{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,13]],"date-time":"2026-02-13T17:15:51Z","timestamp":1771002951182,"version":"3.50.1"},"reference-count":18,"publisher":"Wiley","issue":"S3","license":[{"start":{"date-parts":[[2008,1,5]],"date-time":"2008-01-05T00:00:00Z","timestamp":1199491200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J of Inher Metab Disea"],"published-print":{"date-parts":[[2008,12]]},"abstract":"<jats:title>Summary<\/jats:title><jats:p>Glycogen storage disease (GSD) type Ib is a congenital disorder of glycogen metabolism that is associated with neutropenia, neutrophil dysfunction, and an inflammatory bowel disease that mimics a Crohn phenotype. Gastrointestinal inflammation in GSD Ib has been successfully treated with 5\u2010aminosalicylic acid and granulocyte colony\u2010stimulating factor (G\u2010CSF). However, therapeutic options for patients not responding to traditional therapies have been limited owing to untoward effects of glucocorticoids and immunomodulators in this metabolic disorder. Adalimumab is a monoclonal antibody targeting tumour necrosis factor\u2010\u03b1 that has shown promise for the treatment of patients with Crohn disease. Due to the limited options for treating GSD\u2010associated inflammatory bowel disease, use of adalimumab was attempted in a case unresponsive to aminosalicylate, G\u2010CSF, and antibiotic therapy. Significant clinical and histological improvement was observed in our patient, and the medication was well tolerated.<\/jats:p>","DOI":"10.1007\/s10545-007-0774-9","type":"journal-article","created":{"date-parts":[[2008,1,2]],"date-time":"2008-01-02T15:32:09Z","timestamp":1199287929000},"page":"505-509","source":"Crossref","is-referenced-by-count":26,"title":["Adalimumab for the treatment of Crohn\u2010like colitis and enteritis in glycogen storage disease type Ib"],"prefix":"10.1002","volume":"31","author":[{"given":"M. K.","family":"Davis","sequence":"first","affiliation":[{"name":"Division of Pediatric Gastroenterology, Department of Pediatrics University of Florida College of Medicine Gainesville Florida USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"P. A.","family":"Rufo","sequence":"additional","affiliation":[{"name":"Division of Gastroenterology and Nutrition and Center for Inflammatory Bowel Disease Children's Hospital Boston Boston Massachusetts USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"S. F.","family":"Polyak","sequence":"additional","affiliation":[{"name":"Division of Gastroenterology, Hepatology &amp; Nutrition, Department of Medicine University of Florida College of Medicine Gainesville Florida USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"D. A.","family":"Weinstein","sequence":"additional","affiliation":[{"name":"Division of Pediatric Endocrinology, Department of Pediatrics University of Florida College of Medicine Gainesville Florida USA"},{"name":"Glycogen Storage Disease Program University of Florida College of Medicine Gainesville FL 32610 USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2008,1,5]]},"reference":[{"key":"e_1_2_5_2_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF02072090"},{"key":"e_1_2_5_3_1","first-page":"1521","volume-title":"The Metabolic and Molecular Bases of Inherited Disease","author":"Chen Y\u2010T","year":"2001"},{"key":"e_1_2_5_4_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.MIB.0000185402.65288.19"},{"key":"e_1_2_5_5_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF02680002"},{"key":"e_1_2_5_6_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF02072085"},{"issue":"12","key":"e_1_2_5_7_1","first-page":"535","article-title":"The leukocyte count predicts the efficacy of treatment with azathioprine in inflammatory bowel disease.","volume":"10","author":"Glas J","year":"2005","journal-title":"Eur J Med Res"},{"key":"e_1_2_5_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(02)08512-4"},{"key":"e_1_2_5_9_1","doi-asserted-by":"publisher","DOI":"10.1053\/j.gastro.2005.11.030"},{"key":"e_1_2_5_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2036.2005.02287.x"},{"key":"e_1_2_5_11_1","article-title":"Tumour necrosis factor alpha blocking agents for induction of remission in ulcerative colitis.","volume":"3","author":"Lawson MM","year":"2006","journal-title":"Cochrane Database Syst Rev"},{"key":"e_1_2_5_12_1","doi-asserted-by":"publisher","DOI":"10.1172\/JCI117645"},{"key":"e_1_2_5_13_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.8211187"},{"key":"e_1_2_5_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3476(86)80572-8"},{"key":"e_1_2_5_15_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa050516"},{"key":"e_1_2_5_16_1","doi-asserted-by":"publisher","DOI":"10.1067\/mpd.2000.105232"},{"key":"e_1_2_5_17_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF02680008"},{"key":"e_1_2_5_18_1","doi-asserted-by":"publisher","DOI":"10.1093\/ajcn\/65.5.1507"},{"key":"e_1_2_5_19_1","doi-asserted-by":"publisher","DOI":"10.1097\/00005176-199908000-00008"}],"container-title":["Journal of Inherited Metabolic Disease"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1007\/s10545-007-0774-9","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,15]],"date-time":"2023-09-15T04:30:08Z","timestamp":1694752208000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1007\/s10545-007-0774-9"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2008,1,5]]},"references-count":18,"journal-issue":{"issue":"S3","published-print":{"date-parts":[[2008,12]]}},"alternative-id":["10.1007\/s10545-007-0774-9"],"URL":"https:\/\/doi.org\/10.1007\/s10545-007-0774-9","archive":["Portico"],"relation":{},"ISSN":["0141-8955","1573-2665"],"issn-type":[{"value":"0141-8955","type":"print"},{"value":"1573-2665","type":"electronic"}],"subject":[],"published":{"date-parts":[[2008,1,5]]}}}