Primary sclerosing cholangitis in children with inflammatory bowel disease: An ESPGHAN position paper from the Hepatology Committee and the IBD Porto group

Objective We aimed to provide an evidence‐supported approach to diagnose, monitor, and treat children with inflammatory bowel disease (IBD) and primary sclerosing cholangitis (PSC). Methods The core group formulated seven PICO‐structured clinical questions. A systematic literature search from incept...

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Vydáno v:Journal of pediatric gastroenterology and nutrition Ročník 80; číslo 2; s. 374 - 393
Hlavní autoři: Rheenen, Patrick F., Kolho, Kaija‐Leena, Russell, Richard K., Aloi, Marina, Deganello, Annamaria, Hussey, Séamus, Junge, Norman, De Laffolie, Jan, Deneau, Mark R., Fitzpatrick, Emer, Griffiths, Anne M., Hojsak, Iva, Nicastro, Emanuele, Nita, Andreia, Pakarinen, Mikko, Ricciuto, Amanda, Ridder, Lissy, Sonzogni, Aurelio, Tenca, Andrea, Samyn, Marianne, Indolfi, Giuseppe
Médium: Journal Article
Jazyk:angličtina
Vydáno: United States 01.02.2025
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ISSN:0277-2116, 1536-4801, 1536-4801
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Abstract Objective We aimed to provide an evidence‐supported approach to diagnose, monitor, and treat children with inflammatory bowel disease (IBD) and primary sclerosing cholangitis (PSC). Methods The core group formulated seven PICO‐structured clinical questions. A systematic literature search from inception to December 2022 was conducted by a medical librarian using MEDLINE and EMBASE. Core messages from the literature were phrased as position statements and then circulated to a sounding board composed of international experts in pediatric gastroenterology and hepatology, histopathology, adult gastroenterology and hepatology, radiology, and surgery. Statements reaching at least 80% agreement were considered as final. The other statements were refined and then subjected to a second online vote or rejection. Results Regular screening for gamma‐glutamyltransferase (GGT) is essential for detecting possible biliary disease in children with IBD. MR cholangiopancreatography is the radiological modality of choice for establishing the diagnosis of PSC. Liver biopsy is relevant in the evaluation of small duct PSC or autoimmune hepatitis. Children who do not have known IBD at the time of PSC diagnosis should undergo initial screening with fecal calprotectin for asymptomatic colitis, and then at least once yearly thereafter. Children with a cholestatic liver enzyme profile can be considered for treatment with ursodeoxycholic acid and can continue if there is a meaningful reduction or normalization in GGT. Oral vancomycin may have a beneficial effect on GGT and intestinal inflammation, but judicious use is recommended due to the lack of long‐term studies. Children with PSC–IBD combined with convincing features of autoimmune hepatitis may benefit from corticosteroids and antimetabolites. Conclusions We present state‐of‐the‐art guidance on the diagnostic criteria, follow‐up strategies, and therapeutic strategies and point out research gaps in children and adolescents with PSC–IBD. What is Known • Primary sclerosing cholangitis (PSC) is an uncommon condition in childhood‐onset inflammatory bowel disease (IBD) with severe outcomes, and there is no consensus on diagnostic and treatment approaches. What is New • The European Society for Paediatric Gastroenterology, Hepatology and Nutrition Hepatology Committee and the Pediatric IBD Porto group formulated clinical questions considered to be relevant for the care of children with PSC‐IBD. • An evidence‐supported approach to diagnose, monitor, and treat children with PSC–IBD is presented here, as well as suggestions for future studies based on current knowledge gaps.
