Displaying publications 1 - 20 of 21 in total

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  1. Azizan EA, Brown MJ
    Curr Opin Endocrinol Diabetes Obes, 2016 06;23(3):209-17.
    PMID: 26992195 DOI: 10.1097/MED.0000000000000255
    PURPOSE OF REVIEW: Aldosterone regulation in the adrenal plays an important role in blood pressure. The commonest curable cause of hypertension is primary aldosteronism. Recently, mutations in novel genes have been identified to cause primary aldosteronism. Elucidating the mechanism of action of these genetic abnormalities may help understand the cause of primary aldosteronism and the physiological regulation of aldosterone in the zona glomerulosa.

    RECENT FINDINGS: KCNJ5, ATP1A1, ATP2B3, CACNA1D, CTNNB1, and CACNA1H mutations are causal of primary aldosteronism. ARMC5 may cause bilateral lesions resulting in primary aldosteronism.LGR5, DACH1, and neuron-specific proteins are highly expressed in the zona glomerulosa and regulate aldosterone production.

    SUMMARY: Most mutations causing primary aldosteronism are in genes encoding cation channels or pumps, leading to increased calcium influx. Genotype-phenotype analyses identified two broad subtypes of aldosterone-producing adenomas (APAs), zona fasciculata-like and zona glomerulosa-like, and the likelihood of under-diagnosed zona glomerulosa-like APAs because of small size. Zona fasciculata-like APAs are only associated with KCNJ5 mutations, whereas zona glomerulosa-like APAs are associated with mutations in ATPase pumps, CACNA1D, and CTNNB1. The frequency of APAs, and the multiplicity of causal mutations, suggests a pre-existing drive for these mutations. We speculate that these mutations are selected for protecting against tonic inhibition of aldosterone in human zona glomerulosa, which express genes inhibiting aldosterone production.

    Matched MeSH terms: Hyperaldosteronism/genetics*; Hyperaldosteronism/metabolism*
  2. Naruse M, Murakami M, Katabami T, Kocjan T, Parasiliti-Caprino M, Quinkler M, et al.
    Eur J Endocrinol, 2023 Jan 10;188(1).
    PMID: 36726325 DOI: 10.1093/ejendo/lvac002
    OBJECTIVE: Primary aldosteronism (PA) is one of the most frequent causes of secondary hypertension. Although clinical practice guidelines recommend a diagnostic process, details of the steps remain incompletely standardized.

    DESIGN: In the present SCOT-PA survey, we have investigated the diversity of approaches utilized for each diagnostic step in different expert centers through a survey using Google questionnaires. A total of 33 centers from 3 continents participated.

    RESULTS: We demonstrated a prominent diversity in the conditions of blood sampling, assay methods for aldosterone and renin, and the methods and diagnostic cutoff for screening and confirmatory tests. The most standard measures were modification of antihypertensive medication and sitting posture for blood sampling, measurement of plasma aldosterone concentration (PAC) and active renin concentration by chemiluminescence enzyme immunoassay, a combination of aldosterone-to-renin ratio with PAC as an index for screening, and saline infusion test in a seated position for confirmatory testing. The cutoff values for screening and confirmatory testing showed significant variation among centers.

    CONCLUSIONS: Diversity of the diagnostic steps may lead to an inconsistent diagnosis of PA among centers and limit comparison of evidence for PA between different centers. We expect the impact of this diversity to be most prominent in patients with mild PA. The survey raises 2 issues: the need for standardization of the diagnostic process and revisiting the concept of mild PA. Further standardization of the diagnostic process/criteria will improve the quality of evidence and management of patients with PA.

