Fecal microbiota transplantation for indolent T-cell lymphoproliferative disorder of gastrointestinal tract: A case report and review of literature

Abstract

BACKGROUND

Indolent T-cell lymphoproliferative disorder of the gastrointestinal tract (ITLPD-GI) is a rare clonal T-cell proliferation characterized by chronic diarrhea, abdominal pain, and weight loss. Its non-specific clinical and endoscopic presentation often leads to misdiagnosis as inflammatory bowel disease. Fecal microbiota transplantation (FMT), which aims to restore gut microbial homeostasis, has demonstrated efficacy in recurrent Clostridioides difficile infections, and its immunomodulatory potential is being explored in other conditions. However, the role of the gut microbiome in the pathogenesis of ITLPD-GI remains unknown.

CASE SUMMARY

A male patient presented with recurrent oral ulcers, chronic diarrhea, and significant weight loss. He was initially diagnosed with and treated for ulcerative colitis, without a successful response. A comprehensive re-evaluation, including systematic histopathological and immunohistochemical analyses of mucosal biopsies, confirmed a definitive diagnosis of ITLPD-GI. Following this diagnosis, the patient underwent a series of FMT procedures via colonoscopy. This intervention resulted in a remarkable and sustained clinical remission, with the complete resolution of diarrhea and oral ulcers, and significant weight gain. Follow-up endoscopy demonstrated substantial mucosal improvement, with a marked reduction in previous congestion and edema. The patient reported a significant improvement in his quality of life.

CONCLUSION

This report presents the first documented case in which FMT induced clinical and endoscopic remission in a patient with ITLPD-GI. The findings underscore the necessity to accurately differentiate ITLPD-GI from inflammatory bowel disease histopathologically. The significant therapeutic response suggests a pathogenic role of gut dysbiosis in ITLPD-GI and establishes FMT as a promising microbiota-targeting intervention. Further clinical studies are needed to validate these findings and elucidate the precise immunomodulatory mechanisms involved.

Keywords: Indolent T-cell lymphoproliferative disorder of the gastrointestinal tract; Fecal microbiota transplantation; Oral ulcers; diarrhea; Weight loss; Clinical manifestations; Endoscopic presentation; Case report

Core Tip: Indolent T-cell lymphoproliferative disorder of the gastrointestinal tract is frequently misdiagnosed as inflammatory bowel disease, leading to ineffective treatments. This first successful use of fecal microbiota transplantation in a patient with indolent T-cell lymphoproliferative disorder of the gastrointestinal resulted in sustained clinical and endoscopic remission, suggesting that gut dysbiosis may play a key pathogenic role and positioning fecal microbiota transplantation as a novel, microbiota-targeting therapeutic strategy for this rare condition.