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Twenty-Year Follow-Up of 50 ConsecutivePatients Born with Unilateral Complete CleftLip and Palate Treated by the Oslo CleftTeam, NorwayGunvor Semb, Elisabeth Rønning, and Frank Åbyholm

Long-term follow-up of patients with complete clefts provides a morecertain indication of treatment outcome than short-term studies. Rela-tively few published reports, however, describe outcomes at age 20 yearsor beyond. This retrospective cohort study involved 50 patient (17 female,33 male) born with complete unilateral cleft lip and palate who wereconsecutively treated by the Oslo Cleft Team. The data were analyzed byinternal and external observers with the use of standardized procedures,ie, for assessment of dental arch relationship, the late adolescent version

of the original Goslon Yardstick; for facial growth, standardized cepha-lometry; and an extension of the Bergland scale for rating alveolar bonegrafting success. The kappa statistic was used to evaluate interraterreliability. The burden of care in numbers of operations and duration oforthodontic treatment was calculated. Results for dental arch relation-ship were as follows: 40% had excellent, 32% good, 18% fair, and 10% hadpoor outcome. The cephalometric measurements are comparable withother published results. A completely normal interdental septum afterbone grafting was observed in 80%, a slightly reduced septum in 18%,and a failed graft in 2%. Long-term follow-up suggests that the Oslotreatment protocol for unilateral cleft lip and palate achieves a satisfac-tory balance between the burden of care and dentofacial outcome. (Se-min Orthod 2011;17:207-224.) © 2011 Elsevier Inc. All rights reserved.

L ong-term follow-up to adulthood of a spe-cific treatment protocol is a desirable but

relatively uncommon occurrence in the cleft literature because the following circumstancesmay not commonly coexist: adequate caseload, commitment to research, consistency over time of treatment methods, record collec-tion, team membership, and patient atten-

dance.Enemark et al1 appear to have reported thefirst major longitudinal long-term follow-up.The study by Enemark et al involved multidis-ciplinary evaluation of 57 patients with unilat-eral cleft lip and palate (UCLP) at 21 years of age and included skeletal and soft-tissue facialgrowth (cephalometry), occlusion, speech,and need for secondary surgery. Fifty-one of 57 patients had an acceptable occlusion.

Senior Lecturer in Craniofacial Anomalies, Dental School, Uni- versity of Manchester and affiliated with the Oslo Cleft Team, Department of Plastic Surgery, Oslo University Hospital, and Bredt- vet Resource Center and Adjunct Professor at the Faculty of Odon-

tology, University of Oslo, Norway; Head of Dental Unit, Depart- ment of Plastic Surgery, Oslo University Hospital, Norway; Professor, Department of Plastic Surgery, Oslo University Hospital,Norway.

This article is based on the work of the Oslo Cleft Team, Oslo University Hospital, Norway.

Address correspondence to Gunvor Semb, Dental School, Univer- sity of Manchester, Higher Cambridge Street, Manchester M15 6FH,United Kingdom. E-mail: [email protected]

© 2011 Elsevier Inc. All rights reserved.1073-8746/11/1703-0$30.00/0 doi:10.1053/j.sodo.2011.02.005

207Seminars in Orthodontics, Vol 17, No 3 (September), 2011: pp 207-224

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Cleft care in Norway has been centralized in 2multidisciplinary teams for more than 50 years,one in Oslo and the other in Bergen. Approxi-mately 70-80 new patients with clefts are referredto the Oslo Team annually. The Norwegian pop-

ulation is fairly homogenous, and staff at theCleft Centre have tended to remain on staff fora long time. All treatment and travel is free forthe patient and one parent, and patient atten-dance has been very good. One principle sincethe 1960s has been that the Team’s specialistsshould do all key treatment. With long distancesto travel and a desire to minimize the burden of care for the patients and the family, treatment

with no proven long-term benefit has not beenadopted, and treatment periods have been con-centrated to keep visits to a minimum. Since the

1960s, standardized record keeping has alsobeen seen as very important so that outcomescan be monitored and protocols revised as nec-essary. All attempts are made to have the finalrecord collection for patients with completeclefts at 21 years of age. This article will focus onpatients born with UCLP.

The Surgical Protocol for UCLP

Apart from the introduction of alveolar bonegrafting, changes to primary surgery protocols

have been modest. Since 1968, in patients withUCLP, the lip was closed at 3 months of age by use of the Millard procedure2 and at the sametime the hard palate was closed by a single layer

vomer flap. A modified von Langenbeck proce-dure3 was used to close the soft palate at 18months, then the timing was changed to 12months in 1993. Alveolar bone grafting usingcancellous bone from the iliac crest was intro-duced in 1977 and soon became a routine pro-cedure for all patients with alveolar clefts. Sec-ondary surgery (pharyngoplasty, sulcoplasty, lipand/or nose corrections) was performed ac-cording to individual needs.

The Orthodontic Protocol for UCLP

No presurgical orthopedics or treatment in thedeciduous dentition has ever been undertakenin Oslo because of the absence of evidence inpast decades or until the present time. Since theintroduction of alveolar bone grafting, orth-

odontic treatment has been provided in 2 dis-tinct stages:

Pregrafting Orthodontic Preparation (When

Necessary) Anterior cross-bites and severe rotations of maxillary incisors are corrected. This is mostly done if the patients are very motivated to havetheir new front teeth aligned and sometimesto move a retroclined cleft side incisor out of the alveolar cleft region to improve surgicalaccess during bone grafting. Segmental dis-placement, if sufficiently severe, is corrected

just before bone grafting using a removablequad helix, which is kept in place for 3 monthspostoperatively.

