Primary Aldosteronism Surgical Outcome Research Articles

ACTH stimulation and oral salt loading tests detect biochemical outcome early after primary aldosteronism surgery.

In primary aldosteronism (PA), the biochemical outcomes of the Primary Aldosteronism Surgical Outcome study are used to assess aldosterone hypersecretion 6-12 months after surgery. However, few studies have investigated whether the outcomes can be predicted in the early postoperative period. In this retrospective study, we evaluated whether the adrenocorticotropin stimulation test (AST) and oral salt loading test (OST) performed immediately after surgery could predict biochemical outcomes 1 year after surgery. We assessed 268 patients with PA who underwent adrenalectomy at our hospital between 2008 and 2020, underwent AST and OST within 15 days of surgery, and were assessed for biochemical outcomes 1 year after surgery. Patients were divided into two groups: biochemical complete success (B-com; n = 219) and incomplete success (B-inc; n = 49). Patients were divided into clinical complete and partial success and absent success groups. The relationships between various AST and OST values and outcomes were analyzed. The B-inc group had significantly higher plasma aldosterone concentration (PAC) and PAC/serum cortisol ratio (PAC/Cort) at baseline and after ACTH loading in AST and 24-hour urine aldosterone in OST than the B-com group. PAC/Cort at 30 min after ACTH loading (area under the curve (AUC) = 0.76) and 24-hour urine aldosterone (AUC = 0.77) were relatively superior predictors of the outcome. Parameters after ACTH loading were better predictors of biochemical and clinical outcomes than baseline. AST and OST immediately after surgery can predict biochemical and clinical outcomes 1 year after surgery in patients with PA.

7211 Clinical and Molecular Characterization Of An Aldosterone-Producing Adenoma Harboring Double Somatic Mutations In CTNNB1 and GNA11

Abstract Disclosure: A. Blinder: None. K. Nanba: Grant Recipient; Self; AstraZeneca. A. Udager: None. Y. Hirokawa: None. T. Miura: None. H. Okuno: None. K. Moriyoshi: None. Y. Yamazaki: None. H. Sasano: None. A. Yasoda: None. N. Satoh-Asahara: None. W.E. Rainey: None. T. Tagami: None. Background: Somatic activating mutations in CTNNB1, encoding β-catenin, have been detected in a small subset of aldosterone-producing adenomas (APAs). As a possible pathogenesis, an association of CTNNB1-mutated APA with pregnancy and menopause has been proposed. A recent study reported double somatic mutations in CTNNB1 and GNA11/Q, encoding guanine nucleotide-binding protein subunits alpha-11 and alpha-Q, in APA. However, due to its rare incidence, clinical and molecular features of APA associated with these mutations have not been well characterized. Herein, we report a case of APA harboring somatic CTNNB1 and GNA11 mutations. Clinical Case: A 46-year-old Japanese woman presented with hypertension and hypokalemia. She had two pregnancies in the past but had no history of pregnancy-induced hypertension. She had regular menstrual cycles at presentation. Laboratory testing showed elevated plasma aldosterone concentration with suppressed plasma renin activity. She was diagnosed as having primary aldosteronism based on the results of a captopril challenge test. Adrenal computed tomography revealed a 2 cm right adrenal mass. Adrenal venous sampling indicated excess aldosterone production from the right adrenal gland. She underwent right laparoscopic adrenalectomy. Histologic diagnosis of the resected tumor was adrenocortical adenoma based on the criteria of Weiss. Notably, Ki-67 labeling index was high (6% at hotspots). After surgery, her blood pressure and serum potassium both normalized. According to the primary aldosteronism surgical outcome (PASO) study criteria, PA remained clinically and biochemically cured at follow-up 2.5 years after surgery. Results: Immunohistochemistry (IHC) showed high and diffuse expression of aldosterone synthase (CYP11B2) within the tumor, whereas immunoreactivity of 17α-hydroxylase (CYP17A1) and 11β-hydroxylase (CYP11B1), both required for cortisol biosynthesis, was markedly low. VSNL1, a marker for the zona glomerulosa (ZG) where physiologic aldosterone biosynthesis occurs, was highly expressed within the tumor. CYP11B2 IHC-guided tumor capture followed by targeted next-generation sequencing identified double somatic CTNNB1 (p.D32Y) and GNA11 (p.Q209H) mutations. Quantitative real-time RT-PCR revealed high tumor expression of LHCGR (LH receptor) and GNRHR (GNRH receptor) mRNA levels compared with those in adjacent normal adrenal (148-fold and 56-fold, respectively). Immunofluorescence staining of the tumor revealed the presence of activated β-catenin, consistent with features of the normal ZG. Conclusions: Our study indicates that an APA with double somatic mutations in CTNNB1 and GNA11 has characteristics of the ZG and could occur in adults with no clear association with pregnancy or menopause. Presentation: 6/3/2024

Safety and efficacy of bilateral superselective adrenal arterial embolization for treatment of idiopathic hyperaldosteronism: a prospective single-center study

