Chronic intestinal pseudo-obstruction caused by an intestinal inflammatory myopathy: case report and review of the literature

Neurogastroenterol Motil. 2008 Apr;20(4):343-8. doi: 10.1111/j.1365-2982.2007.01033.x. Epub 2007 Nov 14.

Abstract

Chronic intestinal pseudo-obstruction (CIP) is an uncommon disorder that may be of primary or secondary origin. We report a case of a 37-year-old woman with CIP due to inflammatory disorder of unknown origin involving the skin (eosinophilic fasciitis), the lungs (decreased diffusion capacity) and the gastrointestinal tract. History, clinical examination, plain abdominal film, barium X-ray and colonoscopy established a diagnosis of recurrent pseudo-obstruction. A full-thickness biopsy was performed during explorative laparotomy, and histological examination revealed findings compatible with an inflammatory myopathy due to a dense lymphoid infiltrate and extensive loss of the muscularis propria layers. Immunosuppressive therapy with cyclosporin was initiated, with significant clinical improvement. This case illustrates another form of CIP, characterized by an inflammatory myopathy, which is histologically distinct from other known visceral myopathies and neuropathies.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Appendectomy
  • Chronic Disease
  • Cyclosporine / therapeutic use
  • Eosinophilia / pathology
  • Fasciitis / pathology
  • Female
  • Humans
  • Immunosuppressive Agents / therapeutic use
  • Intestinal Pseudo-Obstruction / etiology*
  • Intestinal Pseudo-Obstruction / physiopathology*
  • Intestinal Pseudo-Obstruction / therapy
  • Intestines / pathology*
  • Jejunostomy
  • Laparoscopy
  • Myositis / complications*
  • Myositis / physiopathology
  • Myositis / therapy
  • Sterilization, Tubal

Substances

  • Immunosuppressive Agents
  • Cyclosporine