    Matched MeSH terms: Hyperaldosteronism*
  3. Sukor N
    Endocrine, 2012 Feb;41(1):31-9.
    PMID: 22042487 DOI: 10.1007/s12020-011-9553-3
    Primary aldosteronism is now thought to be the commonest potentially curable and specifically treatable form of hypertension. The detection of primary aldosteronism is of utmost importance not only because it provides an opportunity for a targeted treatment, but also because it has been demonstrated that patients with primary aldosteronism are more prone to cardiovascular events and target organ damage than essential hypertensives. Normalization of blood pressure and hypokalemia should not be the only goal of treatment. Normalization of circulating aldosterone or mineralocorticoid blockade is necessary to prevent aldosterone-induced tissue damage that occurs independent of blood pressure. This review will focus on the current understanding and comprehensive management review of primary aldosteronism, highlighting the new evidence that has become recently available.
    Matched MeSH terms: Hyperaldosteronism/complications; Hyperaldosteronism/diagnosis*; Hyperaldosteronism/therapy*
  4. Zhou J, Lam B, Neogi S, Yeo G, Azizan E, Brown M
    J Hypertens, 2016 Sep;34 Suppl 1 - ISH 2016 Abstract Book:e40.
    PMID: 27753883
    Primary aldosteronism (PA) is the most common type of secondary hypertension occurring in ∼10% of hypertensive patients. Up to 50% of PA is caused by aldosterone-producing adenomas (APA). This study is to identify the potential biological processes and canonical pathways involved with aldosterone regulation, APA formation, or APA and ZG cell functions.
    Matched MeSH terms: Hyperaldosteronism
  5. Wu X, Azizan EAB, Goodchild E, Garg S, Hagiyama M, Cabrera CP, et al.
    Nat Genet, 2023 Jun;55(6):1009-1021.
    PMID: 37291193 DOI: 10.1038/s41588-023-01403-0
    Aldosterone-producing adenomas (APAs) are the commonest curable cause of hypertension. Most have gain-of-function somatic mutations of ion channels or transporters. Herein we report the discovery, replication and phenotype of mutations in the neuronal cell adhesion gene CADM1. Independent whole exome sequencing of 40 and 81 APAs found intramembranous p.Val380Asp or p.Gly379Asp variants in two patients whose hypertension and periodic primary aldosteronism were cured by adrenalectomy. Replication identified two more APAs with each variant (total, n = 6). The most upregulated gene (10- to 25-fold) in human adrenocortical H295R cells transduced with the mutations (compared to wildtype) was CYP11B2 (aldosterone synthase), and biological rhythms were the most differentially expressed process. CADM1 knockdown or mutation inhibited gap junction (GJ)-permeable dye transfer. GJ blockade by Gap27 increased CYP11B2 similarly to CADM1 mutation. Human adrenal zona glomerulosa (ZG) expression of GJA1 (the main GJ protein) was patchy, and annular GJs (sequelae of GJ communication) were less prominent in CYP11B2-positive micronodules than adjacent ZG. Somatic mutations of CADM1 cause reversible hypertension and reveal a role for GJ communication in suppressing physiological aldosterone production.
    Matched MeSH terms: Hyperaldosteronism*
  6. Meah FA, Abdullah T, Jasmi AY, Hisham AN, Tan TT, Khalid BA, et al.
    Ann Acad Med Singap, 1996 Mar;25(2):251-4.
    PMID: 8799016
    Between January 1978 to December 1993, 130 cases of adrenal diseases were diagnosed and surgically treated at the National University of Malaysia. They were 58 cases (44.6%) of Conn's syndrome, 40 cases (30.7%) of Cushing's syndrome, 20 cases (15.3%) of phaeochromocytoma and 12 cases (9.2%) of adrenocortical carcinoma (ACC). The commonest cause of Conn's syndrome was an adenoma (96.5%) which affected the left gland four times more than the right gland. Cushing's syndrome was caused by adrenocortical adenoma (32.5%), diffuse bilateral adrenal hyperplasia (40.0%), pigmented macronodular hyperplasia (20.0%) and adrenal carcinoma (7.5%). Twenty-five percent of the phaeochromocytomas were extraadrenal in origin arising mainly from the abdominal sympathetic chain. More than 50% of ACCs were non-functioning tumours. Fifty percent of the patients with ACC had inoperable tumours. The prognosis was poor even with adjuvant chemoradiotherapy. The main surgical approach was the anterior transabdominal route. There was no operative mortality or morbidity in all operated cases.
    Matched MeSH terms: Hyperaldosteronism/diagnosis; Hyperaldosteronism/epidemiology; Hyperaldosteronism/surgery*
  7. Saladina, J.J., Rohaizak, M., Jasmi, A.Y., Sellymiah, A., Aishah, M.A.S., Das, S., et al.
    JUMMEC, 2011;14(1):23-25.
    MyJurnal
    Presence of a hypofunctioning pigmented adenoma are commonly asymptomatic and is usually only found during an autopsy. In contrast, hyperfunctioning pigmented adenoma is a rare clinical entity and in the majority of cases results in Cushing's syndrome. In this case study, we report a 66-year-old male who presented instead with the clinical and biochemical features of Conn's syndrome. On laparoscopic adrenalectomy, it was found that the tumour had a functioning black adenoma which does not usually present with Conn's syndrome but rather to that of a Cushing's. The intraoperative changes and histopathological findings are discussed.
    Matched MeSH terms: Hyperaldosteronism
  8. Loh HH, Kamaruddin NA, Zakaria R, Sukor N
    Minerva Endocrinol., 2018 Jun;43(2):117-125.
    PMID: 28001017 DOI: 10.23736/S0391-1977.16.02553-0
    BACKGROUND: Recent studies showed association between hyperaldosteronism and low bone density among patients with primary aldosteronism (PA) due to secondary hyperparathyroidism. Our objective is to assess bone turnover markers (BTM) and bone mineral density (BMD) of PA patients compared to essential hypertension.

