{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,10]],"date-time":"2026-06-10T14:39:11Z","timestamp":1781102351057,"version":"3.54.1"},"reference-count":27,"publisher":"The Endocrine Society","issue":"5","license":[{"start":{"date-parts":[[2024,6,28]],"date-time":"2024-06-28T00:00:00Z","timestamp":1719532800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100011597","name":"Italian Ministry of Health to Fondazione","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100011597","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,4,22]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Context<\/jats:title>\n                  <jats:p>Pseudohypoparathyroidism (PHP) refers to a group of rare hereditary disorders associated with resistance to parathyroid hormone (PTH) and other hormones now termed inactivating PTH\/PTHrP disorders (iPPSD). Hypercalcitoninemia has been seldom reported in small series.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Objective<\/jats:title>\n                  <jats:p>Our aim was to investigate the characteristics of hypercalcitoninemia in pediatric and adult patients with PHP\/iPPSD.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>We retrospectively collected data from 2 cohorts from 2 European endocrinology tertiary centers: the pediatric cohort comprised 88 children with available calcitonin (CT) measurements; the adult cohort included 43 individuals with simultaneous CT and PTH measurements.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>In the pediatric cohort, 65.9% had hypercalcitoninemia (median CT 15 ng\/L); in the adult cohort 53.5% (mean CT 21.6 ng\/L). There was no difference between CT in pediatric and adult population; we observed stable CT levels over a median follow-up of 134.5 months in adults. Notably, no correlations were detected between CT and PTH levels. Other etiologies of hypercalcitoninemia were excluded; adult patients underwent regular thyroid ultrasound to screen for medullary thyroid cancer (MTC). We performed 20 calcium stimulation tests in adult patients. While there was a significant difference in basal and peak CT between our patients, healthy subjects, and subjects with MTC, there was no difference with patients with C-cell hyperplasia.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusion<\/jats:title>\n                  <jats:p>This study underscores the common occurrence of hypercalcitoninemia in both pediatric and adult patients with PHP\/iPPSD, in particular with subtypes iPPSD2 and iPPSD3. Furthermore, these patients show hyperresponsiveness to calcium stimulation tests falling between healthy subjects and patients with MTC. These findings contribute to the understanding of CT dynamics in the context of PHP\/iPPSD.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1210\/clinem\/dgae416","type":"journal-article","created":{"date-parts":[[2024,6,28]],"date-time":"2024-06-28T12:01:12Z","timestamp":1719576072000},"page":"1334-1341","source":"Crossref","is-referenced-by-count":1,"title":["High Prevalence of Hypercalcitoninemia in a Large Cohort of Adult and Pediatric Patients With PTH Resistance Syndromes"],"prefix":"10.1210","volume":"110","author":[{"ORCID":"https:\/\/orcid.org\/0009-0003-2177-8014","authenticated-orcid":false,"given":"Arianna","family":"Cremaschi","sequence":"first","affiliation":[{"name":"Endocrinology Unit, Fondazione IRCCS Ca\u2019 Granda Ospedale Maggiore Policlinico , 20122 Milan ,","place":["Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9050-0150","authenticated-orcid":false,"given":"Giulia","family":"Del Sindaco","sequence":"additional","affiliation":[{"name":"Endocrinology Unit, Fondazione IRCCS Ca\u2019 Granda Ospedale Maggiore Policlinico , 20122 Milan ,","place":["Italy"]},{"name":"Department of Clinical Sciences and Community Health, University of Milan , 20122 Milan ,","place":["Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Angela","family":"Pagnano","sequence":"additional","affiliation":[{"name":"Endocrinology Unit, Fondazione