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Kothari ST, Shah AM, Botu D, et al. Isolated angioedema of the bowel due to C1 esterase inhibitor deficiency: a case report and review of literature. J Med Case Rep. 2011;5:62doi: 10.1186/1752-1947-5-62.
Kothari, S. T., Shah, A. M., Botu, D., Spira, R., Greenblatt, R., & Depasquale, J. (2011). Isolated angioedema of the bowel due to C1 esterase inhibitor deficiency: a case report and review of literature. Journal of medical case reports, 562. https://doi.org/10.1186/1752-1947-5-62
Kothari, Shivangi T, et al. "Isolated angioedema of the bowel due to C1 esterase inhibitor deficiency: a case report and review of literature." Journal of medical case reports vol. 5 (2011): 62. doi: https://doi.org/10.1186/1752-1947-5-62
Kothari ST, Shah AM, Botu D, Spira R, Greenblatt R, Depasquale J. Isolated angioedema of the bowel due to C1 esterase inhibitor deficiency: a case report and review of literature. J Med Case Rep. 2011 Feb 14;5:62. doi: 10.1186/1752-1947-5-62. PMID: 21320328; PMCID: PMC3089795.
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J Med Case Rep. 2011 Feb 14;5:62. doi: 10.1186/1752-1947-5-62.

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

Journal of medical case reports

Shivangi T Kothari, Anish M Shah, Deviprasad Botu, Robert Spira, Robert Greenblatt, Joseph Depasquale

Affiliations

  1. Department of Gastroenterology, School of Health and Medical Sciences Seton Hall University, South Orange, NJ, USA. [email protected].

PMID: 21320328 PMCID: PMC3089795 DOI: 10.1186/1752-1947-5-62

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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