Intest Res.  2017 Jan;15(1):133-137. 10.5217/ir.2017.15.1.133.

Crohn's disease with ankylosing spondylitis in an adolescent patient who had undergone long ileo-colonic anastomosis for Hirschsprung's disease as an infant

• Kim HY
• Kim TW

• KMID: 2368497
• DOI: http://doi.org/10.5217/ir.2017.15.1.133

Abstract

Crohn's disease (CD) is a chronic, idiopathic, inflammatory disorder of the gastrointestinal tract. In rare cases, CD has been associated with Hirschsprung's disease (HD); however, the underlying pathophysiology of this and other comorbidities is not yet fully understood. In this report, we describe the case of a 17-year-old patient who was diagnosed with both CD and ankylosing spondylitis (AS), having undergone a long ileo-colonic anastomosis to treat HD at 12 months of age. To our knowledge, this is the first documented case of CD combined with AS in a patient with HD.

Keyword

Crohn disease; Spondylitis, ankylosing; Hirschsprung disease; Inflammatory bowel diseases; Long ileo-colonic anastomosis

MeSH Terms

Adolescent*
Comorbidity
Crohn Disease*
Gastrointestinal Tract
Hirschsprung Disease*
Humans
Infant*
Inflammatory Bowel Diseases
Spondylitis, Ankylosing*

Figure

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  Fig. 1 Abdominal CT findings. Multiple strictures were accompanied by dilation of the small bowel. (A) A thickened mucosal wall was seen at the neoascending colon portion of ileum (arrow). (B) A thickened mucosal wall was also noted at the neosigmoid colon portion of ileum and anastomosis site (arrow).

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  Fig. 2 Colonoscopic findings. Edematous mucosa and a superficial ulcerative lesion were observed above 10 cm from the anal verge. The scope could not pass through because of luminal narrowing.

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  Fig. 3 Small bowel series findings. (A) Luminal narrowing was seen at neoterminal ileum (white arrow). (B) An irregular and thickened wall was observed at neoascending colon portion of ileum (white arrow). (C) A suspected focal stricture was observed in the neosigmoid colon portion of ileum (black arrow).

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