{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,27]],"date-time":"2026-05-27T21:24:47Z","timestamp":1779917087864,"version":"3.53.1"},"reference-count":22,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2020,12,26]],"date-time":"2020-12-26T00:00:00Z","timestamp":1608940800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100000060","name":"National Institute of Allergy and Infectious Diseases","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000060","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Clinical Endocrinology"],"published-print":{"date-parts":[[2021,3]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Context<\/jats:title><jats:p>The monogenic disorder autoimmune polyendocrine syndrome type 1 (APS\u20101) or autoimmune polyendocrinopathy\u2010candidiasis\u2010ectodermal dystrophy (APECED) manifests frequently with hypoparathyroidism, which requires treatment with oral supplementation with calcium and active vitamin D analogs. The majority of APS\u20101\/APECED patients also suffer from intestinal malabsorption, which complicates the management of hypoparathyroidism and may lead to refractory severe hypocalcaemia. In such situations, reliance on intravenous calcium carries a high risk of nephrocalcinosis and renal damage.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Here, we report our experience of periprocedural subcutaneous administration of recombinant human parathyroid hormone (rhPTH 1\u201034) in APS\u20101\/APECED patients. Serum calcium was measured up to five times within the 36\u2010hour period starting the evening before the scheduled procedure and ending the morning following the procedure.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Twenty\u2010seven APS\u20101\/APECED patients with hypoparathyroidism (aged 4\u201067\u00a0years) underwent 31 invasive gastrointestinal and\/or pulmonary procedures. The patients received an average rhPTH1\u201034 dose of 9.6\u00a0\u00b1\u00a01.4\u00a0\u00b5g by subcutaneous injection. 92% of the adults and 54% of children in our cohort had evidence of nephrocalcinosis. Mean calcium levels remained stable and ranged from 2.06 to 2.17\u00a0mmol\/L with minimal fluctuation. None of our patients experienced periprocedural adverse events connected with hypocalcaemia.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>rhPTH 1\u201034 is an alternative to conventional therapy in patients with APS\u20101\/APECED and hypoparathyroidism undergoing invasive procedures. Subcutaneous PTH1\u201034 given directly before and after procedures resulted in well\u2010controlled serum calcium levels maintained in the low\u2010normal range and avoided the need for intravenous calcium which may contribute to renal calcifications and tubular damage.<\/jats:p><\/jats:sec>","DOI":"10.1111\/cen.14335","type":"journal-article","created":{"date-parts":[[2020,9,21]],"date-time":"2020-09-21T19:18:05Z","timestamp":1600715885000},"page":"377-383","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Impact of periprocedural subcutaneous parathyroid hormone on control of hypocalcaemia in APS\u20101\/APECED patients undergoing invasive procedures"],"prefix":"10.1111","volume":"94","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-8261-5188","authenticated-orcid":false,"given":"Karen K.","family":"Winer","sequence":"first","affiliation":[{"name":"Eunice Kennedy Shriver National Institutes of Child Health and Human Development (NICHD) NIH Bethesda MD USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Monica M.","family":"Schmitt","sequence":"additional","affiliation":[{"name":"Fungal Pathogenesis Section Laboratory of Clinical Immunology and Microbiology (LCIM) National Institute of Allergy and Infectious Diseases (NIAID) NIH Bethesda MD USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elise M. N.","family":"Ferre","sequence":"additional","affiliation":[{"name":"Fungal Pathogenesis Section Laboratory of Clinical Immunology and Microbiology (LCIM) National Institute of Allergy and Infectious Diseases (NIAID) NIH Bethesda MD USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kevin P.","family":"Fennelly","sequence":"additional","affiliation":[{"name":"Laboratory of Chronic Airway Infection Pulmonary Branch National Heart, Lung, and Blood Institute (NHLBI) Bethesda MD USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kenneth N.","family":"Olivier","sequence":"additional","affiliation":[{"name":"Laboratory of Chronic Airway Infection Pulmonary Branch National Heart, Lung, and Blood Institute (NHLBI) Bethesda MD USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Theo","family":"Heller","sequence":"additional","affiliation":[{"name":"Translational Hepatology Section Liver Diseases Branch National Institute of Diabetes and Digestive and Kidney Diseases (NIDDK) NIH Bethesda MD USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michail S.","family":"Lionakis","sequence":"additional","affiliation":[{"name":"Fungal Pathogenesis Section Laboratory of Clinical Immunology and Microbiology (LCIM) National Institute of Allergy and Infectious Diseases (NIAID) NIH Bethesda MD USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2020,12,26]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bone.2018.09.018"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrdp.2017.55"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00198-012-2230-4"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1111\/imr.12714"},{"key":"e_1_2_8_6_1","first-page":"1","article-title":"Redefined clinical features and diagnostic criteria in autoimmune 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