{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,20]],"date-time":"2026-05-20T20:07:00Z","timestamp":1779307620285,"version":"3.51.4"},"reference-count":31,"publisher":"Endocrinology Research Centre","issue":"2","license":[{"start":{"date-parts":[[2026,5,20]],"date-time":"2026-05-20T00:00:00Z","timestamp":1779235200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/www.probl-endojournals.ru\/jour\/about\/editorialPolicies#openAccessPolicy"}],"content-domain":{"domain":["www.probl-endojournals.ru"],"crossmark-restriction":true},"short-container-title":["Probl Endokrinol (Mosk)"],"abstract":"<jats:p>\n                    <jats:bold>BACKGROUND<\/jats:bold>\n                    : Hypoparathyroidism (HypoPT) and pseudohypoparathyroidism (PHP) are rare, predominantly genetically determined diseases in children with similar complications, characterized by hypocalcemia and hyperphosphatemia. PHP and HypoPT have similar complications: brain and lens calcification. In HypoPT, there is also an increased risk of developing nephrocalcinosis. Data on the frequency and structure of complications in affected children in Russia are limited, and comparative studies are lacking.\n                  <\/jats:p>\n                  <jats:p>\n                    <jats:bold>AIM<\/jats:bold>\n                    : To compare the prevalence of chronic complications and to identify factors associated with their development in children with PHP and congenital forms of HypoPT.\n                  <\/jats:p>\n                  <jats:p>\n                    <jats:bold>MATERIALS AND METHODS<\/jats:bold>\n                    : A retrospective study with a prospective component included 135 children with PHP and congenital forms of HypoPT. The results of laboratory and instrumental studies were evaluated.\n                  <\/jats:p>\n                  <jats:p>\n                    <jats:bold>RESULTS<\/jats:bold>\n                    : At least one complication associated with HypoPT\/PHP was identified in 82% of children. Nephrocalcinosis was most frequently detected in patients with autoimmune polyendocrine syndrome type 1 (APS-1) (67%) and autosomal dominant hypocalcemia type 1 (ADH1), and much less often in patients with PHP (22%) and unspecified forms of HypoPT (18%). Disease duration, as well as the duration of therapy with active vitamin D analogs and calcium supplements, was significantly longer in patients with nephrocalcinosis (p &lt; 0.001). An association between nephrocalcinosis and cataracts was established (p=0.005). Hypercalciuria persisted in 60% of patients despite medical compensation, was most prevalent in APS-1 and ADH1, and was not observed in PHP (p &lt; 0.001). No statistically significant relationship was identified between hypercalciuria and the development of nephrocalcinosis (p=0.567). A decrease in eGFR corresponding to CKD stage 2 was observed in 48.9% of patients. Fahr\u2019s syndrome was detected in 76% of cases, predominantly involving the basal ganglia, and the presence of mineral deposits was associated with hyperphosphatemia (p=0.010). Cataract and micronephrolithiasis were less frequent (18.6% and 3.9%, respectively), with no significant differences between nosological groups. The most common lens opacities observed were cortical (68%) and posterior subcapsular (41%). The duration of the disease was longer in patients with cataracts (p = 0.018).\n                  <\/jats:p>\n                  <jats:p>\n                    <jats:bold>CONCLUSION<\/jats:bold>\n                    : Among the complications observed in children, nephrocalcinosis, hypercalciuria, and Fahr\u2019s syndrome were the most frequent. Renal complications predominated in patients with APS-1 and ADH1. Hypercalciuria persisted despite optimal serum calcium levels in most patients.