Internal Rotation Moment at the affected side (r =0,805) and for IOWA function score and
Balance Path Length of the affected side (r =0,823).
37
4 Discussion
The research question 4.1Until today there is no uniformly accepted treatment protocol for Legg-Calvé-Perthes disease
(LCPD). The different stages of the disease have been defined, but the etiological background
remains not fully understood. Treatment has therefore been based more on an empirical basis
and historical series. Most of current management however is based on the ‘principle of
containment’ (4).
So far, there is a lack of full understanding on how specific treatment types for LCPD can lead to
an optimal (long-term) clinical outcome. Treatment is usually guided by clinical examination and
radiological follow-up, focusing on maintaining a spherical femoral head in the end stages of the
disease (43).
Studies focusing on the functional outcome of surgical treatment for Perthes are very sparse
(43). Our research aims to fill this void. Furthermore, this series studied is unique since it
focuses on a novel surgical technique alleviating the use of cumbersome hip spica casts (32).
Our thesis therefore focuses on the functional (more than radiological) outcome of modified
Shelf Acetabuloplasty (mSA) in the treatment of LCPD by collecting objective data, derived from
gait analysis.
Discussion of the results 4.2
4.2.1 Gait model
In this gait analysis study, two models are simultaneously applied, (a) the standard Plug-in-Gait
model and (b) a functional version of the Plug-in-Gait model (based on Davis’s model) (35). Both
models are compared, in order to allow for a functional evaluation of the modified Shelf
Acetabuloplasty, based on the clinically most valid model. In the PiG model, joint centres are
empirically estimated based on cadaveric studies (39). However, caution is needed when
applying the model to LCPD patients. LCPD causes an extrusion of the femoral head and
therefore leads to a more lateral position of the hip joint centre (51). Therefore, the empirical
based location of the hip joint centre should be questioned. For this reason, within this study
both models (a and b) are applied and derived measurement results compared.
In the comparison of these models, first the knee variables: Maximum Varus, Maximum Valgus
38
and Mean Varus are compared as a quality measure for the Knee Alignment Device (KAD)
placement. All three variables are shown to be significantly different (0,001 < P-value < 0,006) in
an overall comparison between both models, including all subjects. In order to determine the
clinical meaning of this significant difference, the control group is used to compare the absolute
results. A threshold of 5° is set to define clinically relevant differences between both models12.
The differences found for the knee variables, do vary from 6,8° to 18,0° (cfr. Table 5
(Appendix)). As the differences between the two models exceed the threshold, they are
considered to be clinically relevant. Based on these findings one should question the placement
of the KAD and its benefit when studying (paediatric) patients with LCPD.
Secondly, the variables important to this study are compared: Hip Maximum Flexion (P-value:
0,004) (-2,5°), Hip Maximum Abduction (P-value < 0,001) (7,7°) and Hip Internal Rotation (P-
value < 0,001) (14,2°) are shown to be significantly different (cfr. Appendix: Table 5). Note that
Hip Maximum Flexion is statistically significant, but considered not to be clinically relevant,
because of the threshold set at 5°.
In the further report of this study the functional model, i.e. PiG-Matlab, will be used. This decision
is based on the following:
- The aberrant hip joint centre location in LCPD (due to extrusion of the hip and femoral
head deformation (51), using the functional model, is defined based on the functional
joint centre and not an empirical estimation thereof.
- Using the PiG model, varus angles are observed exceeding 17º, for the healthy subjects.
This does not correspond with the physiologic values during gait in healthy subjects. The
aberrant varus angles, make us question the accuracy of the KAD.
- In contrast with other diseases often studied by gait analysis, such as cerebral palsy, the
LCPD patients included in this study do generate sufficient range of motion and muscle
power in hip and knee. This enables one to calculate the hip and knee axis based on
joint motion, and alleviates the use of the KAD.
- The gait lab, used in this study, does provide with sufficiently accurate and sensitive
equipment to apply the functional model (PiG-Matlab) correctly (52).