    METHODS: This was an open-label, prospective, case-controlled study, conducted over 12 months. Fifty-two consecutive patients referred for secondary hypertension were screened. Eighteen patients with confirmed PA (diagnosis based on the Endocrine Society clinical guideline) and seventeen matched controls with essential hypertension were recruited. BTM (CTX and P1NP), BMD, intact parathyroid hormone (iPTH), and bone profile were assessed at baseline and three months following treatment among the PA patients. Calcium intake was assessed using a validated questionnaire. Primary outcomes were the changes of bone markers and BMD following treatment of PA, and their relation to other parameters.

    RESULTS: PA patients had significantly lower serum calcium and higher iPTH despite comparable vitamin D levels with control group. Both BTM were significantly higher among the PA group. BMD of lumbar spine, neck of femur and distal radius did not differ between groups. Three months following treatment, there were significant: 1) reduction in BTM; 2) improvement in the lumbar spine BMD; 3) reduction in iPTH level; and 4) increment of serum 25-OH vitamin D level.

    CONCLUSIONS: Our findings support that bone loss and potential fracture risk among PA patients are likely a result of aldosterone-mediated secondary hyperparathyroidism. Patients with early PA may already exhibit increased bone turnover despite no significant changes in BMD.

    Matched MeSH terms: Hyperaldosteronism/blood*; Hyperaldosteronism/drug therapy*
  9. Loh KC, Koay ES, Khaw MC, Emmanuel SC, Young WF
    J Clin Endocrinol Metab, 2000 Aug;85(8):2854-9.
    PMID: 10946893
    Recent studies using the ratio of plasma aldosterone concentration (PAC) to PRA as the screening test for primary aldosteronism in hypertensive populations suggested that the prevalence may be as high as 5-15%, with well over half of the subjects having normal serum potassium concentrations. Despite an increasing clinical awareness of this entity, many clinicians are reluctant to consider routine screening for primary aldosteronism in essential hypertensive patients because there are few community-based prevalence studies of primary aldosteronism in different populations. Furthermore, genetic and environmental differences may affect the prevalence and presentation of primary aldosteronism in distinct populations. This study was designed to determine the prevalence of primary aldosteronism in the predominantly Chinese population in Singapore. Three hundred and fifty unselected adult hypertensive patients attending two primary care clinics had random ambulatory measurements for PAC (nanograms per dL) and PRA (nanograms per mL/h). Serum urea, creatinine, and electrolyte measurements were obtained simultaneously. Subjects with renal insufficiency (serum creatinine, >140 micromol/L) and those treated with glucocorticoids or spironolactone were excluded. Screening was considered positive if the PAC: PRA ratio was more than 20 and the PAC was more than 15 ng/dL (>416 pmol/L). Primary aldosteronism was confirmed with the determination of PAC after 2 L saline administered iv over 4 h. Adrenal computed tomographic (CT) scans were performed in biochemically