Permanent dentition orthodontics is dis-tinctly different for patients with complete cleftscompared with noncleft patients for many rea-sons. Some degree of reduced maxillary growthpotential is the rule (Fig 1),4 and early determi-nation of the eventual need for maxillary osteot-omy is a challenge, requiring borderline cases tobe assessed carefully. There is a tendency for themaxillary arch midline to be displaced to thecleft side. The permanent lateral incisor is miss-ing in 45% of Norwegian patients with alveolarclefts,5 and many laterals that are present are

malformed or erupt ectopically and cannot bekept with a good long-term prognosis. Otherteeth are more frequently missing in patients

with clefts.6

The orthodontist will choose whether orth-odontic space closure is the best option or

whether the lateral incisor space should be pre-served for replacements of various kinds. In Osloorthodontic space closure has been favored overprosthodontic restorative space closure for rea-sons discussed in this article. Protraction head-gear/facemask is sometimes used to stabilize the

incisor position while posterior teeth are movedmesially. However, we do not consider protrac-tion to achieve significant or lasting skeletalchange in the position of the maxilla. Facialesthetics takes precedence over “normal” toothpositioning, and slight proclination may help tosupport the upper lip.

Cleft side canine impaction occurs in 25%of 191 Norwegian patients with UCLP whohave had alveolar bone grafting.7 This “com-

208 Semb, Rønning, and Åbyholm



plication” calls for an extra oral surgery pro-

cedure and an increase in the duration of theorthodontic treatment. The relapse tendency in patients with complete clefts is greater whencompared with noncleft patients, and this isrelated to the scar tissues from the surgeries. A tight upper lip and scars in the alveolus and inthe palate will encourage the migration of teeth into crossbite.8,9 The continued impair-ment of maxillary growth together with con-tinued mandibular growth in the late teens(especially for males)4,10 may be factors in the

worsening of the occlusion seen in some pa-

tients in the late teens or early twenties.The purpose of the present paper is to pres-ent dentofacial outcomes at the mean age of 20

years for a cohort of consecutively treated pa-tients with UCLP using study models, cephalo-grams and occlusal radiographs of the bone-grafted region. The occlusal changes fromdebonding to follow-up at about 5 years later isdescribed as is the total amount of surgical in-terventions and orthodontic treatment.

Participants

The inclusion criteria for participants withUCLP of this study were as follows:

● nonsyndromic clefting and no other malfor-mation;

● all surgery and treatment follow-up by theOslo Cleft Team;

● full records (cephalograms, study models andocclusal radiographs of the bone grafted cleft region, and clinical case notes) available at approximately 20 years of age; and

● complete bony cleft, although patients with

a soft tissue band (Simonart’s band) wereincluded.

The sample consisted of the first 50 patientsborn from January 1, 1975, who met the inclu-sion criteria. It included 17 female and 33 malepatients born between January 1, 1975, and Oc-tober 1979. None of the patients in the samplehad chosen to have orthognathic surgery by thetime of record collection, although surgery had

Figure 1. Changes in maxillary prominence (s-n-ss [sella-nasion-subspinale] or SNA angle) from 5 to 18 years in257 patients with UCLP and a noncleft group (all definitions in Appendix).2

209Twenty-Year Follow-Up of UCLP



been discussed with several of them. The detailsof the study sample are presented in Table 1.Thirty-three patients (66%) had a cleft on theleft side, with 17 (34%) on right side. Twenty subjects (40%) had a soft-tissue band across thecleft, either under the nostril or between the

alveolar processes. The mean age and range of records for the models and radiographs is alsofound in Table 1. The time from debondinguntil follow-up models was a mean of 4.9 years(range, 0.5-10.4 years) and the follow-up timeafter bone grafting was at a mean of 10.4 years(range, 3.5-12.9 years).

All patients had the same primary surgery: lipclosure (Millard’s technique) and simultaneoushard palate closure with a single layer vomer flapat a mean age of 3.2 months (range, 2.2-4.9months), posterior palate closure (modified von

Langenbeck technique) at 18.2 months (range,15.7-31.1 months) and alveolar bone grafting at mean age of 9.7 years (range, 8.8-12.6 years).The dental status of the patients, number of missing lateral incisors and missing teeth beyondthe cleft region is presented in Tables 2 and 3.

Methods

Assessing Dental Arch Relationship

The dental arch relationship was assessed with amodification of the Goslon Yardstick,11 whichranks dental study casts of subjects with UCLP in

the late mixed dentition/early permanent denti-tion into 5 categories: group 1 (excellent results);group 2 (good results); group 3 (fair results);group 4 (poor results); and group 5 (very poorresults). The Yardstick has been used in many studies, including the Eurocleft comparative stud-ies of 9- and 12-year-old patients with UCLP.12,13

The Yardstick was adapted for the Eurocleft 17- year-old assessment,13 and this modification wasused in the present study. Three judges (2 externaland 1 internal) scored the models by using thefollowing definitions: group 1 (excellent result):

good horizontal and vertical relations. One toothin the lateral segment in cross bite is accepted. Anexample of a study model in group 1 is seen in Fig2 A (left; from the Eurocleft Study 13). Group 2(good result): positive overjet and overbite.Crossbite in one lateral segment is accepted.Group 3 (fair result): edge-to-edge anterior oc-clusion, inversion of one tooth in the frontalregion (including canine) is accepted, and uni-lateral or bilateral cross bite also is accepted.Group 4 (poor result): negative incisal overjet

with symmetric form of the upper arch, unilat-eral or bilateral cross bite is accepted. Group 5(very poor result): severe negative overjet with anarrow upper arch. An example of a study model in group 5 from the Eurocleft Study 13 isseen in Figure 2B (right). The interrater reliabil-ity was assessed by the Kappa statistics.14