12 McGinley et al. suggested that errors in excess of 5° should raise concern and may be large enough to mislead clinical interpretation (38).
39
4.2.2 Overall group comparison13
Westhoff et al. did describe gait pattern differences in LCPD patients (during the florid stage of
the disease) when compared to a control group.
The affected side showed an increased anterior pelvic tilt, decreased range of motion (ROM)
and a decreased extension, both of the hip. In the unaffected side an increased ROM was
observed, due to increased maximal hip flexion (53).
At the contrary, a study of Stief et al. observed a normalisation of hip flexion and extension after
containment therapy (i.e. femoral varus osteotomy combined with a Salter osteotomy) (54).
The results observed within our study do support the findings of Westhoff et al (53). In the
overall group comparison, both the affected and unaffected side show a significantly increased
Anterior Pelvic Tilt. After post hoc analysis, a significant difference for the affected side (+9,2° )
and a trend for the unaffected side (+9,6° ) were shown to exist between the controls and the
ones classified as ‘bad outcome patients’.
When compared to the control group, a significant reduction of Hip ROM (-11,8°) was detected
for the affected side of the patients with bad outcome. This is in agreement with Whesthoff et al.
(55).
Our results do show a reduced Hip ROM, due to a significant reduction of Hip Maximum
Extension of the affected side (-15,3°). The Hip Maximum Extension of the bad outcome patients
is borderline-missed significantly reduced (-9,3° ) when compared with the good outcome
patients.
Although no firm conclusions can be made, there seems to be a gradient in the degree of Hip
ROM reduction of the affected side, dependent on the radiographic outcome of LCPD (Graph 5).
Westhoff et al did hypothesize that the decreased Hip ROM and Hip Maximum Extension in the
affected hip in LCPD are part of (developed) strategies:
- Hip ROM is reduced in order to lessen hip joint loading.
- The decrease in Hip Extension prevents the intra-articular pressure from peaking.
- An aberrant gait pattern, primarily caused by the LCPD, becomes permanent due to soft
13 Differences between both legs of the controls should be taken into account if there are greater than 1°, and therefore, any difference greater than 1° shall be reported.
40
tissue changes at the ventral side of the hip. Even if the primary cause is resolved (55).
According to Whesthoff’s findings, a reduction of Hip Maximum Extension, should lead to a
smaller step length, if not compensated by an increased Hip ROM of the unaffected hip and
increased Anterior Pelvic Tilt (55). In our study however, neither an increase nor decrease in Hip
ROM could be detected for the unaffected side. We can therefore only partially support the
findings of Westhoff et al.
Hip abduction is almost always affected in LCPD. Hip abductor muscle weakness has been
reported as causal factor. The phenomenon of hip abductor weakness has been confirmed by
Westhoff et al. (53). It is often stated as one of the primary mobility problems in LCPD and is
often accompanied by the presence of adductor tightness (and Trendelenburg gait) (24,50,52–
55). In the overall group comparison significant differences are found for Hip Maximum
Abduction, only on the unaffected side. After post hoc evaluation no significant result can be
withheld. However, a trend of reduction could be detected for the bad outcome patients (-6,9°)
(Graph 3). The reduction of Hip Maximum Abduction of the unaffected side seems clinically
relevant (> 5° difference) (cfr. Table 5). This observation is not supported by the results for
Minimum Pelvic Obliquity, Standing Balance and Hip Maximum Abduction Moment and current
literature (53,54). For the three indirect abduction measures, i.e. Minimum Pelvic Obliquity,
Standing Balance and Hip Maximum Abduction Moment, no significant differences, nor trends,
are found for the unaffected side. The findings for the indirect abduction measures are confirmed
by the study of Stief et al. In their study a resolution of differences in Pelvic Obliquity, through the
healing process, is observed (54). Regarding the affected side, of subjects included in this study,
differences in Hip Maximum Abduction, between the controls and both the good and bad
outcome patients, are not significant. The reduction varies between <0,1° and 2,4°. This
indicates a clear absence of reduction for the affected side.