confirmed cases of primary aldosteronism. Further localization with adrenal vein sampling was carried out in selected patients with equivocal findings on adrenal CT scan. Sixty-three (18%) of the 350 hypertensive patients (215 women and 135 men; age range, 23-75 yr) were screened positive for primary aldosteronism. Only 13 of these 63 subjects (21%) were hypokalemic (serum potassium, <3.5 mmol/L). Confirmatory studies were carried out in 56 (89%) of the subjects with a positive PAC:PRA ratio. Using a PAC above 10 ng/dL (>277 pmol/L) after saline infusion as the diagnostic cut-off, 16 of the 56 patients had biochemically confirmed primary aldosteronism. Hypokalemia was found in 6 of the 16 patients (37.5%) with primary aldosteronism. Subtype evaluation with adrenal CT scan and adrenal vein sampling indicated that half of the patients with primary aldosteronism may have had potentially curable unilateral adrenal adenoma. Our data suggest that primary aldosteronism occurs in at least 5% of the adult Asian hypertensive population, and approximately half of these individuals may have potentially curable, unilateral, aldosterone-producing adrenal adenoma. Our findings also confirm the poor predictive value of hypokalemia in both the diagnosis and the exclusion of primary aldosteronism.
    Matched MeSH terms: Hyperaldosteronism/complications*; Hyperaldosteronism/diagnosis; Hyperaldosteronism/epidemiology*
  10. Zhou J, Lam BY, Neogi SG, Yeo GS, Teo AE, Maniero C, et al.
    J Hypertens, 2016 Sep;34 Suppl 2:e26.
    PMID: 27508643 DOI: 10.1097/01.hjh.0000491398.48468.bf
    Primary aldosteronism (PA) is the most common type of secondary hypertension occurring in ∼10% of hypertensive patients. Up to 50% of PA is caused by aldosterone-producing adenomas (APA). We recently performed a microarray assay using 21 pairs of zona glomerulosa (ZG) and zona fasciculata (ZF), and 14 paired APAs. This study is to identify the potential biological processes and canonical pathways involved with aldosterone regulation, APA formation, or APA and ZG cell functions.
    Matched MeSH terms: Hyperaldosteronism
  11. Loh HH, Yee A, Loh HS
    Minerva Endocrinol., 2019 Dec;44(4):387-396.
    PMID: 30482008 DOI: 10.23736/S0391-1977.18.02867-5
    INTRODUCTION: Recent studies showed a possible association between hyperaldosteronism and secondary hyperparathyroidism leading to reduced bone health, however results are conflicting.

    EVIDENCE ACQUISITION: We conducted a meta-analysis to evaluate the relationship between primary aldosteronism (PA) with bone biochemical markers and to assess bone mineral density in patients with primary aldosteronism.

    EVIDENCE SYNTHESIS: A total of 939 subjects were examined (37.5% with PA). Patients with PA had significantly higher serum parathyroid hormone, lower serum calcium, higher urine calcium excretion and higher serum alkaline phosphatase compared to patients without PA, with no significant difference in serum vitamin D between both groups. Bone mineral density of lumbar spine, femoral neck and total neck of femur were similar between two groups. With PA treatment, there was a significant increment in serum calcium and reduction in serum parathyroid hormone.