Cephalometry

Standard cephalometric analysis also was per-formed. All cephalograms were traced by one

Table 1. Overview of the Sample: Sex, Cleft Side, Soft-Tissue Band, Age at Models, Cephalogram and BoneGraft Radiographs

Patients

Cleft Side

Soft-Tissue Band Age at Model, yr Age at

Cephalogram, yr Age at Bone Graft

Radiograph, yr Left Right

n % n % n % Yes % No % Mean Range Mean Range Mean Range

Females 17 34 10 59 7 41 8 47 9 53 19.9 17.7-22.8 20.0 18.2-22.8 20.0 18.2-22.8Males 33 66 23 70 10 30 12 36 21 64 20.1 17.4-22.1 20.1 17.4-22.3 20.5 17.4-22.3Total 50 100 33 66 17 34 20 40 30 60 20.0 17.4-22.8 20.1 17.4-22.8 20.3 17.4-22.8

Table 2. Status of Cleft Side Lateral Incisor, Present or Absent, and the Number of Lateral Incisors Kept for Alignment

Status of Cleft Side Lateral

Number of Laterals Kept

n % n %

Missing 17 34 — —Present

On larger segment 7 14 5 71On smaller

segment 15 30 4 26

On both segments 11 22 On largersegment, 8

73

On smallersegment, 1

9




investigator (G.S.). Fourteen angular measure-ments were calculated.

Success of Alveolar Bone Grafting

The height of the interdental septum after alve-olar bone grafting in the mixed dentition wasevaluated by a panel of 4 judges (2 internal and2 external) who used the Bergland/Åbyholmscale (Fig 3).15,16 This is a 4-category scale: typeI, completely normal interdental septum; type II,the septum reaches 75% of normal septumheight; type III, the septum reaches less that 75% of normal septum; and type IV, failure, nobony bridge across the cleft. In addition, a 3-cat-egory scale for scoring the naso-apical aspect of the bone graft was used (Fig 4).17 The defini-tions are score 1: no naso-apical defect; score 2:defect in apical area, but with sufficient bonepresent to allow uprighting of teeth and closingthe gap; and score 3: defect in apical area withinsufficient bone to allow uprighting of teeth.

Amount of Surgical and Orthodontic Treatment

A survey of the written hospital records was un-dertaken to calculate the average number of treatment episodes and days in hospital for sur-gery. Forty-five of the patients had had all orth-

odontic treatment at the Cleft Centre, and visitsand duration of orthodontic treatment could becalculated.

Results

Dental Arch Relationship

The 3 judges had very good inter-rater agree-ment. The Kappa values ranged from 0.845 to

0.915. The interpretation of the Kappa score isfound in Table 4. As indicated in Figure 5, 40% were in the very good category of the dental archrelationship and no patients were in the very poor category. The mean score for the sample

was 2.03.

Change in Occlusion from 16 to 20 Years of Age

The change in scores from 16 to 20 years is alsoseen in Figure 5. Thirty-five patients (70%)scored the same at 16 and 20 years. In 15 pa-

tients (30%), the score had worsened:● Five patients slipped from score 1-2 because

more than one premolar had assumed a lat-eral crossbite.

● Four patients changed from score 2 to score 3.For 3, the reason was continued mandibular

Table 3. Overview of Patients (n 18) With Missing Teeth Beyond the Cleft Region

Missing Teeth in Upper Jaw Missing Teeth in Lower Jaw

Cleft Side Central Incisor, n

Cleft Side Second Premolar, n

Noncleft Side Second Premolar, n

Both Second Premolars, n

Three Upper Premolars, n

One Second Premolar, n

Two Second Premolars, n

1 3 7 3 1 2 3

Figure 2. Left: A representative case (lateral view) from the 17-year Yardstick group 1: excellent result. 11 Right: A representative case (lateral view) from the 17-year Yardstick group 5: very poor result.11




growth resulting in a change from positive

overjet/verbite into an edge to edge bite.● Three patients had changed from score 3 toscore 4. The edge-to-edge bite had become ananterior crossbite.

● Two patients changed from 1 to 3; one be-cause the premolars, the first molar, and thecleft side canine (in the lateral incisor’s posi-tion) had all assumed a crossbite. The otherpatient’s continued mandibular growth hadresulted in edge-to-edge bite.

● One patient changed from 1 to 4 because of marked continued mandibular growth.

Interestingly, 2 patients without evidence of an-terior crossbite in the deciduous dentition haddeteriorated to category 4 by age 20 years.

An example of a patient with a relatively sta-

ble occlusion (found in 70% of the sample)from 12.4 to 22.8 years of age is illustrated inFigure 6. Figure 6F was taken at 13.9 years, andFigure 6H was taken at 22.8 years. This patient isused as an example of reasonable stability of theocclusion after orthodontic treatment. An exam-ple of a patient with deterioration of occlusionfrom debonding at 15.2 years (score 1) to 20.1

years of age(score 3) is illustrated in Figure 7.Figure 7G was taken at 16.1 years and Figure 7Iat 20.1 years, and this is an example of relapse of the crossbite after orthodontic treatment. The

outcome was mostly attributable to relapse of the cleft side crossbite in addition to one front tooth also in crossbite. An example of a patient

Figure 3. The Bergland/Åbyholm13,14 scale for assessing the height of the interdental septum achieved afteralveolar bone grafting: Type 1: interdental septum height approximately normal; Type 2: height at least three-quarters of normal height; Type 3: height less than three-quarters of normal height; Type 4: failure. Nocontinuous bony bridge across the cleft achieved.

