{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,7]],"date-time":"2026-07-07T16:17:59Z","timestamp":1783441079638,"version":"3.54.6"},"reference-count":29,"publisher":"Bioscientifica","issue":"3","license":[{"start":{"date-parts":[[2026,2,10]],"date-time":"2026-02-10T00:00:00Z","timestamp":1770681600000},"content-version":"vor","delay-in-days":375,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["journals.bioscientifica.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2025,3,1]]},"abstract":"<jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Data on bilateral macronodular adrenocortical disease (BMAD) with respect to computed tomography (CT) scan characteristics (attenuation and washout) and long-term follow-up are limited. This study aims to describe BMAD patients managed in a single centre.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>BMAD was defined by the presence of bilateral adrenal macronodules (&amp;gt;1 cm) on CT. Clinical, biochemical, radiological, genetic characteristics, management and follow-up of 22 BMAD patients were studied retrospectively.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>The median age (range) at presentation was 49.5 (23\u201383) years, predominantly observed in females (16\/22). Eighteen (82%) patients were incidentally diagnosed (11 with mild autonomous cortisol secretion (MACS) and seven non-secretory), three (13.7%) presented with overt Cushing\u2019s syndrome (CS), and one (4.5%) had androgen excess (without CS features). On CT, the dominant nodule\u2019s median (range) size was 2.6(1.6\u20139.5) cm. 77.8% (14\/18) of adrenal nodules were lipid-rich, and 93.3% (14\/15) of the nodules exhibited good washout. Genetic analysis was available for eight patients; one had a novel germline ARMC5 variant, and two had MEN-1 gene mutations. Three overt CS and one androgen-secreting patient underwent total bilateral adrenalectomy; histopathology showed macronodular hyperplasia with internodular hypertrophy. Only one (1\/8) patient from the MACS group developed a new comorbidity (diabetes mellitus) after a median follow-up of 6.4 (0.5\u201312.4) years, while none of the non-secretory group patients developed new comorbidities after a median follow-up of 1.4 (0.8\u201312.2) years.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Most BMAD patients presented without overt hormonal excess, and none developed overt CS on follow-up. Detailed CT characteristics of BMAD nodules may help in radiological diagnosis in bilateral adrenal incidentalomas.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1530\/ec-24-0664","type":"journal-article","created":{"date-parts":[[2025,1,31]],"date-time":"2025-01-31T10:10:20Z","timestamp":1738318220000},"update-policy":"https:\/\/doi.org\/10.1530\/crossmarkpolicy-10","source":"Crossref","is-referenced-by-count":2,"title":["Bilateral macronodular adrenocortical disease: a single centre experience"],"prefix":"10.1530","volume":"14","author":[{"given":"Anuj","family":"Ban","sequence":"first","affiliation":[{"name":"Department of Endocrinology and Metabolism, Seth G S Medical College and KEM Hospital 1 , ,","place":["Mumbai, India"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7612-506X","authenticated-orcid":true,"given":"Rohit","family":"Barnabas","sequence":"additional","affiliation":[{"name":"Department of Endocrinology and Metabolism, Seth G S Medical College and KEM Hospital 1 , ,","place":["Mumbai, India"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Manjiri","family":"Karlekar","sequence":"additional","affiliation":[{"name":"Department of Endocrinology and Metabolism, Seth G S Medical College and KEM Hospital 1 , ,","place":["Mumbai, India"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9623-4471","authenticated-orcid":true,"given":"Anurag Ranjan","family":"Lila","sequence":"additional","affiliation":[{"name":"Department of Endocrinology and Metabolism, Seth G S Medical College and KEM Hospital 1 , ,","place":["Mumbai, India"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chethan","family":"Yami Channaiah","sequence":"additional","affiliation":[{"name":"Department of Endocrinology and Metabolism, Seth G S Medical College and KEM Hospital 1 , ,","place":["Mumbai, India"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Saba Samad","family":"Memon","sequence":"additional","affiliation":[{"name":"Department of Endocrinology and Metabolism, Seth G S Medical College and KEM Hospital 1 , ,","place":["Mumbai, India"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Virendra A","family":"Patil","sequence":"additional","affiliation":[{"name":"Department of Endocrinology and Metabolism, Seth G S Medical College and KEM Hospital 1 , ,","place":["Mumbai, India"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vijaya","family":"Sarathi","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Vydehi Institute of Medical Sciences and Research Centre 2 , ,","place":["Bangalore, India"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gwendolyn","family":"Fernandes","sequence":"additional","affiliation":[{"name":"Department of Pathology, Seth GSMC and KEMH 3 , ,","place":["Mumbai, India"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hemangini","family":"Thakkar","sequence":"additional","affiliation":[{"name":"Department of Radiology, Seth GSMC and KEMH 4 , ,","place":["Mumbai, India"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sameer","family":"Rege","sequence":"additional","affiliation":[{"name":"Department of General Surgery, Seth GSMC and KEMH 5 , ,","place":["Mumbai, India"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nalini S","family":"Shah","sequence":"additional","affiliation":[{"name":"Department of Endocrinology and Metabolism, Seth G S Medical College and KEM Hospital 1 , ,","place":["Mumbai, India"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tushar","family":"Bandgar","sequence":"additional","affiliation":[{"name":"Department of Endocrinology and Metabolism, Seth G S Medical College and KEM Hospital 1 , ,","place":["Mumbai, India"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"416","published-online":{"date-parts":[[2025,1,31]]},"reference":[{"key":"2026070710570265100_bib1","doi-asserted-by":"crossref","first-page":"245","DOI":"10.1016\/j.beem.2008.10.011","article-title":"ACTH-independent macronodular adrenal hyperplasia","volume":"23","author":"Lacroix","year":"2009","journal-title":"Best Pract Res Clin Endocrinol Metab"},{"key":"2026070710570265100_bib2","doi-asserted-by":"crossref","first-page":"E926","DOI":"10.1210\/jc.2014-4204","article-title":"ARMC5 mutations in a large cohort of primary macronodular adrenal hyperplasia: clinical and functional consequences","volume":"100","author":"Espiard","year":"2015","journal-title":"J Clin Endocrinol Metab"},{"key":"2026070710570265100_bib3","doi-asserted-by":"crossref","first-page":"123","DOI":"10.1530\/EJE-21-1032","article-title":"Identification of predictive criteria for pathogenic variants of primary bilateral macronodular adrenal hyperplasia (PBMAH) gene ARMC5 in 352 unselected patients","volume":"187","author":"Bouys","year":"2022","journal-title":"Eur J Endocrinol"},{"key":"2026070710570265100_bib4","doi-asserted-by":"crossref","first-page":"913","DOI":"10.1016\/S0140-6736(14)61375-1","article-title":"Cushing\u2019s syndrome","volume":"386","author":"Lacroix","year":"2015","journal-title":"Lancet"},{"key":"2026070710570265100_bib5","doi-asserted-by":"crossref","first-page":"567","DOI":"10.1210\/endrev\/bnac034","article-title":"Clinical, pathophysiologic, genetic, and therapeutic progress in primary bilateral macronodular adrenal hyperplasia","volume":"44","author":"Bertherat","year":"2023","journal-title":"Endocr Rev"},{"key":"2026070710570265100_bib6","doi-asserted-by":"crossref","first-page":"430","DOI":"10.1016\/j.juro.2012.07.139","article-title":"Adrenal nodular hyperplasia in hereditary leiomyomatosis and renal cell cancer","volume":"189","author":"Shuch","year":"2013","journal-title":"J Urol"},{"key":"2026070710570265100_bib7","doi-asserted-by":"crossref","first-page":"679","DOI":"10.1097\/DCR.0000000000001008","article-title":"Characteristics of adrenal masses in familial adenomatous polyposis","volume":"61","author":"Shiroky","year":"2018","journal-title":"Dis Colon Rectum"},{"key":"2026070710570265100_bib8","doi-asserted-by":"crossref","first-page":"2990","DOI":"10.1210\/jc.2012-1230","article-title":"Clinical