    CONCLUSIONS: PA is associated with hypercalciuria with subsequent secondary hyperparathyroidism. This potentially affects bone health. We recommend this to be part of complication screening among patients with PA.

    Matched MeSH terms: Hyperaldosteronism/blood; Hyperaldosteronism/complications*; Hyperaldosteronism/drug therapy
  12. Kang W, Singarayar C, Wahab NA, Sukor N, Kamaruddin NA
    J ASEAN Fed Endocr Soc, 2019;34(1):95-98.
    PMID: 33442143 DOI: 10.15605/jafes.034.01.15
    An adrenal mass can be a diagnostic challenge as it is not easy to differentiate the adrenal glands from other adrenal pseudotumours with only radio-imaging. We report a 28-year-old patient who was diagnosed radiologically as an adrenal cortical carcinoma after he presented with abdominal pain and fullness. Biochemically, he demonstrated secondary hyperaldosteronism. Intra-operatively there was a huge mass, inferior to a normal right adrenal, which was histopathologically proven to be a dedifferentiated liposarcoma.
    Matched MeSH terms: Hyperaldosteronism
  13. Sukor N
    Postgrad Med J, 2011 Oct;87(1032):706-13.
    PMID: 21746730 DOI: 10.1136/pgmj.2011.118661
    Hypertension is a chronic disorder which often entails debilitating cardiovascular and renal complications. Hypertension mostly arises as a complex quantitative trait that is affected by varying combinations of genetic and environmental factors. Secondary hypertension has been encountered with increasing frequency. The common causes of secondary hypertension include renal parenchymal disease, renal artery stenosis, primary aldosteronism, phaeochromocytoma, and Cushing's syndrome. The detection of a secondary cause is of the utmost importance because it provides an opportunity to convert an incurable disease into a potentially curable one. Early identification and treatment will provide a better opportunity for cure, prevent target organ damage, reduce socioeconomic burden and health expenditure associated with drug costs, and improve patients' quality of life. Hence, it is a condition not to be missed.
    Matched MeSH terms: Hyperaldosteronism/complications*
  14. Hamidon BB, Jeyabalan V
    Singapore Med J, 2006 Feb;47(2):156-8.
    PMID: 16435060
    A 31-year-old woman presented with a one-week history of headache, generalised lethargy, weakness and poor appetite. Clinical examination showed that her blood pressure was 200/120 mmHg. On an earlier occasion, her blood pressure was found to be normal by a general practitioner whom she last visited three months earlier when she had an upper respiratory tract infection. Investigations showed hypokalaemia, suppressed serum renin and aldosterone. Further history was taken and revealed that she had been craving for guava fruits which she ate with flavoured "asam boi" (containing glycyrrhizic acid) at least three spoonfuls twice a day for the past six weeks. The hypertension and hypokalaemia resolved after two weeks of stopping the "asam boi". Her clinical picture was compatible with exogenously-induced hypermineralocortoidism.
    Matched MeSH terms: Hyperaldosteronism/etiology*
  15. Zhou J, Lam B, Neogi SG, Yeo GS, Azizan EA, Brown MJ
    Hypertension, 2016 12;68(6):1424-1431.
    PMID: 27777363
    Primary aldosteronism is present in ≈10% of hypertensives. We previously performed a microarray assay on aldosterone-producing adenomas and their paired zona glomerulosa and fasciculata. Confirmation of top genes validated the study design and functional experiments of zona glomerulosa selective genes established the role of the encoded proteins in aldosterone regulation. In this study, we further analyzed our microarray data using AmiGO 2 for gene ontology enrichment and Ingenuity Pathway Analysis to identify potential biological processes and canonical pathways involved in pathological and physiological aldosterone regulation. Genes differentially regulated in aldosterone-producing adenoma and zona glomerulosa were associated with steroid metabolic processes gene ontology terms. Terms related to the Wnt signaling pathway were enriched in zona glomerulosa only. Ingenuity Pathway Analysis showed "NRF2-mediated oxidative stress response pathway" and "LPS (lipopolysaccharide)/IL-1 (interleukin-1)-mediated inhibition of RXR (retinoid X receptor) function" were