Figure 4. A 3-category scale for scoring the nasolabial aspect of the bone grafted region. Score 1: no defect.Score 2: defect in apical area, but with sufficient bone present to allow uprighting of teeth. Score 3: defect inapical area with insufficient bone present to allow uprighting of teeth.




with deterioration of occlusion from score 1 at 17.5 years to score 3 at 21.3 years mostly attrib-utable to continued mandibular growth is illus-trated in Figure 8.

Facial Growth

The results of the cephalometric analysis are

seen in Table 5. The mandibular prominence was statistically significantly larger in male thanin female patients, as was soft-tissue maxillary prominence.

Success of Alveolar Bone Grafting

Scores for the height of the interdental bone inthe bone grafted area are presented in Figure 9.Eighty percent had a completely normal septum(score 1), 18% had 75% of normal bone height,

whereas one bone graft failed. This patient un-derwent repeat surgery and subsequently ob-

tained a score 1. Examples of the scores 1 and 2in patients in this study can be seen in Figure 10.The scores of the naso-apical aspect are as fol-lows: 96% had no naso-apical defect, 2% (1 pa-tient) had a small defect, and 2% (1 patient)

with insufficient bone to allow uprighting of teeth.

Burden of Care—Surgery

Tables 6 and 7 summarize the proportion of patients who received different surgeries, aver-age age of surgeries, and the distribution of surgeries across patients. The average number of surgeries per patient was 4.8 and the mean num-ber of days in hospital was 24.1 (Table 8).

Burden of Care—Orthodontic Treatment

Tables 8 and 9 show the number of patientsreceiving orthodontic interventions, average du-ration, age at completion and number of visitsfor orthodontic treatment and follow-up. Cor-

rection of rotated and/or retroclined incisors inanterior crossbite was performed in 74% of pa-tients in the present sample, usually commenc-ing at age 7-8 years. Transverse expansion beforebone grafting was done in 16% of this sample,

usually starting 6-8 months before bone grafting(in unpublished data of a larger survey of 485patients with UCLP the number of incisal cor-rections was lower and the number of expan-sions was higher, 64% and 28%, respectively).

All patients in this sample had orthodontictreatment in the permanent dentition. This treat-ment was done by the Center’s orthodontistsin 45 of the 50 patients. The orthodontic treat-ment of the permanent dentition started at amean age of 12.7 years (range, 9.3-15.6 years)and lasted on average 2.4 years (Table 9). All

but 3 patients had fixed appliances in botharches, except for 3 who did not want appli-ances in the lower jaw. The number of visitsfor treatment was on average 24.3 per patient and nontreatment reviews from 6 to 21 years

were on average 11.9 (Table 8). In many of these follow-up visits, the patient was seen by several members of the Cleft Team.

In this sample 58% of the patients had surgi-cal exposure of the cleft side canine, which ismore than twice as high than the frequency found in the unpublished data of 485 patients

with UCLP where 27% of patients had surgical

uncovering of cleft side canine. The duration of the orthodontic treatment is usually longer

Figure 5. Modified Goslon Yardstick scores of dentalarch relationship at mean age of 16 years (cross-hatched columns) and at 20 years (plane columns).Score 1: excellent results. Score 2: good results. Score3: fair results. Score 4: poor results. Score 5: very poorresults.

Table 4. Interpretation of Kappa Values14

Value of Kappa Strength of Agreement

0.20 Poor0.21-0.40 Fair0.41-0.60 Moderate

0.61-0.80 Good0.81-1.00 Very good1.00 Perfect agreement




when canines need traction to be aligned in thearch. In 3 patients the canine had assumed atransposition with the first premolar.

In 8 patients (18%), both lower first premo-lars were extracted because of moderate-to-se-

vere crowding and in another 8 (18%) patients alower incisor was extracted because of mildcrowding (6 patients) and because a better over-bite/overjet could be obtained (2 patients). In 2patients one lower premolar was extracted and

Figure 6. Example of a patient with fairly stable occlusion after debonding (score 1). The patient was born witha left-sided complete cleft lip and palate with soft tissue bridge. (A) Occlusal view of the maxilla on the day of lip and hard palate closure at 3.4 months. (B, C) After eruption of permanent incisors, 6.1 years. (D) Occlusalx-ray of the alveolar cleft before bone grafting at 9.6 years. Missing cleft side lateral incisor. (E, F, G) Occlusion1.5 years after debonding, age 13.9 years. Total duration of orthodontic treatment was 2.2 years. (H, I) Occlusionat 22.8 years. (J) Occlusal radiography of the cleft region 8.4 years after bone grafting. (K) Lateral cephalogramat 22.8 years.




Figure 7. Example of a patient who had scored 1 (excellent result) at 16 years of age but who had experienceddeterioration of the occlusion at age 20.1 years resulting in score 3 (fair result). The patient was born with aleft-sided complete cleft lip and palate. (A) Occlusal view of the maxilla on the day of lip and hard palate closureat 2.9 months. (B, C) After eruption of permanent incisors, at 8.1 years. (D) Occlusal radiograph of the alveolarcleft before bone grafting at 10.2 years. (E) Occlusion at 11.9 years. Awaiting eruption of permanent teeth. (F,G, H) Occlusion 8 months after debonding at 16.1 years. Total duration of orthodontic treatment was 2.5 years.(I, J) Occlusion at 20.1 years. Crossbite of premolars and first molar on cleft side and of the cleft side canine (inthe laterals incisor’s place). (K) Occlusal radiograph of the cleft region 11.4 years after bone grafting. (L) Lateralcephalogram at 20.1 years.




