{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,17]],"date-time":"2026-08-17T22:15:13Z","timestamp":1787004913660,"version":"build-2736575974"},"reference-count":40,"publisher":"Georg Thieme Verlag KG","issue":"05","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Eur J Pediatr Surg"],"published-print":{"date-parts":[[2023,10]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Background\u2003The gold standard for diagnosing Hirschsprung disease (HD) in patients younger than 6 months is pathological examination of rectal suction biopsy (RSB). The aim of this study was to gain insight into the following: (1) complications following RSB, (2) final diagnosis of patients referred for RSB, and (3) factors associated with HD.<\/jats:p>\n                  <jats:p>Methods\u2003Patients suspected of HD referred for RSB at our center were analyzed retrospectively. Severity of complications of RSB was assessed using Clavien\u2013Dindo (CD) grading. Factors associated with HD were tested using multivariate logistic regression analysis.<\/jats:p>\n                  <jats:p>Results\u2003From 2000 to 2021, 371 patients underwent RSB because of infrequent defecation, at a median age of 44 days. Three patients developed ongoing rectal bleeding (0.8%) graded CD1. Most frequent final diagnoses were: HD (n\u2009=\u2009151, 40.7%), functional constipation (n\u2009=\u2009113, 31%), idiopathic meconium ileus (n\u2009=\u200911, 3%), and food intolerance (n\u2009=\u200911, 3%). Associated factors for HD were male sex (odds ratio [OR], 3.19; confidence interval [CI], 1.56\u20136.53), presence of syndrome (OR, 7.18; CI, 1.63\u201331.69), younger age at time of RSB (OR, 0.98; CI, 0.85\u20130.98), meconium passage for more than 48\u2009hours (OR, 3.15; CI, 1.51\u20136.56), distended abdomen (OR, 2.09; CI, 1.07\u20134.07), bilious vomiting (OR, 6.39; CI, 3.28\u201312.47), and failure to thrive (OR, 8.46; CI, 2.11\u201334.02) (model R\n          2\u2009=\u20090.566).<\/jats:p>\n                  <jats:p>Conclusion\u2003RSB is a safe procedure with few and only minor complications. In the majority of patients referred for RSB under the age of 6 months, HD was found followed by a functional cause for the defecation problems. RSB should be obtained on a low threshold in all patients under the age of 6 months with the suspicion of HD.<\/jats:p>","DOI":"10.1055\/s-0043-1760839","type":"journal-article","created":{"date-parts":[[2023,2,1]],"date-time":"2023-02-01T19:06:08Z","timestamp":1675278368000},"page":"360-366","source":"Crossref","is-referenced-by-count":5,"title":["Diagnosing Hirschsprung Disease in Children Younger than 6 Months of Age: Insights in Incidence of Complications of Rectal Suction Biopsy and Other Final Diagnoses"],"prefix":"10.1055","volume":"33","author":[{"given":"Lieke","family":"Beltman","sequence":"additional","affiliation":[{"name":"Department of Pediatric Surgery, Amsterdam UMC location University of Amsterdam, Amsterdam, the Netherlands"},{"name":"Department of Pediatrics, Emma Children's Hospital Amsterdam UMC Follow-Me Program & Emma Neuroscience Group, Amsterdam UMC location University of Amsterdam, Amsterdam, the Netherlands"},{"name":"Amsterdam Gastroenterology Endocrinology and Metabolism Research Institute, Amsterdam, the Netherlands"},{"name":"Amsterdam Reproduction and Development Research Institute, Amsterdam, the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0599-7183","authenticated-orcid":false,"given":"Hosnieya","family":"Labib","sequence":"additional","affiliation":[{"name":"Department of Pediatric Surgery, Amsterdam UMC location University of Amsterdam, Amsterdam, the Netherlands"},{"name":"Amsterdam Gastroenterology Endocrinology and Metabolism Research Institute, Amsterdam, the