\n                  <\/jats:p>","DOI":"10.14341\/probl13704","type":"journal-article","created":{"date-parts":[[2026,5,20]],"date-time":"2026-05-20T19:22:08Z","timestamp":1779304928000},"page":"86-97","update-policy":"https:\/\/doi.org\/10.14341\/crossmarkpolicy2018","source":"Crossref","is-referenced-by-count":0,"title":["Complications of Pseudohypoparathyroidism and Congenital Forms of Hypoparathyroidism in Children"],"prefix":"10.14341","volume":"72","author":[{"ORCID":"https:\/\/orcid.org\/0009-0006-9482-5901","authenticated-orcid":true,"given":"A. A.","family":"Dzhamaludinova","sequence":"first","affiliation":[{"name":"Endocrinology Research Centre"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5650-1440","authenticated-orcid":true,"given":"L. S.","family":"Sozaeva","sequence":"additional","affiliation":[{"name":"Endocrinology Research Centre"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"N. V.","family":"Makazan","sequence":"additional","affiliation":[{"name":"Endocrinology Research Centre"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"A. A.","family":"Kolodkina","sequence":"additional","affiliation":[{"name":"Endocrinology Research Centre"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0434-9088","authenticated-orcid":true,"given":"K. S.","family":"Kulikova","sequence":"additional","affiliation":[{"name":"Endocrinology Research Centre; Research Center for Medical Genetics; \r\nRussian Children's Clinical Hospital, Pirogov RNIMU"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2000-7694","authenticated-orcid":true,"given":"N. Yu.","family":"Kalinchenko","sequence":"additional","affiliation":[{"name":"Endocrinology Research Centre"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3127-5974","authenticated-orcid":true,"given":"A. V.","family":"Bolmasova","sequence":"additional","affiliation":[{"name":"Endocrinology Research Centre"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6505-7548","authenticated-orcid":true,"given":"E. E.","family":"Novokreshennih","sequence":"additional","affiliation":[{"name":"Endocrinology Research Centre"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5507-4627","authenticated-orcid":true,"given":"V. A.","family":"Peterkova","sequence":"additional","affiliation":[{"name":"Endocrinology Research Centre"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9717-9742","authenticated-orcid":true,"given":"N. G.","family":"Mokrysheva","sequence":"additional","affiliation":[{"name":"Endocrinology Research Centre"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"5458","published-online":{"date-parts":[[2026,5,20]]},"reference":[{"key":"ref1","doi-asserted-by":"crossref","unstructured":"Powers J, Joy K, Ruscio A, Lagast H. Prevalence and incidence of hypoparathyroidism in the United States using a large claims database. J Bone Miner Res. 2013;28(12):2570-6. doi: https:\/\/doi.org\/10.1002\/jbmr.2004","DOI":"10.1002\/jbmr.2004"},{"key":"ref2","unstructured":"A Practical Approach to Hypocalcaemia in Children | Calcium and Bone Disorders in Children and Adolescents | Books Gateway | Karger Publishers [\u042d\u043b\u0435\u043a\u0442\u0440\u043e\u043d\u043d\u044b\u0439 \u0440\u0435\u0441\u0443\u0440\u0441]. URL: https:\/\/karger.com\/books\/book\/2660\/chapter-abstract\/5751562\/A-Practical-Approachto-Hypocalcaemia-in-Children?redirectedFrom=fulltext (\u0434\u0430\u0442\u0430 \u043e\u0431\u0440\u0430\u0449\u0435\u043d\u0438\u044f: 22.11.2023)"},{"key":"ref3","doi-asserted-by":"crossref","unstructured":"Takatani R., et al. Prevalence