practice guidelines for multiple endocrine neoplasia type 1 (MEN1)","volume":"97","author":"Thakker","year":"2012","journal-title":"J Clin Endocrinol Metab"},{"key":"2026070710570265100_bib9","doi-asserted-by":"crossref","first-page":"3773","DOI":"10.1210\/jc.2004-2377","article-title":"Hereditary leiomyomatosis associated with bilateral, massive, macronodular adrenocortical disease and atypical cushing syndrome: a clinical and molecular genetic investigation","volume":"90","author":"Matyakhina","year":"2005","journal-title":"J Clin Endocrinol Metab"},{"key":"2026070710570265100_bib10","doi-asserted-by":"crossref","first-page":"2105","DOI":"10.1056\/NEJMoa1304603","article-title":"ARMC5 mutations in macronodular adrenal hyperplasia with Cushing\u2019s syndrome","volume":"369","author":"Assi\u00e9","year":"2013","journal-title":"N Engl J Med"},{"key":"2026070710570265100_bib11","doi-asserted-by":"crossref","first-page":"813","DOI":"10.1016\/S2213-8587(21)00236-9","article-title":"Loss of KDM1A in GIP-dependent primary bilateral macronodular adrenal hyperplasia with Cushing\u2019s syndrome: a multicentre, retrospective, cohort study","volume":"9","author":"Chasseloup","year":"2021","journal-title":"Lancet Diabetes Endocrinol"},{"key":"2026070710570265100_bib12","doi-asserted-by":"crossref","DOI":"10.1210\/jendso\/bvaa162","article-title":"Volumetric modeling of adrenal gland size in primary bilateral macronodular adrenocortical hyperplasia","volume":"5","author":"Wurth","year":"2021","journal-title":"J Endocr Soc"},{"key":"2026070710570265100_bib13","doi-asserted-by":"crossref","first-page":"1173","DOI":"10.21037\/atm-20-5963","article-title":"Analysis of clinical and pathological features of primary bilateral macronodular adrenocortical hyperplasia compared with unilateral cortisol-secreting adrenal adenoma","volume":"8","author":"Zhang","year":"2020","journal-title":"Ann Transl Med"},{"key":"2026070710570265100_bib14","doi-asserted-by":"crossref","first-page":"458","DOI":"10.1016\/j.rce.2022.01.003","article-title":"Prevalence and phenotype of primary bilateral macronodular adrenal hyperplasia with autonomous cortisol secretion: a study of 98 patients","volume":"222","author":"Bengoa-Rojano","year":"2022","journal-title":"Rev Clin Esp"},{"key":"2026070710570265100_bib15","doi-asserted-by":"crossref","first-page":"898","DOI":"10.1530\/EC-19-0198","article-title":"Radiological differentiation of phaeochromocytoma from other malignant adrenal masses: importance of wash-in characteristics on multiphase CECT","volume":"8","author":"Goroshi","year":"2019","journal-title":"Endocr Connect"},{"key":"2026070710570265100_bib16","doi-asserted-by":"crossref","DOI":"10.1259\/bjr.20151018","article-title":"Adrenal imaging for adenoma characterization: imaging features, diagnostic accuracies and differential diagnoses","volume":"89","author":"Park","year":"2016","journal-title":"Br J Radiol"},{"key":"2026070710570265100_bib17","doi-asserted-by":"crossref","first-page":"G1","DOI":"10.1093\/ejendo\/lvad066","article-title":"European Society of Endocrinology clinical practice guidelines on the management of adrenal incidentalomas, in collaboration with the European Network for the Study of Adrenal Tumors","volume":"189","author":"Fassnacht","year":"2023","journal-title":"Eur J Endocrinol"},{"key":"2026070710570265100_bib18","doi-asserted-by":"crossref","first-page":"129","DOI":"10.1530\/EJE-10-0195","article-title":"Aberrant cortisol regulations in bilateral macronodular adrenal hyperplasia: a frequent finding in a prospective study of 32 patients with overt or subclinical Cushing\u2019s syndrome","volume":"163","author":"Lib\u00e9","year":"2010","journal-title":"Eur J Endocrinol"},{"key":"2026070710570265100_bib19","doi-asserted-by":"crossref","first-page":"959","DOI":"10.1007\/s12020-016-0956-z","article-title":"A multicenter experience on the prevalence of ARMC5 mutations in patients with primary bilateral macronodular adrenal hyperplasia: from genetic characterization to clinical phenotype","volume":"55","author":"Albiger","year":"2017","journal-title":"Endocrine"},{"key":"2026070710570265100_bib20","doi-asserted-by":"crossref","first-page":"E1501","DOI":"10.1210\/jc.2013-4237","article-title":"ARMC5 