Please use this identifier to cite or link to this item: https://ahro.austin.org.au/austinjspui/handle/1/21703
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dc.contributor.authorGunjur, Ashray-
dc.contributor.authorKlein, Oliver-
dc.contributor.authorKee, Damien-
dc.contributor.authorCebon, Jonathan S-
dc.date2019-09-05-
dc.date.accessioned2019-09-09T00:42:27Z-
dc.date.available2019-09-09T00:42:27Z-
dc.date.issued2019-09-05-
dc.identifier.citationJournal for Immunotherapy of Cancer 2019; 7(1): 241en
dc.identifier.urihttp://ahro.austin.org.au/austinjspui/handle/1/21703-
dc.description.abstractAutoimmune polyendocrine syndrome type II (APS-2) is a rare constellation of autoimmune hypoadrenalism, thyroid dysfunction and/or type 1 diabetes (T1DM), usually occurring in the 3rd or 4th decades and associated with a human leukocyte antigen (HLA) DR3 or DR4 serotype. We detail the first report of an elderly woman developing the full triad of APS-2 shortly after commencing anti-programmed cell death protein 1 (anti-PD1) immune checkpoint inhibition for unresectable melanoma and review the literature for similar presentations secondary to anti-PD1 axis therapy. A 78-year-old female with advanced unresectable BRAF wild-type melanoma was treated with pembrolizumab (2 mg/kg 3-weekly). Three weeks following the first dose she developed fulminant autoimmune diabetes, with an initially low C-peptide denoting rapid destruction of ß-islet cells. Following stabilisation of her diabetes, two further doses of pembrolizumab was administered. She then represented with symptomatic hypoadrenalism and hypothyroidism, consistent with APS-2. Her HLA class II genotype was HLA-DRB1*04.16 (DR4 serotype), a recognised association with this syndrome. Her melanoma responded rapidly to anti-PD1 therapy, and a complete response (CR) was attained after four doses of pembrolizumab. Treatment was discontinued and her CR is ongoing. This is the first report of the full triad of APS-2 developing in a genetically susceptible individual at the age of 78 after treatment with an anti-PD1 agent. Although scarcely reported, a literature review of similar reports seems to indicate a predilection for this syndrome in patients with HLA-DR4 serotypes. HLA Class II typing is not routinely recommended, but may provide useful predictive information for patients at risk of poly-endocrinopathy even in patients without a relevant personal or family history. Additional studies are required to determine if such testing would be useful and/or cost effective.en
dc.language.isoeng-
dc.subjectDiabetes mellitusen
dc.subjectHypoadrenalismen
dc.subjectHypothyroidismen
dc.subjectImmune checkpoint inhibitoren
dc.subjectPD1 inhibitoren
dc.subjectPembrolizumaben
dc.subjectPoly-endocrinopathyen
dc.titleAnti-programmed cell death protein 1 (anti-PD1) immunotherapy induced autoimmune polyendocrine syndrome type II (APS-2): a case report and review of the literature.en
dc.typeJournal Articleen
dc.identifier.journaltitleJournal for Immunotherapy of Canceren
dc.identifier.affiliationDepartment of Medical Oncology, Austin Health, Heidelberg, Victoria, Australiaen
dc.identifier.affiliationDepartment of Medicine, University of Melbourne, Melbourne, Australiaen
dc.identifier.affiliationSchool of Cancer Medicine, La Trobe University, Melbourne, Australiaen
dc.identifier.affiliationOlivia Newton-John Cancer Research Institute, Heidelberg, Victoria, Australiaen
dc.identifier.doi10.1186/s40425-019-0713-yen
dc.type.contentTexten
dc.identifier.orcid0000-0001-9713-1872en
dc.identifier.orcid0000-0002-3898-950Xen
dc.identifier.pubmedid31488221-
dc.type.austinJournal Article-
item.cerifentitytypePublications-
item.fulltextNo Fulltext-
item.grantfulltextnone-
item.openairecristypehttp://purl.org/coar/resource_type/c_18cf-
item.openairetypeJournal Article-
item.languageiso639-1en-
crisitem.author.deptMedical Oncology-
crisitem.author.deptMedical Oncology-
crisitem.author.deptMedical Oncology-
crisitem.author.deptOlivia Newton-John Cancer Research Institute-
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