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Journal club review; Pediatrics

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Journal club review: Impact of Screening for Hepatic Hemangiomas in Patients with Multiple Cutaneous Infantile Hemangiomas

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Journal club review; Pediatrics

  1. 1. Journal Club Presentation Impact of Screening for Hepatic Hemangiomas in Patients with Multiple Cutaneous Infantile Hemangiomas Pediatric Dermatology 1-5, 2015 Authors: Kristy L. Rialon, Rudy Murillo, Rebecca D. Fevurly, et. al. By Ext. Sripada Kriangkhajorn Faculty of Medicine, Srinakharinwirot University
  2. 2. 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
  3. 3. Abstract • The study aim to determine whether the hemangiomas found through screening had improved clinical outcomes in children with multiples cutaneous IHs
  4. 4. BACKGROUND
  5. 5. 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)
  6. 6. 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
  7. 7. 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 IHs Multifocal Diffuse
  8. 8. Study Objective ‘To determine whether the hemangiomas found through screening had improved clinical outcomes in children with multiples cutaneous IHs’
  9. 9. METHODS
  10. 10. 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
  11. 11. Methods Study 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
  12. 12. 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
  13. 13. RESULTS
  14. 14. Results Initial Abdominal Ultrasound Screening 43(60%) had HHs detected by screening 29(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
  15. 15. 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
  16. 16. 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
  17. 17. 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
  18. 18. 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)
  19. 19. 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
  20. 20. 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.
  21. 21. 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
  22. 22. DISCUSSION
  23. 23. 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
  24. 24. 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.
  25. 25. 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.
  26. 26. Discussion Weak 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.
  27. 27. CONCLUSION
  28. 28. 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‘
  29. 29. STUDY APPLICATIONS
  30. 30. 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
  31. 31. THANK YOU
  32. 32. References • คู่มือโรคผิวหนังเด็ก; อมรศรี ชุณหรัศมิ์. ชมรมแพทย์ผิวหนังเด็กแห่งประเทศไทย. พิมครั้งที่ 1 มกราคม 2559 • ตารา โรคผิวหนังเด็ก เล่ม 1; ศิริวรรณ วนานุกูล. ภาควิชากุมารเวชศาสตร์. คณะแพทยศาสตร์ จุฬาลงกรณ์มหาวิทยาลัย. พิมพ์ครั้งที่ 1 มกราคม 2557

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