The Journal of Clinical Endocrinology & Metabolism · 2016 · 238 citations · 47 references
Life SciencesEndocrinologyGeneticsAutoimmunityEndocrine GeneticsMedical GeneticsAutoimmune DiseasesEndocrine DiseaseGenetic DisordersInherited Endocrine SyndromesParathyroid DisordersParathyroid GeneticsAutoimmune Polyendocrine Syndrome Type 1Autoimmune Polyendocrine SyndromesDigeorge SyndromeGenetic Syndromes Of HyperparathyroidismHospital-based RegistriesMedian AgePrevalence EstimationSyndromic Hypoparathyroidism
OBJECTIVE: The epidemiology of hypoparathyroidism (HP) is largely unknown. We aimed to determine prevalence, etiologies, health related quality of life (HRQOL) and treatment pattern of HP. METHODS: Patients with HP and 22q11 deletion syndrome (DiGeorge syndrome) were identified in electronic hospital registries. All identified patients were invited to participate in a survey. Among patients who responded, HRQOL was determined by Short Form 36 and Hospital Anxiety and Depression scale. Autoantibodies were measured and candidate genes (CaSR, AIRE, GATA3, and 22q11-deletion) were sequenced for classification of etiology. RESULTS: We identified 522 patients (511 alive) and estimated overall prevalence at 102 per million divided among postsurgical HP (64 per million), nonsurgical HP (30 per million), and pseudo-HP (8 per million). Nonsurgical HP comprised autosomal dominant hypocalcemia (21%), autoimmune polyendocrine syndrome type 1 (17%), DiGeorge/22q11 deletion syndrome (15%), idiopathic HP (44%), and others (4%). Among the 283 respondents (median age, 53 years [range, 9-89], 75% females), seven formerly classified as idiopathic were reclassified after genetic and immunological analyses, whereas 26 (37% of nonsurgical HP) remained idiopathic. Most were treated with vitamin D (94%) and calcium (70%), and 10 received PTH. HP patients scored significantly worse than the normative population on Short Form 36 and Hospital Anxiety and Depression scale; patients with postsurgical scored worse than those with nonsurgical HP and pseudo-HP, especially on physical health. CONCLUSIONS: We found higher prevalence of nonsurgical HP in Norway than reported elsewhere. Genetic testing and autoimmunity screening of idiopathic HP identified a specific cause in 21%. Further research is necessary to unravel the causes of idiopathic HP and to improve the reduced HRQOL reported by HP patients.
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John P. Bilezikian, Aliya Khan, John T. Potts et al. · Journal of Bone and Mineral Research · 2011 · 616 citations · Full text
Medicine, Endocrinology, Pathology +16
Anti-Interferon Autoantibodies in Autoimmune Polyendocrinopathy Syndrome Type 1
Anthony Meager, Kumuthini Visvalingam, Pärt Peterson et al. · PLoS Medicine · 2006 · 452 citations · Full text
Life Sciences, Genetics, Immunology +17