Type 1 Refractory Celiac Disease: Report of Two Cases with Different Therapeutic Approaches
DOI:
https://doi.org/10.22516/25007440.1271Keywords:
Celiac disease, Treatment, Diagnosis, gluten-free diet, budesonide, azathioprineAbstract
Background: Lifelong strict adherence to a gluten-free diet (GFD) is the effective treatment for celiac disease (CD), leading to symptom remission and mucosal healing. Refractory celiac disease (RCD) is defined as the persistence or relapse of symptoms and intestinal damage in individuals previously diagnosed with CD after at least 12 months of strict GFD adherence, occurring in a minority of CD patients. Diagnosis and differentiation of RCD type are performed via specific immunohistochemistry on duodenal biopsy.
Case 1: A 52-year-old male with a prior CD diagnosis presented with persistent symptoms even after five years of strict GFD adherence. He was diagnosed with type 1 refractory RCD and treated with oral budesonide (9 mg/day for 8 months), achieving clinical remission, with normalization of duodenal mucosal histopathology.
Case 2: A 62-year-old female with a prior CD diagnosis and two years of strict GFD adherence presented with severe symptoms. She was diagnosed with type 1 RCD and treated with azathioprine at 2 mg/kg/day for 24 months, resulting in complete symptom remission and restoration of duodenal mucosal integrity.
Conclusions: In addition to strict adherence to a healthy gluten-free diet, both oral budesonide and azathioprine were effective in treating type 1 RCD, as patients achieved and maintained clinical remission without drug-related adverse effects. Histological response, demonstrating complete normalization of duodenal mucosal architecture, confirmed the success of therapy.
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