ObjectiveThis study aimed to assess the efficacy and safety of bilateral superselective adrenal arterial embolization (SAAE) in patients with bilateral idiopathic hyperaldosteronism (IHA), a subtype of PA.MethodsNinety-eight patients with bilateral IHA underwent bilateral SAAE between August 2022 and August 2023. Sixty-eight patients were followed up for up to 12 months. The study outcomes were evaluated using the criteria provided by the Primary Aldosteronism Surgical Outcome (PASO) guidelines.ResultsThe mean reductions in systolic and diastolic blood pressure were 27.4 ± 21.3 mmHg and 23.1 ± 17.4 mmHg, respectively (p < 0.001). The rates of clinical success and biochemical success after adrenal artery ablation were 63.2% (43/68) and 39.7% (27/68), respectively. Overall, there were significant reductions in daily defined doses (DDD), aldosterone/renin ratio (ARR), and plasma aldosterone levels (p < 0.001). Plasma renin levels increased by a mean value of 10.4 ± 39.0 pg/mL (p = 0.049), and potassium levels increased by 0.40 ± 0.63 mmol/L (p < 0.001). No significant adverse events were reported during SAAE or the follow-up period of up to one year. Additionally, no abnormalities were detected by adrenal 68Ga-Pentixafor PET/CT scans before or after SAAE.ConclusionBilateral SAAE appears to lead to sustained improvements in blood pressure and biochemical parameters in patients with bilateral PA, with minimal adverse effects. This suggests that bilateral SAAE could serve as an effective alternative approach for treating bilateral IHA, potentially curing this condition.

Rationality and implication of catheter-based adrenal ablation for bilateral primary aldosteronism.

Mineralocorticoid receptor antagonists (MRAs) for bilateral primary aldosteronism (PA) are the mainstay option recommended by guidelines, but poor compliance occurs due to numerous side effects. We aimed to examine whether catheter-based adrenal ablation could be an alternative treatment for bilateral PA.644 PA patients were included from a total of 6054 hypertensive patients. Adrenal CT scan and adrenal venous sampling (AVS) were both performed for PA subtype classification. Clinical and biochemical outcomes were assessed at 6 months after treatment according to the Primary Aldosteronism Surgical Outcome (PASO) criteria.93 patients with PA were recruited to be treated by adrenal ablation, including 25 bilateral PA and 68 unilateral PA according to AVS results. Office SBP and DBP significantly decreased from baseline levels, serum potassium levels increased and ARR significantly decreased (p < 0.01) in both the bilateral and unilateral groups. In the bilateral group, complete, partial and absent clinical success was achieved in 6 (24.0%), 11 (44.0%) and 8(32.0%) patients, respectively. In the unilateral group, complete, partial and absent clinical success was achieved in 12 (17.6%), 37 (54.4%), and 19 (27.9%) patients, respectively. The numbers of patients achieving complete, partial, and absent biochemical success were 15 (60.0%), 6 (24.0%), and 4 (16.0%), respectively, in the bilateral group versus 37 (54.4%), 9 (13.2%), and 22 (32.3%), respectively, in the unilateral group. In conclusion, we provide evidence for the beneficial outcomes of unilateral adrenal ablation for patients with bilateral PA. Our findings provide insight into an alternative option for patients with bilateral excess aldosterone.

A comparison of the performance of 68Ga-Pentixafor PET/CT versus adrenal vein sampling for subtype diagnosis in primary aldosteronism.

To investigate the diagnostic efficiency and prognostic value of 68Ga-Pentixafor PET/CT in comparison with adrenal vein sampling (AVS) for functional lateralization in primary aldosteronism (PA). Histology and long-term clinical follow-up normally serve as the gold standard for such diagnosis. We prospectively recruited 26 patients diagnosed with PA. All patients underwent 68Ga-Pentixafor PET/CT and AVS. Postsurgical biochemical and clinical outcomes of patients with unilateral primary aldosteronism (UPA), as diagnosed by PET/CT or AVS, were assessed by applying standardized Primary Aldosteronism Surgical Outcome (PASO) criteria. Immunohistochemistry (IHC) was performed to detect the expression of aldosterone synthase (CYP11B2) and CXCR4. On total, 19 patients were diagnosed with UPA; of these, 13 patients were lateralized by both PET/CT and AVS, four patients were lateralized by PET-only, and two by AVS-only. Seven subjects with no lateralization on AVS and PET received medical therapy. All patients achieved complete biochemical success except one with nodular hyperplasia lateralized by AVS alone. The consistency between PET/CT and AVS outcomes was 77% (20/26). Moreover, CYP11B2-positive nodules were all CXCR4-positive and showed positive findings on PET. Patients who achieved complete biochemical and clinical success had a higher uptake on PET as well as stronger expression levels of CXCR4 and CYP11B2. Our analysis showed that 68Ga-Pentixafor PET/CT could enable non-invasive diagnosis in most patients with PA and identify additional cases of unilateral and surgically curable PA which could not be classified by AVS. 68Ga-Pentixafor PET/CT should be considered as a first-line test for the future classification of PA.

