{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,11,13]],"date-time":"2025-11-13T04:04:44Z","timestamp":1763006684096,"version":"3.45.0"},"reference-count":15,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2025,6,22]],"date-time":"2025-06-22T00:00:00Z","timestamp":1750550400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["JPGN rep."],"published-print":{"date-parts":[[2025,11]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Gastrointestinal dysmotility is commonly reported among patients with Mowat\u2010Wilson syndrome (MWS) and poses a significant symptomatic burden. Unfortunately, there remains a knowledge gap regarding effective treatment strategies. A 2\u2010year\u2010old male with MWS presented with chronic paradoxical abdominal pain, constipation, and progressively restricted oral intake. He was diagnosed with intestinal pseudo\u2010obstruction, due to recurrent transient small bowel intussusception identified on ultrasound. Pyridostigmine, an acetylcholinesterase inhibitor, was successfully used to promote gastrointestinal motility, improving the child's pain symptoms and allowing return to his baseline activities. This case adds to emerging evidence of low\u2010dose pyridostigmine as a viable treatment to consider for children with symptoms related to impaired gastrointestinal motility, by demonstrating its successful use in a child with MWS for the first time.<\/jats:p>","DOI":"10.1002\/jpr3.70054","type":"journal-article","created":{"date-parts":[[2025,6,22]],"date-time":"2025-06-22T10:49:38Z","timestamp":1750589378000},"page":"416-419","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Pyridostigmine as treatment for chronic gastrointestinal dysmotility in a child with Mowat\u2010Wilson syndrome: A case report and literature review"],"prefix":"10.1002","volume":"6","author":[{"ORCID":"https:\/\/orcid.org\/0009-0007-5997-3009","authenticated-orcid":false,"given":"Grace J.","family":"Lin","sequence":"first","affiliation":[{"name":"Department of Pediatrics, BC Children's Hospital University of British Columbia Vancouver British Columbia Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shreya B.","family":"Kishore","sequence":"additional","affiliation":[{"name":"Department of Pediatrics BC Children's Hospital, British Columbia &amp; Griffith University School of Medicine Vancouver Brisbane Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sally","family":"Samaan","sequence":"additional","affiliation":[{"name":"Department of Pediatrics BC Children's Hospital, British Columbia &amp; Ain Shams University Vancouver Cairo Egypt"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lisa S.","family":"Hackett","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, BC Children's Hospital University of British Columbia Vancouver British Columbia Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kevan","family":"Jacobson","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Division of Gastroenterology, Hepatology and Nutrition, BC Children's Hospital University of British Columbia Vancouver British Columbia Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Gabriella","family":"Horvath","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Division of Biochemical Genetics, BC Children's Hospital University of British Columbia Vancouver British Columbia Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2025,6,22]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.40.5.305"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1186\/1750-1172-2-42"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1177\/1093526620925960"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00383-020-04751-8"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00383-015-3732-x"},{"key":"e_1_2_9_7_1","first-page":"1993","volume-title":"GeneReviews [Internet]","author":"Adam MP","year":"2007"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.3389\/fped.2022.837462"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.3390\/genes12070982"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.31530"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpedsurg.2005.05.040"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.5056\/jnm19078"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.1111\/nmo.12945"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1463-1318.2009.01838.x"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.1007\/s40272-017-0277-6"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.1097\/MPG.0000000000001982"}],"container-title":["JPGN Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/jpr3.70054","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,11,13]],"date-time":"2025-11-13T04:02:18Z","timestamp":1763006538000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/jpr3.70054"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,6,22]]},"references-count":15,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2025,11]]}},"alternative-id":["10.1002\/jpr3.70054"],"URL":"https:\/\/doi.org\/10.1002\/jpr3.70054","archive":["Portico"],"relation":{},"ISSN":["2691-171X","2691-171X"],"issn-type":[{"type":"print","value":"2691-171X"},{"type":"electronic","value":"2691-171X"}],"subject":[],"published":{"date-parts":[[2025,6,22]]},"assertion":[{"value":"2024-11-07","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-06-03","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-06-22","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}