A New Insight into the Surgical Treatment of Primary Macronodular Adrenal Hyperplasia

Tanno, Fabio Y; Srougi, Victor; Almeida, Madson Q.; Yamauchi, Fernando Ide; Coelho, Fernando Morbeck Almeida; Nishi, Mirian Yumie; Zerbini, Maria Cláudia Nogueira; Soares, Iracy Silvia Corrêa; Pereira, Maria Adelaide Albergaria; Charchar, Helaine Laiz Silva; Lacombe, Amanda Meneses Ferreira; Brondani, Vânia Balderrama; Srougi, Miguel; Nahas, Willian; Mendonça, Berenice Bilharinho; Chambô, José Luis; Fragoso, Maria Candida Barisson Villares

doi:10.1210/jendso/bvaa083

Cited by 21 publications

(12 citation statements)

References 32 publications

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“…It is also known to use image techniques such as scintigraphy to decide which side of adrenal glands to be removed ( 39 ), however, the information is only about the size or hormonal activity of adrenal disease and not able to suggest the partial adrenalectomy. The current evidence regarding the use of partial adrenalectomy is limited ( 40 ). If circulating miRNA expression can predict the subtypes of PBMAH in addition to imaging information, partial adrenalectomy could be adopted more often in cases of multiple adenoma types, which can’t be distinguished from hyperplasia types using scintigraphy.…”

Section: Discussionmentioning

confidence: 99%

Heterogeneous circulating miRNA profiles of PBMAH

et al. 2022

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ObjectivePrimary bilateral macronodular adrenal hyperplasia (PBMAH), a rare cause of Cushing syndrome, is often diagnosed as a bilateral adrenal incidentaloma with subclinical cortisol production. Circulating microRNAs (miRNAs) are a characteristic of adrenocortical adenomas, but miRNA expression in PBMAH has not been investigated. We aimed to evaluate the circulating miRNA expression in patients with PBMAH and compare them with those in patients with non-functioning adrenocortical adenoma (NFA) and cortisol-producing adrenocortical adenoma (CPA).MethodsmiRNA profiling of plasma samples from four, five, and five patients with NFA, CPA, and PBMAH, respectively, was performed. Selected miRNA expressions were validated using quantitative RT-PCR.ResultsPBMAH samples showed distinct miRNA expression signatures on hierarchical clustering while NFA and CPA samples were separately clustered. PBMAH was distinguished from the adenoma group of NFA and CPA by 135 differentially expressed miRNAs. Hsa-miR-1180-3p, hsa-miR-4732-5p, and hsa-let-7b-5p were differentially expressed between PBMAH and adenoma (P = 0.019, 0.006, and 0.003, respectively). Furthermore, PBMAH could be classified into two subtypes based on miRNA profiling: subtype 1 with a similar profile to those of adenoma and subtype 2 with a distinct profile. Hsa-miR-631, hsa-miR-513b-5p, hsa-miR-6805-5p, and hsa-miR-548av-5p/548k were differentially expressed between PBMAH subtype 2 and adenoma (P = 0.027, 0.027, 0.027, and 1.53E-04, respectively), but not between PBMAH, as a whole, and adenoma.ConclusionCirculating miRNA signature was identified specific for PBMAH. The existence of subtype-based miRNA profiles may be associated with the pathophysiological heterogeneity of PBMAH.

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Section: Discussionmentioning

confidence: 99%

Heterogeneous circulating miRNA profiles of PBMAH

et al. 2022

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“…Recently, a more promising surgery method was proposed for the treatment of PBMAH, which was called adrenal-sparing surgery. In 2020, Tanno et al [19] first reported this surgery method and demonstrated that most patients undertaken adrenal-sparing surgery had complete hypercortisolism control after 41 months follow-up without the disadvantages of lifetime corticosteroid replacement in a prospective cohort. However, as many patients in this study had mild hypercortisolism, the benefit of this surgery in PBMAH patients with overt CS is needed to be clarified.…”

Section: Discussionmentioning

confidence: 99%

“…BA has been taken for the treatment of choice to achieve cure of hypercortisolism [16], but it can result to adrenal insufficiency permanently, requiring lifelong corticosteroid replacement and causing health care burden, including under-and overreplacement associated with increased metabolic risk and adrenal crisis risk, respectively [4]. Considering these negative effects, UA and even adrenal-sparing surgery has been proposed as a promising and safe alternative to BA in patients with mild hypercortisolism or clear adrenal asymmetry [4,[17][18][19]. However, patients with UA or adrenal-sparing surgery may have the risk of recurrence and required a second surgery according to the CS-related manifestations such as uncontrolled hypertension, diabetes, obesity, and dyslipidemia [12], which correlate in part with the extent and duration of cortisol excess [20].…”

