Saudi Journal of Gastroenterology
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CASE REPORT
Year : 2016  |  Volume : 22  |  Issue : 5  |  Page : 391-395

Pediatric gastrointestinal sarcoidosis: Successful treatment with infliximab


1 Department of Pediatrics, Children's Specialized Hospital, King Fahad Medical City, Riyadh, Saudi Arabia
2 Department of Pathology, King Fahad Medical City, Riyadh, Saudi Arabia

Correspondence Address:
Abdulrahman Al-Hussaini
Division of Pediatric Gastroenterology, Hepatology and Nutrition, Children's Specialized Hospital, King Fahad Medical City, University of King Saud for Health Sciences, P.O. Box: 59046, Riyadh 11525
Saudi Arabia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1319-3767.191146

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Gastrointestinal sarcoidosis is a rare disease with very limited data in children. Here we report the first pediatric case of successful treatment with infliximab. The first case was an 8-year-old Saudi girl who presented with fever, weight loss, and abdominal pain that was followed in a few months with hematemesis and development of hepatosplenomegaly. The second case was a 9-year-old Sudanese boy who manifested with vomiting, epigastric pain, and weight loss. On upper endoscopy, both cases demonstrated severe erosive nodular gastric mucosa. Gastric and esophageal biopsies had shown noncaseating granulomatous inflammation. The first case had histopathological evidence of granulomatous hepatitis, and both cases demonstrated lung nodularity on computed tomography chest. The boy had elevated angiotensin-converting enzyme level. Given the multisystem involvement with significant chest findings, tissue findings of granulomatous disease, and negative workup for other causes of granulomatous inflammation, both cases were diagnosed with active disseminated sarcoidosis, and treated with corticosteroids. The girl continued to be symptom-free for 4 years after tapering steroid therapy. The boy had relapses off steroids and the disease was brought into remission for 5 years off steroid therapy by infliximab. Pediatric GI sarcoidosis is a rare disease that exhibits heterogeneity in natural course. The chronic relapsing progressive form of the disease might benefit from infliximab therapy.


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