Netherlands"},{"name":"Amsterdam Reproduction and Development Research Institute, Amsterdam, the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marit","family":"Masselink","sequence":"additional","affiliation":[{"name":"Department of Pediatric Surgery, Amsterdam UMC location University of Amsterdam, Amsterdam, the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Manouk","family":"Backes","sequence":"additional","affiliation":[{"name":"Department of Pediatric Surgery, Amsterdam UMC location University of Amsterdam, Amsterdam, the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marc A.","family":"Benninga","sequence":"additional","affiliation":[{"name":"Amsterdam Gastroenterology Endocrinology and Metabolism Research Institute, Amsterdam, the Netherlands"},{"name":"Department of Pediatric Gastroenterology and Nutrition, Amsterdam UMC location University of Amsterdam, Amsterdam, the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joris J.T.H.","family":"Roelofs","sequence":"additional","affiliation":[{"name":"Department of Pathology, Amsterdam UMC location University of Amsterdam, Amsterdam, the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J. Patrick","family":"van der Voorn","sequence":"additional","affiliation":[{"name":"Department of Pathology, Amsterdam UMC location Vrije Universiteit Amsterdam, Amsterdam, the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joost","family":"van Schuppen","sequence":"additional","affiliation":[{"name":"Department of Radiology and Nuclear Medicine, Amsterdam UMC location University of Amsterdam, Amsterdam, the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jaap","family":"Oosterlaan","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Emma Children's Hospital Amsterdam UMC Follow-Me Program & Emma Neuroscience Group, Amsterdam UMC location University of Amsterdam, Amsterdam, the Netherlands"},{"name":"Amsterdam Reproduction and Development Research Institute, Amsterdam, the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"L.W. Ernest","family":"van Heurn","sequence":"additional","affiliation":[{"name":"Department of Pediatric Surgery, Amsterdam UMC location University of Amsterdam, Amsterdam, the Netherlands"},{"name":"Amsterdam Gastroenterology Endocrinology and Metabolism Research Institute, Amsterdam, the Netherlands"},{"name":"Amsterdam Reproduction and Development Research Institute, Amsterdam, the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joep P.M.","family":"Derikx","sequence":"additional","affiliation":[{"name":"Department of Pediatric Surgery, Amsterdam UMC location University of Amsterdam, Amsterdam, the Netherlands"},{"name":"Amsterdam Gastroenterology Endocrinology and Metabolism Research Institute, Amsterdam, the Netherlands"},{"name":"Amsterdam Reproduction and Development Research Institute, Amsterdam, the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"194","published-online":{"date-parts":[[2023,2,1]]},"reference":[{"issue":"09","key":"ref1","doi-asserted-by":"crossref","first-page":"695","DOI":"10.1002\/bdra.23269","article-title":"Hirschsprung's disease prevalence in Europe: a register based study","volume":"100","author":"K E Best","year":"2014","journal-title":"Birth Defects Res A Clin Mol Teratol"},{"issue":"15","key":"ref2","doi-asserted-by":"crossref","first-page":"1421","DOI":"10.1056\/NEJMoa1706594","article-title":"Molecular genetic anatomy and risk profile of Hirschsprung's disease","volume":"380","author":"J M Tilghman","year":"2019","journal-title":"N Engl J Med"},{"issue":"03","key":"ref3","doi-asserted-by":"crossref","first-page":"368","DOI":"10.1097\/MOP.0b013e328360c2a0","article-title":"Hirschsprung disease","volume":"25","author":"J C Langer","year":"2013","journal-title":"Curr Opin