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Pediatric gastrointestinal sarcoidosis: Successful treatment with infliximab
Laila Alawdah1, Ahmad Nahari1, Dayel Alshahrani1
1Department of Pediatrics, Children's Specialized Hospital, King Fahad Medical City, Riyadh, Saudi Arabia.
Insights
Gastrointestinal sarcoidosis in children is rare. Infliximab successfully treated a chronic relapsing case, suggesting its potential for severe pediatric gastrointestinal sarcoidosis.
Area of Science:
- Pediatric Gastroenterology
- Immunology
- Rare Diseases
Background:
- Gastrointestinal sarcoidosis is exceptionally rare in pediatric populations, with limited documented cases and treatment data.
- Sarcoidosis is a multisystem granulomatous disease of unknown etiology, often affecting the lungs but can involve any organ system.
Observation:
- Two pediatric cases of gastrointestinal sarcoidosis are presented, an 8-year-old girl and a 9-year-old boy, exhibiting symptoms like fever, weight loss, abdominal pain, and hematemesis.
- Endoscopic and biopsy findings revealed severe erosive nodular gastric mucosa with noncaseating granulomatous inflammation in both patients.
- Multisystem involvement was confirmed with granulomatous hepatitis in one case and lung nodularity in both, alongside elevated angiotensin-converting enzyme levels in one.
Findings:
- Both children were diagnosed with active disseminated sarcoidosis and initially treated with corticosteroids.
- While one patient remained symptom-free after steroid therapy, the other experienced relapses, necessitating treatment with infliximab.
- Infliximab therapy induced a sustained remission for 5 years in the relapsing pediatric patient with gastrointestinal sarcoidosis.
Implications:
- Pediatric gastrointestinal sarcoidosis presents with variable clinical courses.
- Infliximab therapy shows promise as an effective treatment option for severe, chronic, or relapsing forms of pediatric gastrointestinal sarcoidosis.
- Further research is warranted to establish optimal treatment strategies for pediatric sarcoidosis affecting the gastrointestinal tract.
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.
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