affected in both aldosterone-producing adenoma and zona glomerulosa with associated genes having up to 21- and 8-fold differences, respectively. Comparing KCNJ5-mutant aldosterone-producing adenoma, zona glomerulosa, and zona fasciculata samples with wild-type samples, 138, 56, and 59 genes were differentially expressed, respectively (fold-change >2; P<0.05). ACSS3, encoding the enzyme that synthesizes acetyl-CoA, was the top gene upregulated in KCNJ5-mutant aldosterone-producing adenoma compared with wild-type. NEFM, a gene highly upregulated in zona glomerulosa, was upregulated in KCNJ5 wild-type aldosterone-producing adenomas. NR4A2, the transcription factor for aldosterone synthase, was highly expressed in zona fasciculata adjacent to a KCNJ5-mutant aldosterone-producing adenoma. Further interrogation of these genes and pathways could potentially provide further insights into the pathology of primary aldosteronism.
    Matched MeSH terms: Hyperaldosteronism/genetics*; Hyperaldosteronism/physiopathology
  16. Chand R, Tandukar S, Asmil S, Chico M
    Cureus, 2020 Aug 07;12(8):e9599.
    PMID: 32923203 DOI: 10.7759/cureus.9599
    An adrenal incidentaloma is defined as an adrenal mass measuring at least 1 cm that is discovered surreptitiously in an imaging study done for reasons other than the evaluation of adrenal disease. The increase in the prevalence of adrenal incidentalomas has paralleled the increase in diagnostic imaging done for evaluation of other abdominal pathologies. However, most of these adrenal incidentalomas are benign non-hyperfunctioning adenomas. When an adrenal incidentaloma is discovered, the simultaneous presence of hypokalemia, metabolic alkalosis, mild hypernatremia, and mild to severe drug-resistant hypertension may alert a clinician to underlying primary hyperaldosteronism. We present a case of adrenal incidentaloma noted in a patient with end-stage renal disease on hemodialysis which presented a diagnostic challenge due to the correction of metabolic parameters with hemodialysis. The patient was found to have an aldosterone-producing adenoma based on an elevated aldosterone-to-renin ratio and was started on a mineralocorticoid antagonist.
    Matched MeSH terms: Hyperaldosteronism
  17. Aimi Fadilah M, Fatimah MS, Nor Aisyah Z, Nur’Aini EW, Nazimah AM, Effat O, et al.
    MyJurnal
    Primary aldosteronism (PA) causes a persistently elevated blood pressure (BP) due to
    excessive release of the hormone aldosterone from the adrenal glands. Classically, it is called
    Conn’s syndrome and is described as the triad of hypertension and hypokalemia with the
    presence of unilateral adrenal adenoma. It can be cured with surgical resection of the
    aldosterone-secreting adenoma leading to resolution of hypertension, hypokalemia and
    increased cardiovascular risk associated with hyperaldosteronism. We present a case of a man
    with previous ischemic heart disease (IHD) who presented with resistant hypertension.
    Investigations for secondary causes of hypertension revealed an elevated aldosterone level
    and saline suppression test confirmed the diagnosis of PA. Radiological examination revealed
    a left adrenal adenoma and a normal right adrenal gland. However, adrenal venous sampling
    showed lateralization of aldosterone secretion towards the right. He subsequently underwent
    a laparoscopic right adrenalectomy which improved his BP control promptly. This case
    highlights the importance of recognizing the need to investigate for secondary causes of
    hypertension. It also underscores the importance of dynamic tests, which may not be easily
    accessible to most clinicians but should pursue, to allow a definitive diagnosis and effective
    treatment.
    Matched MeSH terms: Hyperaldosteronism
  18. Chan PL, Tan FHS
    Clin Hypertens, 2018;24:15.
    PMID: 30410790 DOI: 10.1186/s40885-018-0100-x
    Background: Hypokalemia in the presence of hypertension is often attributed to primary hyperaldosteronism as a cause of secondary hypertension, however secondary hyperaldosteronism may present similarly. Accessory renal arteries are variants in the vascular anatomy which are often thought to be innocuous but in some circumstances can cause renovascular hypertension leading to secondary hyperaldosteronism.

