Bowel perforation associated with Cushing’s disease: a case report with literature review

Abstract

Although rare, endogenous hypercortisolemia, including Cushing's disease (CD), is known to cause bowel perforation and to mask typical symptoms of bowel perforation, leading to delayed diagnosis. Additionally, elderly patients with CD are considered to be at a higher risk for bowel perforation because intestinal tissue fragility tends to increase in the elderly. Herein, we describe a rare case in which a young adult patient with CD was diagnosed with bowel perforation associated with CD following severe abdominal pain. A 24-year-old Japanese man was admitted to the hospital for the evaluation of ACTH-dependent Cushing's syndrome. He suddenly complained of severe abdominal pain on the 8th day of hospitalization. Computed tomography revealed free air around the sigmoid colon. The patient was diagnosed with bowel perforation, underwent emergency surgery, and was saved. He was subsequently diagnosed with CD, and the pituitary adenoma was resected transsphenoidally. To date, eight cases of bowel perforation due to CD had been reported, with a median age of 61 years at the time of bowel perforation. Hypokalemia was detected in half of the patients, and all had a history of diverticular disease. Nevertheless, not many patients complained of peritoneal irritation. In conclusion, this is the youngest reported case with bowel perforation due to CD and the first report of bowel perforation in a patient without a history of diverticular disease. Bowel perforation may occur in patients with CD, irrespective of age and the presence of hypokalemia, diverticular disease, or peritoneal irritation.

Adrenocorticotropic Hormone-Dependent Cushing's Syndrome Complicated With Gastric Ulcer Perforation in a 30-Year-Old Saudi Female: A Case Report and a Review of the Literature

“…A limited number of studies have addressed gastrointestinal perforations associated with endogenous hypercortisolemia [5][6][7][8][9][10][11][12][13][14][15][16][17]. The correlation between Cushing's syndrome and gastrointestinal perforation is highlighted in our study and in the case reports that have been previously published (Table 2).…”

“…However, there is limited information on gastrointestinal complications associated with endogenous hypercortisolemia [3,4]. Thus far, only 23 instances have been published addressing the co-occurrence of gastrointestinal perforation with endogenous Cushing's syndrome [5][6][7][8][9][10][11][12][13][14][15][16][17]. To the best of our knowledge, this is the first case reporting gastric perforation in an ACTH-dependent Cushing's syndrome, while the vast majority reported diverticular, sigmoid, or duodenal perforation with Cushing's syndrome [5][6][7][8][9][10][11][12][13][14][15][16][17].…”

“…Thus far, only 23 instances have been published addressing the co-occurrence of gastrointestinal perforation with endogenous Cushing's syndrome [5][6][7][8][9][10][11][12][13][14][15][16][17]. To the best of our knowledge, this is the first case reporting gastric perforation in an ACTH-dependent Cushing's syndrome, while the vast majority reported diverticular, sigmoid, or duodenal perforation with Cushing's syndrome [5][6][7][8][9][10][11][12][13][14][15][16][17]. Herein, we describe the medical history, physical examination, and investigatory findings of a 30-year-old female with a recent diagnosis of ACTH-dependent Cushing's syndrome that was complicated by gastric ulcer perforation, necessitating an urgent exploratory laparotomy.…”