Figure 8. Example of a patient who had score 1 (excellent result) at 16 years of age but in whomdeterioration of the occlusion at age 21.3 years resulted in score 3 (fair result). The patient was born witha left-sided complete cleft lip and palate with a soft tissue bridge (collapsed maxillary arches at 3 months,

but no bony bridge across the cleft was confirmed at surgery). (A) Occlusal view of the maxilla on theday of lip and hard palate closure at 3.6 months. (B) After eruption of permanent incisors, at 9.2 years.(C) Occlusal radiograph of the alveolar cleft before bone grafting at 10.3 years. Missing cleft side lateralincisor. (D) Occlusion at 14.5 years. The option of possible orthognathic surgery was discussed, but declined. Orthodontic treatment started non-extraction in lower arch. (E, F) Occlusion 2 months afterdebonding at 17.5 years. Total duration of orthodontic treatment 2.9 years. (G, H) Occlusion at 21.3 years.

Anterior edge-to edge bite and slightly open bite. (I) Occlusal x-ray of the cleft region 11.1 years after bonegrafting. (J) Lateral cephalogram at 21.3 years.




discarded and a second lower premolar wastransplanted to the upper premolar region.

Relapse Treatment

In 4 patients who lost their bonded retainer anddid not wear their soft acrylic retainer a smallgap opened in the cleft region or the teeth inthe cleft area rotated or moved in a palatal di-

rection. The relapse was such that the patientschose to have another period of orthodontictreatment (mean duration, 0.9 years; range, 0.3-1.4 years).

Orthognathic Surgery

The possibility of orthognathic surgery was dis-cussed with 10 patients, at least 5 of whom theorthodontists believed would really benefit. Allpatients, however, declined the offer of thistreatment.

Discussion

Is the Sample Representative?

The 50 patients included were consecutive from1975 to 1979 and they are probably representa-tive of patients who received treatment over sev-

eral decades. The age range in the present study was from 17.4 to 22.8 years, with mean of 20.1 asthe cohort reported was partly a conveniencesample as much of the material was already ac-cumulated for Oslo’s Eurocleft consecutive se-ries that included records to a minimum age of 17 years. Some further records exist in archivesof the Oslo team and it is planned to repeat thestudy with a larger sample where the minimumage for final records are 20 years.

Table 5. Cephalometric Variables, Means, and SD for the Total Sample and Divided by Sex

Total Sample,n 50

Mean Age 20.3 Years

Females,n 17

Mean Age 19.9 Years

Males,n 33

Mean Age 20.5 Years

Mean SD Mean SD Mean SD

Skeletal variabless-n-ss 76.0 3.5 75.1 2.9 76.5 3.7s-n-pg 79.3 4.0 77.5 3.0 80.2 4.2*ss-n-sm 0.9 2.7 0.4 2.6 1.4 2.8n-ss-pg 183.0 16.9 183.6 6.5 182.6 20.3NSL/ML 31.9 6.7 34.0 7.1 30.8 6.2NL/ML 24.7 6.5 25.2 6.7 24.5 6.4

Dental variablesIls/NL 107.7 7.1 108.7 7.0 107.2 7.2Ili/ML 89.5 6.1 89.3 6.4 89.6 6.1Ils/Ili 134.1 20.3 137.3 6.3 132.5 24.5

Soft-tissue variabless-ns-sss 85.3 4.1 83.0 2.4 86.6 4.3†s-ns-sms 82.3 3.7 80.5 2.5 83.2 3.9*Sss-ns-sms 3.1 2.4 2.5 2.0 3.5 2.6ns-sn-pg 176.7 6.2 177.9 7.2 176.0 5.7

gs-sn-pg 178.1 6.5 180.0 7.0 177.2 6.2

For definitions of cephalometric abbreviations, see the Appendix.*P 0.05.†P 0.01.

Figure 9. Graph presenting the outcome of thescores for the interdental septum after alveolar bonegrafting.15,16 Type 1: interdental septum height ap-proximately normal (80% of sample); Type 2: height at least three-quarters of normal height (18% of sam-ple); Type 3: height less than three-quarters of normalheight; Type 6: failure. No continuous bony bridgeacross the cleft achieved (2% of sample).




Figure 10. Examples of the bone graft score achieved in this sample. (A) Before bone grafting at 9.6 years.(B) Cleft site 8.7 years postgrafting. (C) Before bone grafting at 9.9 years. (D) Cleft site 11.2 yearspostgrafting. (E) Before bone grafting at 10.2 years. (F) Cleft site 11.4 years postgrafting. (G) Before bone

grafting at 10.1 years. (H) Cleft site 10.3 years postgrafting. Note that 80% had a bone graft score I, ie,normal interdental septum (B and D) and 18% had a bone graft score II, ie, 75% of normal septum height (F and H) and 2% (1 patient) had a failed bone graft.

Table 6. Surgical Interventions in the Study Sample

Patients n 50

Number % Mean Age, yr Age Range, yr

Cleft closureLip and hard palate closure 50 100 0.27 0.2-0.4Soft palate closure 50 100 1.5 1.3-2.6 Alveolar bone grafting 50 100 9.7 8.8-12.6

Additional surgery Pharyngeal flap 14 28 7.7 6.3-9.4Lip revision 17 34 9.8 4.6-18.9Fistula closure 2 4 9.1 4.5-13.7Surplus mucosa 8 16 7.4 5.3-13.5Sulcoplasty 2 4 8.1 7.6-8.6Second lip revision 4 8 14.5 7.6-20.7Second fistula closure 1 2 15.2Lip/nose revision 18 36 16.2 13.0-21.3Second lip/nose revision 3 6 20.5 17.0-22.5Nose revision 18 36 15.9 6.1-19.1Second nose revision 3 6 16.6 15.3-17.6




This sample had slightly different facial formfrom results presented in the larger cephalomet-ric study of 131 patients (of 257) with UCLP who

were 18 years or more from the same center4

(Table 10). The present sample had a slightly more prominent maxilla and mandible, but thedifference of the maxillomandibular angle be-

tween the 2 samples was only 0.5 degrees.

