{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,17]],"date-time":"2025-10-17T20:06:50Z","timestamp":1760731610944,"version":"3.37.3"},"reference-count":13,"publisher":"Georg Thieme Verlag KG","issue":"02","license":[{"start":{"date-parts":[[2024,2,27]],"date-time":"2024-02-27T00:00:00Z","timestamp":1708992000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Avicenna J Med"],"published-print":{"date-parts":[[2024,4]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Schmidt's syndrome, or autoimmune polyendocrine syndrome type 2 (APS-2), is an uncommon disorder characterized by the co-occurrence of autoimmune thyroiditis and adrenalitis. APS-2 is defined as a combination of Addison's disease, autoimmune thyroid disease, and\/or type 1 diabetes mellitus. It is an autosomal dominantly inherited polygenic disorder with incomplete penetrance; the candidate genes include but are not limited to HLA-DR3, HLA-DR4, CTLA-4, PTPN22, and CD25-IL-2. Autoimmune thyroiditis, often Hashimoto's disease, results in hypothyroidism. Primary adrenal failure results in enhanced secretion of adrenocorticotrophic hormone melanocyte and co-secretion of melanocyte-stimulating hormone, contributing to hyperpigmentation. Mineralocorticoid deficiency results in salt wasting, fatigue and cramps, postural hypotension, and hyperkalemia. Cortisol, an insulin counter-regulatory hormone, plays a pivotal role in maintaining euglycemia; deficiency predisposes to the development of hypoglycemia. We here report a rare presentation of Schmidt's syndrome as hypoinsulinemic hypoglycemia in a middle-aged male patient. Management includes treatment of acute hypoglycemic episodes with glucose or glucagon, long-term glucocorticoids and mineralocorticoids for adrenal insufficiency, and thyroid hormone supplements for hypothyroidism. This case report and brief overview aim to contribute to the scientific understanding of Schmidt's syndrome\/APS-2. Additionally, here we briefly outline the diagnostic challenges in hypoglycemia evaluation, including the utilization of Whipple's triad and the gold standard supervised 72-hour fast and evaluation for primary adrenal and thyroid insufficiencies.<\/jats:p>","DOI":"10.1055\/s-0044-1779745","type":"journal-article","created":{"date-parts":[[2024,2,28]],"date-time":"2024-02-28T00:53:36Z","timestamp":1709081616000},"page":"130-133","source":"Crossref","is-referenced-by-count":1,"title":["Schmidt's Syndrome: An Uncommon Cause of Spontaneous Hypoglycemia"],"prefix":"10.1055","volume":"14","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5705-3275","authenticated-orcid":false,"given":"George Sarin","family":"Zacharia","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine & Gastroenterology, Ahalia Hospital, Mussafah, Abu Dhabi, United Arab Emirates"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anu","family":"Jacob","sequence":"additional","affiliation":[{"name":"Department of Anaesthesiology, Ahalia Hospital, Mussafah, Abu Dhabi, United Arab Emirates"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Binu Mary","family":"Bose","sequence":"additional","affiliation":[{"name":"Department of Pathology, Malankara Orthodox Syrian Church Medical College, Ernakulum, Kerala, India"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"194","published-online":{"date-parts":[[2024,2,27]]},"reference":[{"key":"ref1","first-page":"153","article-title":"Schmidt's syndrome (thyroid and adrenal insufficiency). A review of the literature and a report of fifteen new cases including ten instances of coexistent diabetes mellitus","volume":"43","author":"C C Carpenter","year":"1964","journal-title":"Medicine (Baltimore)"},{"issue":"01","key":"ref2","doi-asserted-by":"crossref","first-page":"91","DOI":"10.1007\/s40618-017-0740-9","article-title":"Polyglandular