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

      case-report

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          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.

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          Most cited references26

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          Morbidity and mortality in Cushing's disease: an epidemiological approach.

          Although Cushing's disease is a well documented clinical entity, there is no epidemiological information about it. The present study tries to obtain this information. Forty-nine patients affected by Cushing's disease living in Vizcaya (Spain) between 1975 and 1992 were considered for an epidemiological study. The prevalence of known cases at the end of 1992 was 39.1 per million inhabitants. The average incidence of newly diagnosed cases was 2.4 cases per million people per year. Cushing's disease was more frequent in women (n = 46) than in men (n = 3), with a ratio of 15:1. Diabetes mellitus and hypertension were observed in 38.7 and 55.1% of patients, respectively. Remission of Cushing's disease was achieved in 36 out of 41 patients (87.5%). In general, the mortality was higher than that expected for the control population (standardized mortality ratio, SMR 3.8, 95% confidence interval, CI 2.5-17.9, P < 0.03). Concerning the cause of death, the SMR of vascular disease was 5 (95% CI 3.4-48.6, P < 0.05). Higher age, persistence of hypertension and abnormalities of glucose metabolism after treatment, were independent predictors of mortality (multivariate analyses, P < 0.01). Prevalence of Cushing's disease was 39.1 cases/million inhabitants and average incidence was 2.4 cases/million per year. Mortality was elevated, due to vascular disease, associated with higher age, persistence of hypertension and impaired glucose metabolism.
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            Gastrointestinal Tract Perforation: MDCT Findings according to the Perforation Sites

            Our objective is to describe the characteristic CT findings of gastrointestinal (GI) tract perforations at various levels of the gastrointestinal system. It is beneficial to localize the perforation site as well as to diagnose the presence of bowel perforation for planning the correct surgery. CT has been established as the most valuable imaging technique for identifying the presence, site and cause of the GI tract perforation. The amount and location of extraluminal free air usually differ among various perforation sites. Further, CT findings such as discontinuity of the bowel wall and concentrated free air bubbles in close proximity to the bowel wall can help predict the perforation site. Multidetector CT with the multiplanar reformation images has improved the accuracy of CT for predicting the perforation sites.
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              Diverticular disease of the colon, a 20th century problem.

                Author and article information

                Journal
                Endocrinol Diabetes Metab Case Rep
                Endocrinol Diabetes Metab Case Rep
                edm
                EDM Case Reports
                Endocrinology, Diabetes & Metabolism Case Reports
                Bioscientifica Ltd (Bristol )
                2052-0573
                18 November 2013
                2013
                : 2013
                : 130064
                Affiliations
                [1 ]Department of Neurosurgery, Faculty of Medicine University of Tsukuba 1-1-1 Tennodai Tsukuba Ibaraki, Tsukuba, 305-0006Japan
                Author notes
                Correspondence should be addressed to H Akutsu Email: akutsuh@ 123456md.tsukuba.ac.jp
                Article
                EDM130064
                10.1530/EDM-13-0064
                3922247
                24616779
                c4a76112-3fae-425b-93f7-e12076b07e99
                © 2013 The authors

                This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivs 3.0 Unported License.

                History
                : 7 October 2013
                : 30 October 2013
                Categories
                Unique/Unexpected Symptoms or Presentations of a Disease

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