Endocrine Abstracts

Background

Cushing’s syndrome (CS) occurs due to cortisol overproduction. Endogenous hypercortisolemia can be secondary to Adrenocorticotropic hormone (ACTH) dependent as well as independent causes (1). Early identification and prompt definitive management is crucial to minimize mortality. Successful management of CS becomes extremely challenging due to multiple associated complications, especially the thrombotic events which are even more prominent in post-operative period needing close monitoring. We report a case of Cushing’s disease (CD) in a young female managed with trans-sphenoidal surgery, followed by a challenging post-operative period due to multiple thrombotic phenomena, ultimately succumbed.

Case presentation

A 32-year-old Sri Lankan female presented with overt features of CS and diagnosed to have CD with pituitary microadenoma. Pre-operatively she was medically managed with Ketoconazole. She underwent trans-sphenoidal surgery and despite achieving normalization of post-operative day2 cortisol, she developed multiple complications including diverticular rupture and ischemic colitis, needing hemicolectomy, followed by parieto-occipital infarction, which ultimately lead to her death despite aggressive management.

Conclusion

This case highlights important and aggressive complications associated with CS giving rise to a challenging post-operative course. Diverticular rupture had been rarely described in association with hypercortisolemia, due to multiple mechanisms, and this case adds to the existing literature (2). Post-operative ischemic colitis and stroke which lead to the death of this patient are seen with procoagulant state associated with CS, with a high risk during the immediate post-operative period. Venous thrombo-embolism is the most commonly reported thrombotic phenomenon while acute mesenteric ischemia as seen in our patient is only rarely reported (3). This emphasizes the need to consider thromboprophylaxis for patients with CS in the immediate post-operative period, although clear guidelines regarding this do not exist.

References

Sharma ST, Nieman LK, Feelders RA. Cushing’s syndrome: epidemiology and developments in disease management. Clin Epidemiol [Internet]. 2015 Apr 17;7:281–93. Available from: https://www.ncbi.nlm.nih.gov/pubmed/25945066

Sater ZA, Jha S, McGlotten R, Hartley I, El Lakis M, Araque KA, et al. Diverticular Perforation: A Fatal Complication to Forestall in Cushing Syndrome. J Clin Endocrinol Metab [Internet]. 2018 May 28;103(8):2811–4. Available from: https://doi.org/10.1210/jc.2018-00829

Takayasu S, Murasawa S, Yamagata S, Kageyama K, Nigawara T, Watanuki Y, et al. Acute mesenteric ischemia and hepatic infarction after treatment of ectopic Cushing’s syndrome. Endocrinol diabetes Metab case reports [Internet]. 2017 Feb 28;2017:16–144. Available from: https://www.ncbi.nlm.nih.gov/pubmed/28480039