Journal article
Adalimumab for the treatment of Crohn-like colitis and enteritis in glycogen storage disease type Ib
Journal of inherited metabolic disease, Vol.31 Suppl 3(S3), pp.505-509
12/2008
DOI: 10.1007/s10545-007-0774-9
PMID: 18172743
Abstract
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-aminosalicylic acid and granulocyte colony-stimulating factor (G-CSF). 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-α that has shown promise for the treatment of patients with Crohn disease. Due to the limited options for treating GSD-associated inflammatory bowel disease, use of adalimumab was attempted in a case unresponsive to aminosalicylate, G-CSF, and antibiotic therapy. Significant clinical and histological improvement was observed in our patient, and the medication was well tolerated.
Details
- Title: Subtitle
- Adalimumab for the treatment of Crohn-like colitis and enteritis in glycogen storage disease type Ib
- Creators
- M K Davis - Division of Pediatric Gastroenterology, Department of Pediatrics, University of Florida College of Medicine, Gainesville, Florida, USAP A RufoS F PolyakD A Weinstein
- Resource Type
- Journal article
- Publication Details
- Journal of inherited metabolic disease, Vol.31 Suppl 3(S3), pp.505-509
- DOI
- 10.1007/s10545-007-0774-9
- PMID
- 18172743
- ISSN
- 0141-8955
- eISSN
- 1573-2665
- Language
- English
- Date published
- 12/2008
- Academic Unit
- Internal Medicine
- Record Identifier
- 9984094733102771
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