Unusual Case of Psychosis Linked to Adrenal Tumour
In older patients, elevated blood pressure is often observed during visits to general practitioners. However, if it is not measurable, this should raise particular concerns. If a psychotic episode also occurs, prompt action is needed.
Bastien Picut and his colleagues at Valais Hospital in Sion, Switzerland, reported an unusual case of a 60-year-old woman with decompensated metabolic syndrome and psychosis caused by a typical adrenal cortical carcinoma (ACC; adrenocortical oncocytic neoplasm, ACON).
The Patient and Her History
The woman presented to the hospital with symptoms of refractory arterial hypertension, accompanied by dizziness, headaches, and asthenia, and a weight gain of 20 kg within a span of 6 months. Despite maximal antihypertensive therapy, her systolic blood pressure remained at approximately 160 mmHg.
Findings and Diagnosis
Examination revealed the onset of type 2 diabetes, requiring immediate insulin, and mixed hyperlipidaemia associated with hypokalaemia and hypernatremia. While hospitalised under geriatric care, the patient experienced a psychotic breakdown characterised by persecutory delirium, noncritical visual hallucinations, and mistrust of contacts associated with anxiety.
A comprehensive laboratory analysis was performed. Liver function tests were abnormal, with significantly elevated levels of gamma-glutamyl transpeptidase, total bilirubin, alanine aminotransferase, aspartate aminotransferase, and alkaline phosphatase. Additionally, hypernatremia (149 mmol/L) was noted (normal range, 135-145 mmol/L). The calcium and ammonia levels were normal at 2.31 mmol/L and 36 μmol/L, respectively.
Urine analysis revealed proteinuria, ketonuria, urobilinogen, and haematuria. The levels of dehydroepiandrosterone sulphate and urinary catecholamines, including metanephrine, normetanephrine, and methoxytyramine, were marginally elevated.
Following the initial evaluation, an ultrasound was performed, revealing a large, bilobed retro-pancreatic mass near the left kidney and para-aortic region. Subsequent abdominal CT confirmed a bilobed left adrenal mass measuring 8 × 5 cm and 1.8 × 2.2 cm, which was vascularised without calcifications. The CT scan ruled out the presence of any locoregional or distant spread of the disease.
The differential diagnosis of pheochromocytoma was considered. Radiological findings and elevated urinary metanephrines, suggesting the secretion of catecholamine precursors. No other causes for the elevated urinary catecholamines were identified.
Prior to the intervention, the patient was medically managed with haloperidol (1 mg orally three times daily) and clomethiazole (192 mg orally twice daily as needed) for psychiatric decompensation. For diabetes management, metformin 500 mg orally three times daily, linagliptin 5 mg orally, and insulin glargine 30U subcutaneously were administered. Hypertension was managed with metoprolol (50 mg), spironolactone (25 mg), lisinopril (20 mg), and doxazosin (4 mg) orally.
Subsequently, laparoscopic left adrenalectomy was performed without intraoperative complications. The patient required adrenergic support until the fourth postoperative day. Additionally, all antihypertensive medications were discontinued, except for beta blockers.
Discussion
This is the first ACON case described in the literature with such an atypical clinical presentation, according to the authors. Psychotic manifestations of ACC have been previously described; however, they are associated with cortisol-secreting ACC as part of Cushing syndrome.
Furthermore, the doctors could not conclusively determine whether the tumour was a pure non-secreting ACON. The clinical presentation resembled Cushing syndrome, and hypokalaemia could be attributed to vasospasm from secondary hyperaldosteronism.
Aldosterone secretion from the tumour could not be clearly assessed due to the ongoing use of antihypertensive medications (beta blockers, spironolactone, calcium channel blockers, and angiotensin-converting enzyme inhibitors). This may have affected the aldosterone-renin ratio, leading to a potentially false-negative assessment.
The variable clinical presentation and the lack of sensitivity and specificity of preoperative diagnostic procedures, such as imaging in aggressive malignant diseases, make adrenocortical oncocytic neoplasms a significant challenge for clinicians.
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