When discussing all those data one should however realise that one of the objectives of the
mSA is to maintain containment of the hip affected by LCPD (32). In order to meet this
prerequisite, the maintance of (passive) abduction of the hip needs to be seen as the goal of
treatment (in order to maintain the shape of the femoral head in the final stage of LCPD) (53). A
successful containment treatment should restore abduction of the hip in those who tend to lose it
(before the femoral head collapses and loses height of the lateral pillar) (56).
The results for the group comparison of the kinetic abduction variables support our statement
that the hip abduction is not clinically relevant affected. More importantly, these results suggest
41
that the novel surgical technique (mSA) does indeed fulfill its objective.
The comparison of the three groups and the side comparison within the groups did not show any
significant differences for Hip Maximum Abduction Moment and Hip Mean Abduction Moment.
Our findings are somewhat in line with the findings of Plasschaert et al. In their study a
significant reduction is found for maximum abduction moment, when comparing the affected side
to the unaffected side, in a patient group with poor clinical outcome (defined by an IOWA hip
score lower than 90 points) (57). In our study this reduction can also be seen in the bad outcome
patient group (Max Abduction Moment unaffected side: 0,81 Nm/kg; Max Abduction Moment
affected side 0,62 Nm/kg), but it was a non-significant and probably clinically irrelevant
reduction. This does indicate that the patients in our study do not suffer from decreased
abduction capacity at either side, at least in the short-term. It also supports the objective of the
surgical technique not to add any additional trauma to the hip abductors that could weaken them
(32).
Furthermore, we cannot compare to the study of Plasschaert et al. without paying attention to
two important differences between our study and the study of Plasschaert et al. Note that in the
study of Plasschaert et al. the hip abduction was measured in a different way, using a
dynamometer, and that the classification of patients is based on a different method. Plasschaert
et al. used a clinical classification, based on the IOWA hip score, while in our study a radiological
classification is used (57).
Our finding that there is an absence of differences regarding abduction moments, could suggest
that there is no difference in the loading of the hip joint, and that at least in the short-term mSA
has been successful in changing the natural history for those hips, where negative prognosis
was criterium for surgery.
Our findings supporting absence of abductor weakness are in line also with the non-significant
results with respect to the balance variables. The abductor muscles are of great importance in
maintaining good balance (58). In our study balance seems not to be affected, again supporting
a successful procedure in protecting the hip and maintaining its biomechanics.
In our study, no statistical differences are found when comparing the IOWA function and IOWA
pain scores of the good outcome patient group and the bad outcome patient group. The
subjective functional outcome of all patients is quite good (max 32 and min 28, scored on a scale
42
of 35 points). One of the patients is even an elite swimmer. Furthermore, the IOWA scores
makes all our patients lose at least one point, because they cannot drive a car due to age
restriction. None of the patients had already acquired their driver’s license. Pain experience in
our patients ranges from “pain free” to “pain at rest without weight-bearing”.
The relatively good scores for function and pain could support our earlier assumption that
acceptable abduction function is present in all patients included in the study population. It needs
to be noted that no significant correlation is found between the abduction variables and IOWA
function score and pain score.
Svehlik et al. described two abnormal gait patterns in LCPD. The first gait pattern is a Duchenne
gait, presenting a pelvic elevation on the swing side, and increased abduction and external
rotation during stance phase, in order to decrease the hip loading (56). Increased external
rotation is a compensation mechanism to prevent impingement, due to an anterolateral hump.
Such a hump is often present in LCPD patients and was described by Yoo et al. (59). No
statements can be made about possible Duchenne gait patterns in the patient groups, because
of the absence of processed trunk data.
A second gait pattern is the Trendelenburg gait pattern, defined by a pelvic drop to the swing
side. In our study a statistically significant difference in external rotation of the hip, between the
three groups cannot be observed. However, the difference is degrees seems clinically relevant.