Unilateral Primary Aldosteronism: Long-Term Disease Recurrence After Adrenalectomy.

Primary aldosteronism (PA) is frequently caused by a unilateral aldosterone-producing adenoma with a PA-driver mutation. Unilateral adrenalectomy has a high probability of short-term biochemical remission, but long-term postsurgical outcomes are relatively undefined. Our objective was to investigate the incidence of long-term recurrence of PA in individuals with postsurgical short-term biochemical remission. Adrenalectomized patients for unilateral PA were included from a single referral center. Histopathology and outcomes were assessed according to international histopathology of unilateral primary aldosteronism and PASO (Primary Aldosteronism Surgical Outcome) consensuses. Genotyping was performed using CYP11B2 (aldosterone synthase)-guided sequencing. Classical adrenal histopathology, exemplified by a solitary aldosterone-producing adenoma, was observed in 78% of 90 adrenals, compared with 22% with nonclassical histopathology. The classical group displayed higher aldosterone-to-renin ratios (P=0.013) and lower contralateral ratios (P=0.008). Outcome assessments at both short (12 months [7; 12]) and long (89 months [48; 124]) terms were available for 57 patients. At short-term assessment, 53 (93%) displayed complete biochemical success (43 classical and 10 nonclassical), but long-term assessment demonstrated biochemical PA recurrence in 12 (23%) with an overrepresentation of the nonclassical histopathology (6 [60%] of 10 nonclassical histopathology versus 6 [14%] of 43 classical histopathology; P=0.005). PA-driver mutations were identified in 97% of 64 aldosterone-producing adenomas; there was no association of the aldosterone-producing adenoma genotype with PA recurrence. A substantial proportion of individuals display postsurgical biochemical recurrence of PA, which is related to the histopathology of the resected adrenal gland. These findings emphasize the role of histopathology and the requirement for continued outcome assessment in the management of surgically treated patients for PA.

Adrenalectomy for primary aldosteronism and its related surgical characteristics.

Primary aldosteronism (PA) is a common cause of secondary hypertension. Adrenalectomy is an effective treatment for unilateral PA, particularly aldosterone-producing adenoma (APA), resulting in improvements in biochemical parameters and blood pressure in the vast majority of patients. The article provides a comprehensive overview of PA, focusing on the outcomes of adrenalectomy for PA and the factors that may suggest prognostic implications. Analysis of the outcome of different PA patients undergoing adrenalectomy in terms of preoperative factors, vascular and adipose conditions, type of pathology, and somatic variants. In addition, it is recommended to use the histopathology of primary aldosteronism (HISTALDO) consensus to classify the patient's pathological type, with classical and nonclassical pathological types showing a different prognosis and possibly being associated with an unresected contralateral adrenal gland. The primary aldosteronism surgical outcome (PASO) consensus sets uniform standards for postoperative outcomes in unilateral PA, but its setting of thresholds remains controversial. Partial adrenalectomy shows similar surgical results and fewer postoperative complications than total adrenalectomy, but there is a risk of missing the true source of abnormal aldosterone secretion. Steroid profiling and functional imaging techniques offer alternative options to adrenal vein sampling (AVS) for unilateral and bilateral judgments in patients with PA. A combination of factors is needed to predict the prognosis of PA patients undergoing adrenalectomy in order to manage patient expectations of the outcome of the procedure and to closely monitor blood pressure and biochemical parameters in patients who suggest a poorer prognosis.

FRI132 Aldosterone Secretory Capacity Immediately After Surgery Predicts Biochemical Outcome 1 Year After Surgery In Primary Aldosteronism