Pediatr"},{"issue":"06","key":"ref4","doi-asserted-by":"crossref","first-page":"439","DOI":"10.1007\/s00383-002-0934-9","article-title":"The constipated child: how likely is Hirschsprung's disease?","volume":"19","author":"A R Khan","year":"2003","journal-title":"Pediatr Surg Int"},{"issue":"01","key":"ref5","doi-asserted-by":"crossref","first-page":"97","DOI":"10.1055\/s-0038-1675771","article-title":"Too many biopsies performed to rule out Hirschsprung's disease: but it is worth doing them","volume":"29","author":"L AF Phillips","year":"2019","journal-title":"Eur J Pediatr Surg"},{"key":"ref6","volume-title":"Differential diagnosis","author":"A Tanaka","year":"2019"},{"issue":"03","key":"ref7","doi-asserted-by":"crossref","first-page":"245","DOI":"10.1007\/s11894-007-0026-z","article-title":"Symptomatology, pathophysiology, diagnostic work-up, and treatment of Hirschsprung disease in infancy and childhood","volume":"9","author":"F de Lorijn","year":"2007","journal-title":"Curr Gastroenterol Rep"},{"issue":"01","key":"ref8","doi-asserted-by":"crossref","first-page":"8","DOI":"10.1177\/1093526619892351","article-title":"Diagnosis of Hirschsprung disease","volume":"23","author":"L Ambartsumyan","year":"2020","journal-title":"Pediatr Dev Pathol"},{"issue":"01","key":"ref9","doi-asserted-by":"crossref","first-page":"96","DOI":"10.1055\/s-0036-1593991","article-title":"European Paediatric Surgeons' Association survey on the management of Hirschsprung disease","volume":"27","author":"A Zani","year":"2017","journal-title":"Eur J Pediatr Surg"},{"issue":"06","key":"ref10","doi-asserted-by":"crossref","first-page":"1043","DOI":"10.1016\/j.jpedsurg.2006.01.070","article-title":"Rectal suction biopsy in the diagnosis of intestinal dysganglionoses: 5-year experience with Solo-RBT in 389 patients","volume":"41","author":"A Pini-Prato","year":"2006","journal-title":"J Pediatr Surg"},{"key":"ref11","doi-asserted-by":"crossref","first-page":"121","DOI":"10.1007\/s003830050264","article-title":"Rectal biopsy for Hirschsprung's disease: what is the optimum method?","volume":"13","author":"N K Alizai","year":"1998","journal-title":"Pediatr Surg Int"},{"issue":"12","key":"ref12","doi-asserted-by":"crossref","first-page":"716","DOI":"10.1111\/j.1440-1754.2004.00519.x","article-title":"Difficulties in making the diagnosis of Hirschsprung disease in early infancy","volume":"40","author":"Y Nofech-Mozes","year":"2004","journal-title":"J Paediatr Child Health"},{"issue":"02","key":"ref13","doi-asserted-by":"crossref","first-page":"133","DOI":"10.1016\/j.pedneo.2012.01.011","article-title":"Clinical impacts of delayed diagnosis of Hirschsprung's disease in newborn infants","volume":"53","author":"C C Lee","year":"2012","journal-title":"Pediatr Neonatol"},{"issue":"11","key":"ref14","doi-asserted-by":"crossref","first-page":"561","DOI":"10.1016\/j.jpedsurg.2022.02.022","article-title":"Risk factors for complications in patients with Hirschsprung disease while awaiting surgery: beware of bowel perforation","volume":"57","author":"L Beltman","year":"2022","journal-title":"J Pediatr Surg"},{"issue":"09","key":"ref15","doi-asserted-by":"crossref","first-page":"821","DOI":"10.1007\/s00383-015-3742-8","article-title":"Rectal suction biopsy for the diagnosis of Hirschsprung's disease: a systematic review of diagnostic accuracy and complications","volume":"31","author":"F Friedmacher","year":"2015","journal-title":"Pediatr Surg Int"},{"issue":"04","key":"ref16","doi-asserted-by":"crossref","first-page":"494","DOI":"10.1097\/MPG.0000000000003041","article-title":"Rectal biopsy technique for the diagnosis of Hirschsprung disease in children: a systematic review and meta-analysis","volume":"72","author":"G T Comes","year":"2021","journal-title":"J