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01.12.2011 | Case report | Ausgabe 1/2011 Open Access

Journal of Medical Case Reports 1/2011

Isolated angioedema of the bowel due to C1 esterase inhibitor deficiency: a case report and review of literature

Zeitschrift:
Journal of Medical Case Reports > Ausgabe 1/2011
Autoren:
Shivangi T Kothari, Anish M Shah, Deviprasad Botu, Robert Spira, Robert Greenblatt, Joseph Depasquale
Wichtige Hinweise

Electronic supplementary material

The online version of this article (doi:10.​1186/​1752-1947-5-62) contains supplementary material, which is available to authorized users.

Competing interests

The authors declare that they have no competing interests.

Authors' contributions

STK was the primary author, and contributed to patient's diagnosis and treatment in addition to collecting patient information, preparing the framework of the manuscript, tables, illustrations, and writing the entire case report with discussion section. AS and DB contributed to the literature review and data collection. JD worked on the image capturing, legend writing, reference searching and literature review. RG was the primary attending physician on this case, and worked on the data collection. RS worked on the case formatting with grammatical checking and verification of the discussion section with references. All authors read and approved the final manuscript.

Abstract

Introduction

We report a rare, classic case of isolated angioedema of the bowel due to C1-esterase inhibitor deficiency. It is a rare presentation and very few cases have been reported worldwide. Angioedema has been classified into three categories.

Case presentation

A 66-year-old Caucasian man presented with a ten-month history of episodic severe cramping abdominal pain, associated with loose stools. A colonoscopy performed during an acute attack revealed nonspecific colitis. Computed tomography of the abdomen performed at the same time showed a thickened small bowel and ascending colon with a moderate amount of free fluid in the abdomen. Levels of C4 (< 8 mg/dL; reference range 15 to 50 mg/dL), CH50 (< 10 U/mL; reference range 29 to 45 U/ml) and C1 inhibitor (< 4 mg/dL; reference range 14 to 30 mg/dL) were all low, supporting a diagnosis of acquired angioedema with isolated bowel involvement. Our patient's symptoms improved with antihistamine and supportive treatment.

Conclusion

In addition to a detailed comprehensive medical history, laboratory data and imaging studies are required to confirm a diagnosis of angioedema due to C1 esterase inhibitor deficiency.

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