Abstract Disclosure: Y. Ishida: None. K. Nakai: None. Y. Tsurutani: None. K. Watanabe: None. J. Saito: None. T. Nishikawa: None. Background and Objective: In primary aldosteronism (PA), the biochemical outcome of the Primary Aldosteronism Surgical Outcome (PASO) study is used to assess aldosterone hypersecretion six months to one year after surgery. However, there are few reports on whether outcomes can be predicted in the early postoperative period. This retrospective study evaluated aldosterone secretory capacity immediately after surgery using the adrenocorticotropin (ACTH) stimulation test (AST) and oral salt loading test (OST). We hypothesized that these tests would predict biochemical outcomes one year after surgery. Methods: This study assessed 282 patients with PA who underwent adrenalectomy in our hospital between 2008 and 2020 and determined biochemical PASO outcomes 1 year after surgery. Patients with serum cortisol levels ≥ 5 μg/dL after a 1 mg overnight dexamethasone suppression test (n = 9) were excluded. Patients were divided into biochemical complete success (COM) and incomplete (partial + absent) success (INC) groups. AST and OST were performed within 15 days of surgery. The relationships between various AST and OST values and outcomes were analyzed. Results: Two hundred and twenty patients and 53 patients were classified into the COM and INC groups, respectively. The INC group had significantly higher plasma aldosterone concentration (PAC) and higher PAC/cortisol ratio (A/C) at baseline, 30 min, and 60 min after ACTH loading than the COM group. The INC group showed a significantly lower level of plasma renin activity. In the OST, the INC group had a significantly higher 24-hour urine aldosterone level. Of these values, A/C at 30 minutes after ACTH loading (area under the curve (AUC) = 0.77) and 24-hour urine aldosterone (AUC = 0.78) were relatively superior predictors of the outcome, with higher AUCs than plasma renin activity (AUC = 0.63), baseline PAC (AUC = 0.71), baseline ARR (AUC = 0.72), and baseline A/C (AUC = 0.70). A/C at 30 minutes after ACTH loading had 83% sensitivity and 62% specificity when 6.03 was set as the cutoff; 24-hour urine aldosterone had 84% sensitivity and 61% specificity when 2.6 was set as the cutoff. Conclusion: AST and OST immediately after surgery are useful for predicting biochemical outcomes 1 year after surgery in patients with PA. Presentation: Friday, June 16, 2023

Surgery based on computed tomography images might be feasible for primary aldosteronism patients with visible unilateral adenoma.

Primary aldosteronism (PA) with unilateral adrenal disease can be cured or improved by adrenalectomy. Adrenal venous sampling (AVS) is recommended to identify patients for surgical management. However, surgeries based on computed tomography (CT) images are only advocated for PA patients aged <35 with visible unilateral adenoma. Herein, we aimed to compare CT-based and AVS-based surgery outcomes for PA patients with visible unilateral adenomas for different age groups. A total of 178 PA patients who underwent unilateral adrenalectomy between June 2018 and January 2021 were included in the study based on CT (n=54) or AVS (n=124). Demographics, diagnostics, and follow-up data were retrospectively collected. Clinical and biochemical outcomes were analyzed according to Primary Aldosteronism Surgical Outcome (PASO) criteria at 1-year follow-up. Our results showed that complete clinical success (46.3%vs. 47.6%, p=0.875) and complete biochemical success (88.8%vs. 91.9%, p=0.515) were similar between the two groups. Age stratification revealed that patients >55 years old were likely to have worse biochemical outcomes; however, these were still not significantly different (21.4%vs. 8.6%, p=0.220). Of the 114 AVS-based patients who achieved complete biochemical success, 37 (32.4%) with bilateral normal or bilateral abnormal CT images changed treatment options according to AVS results, 1 (0.9%) avoided adrenalectomy on the wrong side. Our results indicated that surgery based on CT images might be feasible for highly selected PA patients with visible unilateral adenomas and less limited by age, while for those with normal adrenal or bilateral adrenal lesions, treatment strategy must be decided by AVS.

Validation of the Aldosteronoma Resolution Score as a Predictive Resolution Score of Hypertension After Unilateral Adrenalectomy for Primary Aldosteronism in a North-African Population.

Unilateral primary aldosteronism (UPA) is the most frequent surgically curable form of endocrine hypertension. Adrenalectomy is the cornerstone of treatment for UPA, but outcomes after surgery are variable. Aldosteronoma Resolution Score (ARS) is a four-item predictive score for the cure of hypertension after adrenalectomy for UPA and has been demonstrated to be valid in different populations. We aimed in this study to validate the accuracy of this score in a North-African population. Between 2000 and 2021, the charts of 71 Tunisian patients who underwent laparoscopic adrenalectomy for UPA were retrospectively reviewed. Postoperative outcomes were assessed using the primary aldosteronism surgical outcome (PASO) criterion. The accuracy of the ARS was determined retrospectively by receiver operating characteristic curve and area under the curve. Thirty-four patients (48%) had complete clinical success according to the PASO criteria. Multivariate regression analysis revealed that the main determinants of complete clinical success were the absence of diabetes (OR: 5.205), a BMI <30 (OR: 4.930), a number of antihypertensive medications ≤2 (OR: 8.667), a plasma ARR >332 (OR: 4.554) and an ARS score ≥3 (OR: 2.056). Cure rates were, respectively, 21.1, 51.6, and 66.6% for patients with a score ARS 0-1, 2-3, and 4-5. The AUC of the ARS was 0.837. The ARS is a sufficiently predictive score in our North-African population. It may be used preoperatively to predict the outcome after adrenalectomy in these populations.

