{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,4]],"date-time":"2026-05-04T23:02:11Z","timestamp":1777935731177,"version":"3.51.4"},"reference-count":90,"publisher":"Walter de Gruyter GmbH","issue":"5","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2017,1,1]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Cushing\u2019s syndrome is rare in childhood and is usually caused by a pituitary adenoma. Primary hyperfunction of adrenal glands is less frequent, particularly primary pigmented nodular adrenocortical disease (PPNAD). It occurs usually in children and adolescents, with female preponderance, while Cushing\u2019s disease has increased frequency in prepubertal males. A case of a 6-year-old boy is presented with isolated non-familiar PPNAD. The clinical pattern involved Cushingoid appearance, hypertension, virilization and depressive mood. Laboratory analyses showed loss of circadian rhythm of cortisol, undetectable adrenocorticotropic hormone (ACTH) level, impaired fasting glucose, polycythemia and elevated white blood count (WBC). Radiology investigation revealed a slightly enlarged medial branch of the left adrenal gland and a normal right one, so a unilateral adrenalectomy was performed. Pathohistology described multiple dark brownish pigmented nodules of various sizes confined to the cortex. Contralateral adrenalectomy was done 3 months later. Follow-up of 3 years was uneventful, except for one adrenal crisis during an intercurrent respiratory illness.<\/jats:p>","DOI":"10.1515\/jpem-2016-0249","type":"journal-article","created":{"date-parts":[[2017,4,9]],"date-time":"2017-04-09T10:01:20Z","timestamp":1491732080000},"source":"Crossref","is-referenced-by-count":5,"title":["Primary pigmented nodular adrenocortical disease: literature review and case report of a 6-year-old boy"],"prefix":"10.1515","volume":"30","author":[{"given":"Dragan","family":"Katani\u0107","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Dejan","family":"Kafka","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mirjana","family":"\u017divojinov","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jovan","family":"Vla\u0161ki","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Zorana","family":"Budakov","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Marija","family":"Kne\u017eevi\u0107 Pogan\u010dev","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ivana","family":"Vorgu\u010din","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tomislav","family":"\u0106uk","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"374","reference":[{"key":"ref61","doi-asserted-by":"crossref","first-page":"4324","DOI":"10.1210\/jc.2002-020592","article-title":"Mutations of the PRKAR1A gene in Cushing\u2019s syndrome due to sporadic primary pigmented nodular adrenocortical disease","volume":"87","year":"2002","journal-title":"J Clin Endocrinol Metab"},{"key":"ref191","doi-asserted-by":"crossref","first-page":"428","DOI":"10.1530\/acta.0.1020428","article-title":"Familial Cushing\u2019s syndrome due to pigmented multinodular adrenocortical dysplasia","volume":"102","year":"1983","journal-title":"Acta Endocrinol"},{"key":"ref21","first-page":"231","volume-title":"An unusual case of McCune Albrights syndrome. 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