of Pseudohypoparathyroidism and Nonsurgical Hypoparathyroidism in Japan in 2017: A Nationwide Survey. Journal of Epidemiology. Japan Epidemiological Association, 2022","DOI":"10.2188\/jea.JE20220152"},{"key":"ref4","doi-asserted-by":"crossref","unstructured":"Underbjerg L, Sikjaer T, Mosekilde L, Rejnmark L. The Epidemiology of Nonsurgical Hypoparathyroidism in Denmark: A Nationwide Case Finding Study. J Bone Miner Res. 2015;30(9):1738-44. doi: https:\/\/doi.org\/10.1002\/jbmr.2501","DOI":"10.1002\/jbmr.2501"},{"key":"ref5","doi-asserted-by":"crossref","unstructured":"Underbjerg L, Sikjaer T, Mosekilde L, Rejnmark L. Pseudohypoparathyroidism - epidemiology, mortality and risk of complications. Clin Endocrinol (Oxf ). 2016;84(6):904-11. doi: https:\/\/doi.org\/10.1111\/cen.12948","DOI":"10.1111\/cen.12948"},{"key":"ref6","doi-asserted-by":"crossref","unstructured":"Zavatta G, Clarke BL. Basal ganglia calcification in hypoparathyroidism and pseudohypoparathyroidism: local and systemic metabolic mechanisms. J Endocrinol Invest. 2021;44(2):245-253. doi: https:\/\/doi.org\/10.1007\/s40618-020-01355-w","DOI":"10.1007\/s40618-020-01355-w"},{"key":"ref7","doi-asserted-by":"crossref","unstructured":"Hansen DW, Nebesio TD, DiMeglio LA, Eugster EA, Imel EA. Prevalence of Nephrocalcinosis in Pseudohypoparathyroidism: Is Screening Necessary? J Pediatr. 2018;199:263-266. doi: https:\/\/doi.org\/10.1016\/j.jpeds.2018.03.003","DOI":"10.1016\/j.jpeds.2018.03.003"},{"key":"ref8","doi-asserted-by":"crossref","unstructured":"Shoback DM, Bilezikian JP, Costa AG, Dempster D, Dralle H, et al. Presentation of Hypoparathyroidism: Etiologies and Clinical Features. J Clin Endocrinol Metab. 2016;101(6):2300-12. doi: https:\/\/doi.org\/10.1210\/jc.2015-3909","DOI":"10.1210\/jc.2015-3909"},{"key":"ref9","doi-asserted-by":"crossref","unstructured":"Levy I, Licht C, Daneman A, Sochett E, Harrington J. The Impact of Hypoparathyroidism Treatment on the Kidney in Children: Long-Term Retrospective Follow-Up Study. J Clin Endocrinol Metab. 2015;100(11):4106-13. doi: https:\/\/doi.org\/10.1210\/jc.2015-2257","DOI":"10.1210\/jc.2015-2257"},{"key":"ref10","unstructured":"Makazan NV. Rol' narushenij postreceptornogo signalinga v razvitii mul'tigormonal'noj rezistentnrosti i avtonomnoj giperfunkcii endokrinnyh zhelez u detej : dis. kand.med. nauk; FGBU \"NMIC endokrinologii\" Minzdrava Rossii, 2017 - 158 s. (In Russ.)"},{"key":"ref11","unstructured":"Orlova EM. Autoimmunnyj poliglandulyarnyj sindrom 1 tipa: klinicheskie varianty, geneticheskie osnovy, immunologicheskie markery, lechenie i prognoz : dis. d-ra med. nauk; FGBU \"NMIC endokrinologii\" Minzdrava Rossii, 2017 -319 s. (In Russ.)"},{"key":"ref12","doi-asserted-by":"crossref","unstructured":"Colantonio DA, Kyriakopoulou L, Chan MK, Daly CH, Brinc D, Venner AA, Pasic MD, Armbruster D, Adeli K. Closing the gaps in pediatric laboratory reference intervals: a CALIPER database of 40 biochemical markers in a healthy and multiethnic population of children. Clin Chem. 2012;58(5):854-68. doi: https:\/\/doi.org\/10.1373\/clinchem.2011.177741","DOI":"10.1373\/clinchem.2011.177741"},{"key":"ref13","doi-asserted-by":"crossref","unstructured":"Schwartz GJ, Mu\u00f1oz A, Schneider MF, Mak RH, Kaskel F, Warady BA, Furth SL. New equations to estimate GFR in children with CKD. J Am Soc Nephrol. 