Cushing's disease presenting with gastrointestinal perforation: a case report

Abstract

Gastrointestinal perforation is a complication associated with steroid therapy or hypercortisolism, but it is rarely observed in patients with Cushing's disease in clinical practice, and only one case has been reported as a presenting symptom. Herein, we report a rare case of Cushing's disease in which a patient presented with gastrointestinal perforation as a symptom. A 79-year-old man complained of discomfort in the lower abdomen for 6 months. Based on the endocrinological and gastroenterological examinations, he was diagnosed with Cushing's disease with a perforation of the descending colon. After consultation with a gastroenterological surgeon, it was decided that colonic perforation could be conservatively observed without any oral intake and treated with parenteral administration of antibiotics because of the mild systemic inflammation and lack of abdominal guarding. Despite the marked elevated levels of serum cortisol, oral medication was not an option because of colonic perforation. Therefore, the patient was submitted to endonasal adenomectomy to normalize the levels of serum cortisol. Subsequently, a colostomy was successfully performed. Despite its rarity, physicians should be aware that gastrointestinal perforation may be associated with hypercortisolism, especially in elderly patients, and immediate diagnosis and treatment of this life-threatening condition are essential. If a perforation can be conservatively observed, endonasal adenomectomy prior to laparotomy is an alternative treatment option for hypercortisolism.Learning points Thus far, only one case of gastrointestinal perforation as a presenting clinical symptom of Cushing's disease has been reported.Physicians should be aware that gastrointestinal perforation might be associated with hypercortisolism in elderly patients because elevated levels of serum cortisol may mask the clinical signs of perforation. Because of this masking effect, the diagnosis of the perforation also tends to be delayed.Although parenteral administration of etomidate is a standard treatment option for decreasing the elevated levels of serum cortisol, endonasal adenomectomy prior to laparotomy is an alternative treatment option if etomidate therapy is unavailable.

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

“…Similar to our case, a female predominance was seen in gastrointestinal perforation among the reported cases of Cushing's syndrome [6,7,12,13,15,16]. Additionally, the average age at which gastrointestinal perforation occurred in patients with endogenous hypercortisolism ranged from 45 to 80, which is a noticeably higher age range than the case we are presenting here (aged 30) [6][7][8][9][10]12]. Furthermore, unlike our case, in which gastrointestinal perforation occurred four months after the onset of Cushing's symptoms, Intestinal perforation occurs approximately 9.8 months after Cushing's symptoms first appear [15].…”

“…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.…”

An Ectopic ACTH Secreting Metastatic Parotid Tumour

“…A whole body CT scan in our patient revealed a lytic lesion on the left ischial bone and patient went on to develop bowel perforation, with minimal features. Exogenous steroid use and CS have been associated with increased risk of intestinal perforation and sepsis [ 21 , 22 ]. It is not uncommon for the cortisol excess to mask signs associated with intestinal perforation delaying the diagnosis which can potentially prove fatal as in our patient.…”