A challenging case of Cushing's disease complicated with multiple thrombotic phenomena following trans-sphenoidal surgery; a case report

BMC Endocr Disord. 2021 Feb 23;21(1):29. doi: 10.1186/s12902-021-00701-0.

Abstract

Background: Cushing's syndrome occurs due to overproduction of cortisol from adrenal glands. Endogenous hypercortisolemia can occur secondary to adrenocorticotropic hormone (ACTH) dependent as well as independent causes. The presence of non-specific symptoms and signs contributes to a delay in diagnosis. Early identification and prompt definitive management is crucial. It is important to be alert about the post-operative complications including multiple thrombotic phenomena, which can add to the mortality. We report a case of Cushing's disease in a young female managed with trans-sphenoidal surgery, followed by a challenging post-operative period complicated with multiple thrombotic phenomena, ultimately succumbed.

Case presentation: A 32-year-old Sri Lankan female presented with overt features of Cushing's syndrome and diagnosed to have ACTH dependent Cushing's disease with pituitary microadenoma. She underwent trans-sphenoidal surgery, following which she developed fatal multiple complications including diverticular rupture and ischemic colitis, needing hemicolectomy, followed by a parieto-occipital infarction.

Conclusion: This case highlights important and aggressive complications associated with Cushing's syndrome giving rise to a challenging post-operative course. Diverticular rupture had been described in association with hypercortisolemia and this case adds to the existing literature. Post-operative ischemic colitis and stroke which contributed to the death of this patient could have been due to the procoagulant state associated with Cushing's syndrome, with a high risk during the immediate post-operative period. This emphasizes the need to consider post-operative thromboprophylaxis in patients undergoing surgery for Cushing's syndrome.

Keywords: Case report; Cushing’s disease; Diverticular rupture; Stroke; Thrombosis.

Publication types

  • Case Reports

MeSH terms

  • ACTH-Secreting Pituitary Adenoma / complications
  • ACTH-Secreting Pituitary Adenoma / diagnostic imaging
  • ACTH-Secreting Pituitary Adenoma / surgery*
  • Adenoma / complications
  • Adenoma / diagnostic imaging
  • Adenoma / surgery*
  • Adult
  • Diverticulitis, Colonic / complications
  • Fatal Outcome
  • Female
  • Humans
  • Intestinal Perforation / etiology
  • Natural Orifice Endoscopic Surgery
  • Pituitary ACTH Hypersecretion / complications*
  • Postoperative Complications / etiology*
  • Stroke / etiology*