Yamini Patel, MD, MS
The Wright Center for Graduate Medical Education
Scranton, Pennsylvania
Disclosure information not submitted.
Navharsh Sekhon, MD
Resident Physician
The Wright Center for Graduate Medical Education
Scranton, Pennsylvania
Disclosure information not submitted.
Nishant Sharma, MD
Pulmonology and Critical Care Fellow
n/a, United States
Disclosure information not submitted.
Srinivasarao Ramakrishna, MD
Specialist in pulmonary and critical care medicine
Geisinger Community Medical Center - Pulmonary Medicine, United States
Disclosure information not submitted.
Pius Ochieng, MD, FCCM
Associate Professor of Medicine, Pulmonary and Critical Care Physician
Geisinger Community Medical Center
Scranton, Pennsylvania, United States
Disclosure information not submitted.
Title: Recurrent bowel angioedema diagnosed retrospectively after oropharyngeal angioedema
Introduction: Angioedema presents as swelling of the face, lips, larynx, extremities, genitalia, or bowel that resolves spontaneously within hours to days. Angiotensin-converting enzyme inhibitors (ACEIs) are commonly used for hypertension, and angioedema is a rare, yet well-known adverse reaction. Bowel wall angioedema secondary to ACEI use is a challenging diagnosis.
Description: A 73-year-old male with hypertension treated with benazepril presented with worsening oropharyngeal edema and gargled speech. He had multiple admissions for colicky abdominal pain of unknown etiology that resolved spontaneously. Prior workup included computed tomography (CT) angiogram of the abdomen and pelvis showing diffuse thickening from the stomach to the sigmoid colon with mesenteric ascites and stranding. General surgery suspected mesenteric ischemia and gastroenterology suspected colitis. A mesenteric duplex showed no significant stenosis of superior mesenteric, inferior mesenteric, and celiac arteries. Esophagogastroduodenoscopy and flexible sigmoidoscopy were notable only for antral gastritis. He was intubated for airway protection and treated with steroids, anti-histamines, and icatibant. Labs were unremarkable. CT abdomen demonstrated thickening of the small intestines with mesenteric ascites. Clostridium difficile, gastrointestinal pathogen, and celiac panel were negative. He received supportive care, and benazepril was held. He was extubated within 24 hours after complete symptom resolution and counseled on avoiding ACEIs. The diagnosis was previously missed despite multi-specialty involvement: general surgery, gastroenterology, emergency department, and internal medicine.
Discussion: ACEIs cause bradykinin-mediated angioedema which may be treated with C1 inhibitor concentrate, fresh frozen plasma, icatibant, or ecallantide. Only a handful of ACEI bowel angioedema cases have been reported in the literature with a diagnosis delay from months to years. This case of ACEI mediated visceral edema highlights the elusive nature of the condition, which typically presents with vague abdominal symptoms and is consequently misdiagnosed or diagnosed late, following an extensive workup or unnecessary procedures. This patient likely had recurrent visceral angioedema which resolved on stopping ACEI.