Remarkably, the smallest external rotation is observed in the affected side of the patients with
good outcome (cfr. Table 5). The good outcome patient group is also the group in which the
largest internal rotations are observed for the affected side. The phenomenon of the decreased
external rotation of the affected side of the good patients, remains unclear. No assertions were
found to state this observation. The absence of an abnormal gait pattern, pelvic abnormalities
and step time could not lead to a causal or suggesting factor for this aberrance. However, the
general trend of increase in external rotation for both affected and unaffected side, in relation to
the radiological outcome of the disease could be in agreement with Svehlik et al.(56). Further
analysis of causality should bring more clarity on the matter.
The absence of differences for Pelvic Obliquity, Hip Maximum Abduction Moments and Standing
Balance do support the assumption that involvement of hip abduction is most likely not clinically
relevant. Pelvic obliquity is an indicator for a Trendelenburg gait pattern. Absence of this gait
pattern is observed in both good and bad outcome patients. The absence of a Trendelenburg
gait pattern results in normal hip loading and does result in the fact that the hip coverage during
43
gait is not compromised (53).
4.2.3 Side by side comparison
Differences between both sides in the control group are analysed as a quality measure, as both
legs are assumed to be normal. As expected, neither significantly, nor clinically relevant
differences could be observed between both sides of the control group.
The patients with good outcome show no side differences.
In the bad outcome patients, significant Hip ROM reductions (-10,7º) are detected together with
a significantly reduced Hip Maximum Flexion (-5,0º). Note the differences in degree of reduction
(-5,8º) between the good and bad outcome group for the Hip ROM (cfr. Table 5). The Hip
Maximum Extension is not analysed for the patients with bad outcome, because the unaffected
side is statistically different from the control group and can therefore not be seen as a matched
healthy control side to compare with the affected side. Therefore, no evaluation can be made
between both sides for the extension and the conclusion for the Hip Max Extension should be
derived from the analysis of the overall group comparison.
Regarding Hip External Rotation Moment, significant side differences in the bad outcome
patients are detected (0,05 Nm/kg). An explanation, based on the findings of the other variables
analysed in the present study, is not found. With respect to external rotation the increase thereof
has been described by Svehlik et al and Yoo et al. (56,59). As no significant results are found for
external rotation in this study, it’s unclear how the anterolateral hump could be an explanation for
the decrease of external rotation moment.
To our knowledge, no articles have yet reported findings for internal and external rotation
moments in LCPD patients or other hip deformities. Further research is needed to explore if hip
rotation moments in LCPD patients are consistently different from healthy controls and to
explore the role of hip rotation moments in the evolution of osteoarthritis in LCPD patients.
In conclusion, the good outcome patients show no compromised gait pattern. This indicates that
the outcome after mSA is an almost perfect gait pattern recovery for the good outcome patients.
In the comparison of both patient groups, a gradient in abnormal gait could be detected,
indicating not all outcomes are superb. The abnormalities observed in the bad outcome patient
44
group are mostly due to aberrant hip extension14. The increased or decreased ROM (depending
on the side), the increased flexion and the increased pelvic tilt are all compensation mechanisms
for reduced extension (55). The secondary soft tissue changes in the hip and their lasting effect
on the gait are the cause of the differences in extension, even if the primary effect (LCPD) is
healed, as described by Westhoff et al. (55). In future studies, the reversibility of the extension
reduction should be evaluated, in order to optimise the long-term effects on gait. The major
clinically important variables, i.e. Hip abduction, Trendelenburg gait and Hip Internal Rotation are
all normal in both patient groups. This is an important result, especially regarding Hip Abduction
and Hip Abduction Moment, because of the consequences. Especially regarding abduction, both
Hip Mean Abduction Moment and Hip Maximum Abduction Moment are not significant, indicating
the absence of both chronical overloading (mean) and peak overloading (maximum) of the hip
joint. This strongly suggests a reduction of long-term complications (such as osteoarthritis), a
major problem in LCPD.