Abstract P301: Predicting Complete Resolution Of Hypertension After Adrenalectomy In Primary Aldosteronism: A Systematic Review And Meta-analysis Of Prognostic Models. Surgical Outcome of PRimary Aldosteronism progNostic mOdels (SOPRANO) Study

Background: Unilateral primary aldosteronism (PA) is the most common surgically curable form of hypertension, despite complete clinical success after adrenalectomy being far from certain. Several presurgical factors were associated with complete resolution of hypertension (CRH). However, it remains unclear which of the available prognostic models combining those factors can be used in clinical practice to predict blood pressure-related outcomes after surgery for PA. Objectives: To identify, describe and appraise all prognostic models developed to predict CRH, and meta-analyse their predictive performances. Methods: We searched MEDLINE, Embase, and Web of Science, for development and validation studies of prognostic models according to the Preferred Reporting Items for Systematic Review and Meta-analysis (PRISMA) statement. The quality assessment used adapted TRIPOD (Transparent Reporting of a multivariable prediction model for Individual Prognosis Or Diagnosis) and PROBAST (Prediction model Risk Of Bias ASsessment Tool) criteria. After selection, we extracted descriptive statistics and aggregated area under the receiver operator curve (AUC) using meta-analysis. Results: From 25 eligible studies, we identified 12 prognostic models used for predicting CRH after total adrenalectomy in PA. We report the results for three models that had available data from at least three external validation studies: the Primary Aldosteronism Surgical Outcome (PASO) score (AUC: 0.81; 95% confidence interval [CI]: 0.74–0.86; 95% predictive interval [PI]: 0.04–1.00), Utsumi nomogram (AUC: 0.79; 95% CI: 0.72–0.85; 95% PI: 0.03–1.00), and the Aldosteronoma Resolution Score (ARS) model (AUC: 0.77; 95% CI: 0.74–0.80; 95% PI: 0.59–0.86 for all studies and AUC: 0.80; 95% CI: 0.75–0.85; 95% PI: 0.57–0.93 for the studies with the same adrenal vein sampling-guided adrenalectomy rate compared to the models meta-analyzed). Conclusions: The PASO score, Utsumi nomogram, and ARS model showed good and comparable discrimination performance to predict CRH after unilateral adrenalectomy in PA. However, unlike the ARS model, the number of external validation studies for the PASO score and the Utsumi nomogram was relatively low to draw definite conclusions.

Predictors of successful outcome after adrenalectomy for unilateral primary aldosteronism.

Unilateral primary aldosteronism (UPA) is the most frequent surgically curable form of endocrine hypertension. Adrenalectomy is the cornerstone of treatment for UPA, but outcomes after surgery are variable. The cause of resistant hypertension after surgery is still a matter of debate. Our aim was to investigate cure rates after surgery and to evaluate preoperative factors that might influence the surgical outcome. Between 2000 and 2021, the charts of 71 Tunisian patients who underwent laparoscopic adrenalectomy for UPA were retrospectively reviewed. Preoperative medical records were collected and follow-up data (1-158 months) were registered. Antihypertensive medication doses were calculated using defined daily doses (DDD) and postoperative outcomes were assessed using the Primary Aldosteronism Surgical Outcome (PASO) criterion. Of 91 enrolled patients, 71 (59% women, mean age 46 years, median length of follow-up 21 months) were suitable for evaluation. Thirty-four patients (48%) had complete clinical success according to the PASO criteria. The most relevant factors associated with complete clinical success on univariate analysis were: absence of diabetes (p= 0.007), low body mass index (BMI) (p= 0.001), lower preoperative DDD (p= 0.01), preoperatively controlled blood pressure (p= 0.024), higher plasma aldosterone to renin ratio (ARR) (p= 0.001), adenoma subtyping (p <0.001) and aldosteronoma resolution score (ARS) (p= 0.002). Multivariate regression analysis showed that the major predictors of complete clinical success were absence of diabetes (OR: 5.205), a BMI < 30 (OR: 4.930), a plasma ARR > 332 (OR: 4.554) and an ARS ≥ 3 (OR: 2.056). Complete and partial clinical response rates were achieved in respectively 48 and 43% of cases. The main predictors of complete resolution of hypertension were absence of diabetes, low BMI, high plasma ARR and high ARS. Taking these factors into account may help identify patients at risk of persistent postoperative hypertension who may require long-term surveillance and medication.

Association of urinary liver-type fatty acid–binding protein with outcomes after adrenalectomy for unilateral primary aldosteronism

Background and AimsThis study explored the association between urinary liver-type fatty acid-binding protein to creatinine (uL-FABP-cre) ratio and postoperative clinical failure in unilateral primary aldosteronism (PA) patients undergoing adrenalectomy. Materials and MethodsData from the Taiwan Primary Aldosteronism Investigation Group database were analyzed, including patients with unilateral PA who had adrenalectomy between December 2015 and October 2018. Statistical methods included generalized additive modeling, logistic regression analysis, net reclassification improvement (NRI), and the C statistic. ResultsIn the study cohort of 131 patients (mean age 52.3 ± 10.8 years; 43.5% male), 117 achieved clinical success, while 14 experienced clinical failure. A uL-FABP-cre ratio ≥5 predicted clinical failure (odds ratio: 6.22, p = 0.005). Subgroup analysis revealed its efficacy in predicting clinical failure in patients with BMI ≥ 24 kg/m2, normokalemia, or <5 years of hypertension.Furthermore, incorporating uL-FABP-cre ratio into the Primary Aldosteronism Surgical Outcome (PASO) score significantly improved predictive ability. The addition increased the C statistic from 0.671 to 0.762 (p < 0.01) and improved category-free NRI by 0.675 (p = 0.014). ConclusionA uL-FABP-cre ratio ≥5 accurately predicted clinical failure post-adrenalectomy in unilateral PA, enhancing PASO score's identification of high-risk patients for postoperative clinical failure.