Pediatr Gastroenterol Nutr"},{"issue":"04","key":"ref17","doi-asserted-by":"crossref","first-page":"51","DOI":"10.47338\/jns.v2.63","article-title":"Hirschsprung's disease in newborns","volume":"2","author":"S N Bhatnagar","year":"2013","journal-title":"J Neonatal Surg"},{"issue":"08","key":"ref18","doi-asserted-by":"crossref","first-page":"618","DOI":"10.1007\/s12098-017-2371-8","article-title":"Hirschsprung disease - current diagnosis and management","volume":"84","author":"K Das","year":"2017","journal-title":"Indian J Pediatr"},{"issue":"02","key":"ref19","first-page":"124","article-title":"Diagnostic efficacy of rectal suction biopsy with regard to weight in children investigated for Hirschsprung's disease","volume":"9","author":"E Fransson","year":"2022","journal-title":"Children (Basel)"},{"issue":"01","key":"ref20","doi-asserted-by":"crossref","first-page":"28","DOI":"10.2350\/13-10-1387-OA.1","article-title":"Calretinin-immunoreactive mucosal innervation in very short-segment Hirschsprung disease: a potentially misleading observation","volume":"17","author":"R P Kapur","year":"2014","journal-title":"Pediatr Dev Pathol"},{"issue":"02","key":"ref21","doi-asserted-by":"crossref","first-page":"245","DOI":"10.1007\/s00428-022-03334-3","article-title":"Diagnostic accuracy of calretinin and acetylcholinesterase staining of rectal suction biopsies in Hirschsprung disease examined by unexperienced pathologists","volume":"481","author":"L Beltman","year":"2022","journal-title":"Virchows Arch"},{"issue":"05","key":"ref22","doi-asserted-by":"crossref","first-page":"496","DOI":"10.1097\/01.mpg.0000214164.90939.92","article-title":"Diagnostic tests in Hirschsprung disease: a systematic review","volume":"42","author":"F de Lorijn","year":"2006","journal-title":"J Pediatr Gastroenterol Nutr"},{"issue":"02","key":"ref23","doi-asserted-by":"crossref","first-page":"205","DOI":"10.1097\/01.sla.0000133083.54934.ae","article-title":"Classification of surgical complications: a new proposal with evaluation in a cohort of 6336 patients and results of a survey","volume":"240","author":"D Dindo","year":"2004","journal-title":"Ann Surg"},{"issue":"02","key":"ref24","first-page":"207","article-title":"Contrast enema for Hirschsprung disease investigation: diagnostic accuracy and validity for subsequent diagnostic and surgical planning","volume":"26","author":"G Frongia","year":"2016","journal-title":"Eur J Pediatr Surg"},{"issue":"01","key":"ref25","doi-asserted-by":"crossref","first-page":"2","DOI":"10.1097\/01.PCC.0000149131.72248.E6","article-title":"International pediatric sepsis consensus conference: definitions for sepsis and organ dysfunction in pediatrics","volume":"6","author":"B Goldstein","year":"2005","journal-title":"Pediatr Crit Care Med"},{"key":"ref26","doi-asserted-by":"publisher","DOI":"10.1053\/j.gastro.2016.02.015","article-title":"Functional disorders: children and adolescents","author":"J S Hyams","year":"2016","journal-title":"Gastroenterology"},{"key":"ref27","doi-asserted-by":"publisher","DOI":"10.1053\/j.gastro.2016.02.016","article-title":"Childhood functional gastrointestinal disorders: neonate\/toddler","author":"M A Benninga","year":"2016","journal-title":"Gastroenterology"},{"issue":"02","key":"ref28","doi-asserted-by":"crossref","first-page":"121","DOI":"10.1055\/s-0037-1609027","article-title":"Meconium ileus","volume":"31","author":"J HT Waldhausen","year":"2018","journal-title":"Clin Colon Rectal Surg"},{"key":"ref29","doi-asserted-by":"crossref","first-page":"f7204","DOI":"10.1136\/bmj.f7204","article-title":"Diagnosis, management, and prevention of rotavirus gastroenteritis in children","volume":"347","author":"U D