The study design choices 4.3In this study, a radiologic classification based on the PCA technique is developed (cfr. 2.4
Radiologic evaluation of the LCPD patients). Both coverage and congruence are stated to be
equally important. After surgery, two important measurements are potentially interesting: (a) the
direct effect of the operation (evaluation of the graft placement) and (b) the long-term effect of
the operation (containment of ‘normal’ congruence).
The graft, when performing an mSA, is placed in order to maximise the coverage and to obtain a
‘normal’ hip congruence (32). Coverage and congruence are considered to be equally important,
because both variables measure a different part of the post-operative evaluation (coverage
measures the direct effect, congruence measures the long-term effect) of containment.
In the analysis of the gait data, both an overall group comparison and a side-by-side comparison
is made. It was our feeling that in a group comparison, less severe gait deviations might not be
detected. Therefore, the side-by-side comparison is used, in which the unaffected leg acts as the
perfectly matched control for the affected leg. No difference, except due to physiological
variation, should be undetected.
14 In the clinical evaluation only the Hip Abduction, Hip Internal Rotation and Trendelenburg gait is disturbed (cfr. 1.2 Clinical presentation). However the gait analysis is more sensitive to abnormalities, therefore more parameters seem to be aberrant.
45
Study limitations 4.4
One of the obvious limitations of this thesis is the relatively small population that was studied.
Only 21 subjects were included (14 patients and seven controls). This is partially due to the fact
that LCPD is a rare disease and only LCPD patients, operated by Prof. Dr. Plasschaert
(paediatric orthopaedics UZ Ghent and St-Jan Bruges) in a timespan of ten years (’04-’14),
treated by a unique mSA are included. In addition, X-ray analysis and gait analyses (incl. the
data processing) are labour-intensive and time-consuming tasks.
Despite the small samplesize, a few factors should be taken into consideration when interpreting
the results. First, in a small samplesize greater variation could be expected. In our study
however, a consistent variation is shown as observed in the graphs in the result section cfr. 3
Results). Second, despite the small samplesize, enough power is obtained because differences
are shown in the analysis. Therefore, we could state, that even if a small samplesize is used, the
results are consistent and meaningful as a first study to evaluate mSA. Additionally, possible
statistical limitations, due to the samplesize, are compensated by a careful interpretation of the
absolute variables.
In order to classify the patients into good and bad outcome groups, X-ray measurements are
used. Because of the lack of an applicable, validated and descriptive classification, this study
had to rely on the Principal Component Analysis (PCA), which is not validated yet in the context
of this study. Possible errors could be caused by incorrect classification of patients and
exclusion of the worst and/or best patients. A third limitation of the technique is the lack of
reproducibility. The PCA derives components out of a set of variables, but the recombination of
the variables to each component is dependent on the sample variation and therefore might slight
differ for each study. Consequently, no repeatable radiologic classification can be extrapolated
based on the result derived in this study. Our results should however be reproducible, even
given the variation, due to the PCA method. A few arguments support the use of PCA. The
radiographic bad outcome patients are indeed shown to have a more aberrant gait pattern than
the good radiographic outcome patients, this could imply that patients are classified
appropriately. The PCA technique is not often used in orthopaedics research, but in psychology
and engineering, it is a frequently applied statistical analysis technique and accepted as a valid
data analysis technique.
The study of Westhoff et al. states the function of the gait could not be evaluated based on
radiologic data (43). As the outcomes of the gait analysis are in line with the radiologic
46
classification, this contradicts the findings of Westhoff et al. Our results are with this respect in
agreement with the study of Svehlik et al. The study of Plasschaert et al. cannot confirm this
statement based on the hip abduction function because no correlation is found between hip
abduction and RX outcome (57).
Future research 4.5This study did focus on gait as a measure of function at an intermediary endpoint of LCPD.
Despite the fact that our work does support the value of mSA in restoring/maintaining function of
the Perthes hip, further long-term evaluation of mSA is needed. This in order to document the
effect of modified Shelf Acetabuloplasty on the development of osteoarthritis. This might allow to
correlate findings regarding osteoarthritis with gait analysis outcome variables.