Prognostic models to predict complete resolution of hypertension after adrenalectomy in primary aldosteronism: A systematic review and meta-analysis.

Complete resolution of hypertension (CRH) after adrenalectomy for primary aldosteronism is far from a certainty. Although several prognostic models have been proposed to predict outcome after adrenalectomy, studies have not clarified which of the available models can be used reliably in clinical practice.To identify, describe and appraise all prognostic models developed to predict CRH, and meta-analyse their predictive performances.We searched MEDLINE, Embaseand Web of Science for development and validation studies of prognostic models. After selection, we extracted descriptive statistics and aggregated area under the receiver operator curve (AUC) using meta-analysis.From 25 eligible studies, we identified 12 prognostic models used for predicting CRH after total adrenalectomy in primary aldosteronism. We report the results for 3models that had available data from at least 3external validation studies: the primary aldosteronism surgical outcome (PASO) score (AUC: 0.81; 95% confidence interval [CI]: 0.74-0.86; 95% predictive interval [PI]: 0.04-1.00), Utsumi nomogram (AUC: 0.79; 95% CI: 0.72-0.85; 95% PI: 0.03-1.00)and the aldosteronoma resolution score (ARS) model (AUC: 0.77; 95% CI: 0.74-0.80; 95% PI: 0.59-0.86 for all studies and AUC: 0.80; 95% CI: 0.75-0.85; 95% PI: 0.57-0.93 for the studies with the same adrenal vein sampling-guided adrenalectomy rate compared to the models meta-analysed).The PASO score, Utsumi nomogramand ARS model showed comparable discrimination performance to predict CRH in primary aldosteronism. Unlike the ARS model, the number of external validation studies for the PASO score and the Utsumi nomogram was relatively low to draw definite conclusions.

The Adrenal Vein Sampling Outcomes Study (AVOS): success rates following adrenalectomy for unilateral primary aldosteronism

The objective was to determine the clinical and biochemical success rates and assess the nature of follow-up after adrenalectomy in patients with unilateral primary aldosteronism (PA), subtyped by adrenal vein sampling (AVS) in West Australia (WA) using the Primary Aldosteronism Surgical Outcome (PASO) criteria. Clinical and biochemical outcomes were retrospectively evaluated in patients with unilateral PA who underwent adrenalectomy according to AVS between September 2017 and September 2020. Pre- and post-surgical data were collected using a standardised questionnaire, review of clinic letters and examination of private and public pathology results and radiological reports. Follow-up data were available for 47 patients post-adrenalectomy; biochemical outcome data were available for 37 patients, clinical outcome data for 40 patients, with 30 patients having both outcomes available. Final assessment was performed between 0 to 3 months in 23/37 (62.2%) patients with biochemical outcomes, 15/40 (37.5%) with clinical outcomes, and 17/30 (56.7%) with both clinical and biochemical outcomes. Complete biochemical success was achieved in 83.8% (31/37) of patients, with 26.7% (8/30) obtaining both complete clinical and biochemical success. Complete clinical success was achieved in 35.0% (14/40) of patients, with 47.5% (19/40) obtaining partial clinical success. Overall, 93.6% (44/47) of patients derived benefit from adrenalectomy. The outcomes of adrenalectomy for unilateral PA in Western Australian using standardised PASO criteria demonstrate highly comparable clinical and biochemical success rates to international data. However, further standardisation of post-operative follow-up care needs to be implemented to ensure the recommended repeat follow-up assessment criteria are collected.