    Case presentation: We report 2 cases of hypertension with secondary hyperaldosteronism associated with accessory renal arteries. Both patients presented with hypokalemia and further investigations revealed hyperaldosteronism with unsuppressed renin levels. Imaging studies showed the presence of accessory renal artery.

    Conclusion: Accessory renal arteries are a potential cause renovascular hypertension which can be detected via CT angiography or magnetic resonance angiography. Hormonal evaluation should be undertaken to determine whether its presence contributes to hypertension in the patient as targeted treatment such as aldosterone antagonist can be initiated. Surgical intervention or renal denervation may be considered in resistant cases.

    Matched MeSH terms: Hyperaldosteronism
  19. Teo AE, Garg S, Shaikh LH, Zhou J, Karet Frankl FE, Gurnell M, et al.
    N Engl J Med, 2015 Oct 08;373(15):1429-36.
    PMID: 26397949 DOI: 10.1056/NEJMoa1504869
    Recent discoveries of somatic mutations permit the recognition of subtypes of aldosterone-producing adenomas with distinct clinical presentations and pathological features. Here we describe three women with hyperaldosteronism, two who presented in pregnancy and one who presented after menopause. Their aldosterone-producing adenomas harbored activating mutations of CTNNB1, encoding β-catenin in the Wnt cell-differentiation pathway, and expressed LHCGR and GNRHR, encoding gonadal receptors, at levels that were more than 100 times as high as the levels in other aldosterone-producing adenomas. The mutations stimulate Wnt activation and cause adrenocortical cells to de-differentiate toward their common adrenal-gonadal precursor cell type. (Funded by grants from the National Institute for Health Research Cambridge Biomedical Research Centre and others.).
    Matched MeSH terms: Hyperaldosteronism/etiology*
  20. Zhou J, Azizan EAB, Cabrera CP, Fernandes-Rosa FL, Boulkroun S, Argentesi G, et al.
    Nat Genet, 2021 Sep;53(9):1360-1372.
    PMID: 34385710 DOI: 10.1038/s41588-021-00906-y
    Most aldosterone-producing adenomas (APAs) have gain-of-function somatic mutations of ion channels or transporters. However, their frequency in aldosterone-producing cell clusters of normal adrenal gland suggests a requirement for codriver mutations in APAs. Here we identified gain-of-function mutations in both CTNNB1 and GNA11 by whole-exome sequencing of 3/41 APAs. Further sequencing of known CTNNB1-mutant APAs led to a total of 16 of 27 (59%) with a somatic p.Gln209His, p.Gln209Pro or p.Gln209Leu mutation of GNA11 or GNAQ. Solitary GNA11 mutations were found in hyperplastic zona glomerulosa adjacent to double-mutant APAs. Nine of ten patients in our UK/Irish cohort presented in puberty, pregnancy or menopause. Among multiple transcripts upregulated more than tenfold in double-mutant APAs was LHCGR, the receptor for luteinizing or pregnancy hormone (human chorionic gonadotropin). Transfections of adrenocortical cells demonstrated additive effects of GNA11 and CTNNB1 mutations on aldosterone secretion and expression of genes upregulated in double-mutant APAs. In adrenal cortex, GNA11/Q mutations appear clinically silent without a codriver mutation of CTNNB1.
    Matched MeSH terms: Hyperaldosteronism/pathology
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