{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,3]],"date-time":"2026-06-03T05:47:59Z","timestamp":1780465679902,"version":"3.54.1"},"reference-count":10,"publisher":"Oxford University Press (OUP)","issue":"6","license":[{"start":{"date-parts":[[2024,6,9]],"date-time":"2024-06-09T00:00:00Z","timestamp":1717891200000},"content-version":"vor","delay-in-days":8,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,6,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Insulin autoimmune syndrome (IAS) is a rare cause of spontaneous hypoglycaemia. We discuss a 91-year-old Caucasian lady who presented with syncope and episodic adrenergic and neuroglycopenic symptoms. Despite significantly elevated insulin, C-peptide, and proinsulin levels with the presence of anti-insulin antibodies, a pancreatic mass was not identified. Serum immunoelectrophoresis demonstrated monoclonal gammopathy of undetermined significance (MGUS). Treatment involved high-dose steroids, diazoxide, corn starch and acarbose, however the patient passed away four months later due to worsening co-morbidities. The management of IAS in the setting of MGUS is challenging.<\/jats:p>","DOI":"10.1093\/omcr\/omae054","type":"journal-article","created":{"date-parts":[[2024,6,9]],"date-time":"2024-06-09T14:04:57Z","timestamp":1717941897000},"source":"Crossref","is-referenced-by-count":2,"title":["A clinical case of insulin autoimmune syndrome with monoclonal gammopathy of uncertain significance; complexity in management"],"prefix":"10.1093","volume":"2024","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-3933-4425","authenticated-orcid":false,"given":"Liam","family":"Clifford","sequence":"first","affiliation":[{"name":"Gosford Hospital Department of Endocrinology, , Gosford, NSW, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8147-7625","authenticated-orcid":false,"given":"Flavian","family":"Joseph","sequence":"additional","affiliation":[{"name":"Gosford Hospital Department of Endocrinology, , Gosford, NSW, Australia"},{"name":"University of Newcastle , Newcastle, NSW, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7840-7597","authenticated-orcid":false,"given":"Tripti","family":"Joshi","sequence":"additional","affiliation":[{"name":"Gosford Hospital Department of Endocrinology, , Gosford, NSW, Australia"},{"name":"University of Newcastle , Newcastle, NSW, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2024,6,7]]},"reference":[{"key":"2024060914003930900_ref1","first-page":"312","article-title":"Insulin autoimmunity in a case with spontaneous hypoglycemia","volume":"13","author":"Hirata","year":"1970","journal-title":"J Jpn Diabet Soc"},{"key":"2024060914003930900_ref2","doi-asserted-by":"crossref","first-page":"141","DOI":"10.1097\/MD.0b013e3181a5b42e","article-title":"Autoimmune forms of hypoglycemia","volume":"88","author":"Lupsa","year":"2009","journal-title":"Medicine"},{"key":"2024060914003930900_ref3","doi-asserted-by":"crossref","first-page":"1351","DOI":"10.1016\/S0140-6736(86)92003-9","article-title":"Islet-cell antibodies and insulin autoantibodies in association with common viral infections","volume":"328","author":"Bodansky","year":"1986","journal-title":"Lancet"},{"key":"2024060914003930900_ref4","doi-asserted-by":"crossref","first-page":"554","DOI":"10.1111\/jdi.13180","article-title":"Nationwide survey of endogenous hyperinsulinemic hypoglycemia in Japan (2017-2018): congenital hyperinsulinism, insulinoma, non-insulinoma pancreatogenous hypoglycemia syndrome and insulin autoimmune syndrome (Hirata\u2019s disease)","volume":"11","author":"Yamada","year":"2019","journal-title":"J Diabetes Investig"},{"key":"2024060914003930900_ref5","doi-asserted-by":"crossref","first-page":"89","DOI":"10.1016\/0168-8227(94)90040-X","article-title":"Insulin autoimmune syndrome (Hirata disease): clinical features and epidemiology in Japan","volume":"22","author":"Uchigata","year":"1994","journal-title":"Diabetes Res Clin Pract"},{"key":"2024060914003930900_ref6","doi-asserted-by":"crossref","first-page":"e19","DOI":"10.1016\/j.diabres.2008.10.015","article-title":"Drug-induced insulin autoimmune syndrome","volume":"83","author":"Uchigata","year":"2008","journal-title":"Diabetes Res Clin Pract"},{"key":"2024060914003930900_ref7","doi-asserted-by":"crossref","first-page":"135","DOI":"10.1016\/j.cca.2014.03.008","article-title":"Development of insulin antibodies and changes in titers over a long-term period in patients with type 2 diabetes","volume":"433","author":"Hattori","year":"2014","journal-title":"Clin Chim Acta"},{"key":"2024060914003930900_ref8","doi-asserted-by":"crossref","first-page":"347","DOI":"10.1111\/cen.13179","article-title":"Diagnosis of insulin autoimmune syndrome using polyethylene glycol precipitation and gel filtration chromatography with ex vivo insulin exchange","volume":"86","author":"Church","year":"2017","journal-title":"Clin Endocrinol"},{"key":"2024060914003930900_ref9","doi-asserted-by":"crossref","first-page":"1","DOI":"10.5041\/RMMJ.10212","article-title":"Unusual manifestations of essential monoclonal Gammopathy. II. Simulation of the insulin autoimmune syndrome","volume":"6","author":"Lichtman","year":"2015","journal-title":"Rambam Maimonides Med J"},{"key":"2024060914003930900_ref10","first-page":"1","article-title":"Steroid-refractory insulin autoimmune syndrome treated with rituximab and continuous glucose monitoring","volume":"13","author":"Batra","year":"2021","journal-title":"Cureus"}],"container-title":["Oxford Medical Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/omcr\/article-pdf\/2024\/6\/omae054\/58161748\/omae054.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/omcr\/article-pdf\/2024\/6\/omae054\/58161748\/omae054.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,6,9]],"date-time":"2024-06-09T14:05:08Z","timestamp":1717941908000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/omcr\/article\/doi\/10.1093\/omcr\/omae054\/7689280"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,6,1]]},"references-count":10,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2024,6,1]]}},"URL":"https:\/\/doi.org\/10.1093\/omcr\/omae054","relation":{},"ISSN":["2053-8855"],"issn-type":[{"value":"2053-8855","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2024,6]]},"published":{"date-parts":[[2024,6,1]]},"article-number":"omae054"}}