PS-BPR01-7: CONCURRENT SOMATIC MUTATIONS IN CTNNB1 AND GNA11 IN AN ALDOSTERONE-PRODUCING ADENOMA

Background: Aldosterone-producing adenoma (APA) is a major subtype of primary aldosteronism (PA), a common form of secondary hypertension. In the past decade, there has been significant progress in the determination of genetic causes of APA. The use of next-generation sequencing (NGS) has led to the identification of somatic mutations in APA that cause excess aldosterone production. Most of the affected genes encode ion channels or transporters. Somatic activating mutations in CTNNB1 (β;-catenin) have also been detected in 2–5% of APA. As a possible pathogenesis, an association of CTNNB1-mutated APA with pregnancy and menopause has been proposed. A recent study reported double somatic mutations in CTNNB1 and GNA11/Q in APA. However, due to its rare incidence, characteristics of APA with these mutations are not well determined. Herein, we report a case of APA harboring somatic CTNNB1 and GNA11 mutations. Case presentation: A 46-year-old Japanese woman presented with hypertension and spontaneous hypokalemia. She had no history of pregnancy-induced hypertension. She had regular menstrual cycle at presentation. Endocrine testing showed elevated plasma aldosterone concentration (677.5 pg/mL) under suppressed renin (plasma renin activity, 0.3 ng/mL/h). Computed tomography revealed a 2 cm right adrenal mass. Adrenal venous sampling indicated excess aldosterone production from the right adrenal gland. NP-59 scintigraphy further demonstrated increased tracer uptake in the right adrenal lesion. She underwent right laparoscopic adrenalectomy. Histologic diagnosis of the resected tumor was adrenocortical adenoma based on the Weiss criteria. Notably, Ki-67 labeling index was high (6% at hotspots). After surgery, her blood pressure and serum potassium both normalized. Based on the PASO (the primary aldosteronism surgical outcome) study criteria, PA remained clinically and biochemically cured at follow-up 18 months after surgery. Methods: Formalin-fixed, paraffin-embedded adrenal tumor tissue was used for immunohistochemical and genetic analysis. Results: Immunohistochemistry (IHC) showed high and diffuse expression of CYP11B2 (aldosterone synthase) within the tumor, whereas 11β;-hydroxylase (CYP11B1, a steroidogenic enzyme for cortisol biosynthesis) was mostly negative. VSNL1, a marker for the zona glomerulosa where physiologic aldosterone biosynthesis occurs, was highly expressed within the tumor. CYP11B2 IHC-guided targeted NGS identified concurrent somatic CTNNB1 (p.D32Y) and GNA11 (p.Q209H) mutations with similar variant allele frequencies (29.5% and 29.7%, respectively). Conclusions: To best of our knowledge, this is the first Asian case of APA with CTNNB1 and GNA11 mutations. Detailed clinical and histologic examination would provide useful information for better characterization of patients with PA due to these rare mutations.

PS-BPC08-3: CATHETER-BASED ADRENAL ABLATION: AN ALTERNATIVE THERAPY FOR PATIENTS WITH ALDOSTERONE-PRODUCING ADENOMA

Objective: Unilateral adrenalectomy is the standard treatment for patients with aldosterone-producing adenoma (APA), but it lacks an option for patients with APA who refuse or are not suitable for surgery. In this study, we studied whether catheter-based adrenal ablation for APA is comparable to adrenalectomy. Design and method: A total of 1180 hypertensive patients were screened, and 112 patients with APA were recruited and counseled on the treatment options. Fifty-two patients opted for catheter-based adrenal ablation, and 60 opted for adrenalectomy. Clinical and biochemical outcomes were assessed at 6 months after treatment. Factors associated with hypertension remission, advantages and limitations of this approach were evaluated. Results: According to the primary aldosteronism surgical outcome (PASO) criteria, complete and partial clinical success was achieved in 21 (40.4%) and 23 (44.2%)patients in the ablation group vs. 33 (55.0%) and 23 (38.3%) patients in the adrenalectomy group. Complete and partial biochemical success was achieved in 30 (57.7%) and 17 (32.7%) patients in the ablation group vs. 51(85.0%)and 5 (8.3%) patients in the adrenalectomy group, respectively. The overall clinical (84.6% vs 93.3%) and biochemical (90.4% vs 93.3%) success rates were not significantly different between the two groups (P &gt; 0.05). Factors associated with adrenal ablation-mediated hypertension remission were hypertension duration and serum potassium level at baseline. Compared with surgery, adrenal ablation requires a shorter operating time and time to resume physical activity. Conclusions: Catheter-based adrenal ablation may bean alternative and feasible option for APA patients unwilling surgical treatment.

Assessment of biochemical outcomes in patients with primary aldosteronism after adrenalectomy based on CT scan diagnosis of unilateral adenoma without adrenal vein sampling.

The purpose of this study was to assess the surgical outcomes of patients with primary aldosteronism when surgery was based only on CT finding of unilateral adenoma without adrenal vein sampling (AVS). This is a retrospective review of the records of patients who had undergone retroperitoneal laparoscopic adrenalectomy for primary aldosteronism based on CT scan finding of unilateral adenoma and had a follow-up of at least 6-12 months from January 2012 to December 2020 in a single center; decision for adrenalectomy was based on CT scan, and AVS was not used. The clinical and biochemical outcomes were accessed using the standardized primary aldosteronism surgical outcome (PASO) criteria. Patient's demographics and preoperative factors were analyzed to assess for independent predictor of surgical success. According to the PASO criteria, 172 patients finally enrolled in the training dataset, and 20 patients enrolled in the validation dataset. In the training dataset, complete clinical success was achieved in 71 patients (41.3%), partial success in 87 (50.6%), and absent success in 14 (8.1%). Biochemical outcomes showed that 151 patients (87.8%) were completely cured, 14 patients (8.1%) got a partial biochemical success, and an absent biochemical success was found in seven patients (4.1%). Multivariate logistic regression analysis showed that age, body mass index (BMI), tumor size, mean arterial pressure (MAP), and serum potassium were the most independent factors for incomplete biochemical success. Based on the results of statistical analysis, our study constructed a nomogram prognostic evaluation model for patients after unilateral primary aldosterone surgery. Laparoscopic adrenalectomy for patients with primary aldosteronism base on CT scan finding of a unilateral adenoma without AVS had a high rate of complete biochemical cure at 12 months. Risk factors for incomplete biochemical success include age, BMI, tumor size, MAP, and serum potassium. Our study constructed a nomogram prognostic evaluation model for patients after unilateral primary aldosterone surgery. The nomogram accurately and reliably predicted the incomplete biochemical success.

