{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,1]],"date-time":"2026-10-01T04:30:11Z","timestamp":1790829011393,"version":"4.1.0"},"reference-count":26,"publisher":"Walter de Gruyter GmbH","issue":"4","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2022,4,26]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec id=\"j_jpem-2022-0050_abs_001\">\n                  <jats:title>Objectives<\/jats:title>\n                  <jats:p>Polyglandular autoimmune syndromes (PAS) are characterized by the association of two or more autoimmune diseases (AID) and are classified into four types. PAS type 1 is more frequently manifested in childhood, but the prevalence of other PAS in children, less described in the literature, seems to be underestimated.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_jpem-2022-0050_abs_002\">\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>This study aimed to evaluate the prevalence of PAS in a selected pediatric population of 879 children with Diabetes mellitus type 1 (DM1), autoimmune thyroid disease (AITD), and Addison\u2019s disease (AD) followed in our hospital for 10 years and describe and classify the manifestations of different PAS.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_jpem-2022-0050_abs_003\">\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>We diagnosed 35 children with PAS, most fulfilled criteria for PAS type 3 (65.7%), and AITD was the AID more frequently detected (74.3%). PAS type 1 was not diagnosed in our sample. Patients with PAS manifested DM1 and AITD at a younger age than children with monoglandular pathology (7.7 vs. 9.3 years, p=0.04 and 7.7 vs. 13.1 years, p&lt;0.01).<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_jpem-2022-0050_abs_004\">\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>This is the first study that analyzes the prevalence of different types of PAS in a pediatric population followed by endocrine pathologies, namely DM1, AD, and AITD. As PAS manifestations are often preceded by a long latency period characterized by the presence of autoantibodies, we reinforce the need to value these markers for timely diagnosis and to screen PAS in patients with AD throughout their lives.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1515\/jpem-2022-0050","type":"journal-article","created":{"date-parts":[[2022,2,16]],"date-time":"2022-02-16T16:05:37Z","timestamp":1645027537000},"page":"477-480","source":"Crossref","is-referenced-by-count":9,"title":["Clinical characteristics of polyglandular autoimmune syndromes in pediatric age: an observational study"],"prefix":"10.1515","volume":"35","author":[{"given":"Bruno","family":"Bou\u00e7a","sequence":"first","affiliation":[{"name":"Endocrinology, Diabetes and Metabolism Department , Centro Hospitalar Universit\u00e1rio Lisboa Central , Lisbon , Portugal"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andreia","family":"Nogueira","sequence":"additional","affiliation":[{"name":"Pediatrics Department , Centro Hospitalar Universit\u00e1rio de Coimbra , Coimbra , Portugal"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joana","family":"Caetano","sequence":"additional","affiliation":[{"name":"Pediatric Endocrinology Diabetes and Growth Department , Centro Hospitalar Universit\u00e1rio de Coimbra , Coimbra , Portugal"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rita","family":"Cardoso","sequence":"additional","affiliation":[{"name":"Pediatric Endocrinology Diabetes and Growth Department , Centro Hospitalar Universit\u00e1rio de Coimbra , Coimbra , Portugal"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Isabel","family":"Dinis","sequence":"additional","affiliation":[{"name":"Pediatric Endocrinology Diabetes and Growth Department , Centro Hospitalar Universit\u00e1rio de Coimbra , Coimbra , Portugal"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alice","family":"Mirante","sequence":"additional","affiliation":[{"name":"Pediatric Endocrinology Diabetes and Growth Department , Centro Hospitalar Universit\u00e1rio de Coimbra , Coimbra , Portugal"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"374","published-online":{"date-parts":[[2022,2,16]]},"reference":[{"key":"2023033123535380557_j_jpem-2022-0050_ref_001","doi-asserted-by":"crossref","unstructured":"Husebye, ES, Anderson, MS, K\u00e4mpe, O. Autoimmune polyendocrine syndromes. N Engl J Med 2018;378:1132\u201341. https:\/\/doi.org\/10.1056\/nejmra1713301.","DOI":"10.1056\/NEJMra1713301"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_002","doi-asserted-by":"crossref","unstructured":"Pham-Dobor, G, Han\u00e1k, L, Hegyi, P, M\u00e1rta, K, P\u00e1rniczky, A, Gergics, M. Prevalence of other autoimmune diseases in polyglandular autoimmune syndromes type II and III. J Endocrinol Invest 2020;43:1\u20139. https:\/\/doi.org\/10.1007\/s40618-020-01229-1.","DOI":"10.1007\/s40618-020-01229-1"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_003","unstructured":"Neufeld, M, Blizzard, RM. Polyglandular autoimmune disease. In: Pinchera, A, Doniach, D, Fenzi, DF, Baschieri, L, editors. Autoimmune aspects of endocrine disorders. London, UK: Academic Press; 1980:357\u201365 pp."