Endocrine Abstracts

Introduction

Some, cases of autoimmune diseases were previously reported after surgical remission of CD. In most cases, autoimmune diseases have generally been reported in younger females. However, some cases of Hashimoto’s thyroiditis, multiple sclerosis, vitiligo, and sarcoidosis in male patients, after the restoration of hypercortisolemia have been reported as well. Nevertheless, an immediate occurrence of severe erosive rheumatoid arthritis (RA) in an elderly male patient after bilateral adrenalectomy while taking glucocorticoid replacement therapy has not been reported so far.

Case

A 67-year-old male patient admitted to our outpatient clinic with classical signs and symptoms of CS. CS was diagnosed by a failure of suppression of cortisol on low and high dose dexamethasone suppression tests, high urinary free cortisol, and high plasma ACTH levels. A pituitary adenoma could not be found on magnetic resonance imaging, therefore, an IPSS (inferior petrosal sinus sampling) was performed and the result was compatible with an ectopic CS. However, an ectopic tumor could not be found on the neck, chest, and abdominal CT scans, as well as on 68Ga- DOTATATE PET-CT. Bilateral adrenalectomy was performed and treatment with hydrocortisone and fludrocortisone was started. Six months later, the patient was admitted with bilateral small joint arthritis of the hands and morning stiffness. The serum Anti – CCP (Cyclic Citrullinated Peptide, erythrocyte sedimentation rate, and C-reactive protein were 6.2 U/ml (< 5 U/ml), 38 mm/h (< 20 mm/h), and 26 mg/l (< 5 mg), respectively. A hand x-ray showed bilateral space narrowing of the proximal interphalangeal joints with marginal erosions of the proximal interphalangeal joints, soft tissue swelling with periarticular osteopenia, and ulnar deviation of the 2. phalanxes. The patient was diagnosed with erosive RA and treatment with methotrexate was started.

Discussion

The occurrence of autoimmune diseases after remission of CS has been reported in some cases. The restoration of the physiological production of cortisol may cause an immune reaction, leading to the onset of autoimmune disease. RA is an autoimmune disease that is generally seen in females between 40–50 years of age. Our case is the first to report an elderly male patient with erosive RA after bilateral adrenalectomy who was taking physiological doses of hydrocortisone. Therefore, all patients with CS, including elderly male patients, should be closely observed for the occurrence of autoimmune disease after successful restoration of hypercortisolemia.