AbstractList We aimed to provide an evidence-supported approach to diagnose, monitor, and treat children with inflammatory bowel disease (IBD) and primary sclerosing cholangitis (PSC).OBJECTIVEWe aimed to provide an evidence-supported approach to diagnose, monitor, and treat children with inflammatory bowel disease (IBD) and primary sclerosing cholangitis (PSC).The core group formulated seven PICO-structured clinical questions. A systematic literature search from inception to December 2022 was conducted by a medical librarian using MEDLINE and EMBASE. Core messages from the literature were phrased as position statements and then circulated to a sounding board composed of international experts in pediatric gastroenterology and hepatology, histopathology, adult gastroenterology and hepatology, radiology, and surgery. Statements reaching at least 80% agreement were considered as final. The other statements were refined and then subjected to a second online vote or rejection.METHODSThe core group formulated seven PICO-structured clinical questions. A systematic literature search from inception to December 2022 was conducted by a medical librarian using MEDLINE and EMBASE. Core messages from the literature were phrased as position statements and then circulated to a sounding board composed of international experts in pediatric gastroenterology and hepatology, histopathology, adult gastroenterology and hepatology, radiology, and surgery. Statements reaching at least 80% agreement were considered as final. The other statements were refined and then subjected to a second online vote or rejection.Regular screening for gamma-glutamyltransferase (GGT) is essential for detecting possible biliary disease in children with IBD. MR cholangiopancreatography is the radiological modality of choice for establishing the diagnosis of PSC. Liver biopsy is relevant in the evaluation of small duct PSC or autoimmune hepatitis. Children who do not have known IBD at the time of PSC diagnosis should undergo initial screening with fecal calprotectin for asymptomatic colitis, and then at least once yearly thereafter. Children with a cholestatic liver enzyme profile can be considered for treatment with ursodeoxycholic acid and can continue if there is a meaningful reduction or normalization in GGT. Oral vancomycin may have a beneficial effect on GGT and intestinal inflammation, but judicious use is recommended due to the lack of long-term studies. Children with PSC-IBD combined with convincing features of autoimmune hepatitis may benefit from corticosteroids and antimetabolites.RESULTSRegular screening for gamma-glutamyltransferase (GGT) is essential for detecting possible biliary disease in children with IBD. MR cholangiopancreatography is the radiological modality of choice for establishing the diagnosis of PSC. Liver biopsy is relevant in the evaluation of small duct PSC or autoimmune hepatitis. Children who do not have known IBD at the time of PSC diagnosis should undergo initial screening with fecal calprotectin for asymptomatic colitis, and then at least once yearly thereafter. Children with a cholestatic liver enzyme profile can be considered for treatment with ursodeoxycholic acid and can continue if there is a meaningful reduction or normalization in GGT. Oral vancomycin may have a beneficial effect on GGT and intestinal inflammation, but judicious use is recommended due to the lack of long-term studies. Children with PSC-IBD combined with convincing features of autoimmune hepatitis may benefit from corticosteroids and antimetabolites.We present state-of-the-art guidance on the diagnostic criteria, follow-up strategies, and therapeutic strategies and point out research gaps in children and adolescents with PSC-IBD.CONCLUSIONSWe present state-of-the-art guidance on the diagnostic criteria, follow-up strategies, and therapeutic strategies and point out research gaps in children and adolescents with PSC-IBD.
