Background: Nodular lymphoid hyperplasia (NLH) of the gastrointestinal (GI) tract is a rare condition in adults. It is usually asymptomatic. Few complications have been described.

Aim:   We report an unusual clinical presentation of focal lymphoid hyperplasia of the GI.

Case Report: A 23-year-old female patient presented with a fistulizingdisease of the terminal ileum and the caecum complicated with an abscess of the lower right quadrant if the abdomen. CT-guided drainage with antibiotic therapy failed to control the abscess. Thus, surgery was undertaken and ileocaecal resection was performed. Focal lymphoid hyperplasia was confirmed by the pathology of the specimen.

Conclusions: NLH is an uncommon condition in adults. To the best of our knowledge, no previous cases have been reported with fistulizing NLH. The management should follow the same algorithm as fistulizing ileitis. Surgery is indicated only in cases of complicated disease after the failure of medical treatment.

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