ACE Inhibitor-Induced Angioedema of the Small Bowel: A Case Report and Review of the Literature

Abstract

Angiotensin-converting enzyme (ACE) inhibitors are known to cause angioedema. Most ACE inhibitor-induced angioedema cases describe swelling in the periorbital region, tongue, and pharynx. We describe a case of a 62-year-old female with presumed angioedema of the small bowel after more than a 2-year history of lisinopril use (with no recent changes in her dose of 40 mg orally twice daily). The patient presented with nausea and intermittent left middle and upper quadrant abdominal pain and denied history of angioedema or swelling with any medications or any history of abdominal pain. On physical examination, bowel sounds, liver, and spleen were normal. Laboratory tests revealed leukocytosis (15 400 per mm) and normal complement 1 esterase inhibitor levels. Abdominal computed tomography (CT) showed segmental small bowel thickening and edema with ascites and surrounding inflammatory changes. There was no lymphadenopathy, obstruction, or ileus. Two days after discontinuation of the lisinopril, the patient reported improvement in symptoms. The Naranjo adverse drug reaction probability scale indicated a probable relationship (score of 7) between the development of angioedema of the small bowel and the lisinopril therapy. This case highlights the unique manner in which ACE inhibitor-induced angioedema may present. A review of published cases of ACE inhibitor-induced angioedema of the small bowel is provided.

An unusual presentation of ACE inhibitor-induced visceral angioedema

“…In a review by Wilin et al , that included 25 unique cases, authors found similar findings as those mentioned previously, in addition, 52% (13/25) of cases included patients who presented on several different occasions, prior to obtaining an official diagnosis, likely secondary to decreased knowledge and awareness of ACEi-induced visceral angioedema 7. Interestingly, 42% (8/19) of patients presented within 1 week of initiation of an ACEi and 32% (6/19) presented at or after 1 year of ACEi initiation 7. In this review, the longest period an individual was on ACEi therapy prior to onset of visceral angioedema was 9 years 7.…”

“…In a review by Scheirey et al that included 20 patients, authors also found a high proportion of obese individuals (65%) 6. In a review by Wilin et al , that included 25 unique cases, authors found similar findings as those mentioned previously, in addition, 52% (13/25) of cases included patients who presented on several different occasions, prior to obtaining an official diagnosis, likely secondary to decreased knowledge and awareness of ACEi-induced visceral angioedema 7. Interestingly, 42% (8/19) of patients presented within 1 week of initiation of an ACEi and 32% (6/19) presented at or after 1 year of ACEi initiation 7.…”

“…Interestingly, 42% (8/19) of patients presented within 1 week of initiation of an ACEi and 32% (6/19) presented at or after 1 year of ACEi initiation 7. In this review, the longest period an individual was on ACEi therapy prior to onset of visceral angioedema was 9 years 7. After being on ACEi therapy for 9 years, this patient had recurrent episodes of gastrointestinal symptoms, in addition to 2 episodes of facial angioedema 8.…”

“…Her enalapril was discontinued after the second episode of facial angioedema, and at the time of the case report, she had been symptom-free, from a gastrointestinal perspective, for 2 years 8. Interestingly, in the review by Wilin et al , 16% of cases reported a prior history of facial swelling before being diagnosed with ACEi-induced visceral angioedema 7…”

“…Labs are relatively unhelpful in diagnosing visceral angioedema and it is mainly a clinical diagnosis though radiographical evidence can be useful. In the review by Wilin et al , of 25 patients, 48% had leucocytosis and 15 of 15 had normal complement 1 esterase levels 7. Normal complement levels (C4, C1i and C1q) help differentiate visceral angioedema caused by ACEi from hereditary angioedema, as complement levels will typically be abnormal in hereditary forms of angioedema 5 7 9…”