A case of adrenal Cushing’s syndrome with bilateral adrenal masses

Abstract

SummaryA functional lesion in corticotrophin (ACTH)-independent Cushing’s syndrome is difficult to distinguish from lesions of bilateral adrenal masses. Methods for distinguishing these lesions include adrenal venous sampling and 131I-6β-iodomethyl-19-norcholesterol (131I-NP-59) scintigraphy. We present a case of a 29-year-old Han Chinese female patient with a history of hypercholesterolaemia and polycystic ovary syndrome. She presented with a 6month history of an 8kg body weight gain and gradual rounding of the face. Serial examinations revealed loss of circadian rhythm of cortisol, elevated urinary free-cortisol level and undetectable ACTH level (<5pg/mL). No suppression was observed in both the low- and high-dose dexamethasone suppression tests. Adrenal computed tomography revealed bilateral adrenal masses. Adrenal venous sampling was performed, and the right-to-left lateralisation ratio was 14.29. The finding from adrenal scintigraphy with NP-59 was consistent with right adrenal adenoma. The patient underwent laparoscopic right adrenalectomy, and the pathology report showed adrenocortical adenoma. Her postoperative cortisol level was 3.2μg/dL, and her Cushingoid appearance improved. In sum, both adrenal venous sampling and 131I-NP-59 scintigraphy are good diagnostic methods for Cushing’s syndrome presenting with bilateral adrenal masses.Learning pointsThe clinical presentation of Cushing’ syndrome includes symptoms and signs of fat redistribution and protein-wasting features.The diagnosis of patients with ACTH-independent Cushing’s syndrome with bilateral adrenal masses is challenging for localisation of the lesion.Both adrenal venous sampling and 131I-NP-59 scintigraphy are good methods to use in these patients with Cushing’s syndrome presenting with bilateral adrenal masses.

Skuteczność jednostronnej adrenalektomii laparoskopowej w hiperkortyzolemii i subklinicznym zespole Cushinga niezależnych od ACTH — badanie retrospektywne na dużej kohorcie

“…When the recurrence occurred, the contralateral adrenal gland was also removed. Such an approach is justified by other studies, and our results correlate with those indicating that, in the long-term, recurrence of hypercortisolaemia after unilateral adrenalectomy is seen in approximately 20% of patients [11,12].…”

ACTH-independent Cushing’s syndrome with bilateral cortisol-secreting adrenal adenomas: a case report and review of literatures

“…In this case, we used the AV: IVC aldosterone ratio to assess the successfulness of catheter insertion since the aldosterone concentration could remain stable during the sampling. There were some researches in which adrenaline concentration was measured to evaluate catheterization accuracy, however, catheterization process itself might lead to stress-induced fluctuation of adrenaline and further result in misjudgment [ 17 , 34 ]. Even though there is still no general agreement on the definition of catheterization success in AVS, aldosterone concentration instead of adrenaline could be a robust assay for this purpose, after the aldosterone overproduction being excluded.…”

Targeted Molecular Imaging in Adrenal Disease—An Emerging Role for Metomidate PET-CT

“…Although early 75 Se-selenomethyl-19-norcholesterol studies showed promising results, scintadren is currently no longer available for clinical studies and has been replaced by 131 I-iodomethyl-norcholesterol (NP-59) [25]. In a declining number of cases, NP-59 continues to be used to investigate the secretory status of adrenal adenomas and aid lesion lateralization [26,27,28,29,30,31]. However, these techniques have significant shortcomings including delivery of a relatively high radiation dose to the adrenal glands, time-consuming acquisition protocols, a requirement for the patient to take a 7–10 day course of high-dose glucocorticoids to suppress non-autonomous hormone synthesis and low sensitivity with poor spatial resolution (traditionally only allowing reliable detection of lesions >2 cm in diameter, although single photon emission computed tomography (SPECT) may afford improved resolution).…”