journal club review; pediatrics
TRANSCRIPT
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Journal Club Presentation
Impact of Screening for Hepatic Hemangiomas in Patients with Multiple
Cutaneous Infantile Hemangiomas
Pediatric Dermatology 1-5, 2015Authors: Kristy L. Rialon, Rudy Murillo, Rebecca D. Fevurly, et. al.
ByExt. Sripada Kriangkhajorn
Faculty of Medicine, Srinakharinwirot University
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Abstract• Several studies have documented the association between
multiple cutaneous IHs with hepatic hemangiomas (HHs)• HHs can be clinically silent or serious symptom like mortality,
congestive heart failure• Screen abdominal ultrasonography has been recommended
for patient with five or more cutaneous hemangioma lesions
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Abstract• The study aim to determine whether the hemangiomas found
through screening had improved clinical outcomes in children with multiples cutaneous IHs
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BACKGROUND
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Infantile Hemangiomas• Most common tumors of infancy• Rarely life threatening, but can be if located in cervicofacial regions,
causing airway obstruction, or within liver, which can cause congestive heart failure, hypothyroidism, hepatic failure, and abdominal compartment syndrome
• Several studies have documented the association between multiple cutaneous IHs with hepatic hemangiomas (HHs)
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Hepatic hemangiomas(HH)• Recently, HH was thought to have no clear patterns of presentation
and natural history• HHs can be clinically silent or can cause serious symptom• Mortality rates have been reported 11%-18%, but historically it has
been much higher• There are many studies about several patterns, predict outcomes,
and guides therapy
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Hepatic hemangiomas(HH)• Focal HHs are consider as rapidly involuting congenital
hemangiomas (RICHs), which are distinct from typical hemangiomas, and less associated with cutaneous hemangiomas
HHs
CHs Focal
IHsMultifocal
Diffuse
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Study Objective
‘To determine whether the hemangiomas found through screening had improved clinical outcomes
in children with multiples cutaneous IHs’
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METHODS
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Population Study
Population Study Setting place at Boston’s Children Hospital
213 patients with HHs were identified from database and online registry
Database from Vascular Anomalies Center and online registry
Study period from January 1, 1995 to December 31, 2012
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MethodsStudy Design Methods
Retrospective Case Control Database from Vascular Anomalies Center and Online Registry between January1, 1995-December 31, 2012
Non-randomized selective case All patients with five or more cutaneous IHs were included
Exclusional Criteria Patients without diagnosed information
Indication for initiative ultrasonography screening
Patients who presented with cutaneous lesions and were suspected of visceral lesions Prior to ultrasound, the patients must had no signs or symptom suggested of HHs
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Methods
Method Study Procedure Analysis
Data Recorded Age, date of presentation, type of HH, symptom, treatment, and outcome(death)
Bivariate analysis
Fisher exact tests, chi-square tests, Mann-Whitney U-tests 95% confidence intervals(CIs) using Wilson’s method Multivariable logistic regression analysis, two-tailed p<0.05, IBM ,IPSS
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RESULTS
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Results
Initial Abdominal Ultrasound Screening
43(60%) had HHs detected by screening29(40%) had HHs be found incidentally, by
development of symptoms, or another indication
72 Patients were case with exposure
Multifocal or diffuse HHs >,= 5 cutaneous IHs Enough data to determine screening status
213 Patients with HHs was identified
Database and Online Registry Jan 1, 1995-Dec 31.2012
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TABLE 1. Analysis of Differences Between Patients with Hepatic Hemangiomas Detected Through Screening Abdominal Ultrasonography and Those Who Were Not Screened Not screened
(n = 29) Screened (n = 43)
p
Variable Age at presentation, days, median (IQR)
53 (18–112) 41 (12–90) 0.26
Diagnosis, n (%) Multifocal Diffuse
18 (62) 11 (38)
40 (93) 3 (7)
0.002
CHF, n (%) Yes No
14 (48) 15 (52)
2 (5)
41 (95)
<0.001
Hypothyroidism, n (%) Yes No Unknown
10 (34) 15 (52) 4 (14)
4 (9)
36 (84) 3 (7)
0.01
Treatment, n(%) Yes No
25 (86)
4 (14)
16 (37) 27 (63)
<0.001
Mortality, n (%) Yes No
8 (28)
21 (72)
0 (0)
43 (100)
<0.001
Follow-up, mos, median (IQR)
12 (3–40) 10 (4–23) 0.87
IQR, interquartile range
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Results• Children identified by screening were less likely to have
serious clinical sequalae from the liver hemangioma• Almost half of children in the non-screened group developed
CHF, while only two who were screen developed CHF• Hypothyroidism was also less prevalent in the screened group
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Results• Three of the diffuse patients on the non-screened group
progressed to abdominal compartment syndrome, compared with none of the diffuse patients who were screened
• None of the patients who screened died• HHs detected through screening were less likely to require
treatment
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Results
TABLE 3. Variations in Treatment Types for Those Who Were Screened and Not Screened
Treatment Not screened (n = 25) Screened (n =16) n (%)
Steroids 21 (84) 9 (56) Propranolol 7 (28) 7 (44) Interferon 3 (12) 1 (6) Vincristine 1(4) 0 (0)
Embolization 4 (16) 1 (6) Radiation 1 (4) 0 (0) Multiple 13 (52) 2 (13)
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Results• Only 1 patient who was screened required embolization for
shunts causing heart failure, while 4 of the non-screened group underwent this procedure
• Multiple treatments were more common in non-screened than in those who were screened
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TABLE 4. Analysis of Differences Between Treated Patients Who Were Screened and Not Screened
Variable Not screened (n = 25)
Screened (n = 16)
p
Age at presentation, days, median (IQR)
45 (15–109)
14 (8–66) 0.08
Diagnosis, n (%) Multifocal Diffuse
14 (56) 11 (44)
14 (88) 2 (12)
0.04
CHF, n (%) Yes No
14 (56) 11 (44)
2 (12)
14 (88)
0.008
Hypothyroidism, n (%) Yes No Unknown
10 (40) 11 (44) 4 (16)
2 (12.5) 12 (75) 2 (12.5)
0.12
Mortality, n (%) Yes No
8 (32)
17 (68)
0 (0)
16 (100)
0.01
Follow-up, mos, median (IQR)
12 (3–40) 9 (3–20) 0.95
IQR, interquartile range.