Parashar","year":"2013","journal-title":"BMJ"},{"issue":"02","key":"ref30","doi-asserted-by":"crossref","first-page":"409","DOI":"10.1016\/S0031-3955(05)70413-2","article-title":"Necrotizing enterocolitis: the search for a unifying pathogenic theory leading to prevention","volume":"43","author":"J Neu","year":"1996","journal-title":"Pediatr Clin North Am"},{"issue":"10","key":"ref31","doi-asserted-by":"crossref","first-page":"1942","DOI":"10.1016\/j.jpedsurg.2018.01.005","article-title":"Full-thickness rectal biopsy in children suspicious for Hirschsprung's disease is safe and yields a low number of insufficient biopsies","volume":"53","author":"N Bj\u00f8rn","year":"2018","journal-title":"J Pediatr Surg"},{"issue":"11","key":"ref32","doi-asserted-by":"crossref","first-page":"729","DOI":"10.1136\/jmg.38.11.729","article-title":"Hirschsprung disease, associated syndromes, and genetics: a review","volume":"38","author":"J Amiel","year":"2001","journal-title":"J Med Genet"},{"key":"ref33","volume-title":"Histopathological diagnosis and differential diagnosis of Hirschsprung's disease","author":"W Meier-Ruge","year":"2008"},{"issue":"03","key":"ref34","first-page":"568","article-title":"A genetic study of Hirschsprung disease","volume":"46","author":"J A Badner","year":"1990","journal-title":"Am J Hum Genet"},{"issue":"06","key":"ref35","doi-asserted-by":"crossref","first-page":"503","DOI":"10.1111\/j.1399-0004.1985.tb00417.x","article-title":"Hirschsprung disease: a genetic study","volume":"28","author":"K L Garver","year":"1985","journal-title":"Clin Genet"},{"key":"ref36","doi-asserted-by":"crossref","DOI":"10.1007\/978-3-030-15647-3","volume-title":"Hirschsprung's Disease and Allied Disorders","author":"P Puri","year":"2019"},{"issue":"01","key":"ref37","doi-asserted-by":"crossref","first-page":"73","DOI":"10.1016\/j.bpg.2010.12.005","article-title":"Childhood constipation; an overview of genetic studies and associated syndromes","volume":"25","author":"B Peeters","year":"2011","journal-title":"Best Pract Res Clin Gastroenterol"},{"issue":"08","key":"ref38","doi-asserted-by":"crossref","first-page":"722","DOI":"10.1136\/archdischild-2016-311872","article-title":"Hirschsprung's disease in the UK and Ireland: incidence and anomalies","volume":"102","author":"T J Bradnock","year":"2017","journal-title":"Arch Dis Child"},{"issue":"08","key":"ref39","doi-asserted-by":"crossref","first-page":"1209","DOI":"10.1053\/jpsu.2000.8728","article-title":"Transanal endorectal coloanal surgery for Hirschsprung's disease: experience in two centers","volume":"35","author":"K R Shankar","year":"2000","journal-title":"J Pediatr Surg"},{"issue":"03","key":"ref40","doi-asserted-by":"crossref","first-page":"412","DOI":"10.1053\/jpsu.2003.50070","article-title":"Hirschsprung's disease: increasing the odds of a positive rectal biopsy result","volume":"38","author":"N A Lewis","year":"2003","journal-title":"J Pediatr Surg"}],"container-title":["European Journal of Pediatric Surgery"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/www.thieme-connect.de\/products\/ejournals\/pdf\/10.1055\/s-0043-1760839.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,10,10]],"date-time":"2023-10-10T19:27:14Z","timestamp":1696966034000},"score":1,"resource":{"primary":{"URL":"http:\/\/www.thieme-connect.de\/DOI\/DOI?10.1055\/s-0043-1760839"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,2,1]]},"references-count":40,"journal-issue":{"issue":"05","published-online":{"date-parts":[[2023,10,10]]},"published-print":{"date-parts":[[2023,10]]}},"URL":"https:\/\/doi.org\/10.1055\/s-0043-1760839","archive":["Portico","CLOCKSS"],"relation":{},"ISSN":["0939-7248","1439-359X"],"issn-type":[{"value":"0939-7248","type":"print"},{"value":"1439-359X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,2,1]]}}}