Despite the fact that gait analysis does already provide data that do support clinical practice, a
further improvement of the reliability of gait analysis outcomes seems mandatory. Further
research is needed to optimise methods. We should improve the interobserver repeatability
(mainly marker placement) and determine the best-suited method for defining the joint axes in
LCPD patients. In the radiological evaluation, an objective and repeatable classification is
needed. These radiological classifications should be applicable in growing patients, since most
LCPD patients are not fully grown during at least the first years of follow-up after (surgical)
treatment.
Future studies should evaluate the effect of physiotherapy and gait training in the prevention of
Hip ROM abnormalities. If gait deviations could be prevented, no aberrant parameters will be
detected in gait analysis. In that case the mid-term outcome of containment therapy would be
optimal. Osteoarthritis, a major but long-term outcome of LCPD could then be evaluated
separate from gait abnormalities. This might enable detecting the exact cause of osteoarthritis of
LCPD: due to gait abnormalities, inherent to the disease or a combination.
Finally, the small sample size is also a limitation of our study. In order to, more reliably,
extrapolate the conclusions to a larger population, the study should be repeated using larger
sample sizes in a longitudinal setting. This would also aid in defining a more reliable definition of
‘normal gait’.
Conclusion 4.6In conclusion, the differences found in our study (Hip ROM, extension, flexion, pelvic tilt), are
47
most likely all due to a reduction of the hip extension. No possible cause for the decreased hip
extension, due to the operation could be detected. Based on the findings of Westhoff et al.,
these results are a secondary effect of LCPD. In the major clinically important variables, no
abnormalities could be detected. Absence of hip overloading, based on findings regarding Hip
Abduction Moment, is an indicator for positive long-term outcome. Therefore, we can state that
the results of mSA regarding gait outcome look very promising for patients with good
radiographic outcome, this applies even for the patients with bad outcome. Based on the
function short term outcome of the involved hip, mSA should be further applied as an operative
technique for severe LCPD patients. Further research is needed to confirm our findings more
profoundly and to further specify the role of mSA and its benefits in the treatment of LCPD.
48
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I
6 Appendix
Schematic illustration of the gait lab 6.1
Figure 3: schematic illustration of the gait lab
II
Radiographic parameters 6.2
6.2.1 Coverage and subluxation
● Sharp’s angle (SA)
The SA is measured using the classic method described by Agus et al. in their article “How
Should the Acetabular Angle of Sharp Be Measured on a Pelvic Radiograph?”: The angle is
formed by a horizontal line connecting the inferior tips of both pelvic teardrops (referred to as the
“reference line”) and by a line connecting the inferior tip of the pelvic teardrop with the lateral
edge of the acetabular roof in the unaffected side or by a line connecting the inferior tip of the
pelvic teardrop with the lateral edge of the shelf in the affected side (Figure 2).
Figure 2: Sharp's angle: SA (Sharp's angle unaffected side), SA' (Sharp's angle affected side).
● Subluxation ratio (SR)
The shortest distance (D and D’) between the most medial point of the femoral head and the
vertical line through the most inferior point of the teardrop, perpendicular to the reference line.
The ratio for the affected to the unaffected side is calculated and expressed as the subluxation
ratio (Figure 3).
III
Figure 3: Subluxation ratio: D (shortest distance unaffected side), D' (shortest distance affected side).
● Femoral head size ratio (FHSR)
The femoral head size ratio is the ratio of the femoral head size of the affected side to the
femoral head size of the unaffected side. The femoral head size is defined as the shortest
distance between the most medial and most lateral point of the femoral head (Figure 4).
A�,��89*�8B�(���8+(��ACDE%:G�,��89*�8B�(��8GG��+�B�(B��8H%
G�,��89*�8B�(��:.8GG��+�(B��8%
Figure 4: Femoral head size ratio: a (femoral head size unaffected side), a' (femoral head size affected side).