The Influence of a Soft-Tissue Band

In this sample 40% of patients had a soft-tissueband across the cleft (but no bony union). Inthe earlier study of 257 patients with UCLP 31%had a soft-tissue band,18 so the number of soft-tissue bands in the present sample was slightly greater than the average for the Oslo cleft pop-ulation. The Oslo Eurocleft sample of 30 con-secutive patients had the greatest frequency of soft-tissue bands among the 6 centers, suggest-

ing that this may be a trait in the Norwegian cleft population.There are some grounds for supposing that

individuals with UCLP and a soft-tissue band would be subject to less disturbed growth anddevelopment because preoperatively maxillary and nasal distortion are generally less severebecause of the restraining influence of the soft tissue band, tissue deficiency is potentially less,and surgery in turn, is arguably facilitated. Inthe cephalometric study the maxillomandibu-lar angle (ss-n-sm/ANB) was slightly more fa-

vorable although the maxilla (s-n-ss/SNA) was

not more prominent in those with soft-tissuebands compared with those without soft-tissuebands.18 However, in the present study there

was no statistically significant difference be-tween the groups with and without a soft-tissue

band for any cephalometric variables or forthe dental arch relationship. It has also beenhypothesized that patients with soft-tissuebands would have fewer missing lateral inci-sors. In this sample patients without a soft-tissue band had missing lateral incisor in 33%of patients and those with a soft-tissue bandhad missing laterals in 35% of patients. How-ever, the sample in this study was rather smallfor subgroup comparisons.

Comparison with Other Centers Cephalometric Comparisons

In Table 10 the cephalometric values from 5other cephalometric studies of young adults arelisted. Three of these presented results of pa-tients with UCLP at 20 or more years,1,10,19 and2 studies20,21 had cephalometric data from pa-tients at 18 years. The results from Oslo on alarger sample of UCLP4 (n 131) are also listedin Table 10. The results of these studies appearcomparable to the Oslo group.

Comparison of Dental Arch Relationship

One large mixed-longitudinal study of UCLPtreated in Gothenburg has been reported, in

which the authors used study casts of 47 19- year-old patients.22 Because the original Gos-lon yardstick was used for the Gothenburgratings, the data are not directly comparable,and they were reported in graphic form. Esti-mating the values for the Gothenburg samplehowever suggest that the Oslo outcomes arebetter at 16 and poorer at 20 years of age.

Table 7. Number of Surgeries Per Patient

Number of Surgeries n %

3 8 164 15 30

5 12 246 9 187 5 108 1 2

Table 8. Visits to Cleft Centre for Surgery and Days in Hospital and Duration of Total OrthodonticTreatment, and Number of Visits for Treatment and Follow-Up

No of Surgeries Days in Hospital Orthodontic Treatment

Duration

Visits for Orthodontic Treatment

Visits for Orthodontic and Team Follow-Up

Mean Range Mean Range Mean Range Mean Range Mean Range

4.8 3-8 24.1 15-40 3.0 years 0.7-5.6 years 24.3 10-47 11.9 2-28




Surgical Interventions

On average, there were 4.8 surgeries per patient

(range, 3-8). The low frequency of fistulae clo-sure is probably explained by the fact that most residual fistulas in the Oslo patients are found inthe region of the alveolus, are asymptomatic andcan be closed during bone grafting.

The patients had stayed in hospital for anaverage of 24.1 days, but recent years have seenan increasing trend to shorter stays and many lipand even nose revisions are today done in theoutpatient clinic. However, geography dictatesthat many Norwegian patients have a long way totravel and for some surgeries with a potential forpostoperative difficulties (palate closure and

pharyngeal flaps), the patients from distant ar-eas still stay a day or 2 longer than those who livecloser to Oslo.

Orthodontic Treatment

As a rule, orthodontic treatment of patients withcomplete clefts is carried out at the Cleft Centreor occasionally when appropriate, in close col-laboration with a selected local orthodontist. Alltreatment is done using fixed appliances.

Anterior Space Management

In this study sample 18 patients (36%) had alateral incisor that could be aligned and kept

with a good long-term prognosis. In the remain-ing 32 patients with missing lateral incisors thegap in the dental arch was closed orthodonti-cally in 29 patients. In 3 patients a resin-bondedbridge was used for space closure, ie, in onepatient in whom the central incisor was missing,in one in whom the cleft side canine showed

cervical root resorption and was lost 7 years afterbone grafting, and in one treated by a localorthodontists who had chosen prosthetic space

closure.The optimal treatment for missing lateral in-

cisors is a controversial issue and has been dis-cussed at length in the orthodontic literature.The major benefit, in the authors’ opinion, isthat at the end of the orthodontic treatment, theoverall dental treatment can be completed. By recontouring the canine to a more ideal lateralincisor shape and size orthodontic space closureprovides long-term results that are as good as, orsuperior to space opening for prosthetic replace-ment.23-25 Other investigators23-27 have shown

that the periodontal conditions are significantly better with orthodontic space closure than withprosthetic replacement, the temporomandibu-lar joint function is not impaired and patient satisfaction is high.