IRCCS Ca\u2019 Granda Ospedale Maggiore Policlinico , 20122 Milan ,","place":["Italy"]},{"name":"Department of Clinical Sciences and Community Health, University of Milan , 20122 Milan ,","place":["Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alessia","family":"Dolci","sequence":"additional","affiliation":[{"name":"Endocrinology Unit, Fondazione IRCCS Ca\u2019 Granda Ospedale Maggiore Policlinico , 20122 Milan ,","place":["Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jugurtha","family":"Berkenou","sequence":"additional","affiliation":[{"name":"AP-HP, Service d\u2019endocrinologie et diab\u00e8te de l\u2019enfant, H\u00f4pital Bic\u00eatre Paris Saclay , 94270 Le Kremlin Bic\u00eatre ,","place":["France"]},{"name":"AP-HP, Centre de R\u00e9f\u00e9rence des maladies rares du m\u00e9tabolisme du Calcium et du Phosphate, fili\u00e8re OSCAR, ERN-BOND, ERN for rare endocrine disorders, Platforme d\u2019expertise des maladies rares Paris Saclay , Paris ,","place":["France"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anya","family":"Rothenbuhler","sequence":"additional","affiliation":[{"name":"AP-HP, Service d\u2019endocrinologie et diab\u00e8te de l\u2019enfant, H\u00f4pital Bic\u00eatre Paris Saclay , 94270 Le Kremlin Bic\u00eatre ,","place":["France"]},{"name":"AP-HP, Centre de R\u00e9f\u00e9rence des maladies rares du m\u00e9tabolisme du Calcium et du Phosphate, fili\u00e8re OSCAR, ERN-BOND, ERN for rare endocrine disorders, Platforme d\u2019expertise des maladies rares Paris Saclay , Paris ,","place":["France"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9300-8516","authenticated-orcid":false,"given":"Andrea","family":"Contarino","sequence":"additional","affiliation":[{"name":"Endocrinology Unit, Fondazione IRCCS Ca\u2019 Granda Ospedale Maggiore Policlinico , 20122 Milan ,","place":["Italy"]},{"name":"Department of Clinical Sciences and Community Health, University of Milan , 20122 Milan ,","place":["Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0556-7650","authenticated-orcid":false,"given":"Emanuele","family":"Ferrante","sequence":"additional","affiliation":[{"name":"Endocrinology Unit, Fondazione IRCCS Ca\u2019 Granda Ospedale Maggiore Policlinico , 20122 Milan ,","place":["Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3988-3616","authenticated-orcid":false,"given":"Maura","family":"Arosio","sequence":"additional","affiliation":[{"name":"Endocrinology Unit, Fondazione IRCCS Ca\u2019 Granda Ospedale Maggiore Policlinico , 20122 Milan ,","place":["Italy"]},{"name":"Department of Clinical Sciences and Community Health, University of Milan , 20122 Milan ,","place":["Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elisa","family":"Giannetta","sequence":"additional","affiliation":[{"name":"Department of Experimental Medicine, Sapienza University of Rome , 00161 Rome ,","place":["Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3455-002X","authenticated-orcid":false,"given":"Agn\u00e8s","family":"Linglart","sequence":"additional","affiliation":[{"name":"AP-HP, Service d\u2019endocrinologie et diab\u00e8te de l\u2019enfant, H\u00f4pital Bic\u00eatre Paris Saclay , 94270 Le Kremlin Bic\u00eatre ,","place":["France"]},{"name":"AP-HP, Centre de R\u00e9f\u00e9rence des maladies rares du m\u00e9tabolisme du Calcium et du Phosphate, fili\u00e8re OSCAR, ERN-BOND, ERN for rare endocrine disorders, Platforme d\u2019expertise des maladies rares Paris Saclay , Paris ,","place":["France"]},{"name":"Universit\u00e9 Paris Saclay, INSERM, U1185 , 94270 Le Kremlin Bic\u00eatre ,","place":["France"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9065-3886","authenticated-orcid":false,"given":"Giovanna","family":"Mantovani","sequence":"additional","affiliation":[{"name":"Endocrinology Unit, Fondazione IRCCS Ca\u2019 Granda Ospedale Maggiore Policlinico , 20122 Milan ,","place":["Italy"]},{"name":"Department of Clinical Sciences and Community Health, University of Milan , 20122 Milan 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