title: Pediatric gastrointestinal sarcoidosis: Successful treatment with infliximab.
abstract: 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.
authors.affiliation: Department of Pediatrics, Children's Specialized Hospital, King Fahad Medical City, Riyadh, Saudi Arabia.
authors.forename: Laila
authors.initials: L
authors.lastname: Alawdah
authors.affiliation: Department of Pediatrics, Children's Specialized Hospital, King Fahad Medical City, Riyadh, Saudi Arabia.
authors.forename: Ahmad
authors.initials: A
authors.lastname: Nahari
authors.affiliation: Department of Pediatrics, Children's Specialized Hospital, King Fahad Medical City, Riyadh, Saudi Arabia.
authors.forename: Dayel
authors.initials: D
authors.lastname: Alshahrani
authors.affiliation: Department of Pathology, King Fahad Medical City, Riyadh, Saudi Arabia.
authors.forename: Musa
authors.initials: M
authors.lastname: Fagih
authors.affiliation: Department of Pediatrics, Children's Specialized Hospital, King Fahad Medical City, Riyadh, Saudi Arabia.
authors.forename: Shahid
authors.initials: S
authors.lastname: Ghazi
authors.affiliation: Department of Pediatrics, Children's Specialized Hospital, King Fahad Medical City, Riyadh, Saudi Arabia.
authors.forename: Abdulrahman
authors.initials: A
authors.lastname: Al-Hussaini
mesh_terms: D002648:Child; D005260:Female; D005765:Gastrointestinal Agents / Q000008:administration & dosage* / Q000627:therapeutic use; D005767:Gastrointestinal Diseases / Q000000981:diagnostic imaging / Q000188:drug therapy*; D006801:Humans; D000069285:Infliximab / Q000008:administration & dosage* / Q000627:therapeutic use; D008297:Male; D012507:Sarcoidosis / Q000000981:diagnostic imaging / Q000188:drug therapy*; D014057:Tomography, X-Ray Computed; D016896:Treatment Outcome
chemical_list: D005765:Gastrointestinal Agents; D000069285:Infliximab