A unique case documented in JCEM Case Reports reveals a rare instance of a hormonal disorder known as Cyclic Cushing Syndrome (CS) relapsing after a patient was infected with COVID-19. This relapse occurred after an impressive 19-year period of remission, raising concerns among researchers regarding the long-term impacts of such viral infections and glucocorticoid treatment on predisposed patients.
The subject of the study, a 49-year-old male, was initially diagnosed with adrenocorticotropic hormone (ACTH)-dependent CS at the age of 30. After begun treatment with steroidogenesis inhibitors such as trilostane and mitotane, he achieved spontaneous remission and remained stable for nearly two decades. During this time, he reported no symptoms indicative of Cushing’s syndrome, and his annual evaluations showed normal hormone levels.
The patient's health status changed significantly when he contracted COVID-19. To manage the viral infection, he was placed on a month-long regimen of high-dose glucocorticoids, including methylprednisolone and prednisolone. Soon after starting this treatment, he began to exhibit typical signs of Cushing's syndrome relapse, including notable fatigue, significant weight gain (11 kg), a characteristic “moon face,” and dangerously elevated blood pressure, which ultimately necessitated his readmission to a healthcare facility.
Researchers from The University of Osaka revealed that this case was particularly unusual, as Cyclic Cushing Syndrome generally operates under a negative feedback loop. However, this patient’s introduction of external glucocorticoids appears to have instigated a positive feedback loop instead, resulting in increased production of ACTH and cortisol, ultimately leading to a relapse of the condition.
The authors hypothesize that the combination of stress due to the COVID-19 infection and the administered steroid therapy acted synergistically to foster this relapse, showcasing the complexities associated with managing patients with a history of cyclic CS, especially during acute illnesses.
Following the relapse, diagnostic tests indicated potential Cushing’s disease originating from a pituitary tumor. However, further imaging failed to identify any such tumor. Subsequent medical scans ultimately identified an 8-mm tumor located in the anterior mediastinum, leading to the diagnosis of an ACTH-secreting thymic typical carcinoid tumor—an example of ectopic CS. This finding was confirmed when a partial thymectomy resulted in the patient achieving a third remission.
This case highlights difficulties in accurately diagnosing Cushing’s syndrome, especially during its natural “trough phase,” when hormone levels fluctuate.
The authors of the study emphasize the importance of monitoring patients with a history of Cyclic Cushing Syndrome, particularly those who may have seemingly resolved their condition over long periods. They assert that clinicians should exercise caution when administering glucocorticoids to such individuals and should closely observe for any emergence of cushingoid symptoms following episodes of infection and glucocorticoid treatment.
In their concluding remarks, the research team strongly advocates for ongoing and meticulous follow-up for cyclic CS patients, even after years of apparent stability. The findings from this case serve as a critical reminder within the medical community regarding the complex interplay between stressors like viral infections and endocrine disorders, particularly in patients with a history of cyclic hormonal fluctuations.
Personalise this feed
Your specialty. Your sources. Your digest.
All set up in under 2 minutes.
Personalise this feed
Your specialty. Your sources. Your digest.
All set up in under 2 minutes.