},{"key":"2023033123535380557_j_jpem-2022-0050_ref_004","unstructured":"Betterle, C, Zanchetta, R. Update on autoimmune polyendocrine syndromes (APS). Acta Biomed 2003;74:9\u201333."},{"key":"2023033123535380557_j_jpem-2022-0050_ref_005","doi-asserted-by":"crossref","unstructured":"Schneller, C, Finkel, L, Wise, M, Hageman, JR, Littlejohn, E. Autoimmune polyendocrine syndrome: a case-based review. Pediatr Ann 2013;42:203\u20138. https:\/\/doi.org\/10.3928\/00904481-20130426-12.","DOI":"10.3928\/00904481-20130426-12"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_006","doi-asserted-by":"crossref","unstructured":"Renzullo, A, Accardo, G, Esposito, D, De Bellis, A, Bizzarro, A, Romano, M. Hashimoto\u2019s thyroiditis and entero-chromaffin-like cell hyperplasia: early detection and somatostatin analogue Treatment. Eur J Inflamm 2013;11:863\u201370. https:\/\/doi.org\/10.1177\/1721727X1301100329.","DOI":"10.1177\/1721727X1301100329"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_007","doi-asserted-by":"crossref","unstructured":"Bain, A, Stewart, M, Mwamure, P, Nirmalaraj, K. Addison\u2019s disease in a patient with hypothyroidism: Autoimmune polyglandular syndrome type 2. In: BMJ Case Reports; 2015, 2015:2\u20134 pp.","DOI":"10.1136\/bcr-2015-210506"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_008","doi-asserted-by":"crossref","unstructured":"Cutolo, M. Autoimmune polyendocrine syndromes. Autoimmun Rev 2014;13:85\u20139. https:\/\/doi.org\/10.1016\/j.autrev.2013.07.006.","DOI":"10.1016\/j.autrev.2013.07.006"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_009","doi-asserted-by":"crossref","unstructured":"Kahaly, GJ. \u2018Polyglandular autoimmune syndrome type II\u2019. Presse Med 2012;41:e663\u2013e670. https:\/\/doi.org\/10.1016\/j.lpm.2012.09.011.","DOI":"10.1016\/j.lpm.2012.09.011"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_010","doi-asserted-by":"crossref","unstructured":"Bahceci, M, Tuzcu, A, Pasa, S, Ayyildiz, O, Tuzcu, S. Polyglandular autoimmune syndrome type III accompanied by common variable immunodeficiency. Gynecol Endocrinol 2004;19:47\u201350. https:\/\/doi.org\/10.1080\/09513590410001725279.","DOI":"10.1080\/09513590410001725279"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_011","doi-asserted-by":"crossref","unstructured":"Turkoglu, Z, Kavala, M, Kolcak, O, Zindanci, I, Can, B. Autoimmune polyglandular syndrome-3C in a child. Dermatol Online J 2010;16:8. https:\/\/doi.org\/10.5070\/d324p9n474.","DOI":"10.5070\/D324P9N474"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_012","unstructured":"Zirilli, G, Santucci, S, Cuzzup\u00e8, C, Corica, D, Pitrolo, E, Salzano, G. Peculiarities of autoimmune polyglandular syndromes in children and adolescents. Acta Biomed 2017;88:271\u20135. https:\/\/doi.org\/10.23750\/abm.v88i3.5898."},{"key":"2023033123535380557_j_jpem-2022-0050_ref_013","doi-asserted-by":"crossref","unstructured":"Kahaly, GJ, Frommer, L. Polyglandular autoimmune syndromes. J\u00a0Endocrinol Invest 2018;41:91\u20138. https:\/\/doi.org\/10.1007\/s40618-017-0740-9.","DOI":"10.1007\/s40618-017-0740-9"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_014","doi-asserted-by":"crossref","unstructured":"Valenzise, M, Aversa, T, Saccomanno, A, De Luca, F, Salzano, G. Epidemiological and clinical peculiarities of polyglandular syndrome type 3 in pediatric age\u2019. Ital J Pediatr 2017;43:4\u20137. https:\/\/doi.org\/10.1186\/s13052-017-0386-4.","DOI":"10.1186\/s13052-017-0386-4"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_015","doi-asserted-by":"crossref","unstructured":"Ruggeri, RM, Trimarchi, F, Giuffrida, G, Certo, R, Cama, E, Campenn\u00ec, A. Autoimmune comorbidities in Hashimoto\u2019s thyroiditis: different patterns of association in adulthood and childhood\/adolescence. Eur J Endocrinol 2017;176:133\u201341. https:\/\/doi.org\/10.1530\/eje-16-0737.","DOI":"10.1530\/EJE-16-0737"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_016","doi-asserted-by":"crossref","unstructured":"Aversa, T, Corica, D, Zirilli, G, Pajno, GB, Salzano, G, De