Three patients had tooth transplantations:one had a small noncleft side second premolarthat was transplanted into the bone-grafted re-gion 6 months after grafting (Fig 11). The sec-ond patient was missing the cleft side lateralincisor and both upper second premolars. A lower second premolar was transplanted into the

cleft side second premolar region. The thirdpatient with tooth transplantation was missingthe cleft side lateral incisor and second premo-lar and also both lower second premolars. Hisnoncleft side second premolar was transplantedto the cleft side second premolar region. Toothtransplantation is a good option in selected pa-tients who otherwise would have had to haveprosthetic space closure and the procedure issafe and reliable if done at an optimal time.28,29

Table 9. Overview of Orthodontic Treatment, Type, and Duration

Number % Mean, yr Age Range, yr

Pregrafting orthodontics n 50Incisor correction 37 74Duration 0.4 0.1-0.9

Age at completion 8.9 7.2-12.5Transverse expansion 8 16Duration 0.6 0.2-1.0 Age at completion 9.6 8.5-10.6

Orthodontic treatment in permanent dentition n 45Duration 2.4 0.7-5.2 Age at completion 15.1 10.7-18.1Relapse treatment 4 9Duration 0.9 0.3-1.4 Age at completion 17.7 13.7-22.4




The survival rate at a mean follow-up of 26.4 years (range, 17-41 years) posttransplantation of 33 teeth in a Norwegian noncleft material was90%.28

Changes in Occlusion from 16 to 20 Years The deterioration in occlusion after debondingas described in this paper has also been found inother studies.8,9,30 Ramstad and Jendal8 found areduction in upper dental arch width in 22 sub-

jects with UCLP who had received a fixed bridgeacross the cleft to replace a missing lateral inci-sor and retain the previous orthodontic expan-sion. The follo w -up time was 13.5 years. Marcus-son and Paulin9 recorded occlusal changes from19 to 25 years in 39 subjects with UCLP. There

was a significant deterioration in the total occlu-

sal score during the follow-up period and this was larger on the cleft side than the noncleft side.

A multidisciplinary follow-up of 20 patients with UCLP with records at 10 and 20 years of agefound a significant shift in the Goslon Yardstickscore of dental arch relationship towards the lessfavorable end of the yardstick and the cephalo-metric measurements showed a more retrusivemaxilla at 20 years compared with the findings at 10 years.30 These studies all highlight the neces-sity for long-term follow-up through to adult-

hood as outcomes cannot be accurately reporteduntil maturity.

Other Outcomes

The present report is confined to dentofacialoutcomes; however, speech, hearing, nasolabialappearance, patient satisfaction, and psychoso-cial well-being are also essential aspects of theoutcome in adulthood. A subgroup of the pres-ent sample has been analyzed for speech at 12

years, and nasolabial appearance and patient satisfaction at age 17 years as part of the Euro-

cleft material.31-33

Speech for the patients fromthe 6 Eurocleft centers w as assessed by a panel of judges at 12 years of age.31 There were no statis-tically significant differences between the 6 cen-ters, but speech for the Oslo group was rankedhigh. Nasolabial appearance was assessed by ablinded panel of judges32 and no significant dif-ferences were found between Oslo and the other4 centers. (At 9 and 12 years, there were 6 cen-ters, but 1 center, Center C, did not participate T

a b l e 1 0 .