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Results• Screened children were more often diagnosed with multifocal
lesions and had lower incidence of CHF• Multivariable logistic regression analysis confirmed that,
independent of the presence of CHF and disease pattern, screening was a significant predictor of reduced mortality
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DISCUSSION
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Discussion
Aim• Because of there had been long known of the association of IHs and HHs,
the recommendation of the screening was created. • This study aim to find the outcome of the recommendation to be
screened in the patients with multiple cutaneous and HHs
Methods• The patients to be studied was grouped based on the recent study, said
that 16% incidence of HHs in infants <6 months with 5 or more cutaneous hemangioma lesions
• The case was grouped to be screened or non-screened depended the screening purposed or for another indication for initial screening
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Strenght
Results Discussion Those who were screened were less likely to develop CHF or hypothyroidism and less likely to have been treated for HHs
It is likely that by early detect the lesions, and referring to specialists, patients are followed, and treated appropriately before the lesions become progress or symptomatic
Screening may detect lesions that would have been clinically silent and never be discovered The most serious type of HH is diffuse, as these can lead to abdominal compartment syndrome and death.
No deaths occurred in the group identified through screening Screening was a significant predictor of lower mortality in treated patients
Ultrasonography is a noninvasive, inexpensive, and widely available for use as a screening tool for children with multiple cutaneous IHs.
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Discussion
Weak Points
Discussion
Retrospective It is impossible to perform a prospective randomized trial because screening would be withheld from some patients.
Careful analysis of the registry provides the best chance to find out, whether screening gives better outcomes than simply detection.
Selective Bias
By separately analyzing just patients who required therapy, the study excluded those that would likely have never been detected in the non-screening.
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DiscussionWeak Points
Discussion
Sampling Error
There were only a few diffuse HHs in the screened group. One may surmise that
some of multifocal lesions patients detected by screening were treated, to prevent progression to the diffuse pattern.
Informative Bias Outside the setting hospital, there are many patients with HHs, but the database were incomplete
We do not know the specific indication for treatment. There may be another selective bias for some patients
The patients were selected with 5 or more cutaneous lesions to match the number that the Hemangioma Investigator Group recommends for screening
The study does not address whether five is the best screening threshold.
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CONCLUSION
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Conclusion
‘Infants HHs detected through screening were less likely to develop serious clinical symptoms and had reduced mortality’
‘Screening was an independent predictor of lower mortality in severe symptomatic children’
‘Screening may result in earlier treatment, early referral, and closure follow-up before the progression of the lesions‘
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STUDY APPLICATIONS
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Study Applications
Study Application
Study Discussions Applicationns Hemangiomas are the most common tumors found in infants
Incidence of the findings was 10%. In Thailand ratio between girl and boy is 2.1:1 and 58% is CHs
Early detection of lesions, early suspect of probable of complication and early referral to appropriated physician may be the best way to reduce morbid & mortality
Clinical characteristics of hemangiomas
Progression of hemangiomas Complication Associated Syndromes Treatment
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THANK YOU
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References• คู่มอืโรคผิวหนังเด็ก; อมรศร ีชุณหรศัมิ.์ ชมรมแพทยผ์ิวหนังเด็ก
แหง่ประเทศไทย. พมิครัง้ท่ี 1 มกราคม 2559 • ตำารา โรคผิวหนังเด็ก เล่ม 1; ศิรวิรรณ วนานุกลู. ภาควชิากมุาร
เวชศาสตร.์ คณะแพทยศาสตรจุ์ฬาลงกรณ์มหาวทิยาลัย. พมิพ์ครัง้ท่ี 1 มกราคม 2557