AI Article Synopsis

  • Gastrointestinal involvement in Langerhans cell histiocytosis (LCH) is rare; a 19-month-old girl presented with severe gastrointestinal and systemic symptoms, leading to multiple medical interventions.
  • The diagnosis of LCH was confirmed after three endoscopies, revealing significant changes in the colorectal region, including ulcers and inflammation, and a BRAF-V600E mutation was identified.
  • Treatment with chemotherapy and a BRAF inhibitor (dabrafenib) resulted in rapid clinical improvement, and the patient was symptom-free with normal bowel movements during a follow-up after one year.

Article Abstract

Gastrointestinal tract involvement in Langerhans cell histiocytosis (LCH) is extremely rare, with limited documentation of endoscopic manifestations. We report a 19-month-old girl who presented with repeated diarrhea and bloody stools, accompanied by recurrent pulmonary infections, anemia, hypoproteinemia, thrombocytopenia, coagulopathy, and hepatosplenomegaly with lymphadenopathy. Initial treatment with antibacterial agents, mesalazine, thalidomide, and prednisone led to temporary improvement; however, the symptoms repeatedly relapsed. She underwent three digestive endoscopies, but until the third endoscopy, a definitive diagnosis of Langerhans cell histiocytosis was established through biopsy. While upper gastrointestinal tract findings were not significant, notable changes were observed in the colorectal region. A colonoscopy revealed progression from erythema to diffuse hyperemia and edema, with erythema, erosion, and superficial ulcers extending into the distal ileal mucosa. Genetic analysis identified a BRAF-V600E mutation. Following treatment with chemotherapy (vincristine and prednisone) and the BRAF inhibitor dabrafenib, the patient demonstrated significant clinical improvement within days. At the 1-year follow-up, the patient had normal bowel movements and a weight gain of 2.5 kg. Early gastrointestinal endoscopy with multiple biopsies in suspected children can facilitate early detection. Dabrafenib is a viable treatment option for Langerhans cell histiocytosis.

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Source
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC11430356PMC
http://dx.doi.org/10.1002/deo2.70023DOI Listing

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