| Abstract [eng] |
Cushing syndrome (CS) is a state caused by chronic hypercortisolemia. CS is associated with low quality of life, high morbidity, and mortality. Diagnosis and treatment of adrenocorticotropic hormone (ACTH) independent CS with bilateral adrenal masses remain a major challenge in current clinical practice. Although adrenal venous sampling (AVS) is increasingly used to lateralize cortisol secretion, the current evidence regarding its impact on clinical decision-making for the syndrome is limited to case reports and small studies, and there is no consensus on treatment strategy. Research aim – to research scientific literature on the diagnosis and treatment of ACTH-independent CS with bilateral adrenal masses, and to conduct a clinical case analysis on this topic. Research objectives. 1) To assess the role of AVS in the selection of treatment strategies for patients with ACTH-independent CS with bilateral adrenal masses. 2) To compare treatment strategies for bilateral adrenal lesions across different centers and evaluate their outcomes. 3) To present diagnostic and treatment approaches applied in a clinical case. 4) To summarise clinical practice recommendations on the diagnosis and treatment of ACTH-independent CS with bilateral adrenal masses. Research methods. The comprehensive literature review was conducted in accordance with PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) criteria: keywords, inclusion and exclusion criteria, and a study selection flowchart. The PubMed database was chosen for the literature search. The most suitable studies published between 2015 and 2025 in English were selected using topic-relevant keyword combinations. The literature review was supplemented with an additional case analysis of bilateral adrenal adenomas. Research results. 16 studies were included in the literature review. Adrenal venous sampling (AVS) was used to lateralize cortisol secretion in 11 publications and was considered a valuable tool for treatment decision-making in 9 studies. Cortisol lateralization on AVS was identified in 38,0% (n = 30) of cases, of whom 29 underwent unilateral adrenalectomy, with disease recurrence only observed in 2 cases (6,9%). In the remaining 51 cases, unilateral adrenalectomy was performed by removing the larger adrenal gland, and disease recurrence was observed in 13 cases (25,5%). Laparoscopic adrenalectomy is the treatment of choice, while data on minimally invasive treatment options are not sufficient. Medical therapy may be a temporary solution for hypercortisolemia control until surgery, whereas observation is used in cases of mild autonomous cortisol secretion. Case report. An 84-year-old woman was evaluated for clinical features of hypercortisolism. ACTH- independent Cushing syndrome was confirmed by biochemical tests, and bilateral adrenal lesions were identified on CT. Due to the patient’s comorbidities and contraindications to surgical treatment, minimally invasive percutaneous radiofrequency ablation of the right adrenal gland was performed after adrenal venous sampling (AVS) demonstrated cortisol lateralization. No recurrence of the disease was observed over a nearly 4-year period. Conclusions. AVS is a valuable diagnostic tool that may prevent radical bilateral adrenalectomy in patients with ACTH-independent CS and bilateral adrenal lesions. Removal of the radiologically larger adrenal gland, when AVS is not performed, or cortisol lateralization is not identified, is associated with higher recurrence and lower remission rates of CS. Percutaneous radiofrequency ablation is an effective alternative to surgical treatment. |
