The simultaneous occurrence of paraganglioma, Takotsubo syndrome, and Markis type I coronary artery ectasia in the same patient is a rare, high-risk clinical syndrome: a case report

BMC Cardiovasc Disord. 2023 Nov 3;23(1):536. doi: 10.1186/s12872-023-03577-1.

Abstract

Background: Population-wide, paraganglioma (PGL) is uncommon. The incidence of Takotsubo syndrome (TTS) ranges from 0.5% to 0.9% and also is an exceedingly rare manifestation of PGL. Coronary artery ectasia (CAE) is also uncommon, with an incidence ranging from 1.2% to 4.9%. Herein, we present a case of PGL, TTS, and Markis type I CAE that occured in the same patient.

Case presentation: A man in his early 40s was admitted to our hospital with a 16-hour history of abdominal colic. Computed tomography and laboratory examination led to the diagnosis of PGL, coronary angiography led to the diagnosis of Markis type I or Chinese type III CAE, and two echocardiographic examinations led to the diagnosis of TTS. When the patient was treated by phenoxybenzamine instead of surgery for the PGL, his blood pressure and glucose level gradually returned to normal. The CAE was treated by thrombolysis, antiplatelet medications, atorvastatin, and myocardial protection therapies. No symptoms of PGL, CAE, or TTS were seen during a 6-month follow-up, and the patient had an excellent quality of life. We confirmed that phenoxybenzamine was the cause of the TTS because paradoxical systolic motion of the apex, inferior wall, left ventricular anterior wall, and interventricular septum were similarly recovered when the PGL was treated by phenoxybenzamine.

Conclusions: To raise awareness of this illness and prevent misdiagnosis, we have herein presented a case of TTS that was brought on by PGL with Markis type I CAE for clinicians' reference. In addition, in clinical practice, we should consider the possibility of a concomitant coronary artery disease even if the TTS is caused by a PGL-induced catecholamine surge.

Keywords: Case report; Coronary artery ectasia; Diagnostic imaging; Paraganglioma; Phenoxybenzamine; Takotsubo syndrome.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Coronary Aneurysm*
  • Coronary Angiography / methods
  • Coronary Vessels
  • Dilatation, Pathologic
  • Humans
  • Male
  • Phenoxybenzamine
  • Quality of Life
  • Risk Factors
  • Syndrome
  • Takotsubo Cardiomyopathy* / diagnostic imaging
  • Takotsubo Cardiomyopathy* / therapy
  • Vascular Diseases*

Substances

  • Phenoxybenzamine