IV
● Femoral head coverage ratio (FHCR)
Femoral head coverage is measured as described by Heyman and Herndon in “Legg-Calvé-
Perthes disease; a method for the measurement of the roëntgenographic result”. Coverage is
calculated as the shortest distance between the vertical line, perpendicular to the reference line,
through the lateral edge of the acetabulum for the unaffected side (A) or through the lateral edge
of the shelf in the affected side (A’). The ratio is calculated by dividing the femoral coverage by
the ipsilateral femoral head size (Figure 5).
A�,��89*�8B��7��8)��8+(�:.8GG��+�B�(B� � G�,��89*�8B��7��8)��I%
G�,��89*�8B�(���8%
A�,��89*�8B��7��8)��8+(�8GG��+�B�(�� � G�,��89*�8B��7��8)��IH%
G�,��89*�8B�(���8H%
Figure 5: a (femoral head size unaffected side), a' (femoral head size affected side), a (femoral head size unaffected side), a' (femoral head size affected side).a (femoral head size unaffected side), a' (femoral head size affected side), A (femoral head coverage unaffected side), A’ (femoral head coverage affected side).
● De center-edge angle (CEA)
To obtain the CEA, as described by hanson et al. in “Discrepancies in measuring acetabular
coverage: revisiting the anterior and lateral center edge angles”. First, the best fitting circle
around the femoral head is drawn. Second, a vertical line, perpendicular to the reference line,
and through the centre of the circle is drawn. The CEA of the unaffected side is defined by the
ventrical line and a line connecting the centre of the circle with the lateral edge of the acetabular
roof. The CEA of the affected side is defined by the vertical line and a line connecting the centre
of the circle with the lateral edge of the shelf in the affected side (Figure 6).
V
Figure 6: Center edge angle: CEA (Center edge angle unaffected side), CEA' (Center edge angle affected side)
VI
6.2.2 Congruence: SDS
The steps and formulas listed in this section are originating from the article “Quantitative
Measures for Evaluating the Radiographic Outcome of Legg-Calvé -Perthes Disease” of Shah et
al.
To calculate the SDS two radiographs are required:
an anteroposterior (AP) radiograph and a frog-leg
radiograph.
The following steps, described by Shah et al., are
applied on the anteroposterior and frog-leg
radiograph:
1. Two reference points are marked: the most
medial and the most lateral margin of the
femoral head (Figure 7-a).
2. A circle touching these reference points is
drawn to match the size of the femoral head
and lines up with the articular surface of the
femoral head (Figure 7-b).
3. If the circle perfectly lines up with the contour
of the articular margin, the radius of the circle
(r ap) is noted.
4. If the articular margin does not line up with the arc of the circle, the size of the circle is
adjusted, so that it just touches the reference points and the articular margin. The circle
does not extend outside the femoral head (maximum inscribed circle = MIC) (Figure 7-b).
5. A second concentric circle is drawn to just touch the outer limits of the articular margin
without extending inside the femoral head (minimum circumscribed circle [MCC]) (Figure
7-c).
6. The radii of the circles are noted: r MIC-ap (radius of the maximum inscribed circle on
the anteroposterior radiograph), r MCC-ap (radius of the maximum circumscribed circle
on the anteroposterior radiograph), r MIC-lat (radius of the maximum inscribed circle on
the frog-leg radiograph) and r MCC-lat (radius of the maximum circumscribed circle on
the frog-leg radiograph)
After measuring the radii, the roundness errors are calculated. They are expressed as ratios and
Figure 7: Radiographs illustrating the technique for measuring the roundness error. Illustration of Shah et al. from “Quantitative Measures for Evaluating the Radiographic Outcome of Legg-Calvé-Perthes Disease”
VII
define the sphericity of the femoral head.