Objective We aimed to provide an evidence‐supported approach to diagnose, monitor, and treat children with inflammatory bowel disease (IBD) and primary sclerosing cholangitis (PSC). Methods The core group formulated seven PICO‐structured clinical questions. A systematic literature search from inception to December 2022 was conducted by a medical librarian using MEDLINE and EMBASE. Core messages from the literature were phrased as position statements and then circulated to a sounding board composed of international experts in pediatric gastroenterology and hepatology, histopathology, adult gastroenterology and hepatology, radiology, and surgery. Statements reaching at least 80% agreement were considered as final. The other statements were refined and then subjected to a second online vote or rejection. Results Regular screening for gamma‐glutamyltransferase (GGT) is essential for detecting possible biliary disease in children with IBD. MR cholangiopancreatography is the radiological modality of choice for establishing the diagnosis of PSC. Liver biopsy is relevant in the evaluation of small duct PSC or autoimmune hepatitis. Children who do not have known IBD at the time of PSC diagnosis should undergo initial screening with fecal calprotectin for asymptomatic colitis, and then at least once yearly thereafter. Children with a cholestatic liver enzyme profile can be considered for treatment with ursodeoxycholic acid and can continue if there is a meaningful reduction or normalization in GGT. Oral vancomycin may have a beneficial effect on GGT and intestinal inflammation, but judicious use is recommended due to the lack of long‐term studies. Children with PSC–IBD combined with convincing features of autoimmune hepatitis may benefit from corticosteroids and antimetabolites. Conclusions We present state‐of‐the‐art guidance on the diagnostic criteria, follow‐up strategies, and therapeutic strategies and point out research gaps in children and adolescents with PSC–IBD. What is Known • Primary sclerosing cholangitis (PSC) is an uncommon condition in childhood‐onset inflammatory bowel disease (IBD) with severe outcomes, and there is no consensus on diagnostic and treatment approaches. What is New • The European Society for Paediatric Gastroenterology, Hepatology and Nutrition Hepatology Committee and the Pediatric IBD Porto group formulated clinical questions considered to be relevant for the care of children with PSC‐IBD. • An evidence‐supported approach to diagnose, monitor, and treat children with PSC–IBD is presented here, as well as suggestions for future studies based on current knowledge gaps.
We aimed to provide an evidence-supported approach to diagnose, monitor, and treat children with inflammatory bowel disease (IBD) and primary sclerosing cholangitis (PSC). The core group formulated seven PICO-structured clinical questions. A systematic literature search from inception to December 2022 was conducted by a medical librarian using MEDLINE and EMBASE. Core messages from the literature were phrased as position statements and then circulated to a sounding board composed of international experts in pediatric gastroenterology and hepatology, histopathology, adult gastroenterology and hepatology, radiology, and surgery. Statements reaching at least 80% agreement were considered as final. The other statements were refined and then subjected to a second online vote or rejection. Regular screening for gamma-glutamyltransferase (GGT) is essential for detecting possible biliary disease in children with IBD. MR cholangiopancreatography is the radiological modality of choice for establishing the diagnosis of PSC. Liver biopsy is relevant in the evaluation of small duct PSC or autoimmune hepatitis. Children who do not have known IBD at the time of PSC diagnosis should undergo initial screening with fecal calprotectin for asymptomatic colitis, and then at least once yearly thereafter. Children with a cholestatic liver enzyme profile can be considered for treatment with ursodeoxycholic acid and can continue if there is a meaningful reduction or normalization in GGT. Oral vancomycin may have a beneficial effect on GGT and intestinal inflammation, but judicious use is recommended due to the lack of long-term studies. Children with PSC-IBD combined with convincing features of autoimmune hepatitis may benefit from corticosteroids and antimetabolites. We present state-of-the-art guidance on the diagnostic criteria, follow-up strategies, and therapeutic strategies and point out research gaps in children and adolescents with PSC-IBD.
Author Indolfi, Giuseppe
Nita, Andreia
Kolho, Kaija‐Leena
Pakarinen, Mikko
Samyn, Marianne
Deganello, Annamaria
Deneau, Mark R.
Rheenen, Patrick F.
Nicastro, Emanuele
Sonzogni, Aurelio
Russell, Richard K.
Hussey, Séamus
Ridder, Lissy
Hojsak, Iva
Fitzpatrick, Emer
Junge, Norman
Tenca, Andrea
De Laffolie, Jan
Ricciuto, Amanda
Aloi, Marina
Griffiths, Anne M.