2009;20(3):629-37. doi: https:\/\/doi.org\/10.1681\/ASN.2008030287","DOI":"10.1681\/ASN.2008030287"},{"key":"ref14","doi-asserted-by":"crossref","unstructured":"Stevens PE, Levin A; Kidney Disease: Improving Global Outcomes Chronic Kidney Disease Guideline Development Work Group Members. Evaluation and management of chronic kidney disease: synopsis of the kidney disease: improving global outcomes 2012 clinical practice guideline. Ann Intern Med. 2013;158(11):825-30. doi: https:\/\/doi.org\/10.7326\/0003-4819-158-11-201306040-00007","DOI":"10.7326\/0003-4819-158-11-201306040-00007"},{"key":"ref15","doi-asserted-by":"crossref","unstructured":"Gosmanova EO, Ayodele O, Chen K, Cook EE, Mu F, Young JA, Rejnmark L. Association of Calcium and Phosphate Levels with Incident Chronic Kidney Disease in Patients with Hypoparathyroidism: A Retrospective Case-Control Study. Int J Endocrinol. 2022;2022:6078881. doi: https:\/\/doi.org\/10.1155\/2022\/6078881","DOI":"10.1155\/2022\/6078881"},{"key":"ref16","doi-asserted-by":"crossref","unstructured":"Swartling O, Evans M, Spelman T, Kamal W, K\u00e4mpe O, et al. Kidney Complications and Hospitalization in Patients With Chronic Hypoparathyroidism: A Cohort Study in Sweden. J Clin Endocrinol Metab. 2022;107(10):e4098-e4105. doi: https:\/\/doi.org\/10.1210\/clinem\/dgac456","DOI":"10.1210\/clinem\/dgac456"},{"key":"ref17","doi-asserted-by":"crossref","unstructured":"Rejnmark L., et al. CALIBRATE: A phase 3, randomized, open-label study evaluating the efficacy and safety of encaleret (CLTX-305) compared to standard of care in participants with autosomal dominant hypocalcemia type 1 [NCT05680818] Endocrine Abstracts. Bioscientifica, 2024;99","DOI":"10.1530\/endoabs.99.OC2.6"},{"key":"ref18","doi-asserted-by":"crossref","unstructured":"Clarke BL, Khan AA, Rubin MR, Schwarz P, Vokes T, Shoback DM, et al. Efficacy and Safety of TransCon PTH in Adults With Hypoparathyroidism: 52-Week Results From the Phase 3 PaTHway Trial. J Clin Endocrinol Metab. 2025;110(4):951-960. doi: https:\/\/doi.org\/10.1210\/clinem\/dgae693. Erratum in: J Clin Endocrinol Metab. 2025;110(4):e1292-e1293. doi: https:\/\/doi.org\/10.1210\/clinem\/dgaf048","DOI":"10.1210\/clinem\/dgaf048"},{"key":"ref19","doi-asserted-by":"crossref","unstructured":"De Coster T, David K, Breckpot J, Decallonne B. Management of autosomal dominant hypocalcemia type 1: Literature review and clinical practice recommendations. J Endocrinol Invest. 2025;48(4):831-844. doi: https:\/\/doi.org\/10.1007\/s40618-024-02496-y","DOI":"10.1007\/s40618-024-02496-y"},{"key":"ref20","doi-asserted-by":"crossref","unstructured":"Kovaleva EV, Eremkina AK, Mokrysheva NG. Daily calcium profile in diagnosis of hypo- and hypercalcemia in patients with chronic hypoparathyroidism. clinical case series. Obesity and metabolism. 2021;18(2):175-179. (In Russ.). doi: https:\/\/doi.org\/10.14341\/omet12729","DOI":"10.14341\/omet12729"},{"key":"ref21","doi-asserted-by":"crossref","unstructured":"Meola A. \u0438 \u0434\u0440. Efficacy and safety of long-term management of patients with chronic post-surgical hypoparathyroidism \/\/ J Endocrinol Invest. 2018. \u0422. 41, \u2116 10. \u0421. 1221\u20131226. Meola A, Vignali E, Matrone A, Cetani F, Marcocci C. Efficacy and safety of long-term management of patients with chronic post-surgical hypoparathyroidism. J Endocrinol Invest. 2018 ;41(10):1221-1226. doi: https:\/\/doi.org\/10.1007\/s40618-018-0857-5","DOI":"10.1007\/s40618-018-0857-5"},{"key":"ref22","doi-asserted-by":"crossref","unstructured":"Mazoni L, Matrone A, Apicella M, Saponaro F, Borsari S, et al. Renal complications and quality of life in postsurgical hypoparathyroidism: a case-control study. J Endocrinol Invest. 