• Roundness error (RE) on the AP radiograph:
EJI6 ��,��8- � �,(�8-
,�8.�G�,��8- K �,(�8-��8-%L 100
• Roundness error on the frog-leg radiograph:
EJO8+ � �,��98+ � �,(�98+
,�8.�G�,��98+ K �,(�98+��98+%L 100
The next step is calculating the Ellipsoid Deformation. The ED is present when the radius of the
femoral head on the AP-radiograph differs from the radius of the femoral head on the frog-leg
radiograph.
• If r lat > r ap
JP ��98+ � �8-
�8-L 100
• If r ap > r lat
JP ��8- � �98+
�98+L 100
The sum of the RE’s and the ellipsoid deformation is the extent to which the shape of the
femoral head deviates from sphericity: SDS = RE AP + RE Lat + ED.
VIII
IOWA hip score (chart) 6.3
IX
Results PiG Model 6.4
6.4.1 Group comparison PiG Model
6.4.1.1 Comparison of the unaffected sides
The Kruskal-Wallis test shows no differences for 13 variables (0,069 < P-value < 0,838). The
following five variables are significant: Hip Maximum flexion, Hip Maximum Extension, Hip
Maximum Internal Rotation, Hip Maximum External Rotation and Knee Mean Varus (0,014 < P-
value < 0,041).
After performing the Mann-Whitney U-test in the PiG model, significant differences between the
control group and the bad outcome patient group are shown for the following variables: Hip
Maximum flexion, Hip Maximum Extension, Hip Maximum Internal Rotation and Hip Maximum
External rotation (0,008 < P-value < 0,014). A trend is detected for Knee mean Varus (P-value:
0.022). The variable Hip Maximum Flexion and Hip Maximum Extension is also significant
different between both patient groups (P-values: 0,014).
No significant differences are detected between the control group and the good outcome patient
group (0,073 < P-value < 0,259). For the PIG model a trend is found for Knee Mean Varus (P-
value: 0,038).
6.4.1.2 Comparison of the affected sides
In the PiG model, the variable Knee Maximum Varus is borderline-missed significant (P-value:
0,054) and the variables Hip Maximum Extension and Hip ROM are shown to be significant
(0,001 < P-value < 0,005). For Hip Maximum Extension significant results are found after post-
hoc testing, when comparing the controls and the patient group with a good radiographic
outcome (P-value: 0,011) and when comparing the controls and the patient group with a bad
radiographic outcome (0,001). When comparing both patient groups a trend is found (P-value:
0,017). For Hip ROM a significant result is found as well after post-hoc comparison of the
controls and the bad outcome patient group (p-value: 0,001). A trend is found when comparing
the controls and the good outcome patient group (P-value: 0,017).
6.4.2 Side comparison
6.4.2.1 Control group
Within the control group, a significant result is detected for Knee Maximum Varus (P-value:
X
0,31).
6.4.2.2 Good outcome patient group
In the PiG model, a significant is found for Hip Maximum Rotation Internal Moment (P-value:
0,043).
6.4.2.3 Bad outcome patient group
Using the PiG model, significant results are found for the variables Hip Range of Motion, Hip
Maximum Flexion Moment, Hip Maximum Internal Rotation Moment and Hip Maximum External
Rotation (P-values: 0,031).
Tables results 6.5
Table 3: overview Abbreviations of variables used in tables
XI
Table 4: overview Signifcance of variables in PiG-Matlab and Pig Models; 1: controls, 2: patients with good outcome, 3: patients with bad outcome; Unaff: unaffected side, Aff: affected side; underlined numbers are significant
XII
Table 5: overview mean values and standard deviations (SD) of Kinematic variables in both PiG-Matlab and PiG Models; unaff: unaffected, aff: affected. Pelvic Tilt and Pelvic Obliquity is identical for both models, and therefore only reported in one table
XIII
Table 6: overview mean values and standard deviations (SD) of Kinetic variables in both PiG-Matlab and PiG Models; unaff: unaffected, aff: affected.
XIV
Table 7: overview additional variables (Balance velocity, Balance path length, step time and IOWA hip score) ; unaff: unaffected, aff: affected.