Luca, F. Phenotypic expression of autoimmunity in children with autoimmune thyroid disorders. Front Endocrinol 2019;10:1\u20137. https:\/\/doi.org\/10.3389\/fendo.2019.00476.","DOI":"10.3389\/fendo.2019.00476"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_017","doi-asserted-by":"crossref","unstructured":"Kahaly, GJ. Polyglandular autoimmune syndromes. Eur J Endocrinol 2009;161:11\u201320. https:\/\/doi.org\/10.1530\/eje-09-0044.","DOI":"10.1530\/EJE-09-0044"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_018","doi-asserted-by":"crossref","unstructured":"Robles, DT, Fain, PR, Gottlieb, PA, Eisenbarth, GS. The genetics of autoimmune polyendocrine syndrome type II. Endocrinol Metab Clin N Am 2002;31:353\u201368. https:\/\/doi.org\/10.1016\/s0889-8529(01)00015-9.","DOI":"10.1016\/S0889-8529(01)00015-9"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_019","doi-asserted-by":"crossref","unstructured":"Maclaren, N. Addison\u2019s disease 2001. J Clin Endocrinol Metab 2001;86:2909\u201322.","DOI":"10.1210\/jcem.86.7.7636"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_020","doi-asserted-by":"crossref","unstructured":"Quinkler, M. Addison\u2019s disease. Med Klin Intensivmed Notfallmed 2012;107:454\u20139. https:\/\/doi.org\/10.1007\/s00063-012-0112-3.","DOI":"10.1007\/s00063-012-0112-3"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_021","doi-asserted-by":"crossref","unstructured":"Schatz, DA, Winter, WE. Autoimmune polyglandular syndrome II: clinical syndrome and treatment. Endocrinol Metab Clin N Am 2002;31:339\u201352. https:\/\/doi.org\/10.1016\/s0889-8529(01)00012-3.","DOI":"10.1016\/S0889-8529(01)00012-3"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_022","doi-asserted-by":"crossref","unstructured":"Fichna, M, Fichna, P, Gryczy\u0144ska, M, Walkowiak, J, Zurawek, M, Sowi\u0144ski, J. Screening for associated autoimmune disorders in Polish patients with Addison\u2019s disease. Endocrine 2010;37:349\u201360. https:\/\/doi.org\/10.1007\/s12020-010-9312-x.","DOI":"10.1007\/s12020-010-9312-x"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_023","doi-asserted-by":"crossref","unstructured":"Zelissen, PMJ, Bast, EJEG, Croughs, RJM. Associated autoimmunity in addison\u2019s disease. J Autoimmun 1995;8:121\u201330. https:\/\/doi.org\/10.1006\/jaut.1995.0009.","DOI":"10.1006\/jaut.1995.0009"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_024","doi-asserted-by":"crossref","unstructured":"Neufeld, M, Maclaren, N, Blizzard, R. Autoimmune polyglandular syndromes. Pediatr Ann 1980;9:154\u201362. https:\/\/doi.org\/10.3928\/0090-4481-19800401-07.","DOI":"10.3928\/0090-4481-19800401-07"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_025","doi-asserted-by":"crossref","unstructured":"Dittmar, M, Kahaly, GJ. Polyglandular autoimmune syndromes: immunogenetics and long-term follow-up. J Clin Endocrinol Metab 2003;88:2983\u201392. https:\/\/doi.org\/10.1210\/jc.2002-021845.","DOI":"10.1210\/jc.2002-021845"},{"key":"2023033123535380557_j_jpem-2022-0050_ref_026","doi-asserted-by":"crossref","unstructured":"Salardi, S, Volta, U, Zucchini, S, Fiorini, E, Maltoni, G, Vaira, B. Prevalence of celiac disease in children with type 1 diabetes mellitus increased in the mid-1990s: an 18-year longitudinal study based on anti-endomysial antibodies. J\u00a0Pediatr Gastroenterol Nutr 2008;46:612\u20134. https:\/\/doi.org\/10.1097\/mpg.0b013e31815d697e.","DOI":"10.1097\/MPG.0b013e31815d697e"}],"container-title":["Journal of Pediatric Endocrinology and Metabolism"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.degruyter.com\/document\/doi\/10.1515\/jpem-2022-0050\/xml","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/www.degruyter.com\/document\/doi\/10.1515\/jpem-2022-0050\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,4,1]],"date-time":"2023-04-01T13:11:50Z","timestamp":1680354710000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.degruyter.com\/document\/doi\/10.1515\/jpem-2022-0050\/html"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,2,16]]},"references-count":26,"journal-issue":{"issue":"4","published-online":{"date-parts":[[2022,2,16]]},"published-print":{"date-parts":[[2022,4,26]]}},"alternative-id":["10.1515\/jpem-2022-0050"],"URL":"https:\/\/doi.org\/10.1515\/jpem-2022-0050","relation":{},"ISSN":["0334-018X","2191-0251"],"issn-type":[{"value":"0334-018X","type":"print"},{"value":"2191-0251","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,2,16]]}}}