2022;45(3):573-582. doi: https:\/\/doi.org\/10.1007\/s40618-021-01686-2","DOI":"10.1007\/s40618-021-01686-2"},{"key":"ref23","doi-asserted-by":"crossref","unstructured":"Underbjerg L, Sikjaer T, Mosekilde L, Rejnmark L. Cardiovascular and renal complications to postsurgical hypoparathyroidism: a Danish nationwide controlled historic follow-up study. J Bone Miner Res. 2013;28(11):2277-85. doi: https:\/\/doi.org\/10.1002\/jbmr.1979","DOI":"10.1002\/jbmr.1979"},{"key":"ref24","doi-asserted-by":"crossref","unstructured":"Kovaleva EV, et al. Complications of chronic hypoparathyroidism according to analysis database Russian Registry. Clinical and experimental thyroidology. 2024;20(1):30-40. (In Russ.). doi: https:\/\/doi.org\/10.14341\/ket12792","DOI":"10.14341\/ket12792"},{"key":"ref25","doi-asserted-by":"crossref","unstructured":"Zavatta G, Clarke BL. Basal ganglia calcification in hypoparathyroidism and pseudohypoparathyroidism: local and systemic metabolic mechanisms. J Endocrinol Invest. 2021 Feb;44(2):245-253. doi: https:\/\/doi.org\/10.1007\/s40618-020-01355-w","DOI":"10.1007\/s40618-020-01355-w"},{"key":"ref26","doi-asserted-by":"crossref","unstructured":"Goswami R, Sharma R, Sreenivas V, Gupta N, Ganapathy A, Das S. Prevalence and progression of basal ganglia calcification and its pathogenic mechanism in patients with idiopathic hypoparathyroidism. Clin Endocrinol (Oxf ). 2012;77(2):200-6. doi: https:\/\/doi.org\/10.1111\/j.1365-2265.2012.04353.x","DOI":"10.1111\/j.1365-2265.2012.04353.x"},{"key":"ref27","doi-asserted-by":"crossref","unstructured":"Wang C, Li Y, Shi L, Ren J, Patti M, Wang T, de Oliveira JR, Sobrido MJ, et al. Mutations in SLC20A2 link familial idiopathic basal ganglia calcification with phosphate homeostasis. Nat Genet. 2012;44(3):254-6. doi: https:\/\/doi.org\/10.1038\/ng.1077","DOI":"10.1038\/ng.1077"},{"key":"ref28","doi-asserted-by":"crossref","unstructured":"Savazzi GM, Cusmano F, Musini S. Cerebral imaging changes in patients with chronic renal failure treated conservatively or in hemodialysis. Nephron. 2001;89(1):31-6. doi: https:\/\/doi.org\/10.1159\/000046040","DOI":"10.1159\/000046040"},{"key":"ref29","doi-asserted-by":"crossref","unstructured":"Roszko KL, Stapleton Smith LM, Sridhar AV, Roberts MS, Hartley IR, Gafni RI, Collins MT, Fox JC, Nemeth EF. Autosomal Dominant Hypocalcemia Type 1: A Systematic Review. J Bone Miner Res. 2022;37(10):1926-1935. doi: https:\/\/doi.org\/10.1002\/jbmr.4659","DOI":"10.1002\/jbmr.4659"},{"key":"ref30","doi-asserted-by":"crossref","unstructured":"Underbjerg L, Sikjaer T, Mosekilde L, Rejnmark L. Postsurgical hypoparathyroidism--risk of fractures, psychiatric diseases, cancer, cataract, and infections. J Bone Miner Res. 2014;29(11):2504-10. doi: https:\/\/doi.org\/10.1002\/jbmr.2273","DOI":"10.1002\/jbmr.2273"},{"key":"ref31","doi-asserted-by":"crossref","unstructured":"Cherchir F, Oueslati I, Yazidi M, Chaker F, Mizouni H, Feki M, Chihaoui M. Long-term complications of permanent hypoparathyroidism in adults: prevalence and associated factors. Endocrine. 2024;84(3):1164-1171. doi: https:\/\/doi.org\/10.1007\/s12020-024-03765-9","DOI":"10.1007\/s12020-024-03765-9"}],"container-title":["Problems of Endocrinology"],"original-title":[],"link":[{"URL":"https:\/\/www.probl-endojournals.ru\/jour\/article\/viewFile\/13704\/10992","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2026,5,20]],"date-time":"2026-05-20T19:22:34Z","timestamp":1779304954000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.probl-endojournals.ru\/jour\/article\/view\/13704"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,5,20]]},"references-count":31,"journal-issue":{"issue":"2","published-online":{"date-parts":[[2026,5,20]]}},"URL":"https:\/\/doi.org\/10.14341\/probl13704","relation":{},"ISSN":["2308-1430","0375-9660"],"issn-type":[{"value":"2308-1430","type":"electronic"},{"value":"0375-9660","type":"print"}],"subject":[],"published":{"date-parts":[[2026,5,20]]}}}