C e p h a l o m e t r i c V a r i a b l e s ( M e a n a n d S D ) f r o m

P r e s e n t S t u d y a n d O t h e r P u b l i s h e d R e p o r t s

f o r C o m p a r i s o n

V a r i a b l e s

P r e s e n t S a m p l e

n

5 0

A g e : 2 0 . 3

Y e a r s

E n e m a r k e t a l 1

n

5 7

A g e : 2 1 Y e a r s

P a u l i n a n d

T h i l a n d e r

1 0

n

2 3 M a l e s

A g e : 2 0 Y e a r s

S m a h e l e t a l 1 9

n

1 6 M a l e s

A g e : 2 4 . 3

Y e a r s

S e m b 4

n

1 3 1

A g e : 1 8

y

N o l l e t e t a l 2 0

n

3 7

A g e : 1 8 Y e a r s

M e a z z i n i e t a l 2 1

n

1 5

A g e : 1 8 Y e a r s

G g a G r o u p

M e a z z i n i e t a l 2 1

n

1 0

A g e : 1 9 Y e a r s

B g G r o u p

M e a n

S D

M e a n

S D

M e a n

S D

M e a n

S D

M e a n

S D

M e a n

S D

M e a n

S D

M e a n

S D

S k e l e t a l

s - n - s s

7 6 . 0

3 . 5

7 2 . 9

4 . 2

7 7 . 2

3 . 5

7 4 . 8

7 4 . 2

3 . 8

7 4 . 3

4 . 5

7 4 . 0

3 . 6

7 6 . 9

3 . 5

s - n - p g

7 9 . 3

4 . 0

7 7 . 5

5 . 5

8 0 . 0

4 . 8

7 6 . 6

7 7 . 9

3 . 6

7 5 . 7

4 . 7

7 8 . 5

3 . 1

7 9 . 7

2 . 1

s s - n - s m

1 . 0

2 . 8

2 . 6

3 . 3

1 . 0

3 . 8

0 . 1

1 . 5

3 . 7

0 . 4

3 . 8

2 . 2

3

1 . 0

3 . 7

n - s s - p g

1 8 3 . 0

1 6 . 9

1 8 5 . 9

8 . 4

1 8 3 . 2

1 8 7 . 5

7 . 9

1 8 7 . 6

7 . 5

1 7 8 . 4

9 . 9

N S L / M L

3 1 . 9

6 . 7

3 2 . 8

7 . 4

3 3 . 2

7 . 6

3 8 . 8

3 4 . 6

5 . 6

3 5 . 7

6 . 9

3 2 . 3

5

3 2 . 7

4 . 4

N L / M L

2 4 . 7

6 . 5

2 4 . 7

7 . 4

3 1 . 5

2 6 . 0

6 . 5

2 3 . 5

5 . 1

2 3 . 2

6 . 5

D e n t a l

I l s / N L

1 0 7 . 7

7 . 1

1 1 1 . 9

1 8 . 2

1 0 9 . 3

7 . 2

1 1 1 . 0

6 . 3

I l i / M L

8 9 . 5

6 . 1

8 6 . 8

5 . 7

8 6 . 8

8 . 0

I l s / I l i

1 3 4 . 1

2 0 . 3

1 3 7 . 8

9 . 5

1 3 1 . 7

1 2 . 1

S o f t t i s s u e

S s s - n s - s m s

3 . 1

2 . 4

0 . 1

3 . 0

3 . 1

3 . 3

3 . 6

4 . 3

2 . 9

4 . 1

3 . 6

n s - s n s - p g

1 7 6 . 7

6 . 2

1 7 4 . 4

1 7 6 . 9

7 . 9

1 7 7 . 5

7 . 7

1 7 5 . 3

4 . 1

g s - s n s - p g

1 7 8 . 1

6 . 5

1 7 8 . 5

7 . 4

8 6 . 8

1 8 3 . 9

9 . 2

F o r d e fi n i t i o n s o f c e p h a l o m e t r i c a b b r e v i a t i o n s , s e e t h e A p p e n d i x .

B g ,

b o n e g r a f t ; G g a , g i n g i v o a l v e o p l a s t y .




in the comparison at 17 years.) Oslo was rankedon a combined third place of 5 centers. In theEurocleft studies a patient questionnaire wasdone at the age of 17 years.33 There were nosignificant differences between centers in therating of satisfaction with overall care.

Conclusions

As noted, long-term follow-up of patients withcleft lip and palate is a major challenge for

cleft teams. It took 25 years to accumulate thematerial for the present study. External com-parison is presently limited because the gen-eral lack of comparative material and the lackof consistency of analysis systems. One key element that should be included in futurestudies in addition to psycho-social well-beingis the need to document the burden of careimposed by different protocols upon childrenand their families. Differences in the complex-ity and costs of different protocols, such asrevealed by a survey of 201 European cleft services,34 are surprisingly large. The relativesimplicity of the Oslo protocols, no presurgicalorthopedics, relatively few primary operations,and limited periods of orthodontics, appearsto have been compatible with acceptable long-term dentofacial treatment outcomes.

AcknowledgmentsThe authors are grateful to Mr Philip Eyres for statisticalassistance and preparation of the manuscript and to Profes-

sor William C. Shaw and Dr Nicky Mandall, University of Manchester, United Kingdom, and Dr Susana Dominguez-Gonzalez, North-West Clinical Network for CLP, UK forscoring models and occlusal x-rays.

Appendix

Definition of Cephalometric Landmarks

s sella. The center of sella tursica.n nasion. The most anterior point on the

fronto-nasal suture.

ss subspinale (A-point). The deepest point on the anterior contour of the upperalveolar arch.

sm supramentale (B-point). The deepest point on the anterior contour of thelower alveolar process.

pg pogonion. The most anterior point onthe mandibular symphysis.

ns soft tissue nasion. The deepest point inthe fronto-nasal curvature.

sss soft tissue subspinale (soft tissue A point). The point of greatest concavity

in the midline of the upper lip.sms soft tissue supramentale (soft tissue B-point). The point of greatest concavity of the midline of the lower lip

sns subnasale. The deepest point in the na-solabial curvature

pgs soft tissue pogonion. The most promi-nent point of the chin

gs soft tissue glabella. The most anteriorpoint on the soft tissue glabella

Figure 11. Patient with a small second premolar from the noncleft side transplanted into the bone graftedregion 6 months after surgery. (A) Before alveolar bone grafting. (B) Six months after bone grafting a smallsecond premolar from the noncleft side was transplanted into the bone graft, 3 months after toothtransplantation. (C) Nine months after tooth transplantation. (D) At 11.8 years after tooth transplantation.




Cephalometric Reference Lines

NSL Nasion-sella line, the line through na-sion and sella

ML Mandibular line, the tangent to thelower border of the mandible throughgn

NL Nasal line. The line through spina na-salis anterior (the apex of the anteriornasal spine) and pterygomaxillare (theintersection between the nasal floorand the posterior contour of the max-illa)

Ils Axis of upper incisors, a line from inci-sion superius (the midpoint of the in-cisal edge of the most prominent uppercentral incisor) and apex superius (theapex of the root of the most prominent

upper central incisor).Ili Axis of the lower incisors, a line from

incision inferius (the midpoint of theincisal edge of the most prominent lower central incisor) and the apex infe-rius (the apex of the root of the most prominent lower central incisor).

Cephalometric Variables

s-n-ss (SNA): maxillary prominence, angle be-tween s-n line at n and subspi-nale ss (A-point)

s-n-pg: mandibular prominence, angle be-tween s-n line at n and pogonion

ss-n-sm: anteroposterior relationship betweenthe maxilla and the mandible

n-ss-pg: the facial convexity angleNSL/ML: angulation between the nasion-

sella line and the mandibular lineNL/ML: angulation between the nasal and

mandibular linesIls/NL: upper incisor inclination to the nasal

lineIli/ML: lower incisors inclination to the man-

dibular lineIls/Ili: interincisal angless-ns-sss: soft tissue maxillary prominence

(soft-tissue SNA)ss-ns-sm: soft tissue mandibular prominence

(soft-tissue SNB)sss-ns-sms: soft tissue anteroposterior relation-

ship between the maxilla and man-dible (soft-tissue ANB)

ns-sns-pgs: soft-tissue facial convexity angle

gs-sns-pgs: soft-tissue facial convexity anglemeasured from glabella

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