autoimmune syndromes","volume":"41","author":"G J Kahaly","year":"2018","journal-title":"J Endocrinol Invest"},{"volume-title":"Polyglandular autoimmune syndrome type II. [Updated 2023 Aug 8]","year":"2023","author":"G Singh","key":"ref3"},{"key":"ref4","doi-asserted-by":"publisher","DOI":"10.1007\/978-1-60327-285-8_50","volume-title":"Autoimmune polyendocrine syndromes","author":"A Tincani","year":"2008"},{"issue":"02","key":"ref5","doi-asserted-by":"crossref","first-page":"92","DOI":"10.1056\/NEJM197801122980209","article-title":"HLA type and occurrence of disease in familial polyglandular failure","volume":"298","author":"G Eisenbarth","year":"1978","journal-title":"N Engl J Med"},{"issue":"03","key":"ref6","doi-asserted-by":"crossref","first-page":"495","DOI":"10.1016\/S0889-8529(05)70084-0","article-title":"Symptoms of hypoglycemia, thresholds for their occurrence, and hypoglycemia unawareness","volume":"28","author":"P E Cryer","year":"1999","journal-title":"Endocrinol Metab Clin North Am"},{"issue":"01","key":"ref7","doi-asserted-by":"crossref","first-page":"261","DOI":"10.1186\/s13256-021-02844-z","article-title":"Transient hypoglycemia as a rare cause of recurring transient loss of consciousness: a case report","volume":"15","author":"M Wester","year":"2021","journal-title":"J Med Case Rep"},{"issue":"03","key":"ref8","doi-asserted-by":"crossref","first-page":"709","DOI":"10.1210\/jc.2008-1410","article-title":"Evaluation and management of adult hypoglycemic disorders: an Endocrine Society Clinical Practice Guideline","volume":"94","author":"P E Cryer","year":"2009","journal-title":"J Clin Endocrinol Metab"},{"issue":"03","key":"ref9","doi-asserted-by":"crossref","first-page":"380","DOI":"10.1111\/j.1532-5415.1999.tb03016.x","article-title":"Recurrent, refractory hypoglycemia presenting as a behavioral disorder in an older woman","volume":"47","author":"T S Dharmarajan","year":"1999","journal-title":"J Am Geriatr Soc"},{"issue":"07","key":"ref11","first-page":"563","article-title":"Addison disease: early detection and treatment principles","volume":"89","author":"A Michels","year":"2014","journal-title":"Am Fam Physician"},{"issue":"03","key":"ref12","first-page":"16222","article-title":"Hashimoto thyroiditis: an evidence-based guide to etiology, diagnosis and treatment","volume":"132","author":"J Klubo-Gwiezdzinska","year":"2022","journal-title":"Pol Arch Intern Med"},{"key":"ref13","doi-asserted-by":"crossref","first-page":"634","DOI":"10.1186\/1756-0500-7-634","article-title":"Recurrent hypoglycaemia in type-1 diabetes mellitus may unravel the association with Addison's disease: a case report","volume":"7","author":"S Passanisi","year":"2014","journal-title":"BMC Res Notes"},{"issue":"06","key":"ref14","first-page":"e26322","article-title":"Atypical case of Schmidt's syndrome in a young male","volume":"14","author":"I Abdullah","year":"2022","journal-title":"Cureus"}],"container-title":["Avicenna Journal of Medicine"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/www.thieme-connect.de\/products\/ejournals\/pdf\/10.1055\/s-0044-1779745.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,11,5]],"date-time":"2024-11-05T10:37:20Z","timestamp":1730803040000},"score":1,"resource":{"primary":{"URL":"http:\/\/www.thieme-connect.de\/DOI\/DOI?10.1055\/s-0044-1779745"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,2,27]]},"references-count":13,"journal-issue":{"issue":"02","published-online":{"date-parts":[[2024,7,1]]},"published-print":{"date-parts":[[2024,4]]}},"URL":"https:\/\/doi.org\/10.1055\/s-0044-1779745","archive":["Portico","CLOCKSS"],"relation":{},"ISSN":["2231-0770","2249-4464"],"issn-type":[{"type":"print","value":"2231-0770"},{"type":"electronic","value":"2249-4464"}],"subject":[],"published":{"date-parts":[[2024,2,27]]}}}