Postinfectious T-lymphocytic enteral leiomyositis as a rare cause of chronic intestinal pseudoobstruction

Abstract

T-lymphocytic enteral leiomyositis (T-lel) is a rare disorder causing chronic intestinal pseudo-obstruction (CIPO), with cases predominantly being reported in the field of veterinary and pediatric medicine. Here, we present a case of T-lel-associated CIPO in an adult female, who initially presented with a paralytic ileus 2 weeks after a common gastroenteritis. The histological diagnosis was established through full-thickness bowel biopsy, exhibiting a dense lymphocytic infiltrate in the lamina muscularis of the intestinal wall. This case shows that T-lel can be a cause of chronic intestinal pseudo-obstruction not only in children but also in adults. A subsequent induction of an immunosuppressive therapy with steroids, azathioprine, and ultimately TNF-alpha-inhibiting antibodies led to a slow recovery and stable disease.

Zusammenfassung

Die T-lymphozytäre enterale Leiomyositis (T-lel) ist eine seltene Erkrankung, die zu einer chronischen intestinalen Pseudoobstruktion (CIPO) führen kann. Die meisten dieser seltenen Fälle sind bei Kindern und in der Veterinärmedizin beschrieben. Hier berichten wir über den Fall einer 42-jährigen Patientin, die sich 2 Wochen nach gastroenterischen Beschwerden mit der Klinik eines paralytischen Ileus vorstellte. Nachdem eine weitreichende Diagnostik mittels laboranalytischer, endoskopischer und radiologischer Verfahren keine wegweisenden Befunde erbrachte, erfolgte die Durchführung einer Dünndarm-Vollwandbiopsie, woraufhin histologisch die Diagnose einer T-lymphozytären Leiomyositis gestellt werden konnte. Eine immunsuppressive Therapie mit initial Prednisolon und Azathioprin wurde aufgrund unzureichenden klinischen Ansprechens auf Adalimumab umgestellt, woraufhin letztlich eine Stabilisierung der Erkrankung und eine Verbesserung der Symptomatik erreicht werden konnten.

Key words

gastrointestinal motility - malassimilation - infectious colitis

Schlüsselwörter

gastrointestinale Motilität - Malassimilation - infektiöse Colitis

Publication History

Received: 23 June 2020

Accepted: 08 November 2020

Article published online:
12 April 2021

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