Section: Introductionmentioning

confidence: 99%

The Relationship Between Baseline Cortisol Levels and Surgery Method of Primary Bilateral Macronodular Adrenal Hyperplasia

Gao

Sun

et al. 2022

Horm Metab Res

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Aim was to explore the associations between baseline cortisol levels and surgery method of primary bilateral macronodular adrenal hyperplasia (PBMAH). We retrospectively reviewed the clinical features and management of 30 patients (18 females and 12 males) who were diagnosed with PBMAH in our center between 2005 and 2019. Based on surgery method, we divided the patients into two groups: unilateral adrenalectomy (UA) group; and bilateral adrenalectomy (BA) group. Serum cortisol rhythm and 24-hour urinary free cortisol (UFC/24 h) levels were assayed using chemiluminescence method. Associations between baseline cortisol levels and BA were assessed using logistic regression. The predictive value of baseline cortisol levels for BA was calculated using receiver operating characteristic (ROC) curves. Twenty patients (66.7%) underwent UAs and ten patients (33.3%) underwent BAs. After adjusting for age, sex, BMI, SBP, and adrenal volume, the concentrations of baseline serum cortisol (8 AM, 4 PM, and 0 AM) and UFC/24 h were associated with bilateral adrenalectomy (all p<0.05). The area under the ROC curve based on 8 AM serum cortisol level model was larger than that in models based on 4 PM, 0 AM serum cortisol levels and UFC/24 h, but the differences were non-significant (all p>0.05). According to maximum Youden index criteria, the optimal cutoffs of 8 AM serum cortisol level and UFC were 26.89 μg/dl and 406.65 μg/24 h, respectively, for BA. The baseline cortisol levels are positively associated with BA. Increased levels of baseline cortisol levels may predict higher possibility of BA, which should be confirmed by prospective studies.

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“…Such patients are prone to acute adrenal crisis as a complication especially under stress or infection, and may have a lower quality of life, so they have to be good compliance 8 12 15 35 . Unilateral total adrenalectomy associated with contralateral partial adrenal (adrenal-sparing surgery) has been observed to achieve good treatment results in PBMAH 55 . This approach avoids the need for lifelong steroid replacement in most cases and has a low rate of adrenal insufficiency and recurrence 56 .…”

Section: Adrenalectomymentioning

confidence: 99%

Treatment of Primary Pigmented Nodular Adrenocortical Disease

et al. 2022

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Primary pigmented nodular adrenocortical disease (PPNAD) is a rare cause of adrenocorticotropin hormone (ACTH)-independent Cushing’s syndrome (CS), which mainly occurs in children and young adults. Treatment options with proven clinical efficacy for PPNAD include adrenalectomy (bilateral or unilateral adrenalectomy) and drug treatment to control hypercortisolemia. Previously, the main treatment of PPNAD is bilateral adrenal resection and long-term hormone replacement after surgery. In recent years, cases reports suggest that unilateral or subtotal adrenal resection can also lead to long-term remission in some patients without the need for long-term hormone replacement therapy. Medications for hypercortisolemia, such as Ketoconazole, Metyrapone and Mitotane et.al, have been reported as a preoperative transition for in some patients with severe hypercortisolism. In addition, tryptophan hydroxylase inhibitor, COX2 inhibitor Celecoxib, somatostatin and other drugs targeting the possible pathogenic mechanisms of the disease are under study, which are expected to be applied to the clinical treatment of PPNAD in the future. In this review, we summarize the recent progress on treatment of PPNAD, in which options of surgical methods, research results of drugs acting on possible pathogenic mechanisms, and the management during gestation, in order to provide new ideas for clinical treatment.

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A New Insight into the Surgical Treatment of Primary Macronodular Adrenal Hyperplasia

Cited by 21 publications

References 32 publications

Heterogeneous circulating miRNA profiles of PBMAH

Heterogeneous circulating miRNA profiles of PBMAH

The Relationship Between Baseline Cortisol Levels and Surgery Method of Primary Bilateral Macronodular Adrenal Hyperplasia

Treatment of Primary Pigmented Nodular Adrenocortical Disease

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