ODP611 Combination Of Seated Saline Suppression Test And Computed Tomography In Predicting Subtype Diagnosis Of Primary Aldosteronism In Chinese Population

Abstract Objective The study aimed to evaluate the accuracy of the seated saline suppression test (SSST) combined with adrenal computed tomography (CT) in determining the primary aldosteronism (PA) subtypes. Methods Each patient underwent a SSST and adrenal CT. Logistic diagnostic model (CT finding + post-SSST plasma aldosterone concentration (PAC)) were built, which evaluated the accuracy of the SSST combined with adrenal CT for a subtype classification of PA. Subtype diagnosis of PA was determined by adrenal venous sampling (AVS), pathology and clinical outcomes (primary aldosteronism surgical outcome(PASO)). Results A total of 72 patients with PA were classified into two groups, including 45 aldosterone-producing adenoma (APA) and 27 idiopathic hyperaldosteronism (IHA) patients. By binary logistic regression analysis, adrenal CT (odds ratio (OR)=16.28, 95% confidence interval (CI) 4.18-63.42, P&amp;lt;0. 05) and 4-hours PAC after SSST (OR =1.13, 95% CI 1. 03 to 1.23, P&amp;lt;0. 05) were significantly independent predictors of APA patients. We have successfully developed a diagnostic model based on logistic regression that is able to predict the probability of APA. The area under the receiver operating characteristic (ROC) curve of logistic diagnostic models(AUC=0.907, 95% CI 0.815 to 0.963) was significantly greater either than that for the adrenal CT (AUC=0.807,95% CI 0.696 to 0.890,respectively,P&amp;lt;0. 05) or SSST (AUC=0.799,95% CI 0.688 to 0.884,respectively,P&amp;lt;0. 05). Positive CT finding combined with a post-SSST PAC≥18.2 ng/dL provided sensitivity of 88.89%,specificity of 74. 07%,and positive predictive value (PPV) of 93.9%(31/33) for predicting APA, that determined patients who required AVS prior to surgery. Conclusion The model combining CT finding with post-SSST PAC has superior accuracy in subtype diagnosis of PA compared with SSST or adrenal CT alone. The current study was the first study to investigate the accuracy of the new method on the subtype classification of PA. We are fortunate to find it may be a safe, convenient, sensitive and noninvasive approach in predicting subtype diagnosis of PA in Chinese individuals, especially in primary care where AVS may be unavailable. The results require further confirmation by RCT clinical trials. Presentation: Saturday, June 11, 2022 1:00 p.m. - 3:00 p.m.

Catheter-based adrenal ablation: an alternative therapy for patients with aldosterone-producing adenoma.

Unilateral adrenalectomy is the standard treatment for patients with aldosterone-producing adenoma (APA), but it lacks an option for patients with APA who refuse or are not suitable for surgery. In this study, we studied whether catheter-based adrenal ablation for APA is comparable to adrenalectomy. A total of 2185 hypertensive patients were screened, and 112 patients with APA were recruited and counselled on the treatment options. Fifty-two patients opted for catheter-based adrenal ablation, and 60 opted for adrenalectomy. Clinical and biochemical outcomes were assessed at 6 months after treatment. Factors associated with hypertension remission and the advantages and limitations of this approach were evaluated. According to the primary aldosteronism surgical outcome (PASO) criteria, complete and partial clinical success was achieved in 21 (40.4%) and 23 (44.2%) patients in the ablation group vs. 33 (55.0%) and 23 (38.3%) patients in the adrenalectomy group, respectively. Complete and partial biochemical success was achieved in 30 (57.7%) and 17 (32.7%) patients in the ablation group vs. 51 (85.0%) and 5 (8.3%) patients in the adrenalectomy group, respectively. The complete clinical success rate was not (P > 0.05), but the complete biochemical success rate was significantly different between the two groups (P < 0.01). Factors associated with adrenal ablation-mediated hypertension remission were hypertension duration and serum potassium level at baseline. Compared with surgery, adrenal ablation requires a shorter operating time and time to resume physical activity. Catheter-based adrenal ablation may be an alternative and feasible option for APA patients unwilling to receive surgical treatment.