mutations are a frequent cause of primary macronodular adrenal hyperplasia","volume":"99","author":"Alencar","year":"2014","journal-title":"J Clin Endocrinol Metab"},{"key":"2026070710570265100_bib21","doi-asserted-by":"crossref","first-page":"269","DOI":"10.1530\/EJE-11-0679","article-title":"Adrenal involvement in MEN1. Analysis of 715 cases from the Groupe d\u2019etude des Tumeurs Endocrines database","volume":"166","author":"Gatta-Cherifi","year":"2012","journal-title":"Eur J Endocrinol"},{"key":"2026070710570265100_bib22","doi-asserted-by":"crossref","first-page":"2985","DOI":"10.1210\/jc.2018-02204","article-title":"Long-term outcome of primary bilateral macronodular adrenocortical hyperplasia after unilateral adrenalectomy","volume":"104","author":"Osswald","year":"2019","journal-title":"J Clin Endocrinol Metab"},{"key":"2026070710570265100_bib23","doi-asserted-by":"crossref","first-page":"690","DOI":"10.1016\/j.eprac.2022.04.011","article-title":"A 30-year, single-center experience of unilateral adrenalectomy for primary bilateral macronodular adrenal hyperplasia","volume":"28","author":"Wang","year":"2022","journal-title":"Endocr Pract"},{"key":"2026070710570265100_bib24","doi-asserted-by":"crossref","first-page":"1206","DOI":"10.2214\/AJR.16.17758","article-title":"Update on CT and MRI of adrenal nodules","volume":"208","author":"Schieda","year":"2017","journal-title":"AJR Am J Roentgenol"},{"key":"2026070710570265100_bib25","doi-asserted-by":"crossref","first-page":"1027","DOI":"10.1007\/s00330-017-5076-5","article-title":"Pheochromocytoma as a frequent false-positive in adrenal washout CT: a systematic review and meta-analysis","volume":"28","author":"Woo","year":"2018","journal-title":"Eur Radiol"},{"key":"2026070710570265100_bib26","doi-asserted-by":"crossref","first-page":"73","DOI":"10.1210\/jc.2002-021292","article-title":"Virilization in bilateral macronodular adrenal hyperplasia controlled by luteinizing hormone","volume":"88","author":"Goodarzi","year":"2003","journal-title":"J Clin Endocrinol Metab"},{"key":"2026070710570265100_bib27","doi-asserted-by":"crossref","first-page":"771","DOI":"10.1530\/EJE-07-0253","article-title":"Food-dependent Cushing\u2019s syndrome: from molecular characterization to therapeutical results","volume":"157","author":"Albiger","year":"2007","journal-title":"Eur J Endocrinol"},{"key":"2026070710570265100_bib28","first-page":"57","article-title":"Functional implications of LH\/hCG receptors in pregnancy-induced cushing syndrome","volume":"1","author":"Pl\u00f6ckinger","year":"2017","journal-title":"J Endocr Soc"},{"key":"2026070710570265100_bib29","doi-asserted-by":"crossref","first-page":"E243","DOI":"10.1210\/jc.2010-1222","article-title":"Clinical, biochemical, and molecular characterization of macronodular adrenocortical hyperplasia of the zona reticularis: a new syndrome","volume":"96","author":"Ghayee","year":"2011","journal-title":"J Clin Endocrinol Metab"}],"container-title":["Endocrine Connections"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/ec.bioscientifica.com\/view\/journals\/ec\/14\/3\/EC-24-0664.xml","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/ec.bioscientifica.com\/downloadpdf\/journals\/ec\/14\/3\/EC-24-0664.xml","content-type":"text\/html","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/journals.bioscientifica.com\/ec\/article-pdf\/doi\/10.1530\/EC-24-0664\/9224\/ec-24-0664.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/journals.bioscientifica.com\/ec\/article-pdf\/doi\/10.1530\/EC-24-0664\/9224\/ec-24-0664.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,7,7]],"date-time":"2026-07-07T15:17:57Z","timestamp":1783437477000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.bioscientifica.com\/ec\/article\/14\/3\/e240664\/1355\/Bilateral-macronodular-adrenocortical-disease-a"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,1,31]]},"references-count":29,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2025,3,1]]}},"URL":"https:\/\/doi.org\/10.1530\/ec-24-0664","relation":{},"ISSN":["2049-3614"],"issn-type":[{"value":"2049-3614","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,1,31]]},"article-number":"e240664"}}