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  orcidid: 0000-0003-3867-2665
  surname: Rheenen
  fullname: Rheenen, Patrick F.
  email: p.f.van.rheenen@umcg.nl
  organization: University Medical Center Groningen, University of Groningen
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  givenname: Kaija‐Leena
  surname: Kolho
  fullname: Kolho, Kaija‐Leena
  organization: Children's Hospital, University of Helsinki and HUS
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  givenname: Richard K.
  surname: Russell
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  organization: Royal Hospital for Children and Young People
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  givenname: Marina
  surname: Aloi
  fullname: Aloi, Marina
  organization: Sapienza University of Rome ‐ Umberto I Hospital
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  organization: King's College Hospital, School of Biomedical Engineering and Imaging Sciences, King's College London
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  organization: Hannover Medical School
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  organization: Justus Liebig University Giessen
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  givenname: Mark R.
  surname: Deneau
  fullname: Deneau, Mark R.
  organization: University of Utah and Intermountain Healthcare Primary Children's Hospital
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  givenname: Emer
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  fullname: Fitzpatrick, Emer
  organization: Children's Health Ireland and University College Dublin
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  organization: University of Toronto
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  fullname: Hojsak, Iva
  organization: University of Zagreb Medical School
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  givenname: Emanuele
  surname: Nicastro
  fullname: Nicastro, Emanuele
  organization: Gastroenterology and Transplantation, Hospital Papa Giovanni XXIII
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  organization: Great Ormond Street Hospital
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  givenname: Mikko
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  fullname: Pakarinen, Mikko
  organization: University of Helsinki and Helsinki University Hospital
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  fullname: Ricciuto, Amanda
  organization: University of Toronto
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  organization: Erasmus University Medical Center Sophia Children's Hospital
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  organization: ASST Bergamo Est
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  organization: Helsinki University and Helsinki University Hospital HUS, Abdominal Center
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  organization: King's College Hospital
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  fullname: Indolfi, Giuseppe
  organization: Meyer Children's Hospital IRCCS
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CitedBy_id crossref_primary_10_1002_jpn3_70180
crossref_primary_10_1016_j_jhep_2025_03_017
crossref_primary_10_1136_archdischild_2024_327998
crossref_primary_10_1002_jpn3_70097
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Keywords colorectal carcinoma
surveillance colonoscopy
ursodeoxycholic acid
MRCP
calprotectin
Language English
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2024 The Author(s). Journal of Pediatric Gastroenterology and Nutrition published by Wiley Periodicals LLC on behalf of European Society for Pediatric Gastroenterology, Hepatology, and Nutrition and North American Society for Pediatric Gastroenterology, Hepatology, and Nutrition.
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Notes CME module may be found at
https://learnonline.naspghan.org/jpgn2
Marianne Samyn and Giuseppe Indolfi contributed equally and share the last authorship.
ESPGHAN is not responsible for the practices of physicians and provides guidelines and position papers as indicators of best practice only. Diagnosis and treatment is at the discretion of the healthcare provider.
Disclaimers
Patrick F. van Rheenen, Kaija‐Leena Kolho, and Richard K. Russell contributed equally and share the first authorship.
Although this paper is produced by the ESPGHAN Hepatology Committee and the IBD Porto Group it does not necessarily represent ESPGHAN policy and is not endorsed by ESPGHAN.
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Snippet Objective We aimed to provide an evidence‐supported approach to diagnose, monitor, and treat children with inflammatory bowel disease (IBD) and primary...
We aimed to provide an evidence-supported approach to diagnose, monitor, and treat children with inflammatory bowel disease (IBD) and primary sclerosing...
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SubjectTerms Adolescent
calprotectin
Child
Cholangiopancreatography, Magnetic Resonance
Cholangitis, Sclerosing - complications
Cholangitis, Sclerosing - diagnosis
Cholangitis, Sclerosing - etiology
Cholangitis, Sclerosing - therapy
colorectal carcinoma
Gastroenterology
Humans
Inflammatory Bowel Diseases - complications
MRCP
surveillance colonoscopy
ursodeoxycholic acid
Title Primary sclerosing cholangitis in children with inflammatory bowel disease: An ESPGHAN position paper from the Hepatology Committee and the IBD Porto group
URI https://onlinelibrary.wiley.com/doi/abs/10.1002%2Fjpn3.12378
https://www.ncbi.nlm.nih.gov/pubmed/39741383
https://www.proquest.com/docview/3150833904
Volume 80
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