PMID- 29087612 OWN - NLM STAT- MEDLINE DCOM- 20191009 LR - 20191010 IS - 1523-4681 (Electronic) IS - 0884-0431 (Linking) VI - 33 IP - 3 DP - 2018 Mar TI - Bone Status Among Patients With Nonsurgical Hypoparathyroidism, Autosomal Dominant Hypocalcaemia, and Pseudohypoparathyroidism: A Cohort Study. PG - 467-477 LID - 10.1002/jbmr.3328 [doi] AB - Nonsurgical hypoparathyroidism (Ns-HypoPT) and pseudohypoparathyroidism (PHP) are both rare diseases, characterized by hypocalcemia. In Ns-HypoPT, PTH levels are low, whereas patients with PHP often have very high levels due to receptor-insensitivity to PTH (PTH-resistance). Accordingly, we hypothesized that indices of bone turnover and bone mineralization/architecture are similar in Ns-HypoPT and PHP despite marked differences in PTH levels. We studied 62 patients with Ns-HypoPT and 31 with PHP as well as a group of age- and sex-matched healthy controls. We found a significantly higher areal BMD (aBMD) by DXA among patients with Ns-HypoPT, both compared with PHP and the background population. Compared with Ns-HypoPT, PHP patients had significantly lower total and trabecular volumetric BMD (vBMD) assessed by quantitative computed tomography (QCT) scans at the spine and hip. High-resolution peripheral quantitative computed tomography (HRpQCT) scans showed a lower trabecular area and vBMD as well as a lower trabecular number at the tibia in PHP compared to Ns-HypoPT and matched controls. In PHP, PTH levels correlated with levels of markers of bone formation (osteocalcin, bone-specific alkaline phosphatase, P1NP), and bone resorption (CTx). In adult males, levels of bone markers were significantly higher in PHP compared with Ns-HypoPT. Levels of procalcitonin and calcitonin were significantly higher in PHP compared with Ns-HypoPT. In conclusion, indices of bone turnover, density, and microarchitecture differ between patients with Ns-HypoPT and PHP. Our data suggest that patients with PHP do not have a complete skeletal resistance to PTH and that the effects of chronically high PTH levels in PHP are mostly confined to the trabecular tissue. (c) 2017 American Society for Bone and Mineral Research. CI - (c) 2017 American Society for Bone and Mineral Research. FAU - Underbjerg, Line AU - Underbjerg L AD - Department of Endocrinology and Internal Medicine, Tage-Hansens Gade (THG), Aarhus University Hospital, Aarhus, Denmark. FAU - Malmstroem, Sofie AU - Malmstroem S AD - Department of Endocrinology and Internal Medicine, Tage-Hansens Gade (THG), Aarhus University Hospital, Aarhus, Denmark. FAU - Sikjaer, Tanja AU - Sikjaer T AD - Department of Endocrinology and Internal Medicine, Tage-Hansens Gade (THG), Aarhus University Hospital, Aarhus, Denmark. FAU - Rejnmark, Lars AU - Rejnmark L AD - Department of Endocrinology and Internal Medicine, Tage-Hansens Gade (THG), Aarhus University Hospital, Aarhus, Denmark. LA - eng PT - Journal Article PT - Research Support, Non-U.S. Gov't DEP - 20171130 PL - England TA - J Bone Miner Res JT - Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research JID - 8610640 RN - Hypercalciuric Hypocalcemia, Familial SB - IM MH - Adult MH - Bone Density MH - Bone and Bones/diagnostic imaging/*pathology MH - Cohort Studies MH - Female MH - Humans MH - Hypercalciuria/*complications/diagnostic imaging MH - Hypocalcemia/*complications/diagnostic imaging MH - Hypoparathyroidism/*complications/*congenital/diagnostic imaging MH - Male MH - Middle Aged MH - Pseudohypoparathyroidism/*complications/diagnostic imaging MH - Radius/diagnostic imaging/pathology MH - Renal Insufficiency, Chronic/complications MH - Tibia/diagnostic imaging/pathology MH - Tomography, X-Ray Computed OTO - NOTNLM OT - DXA OT - HRPQCT OT - NONSURGICAL HYPOPARATHYROIDISM OT - PSEUDOHYPOPARATHYROIDISM OT - QCT EDAT- 2017/11/01 06:00 MHDA- 2019/10/11 06:00 CRDT- 2017/11/01 06:00 PHST- 2017/08/02 00:00 [received] PHST- 2017/10/27 00:00 [revised] PHST- 2017/10/28 00:00 [accepted] PHST- 2017/11/01 06:00 [pubmed] PHST- 2019/10/11 06:00 [medline] PHST- 2017/11/01 06:00 [entrez] AID - 10.1002/jbmr.3328 [doi] PST - ppublish SO - J Bone Miner Res. 2018 Mar;33(3):467-477. doi: 10.1002/jbmr.3328. Epub 2017 Nov 30.