Searchable abstracts of presentations at key conferences on calcified tissues
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43rd Annual European Calcified Tissue Society Congress

Rome, Italy
14 May 2016 - 17 May 2016

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ba0005ht1 | (1) | ECTS2016

Storage disease and neurological phenotype in autosomal dominant osteopetrosis type 2 (ADO2). A preclinical study

Maurizi Antonio , Capulli Mattia , Cortes Juliana , Rito Laura Di , Rucci Nadia , Teti Anna

ADO2 is a debilitating genetic bone disease causing multiple fractures and other severe symptoms. A mouse model of ADO2, harbouring the heterozygous Clcn7G213R gene mutation, phenocopies the human syndrome. The Clc7 gene encodes the ClC7 dimeric 2Cl−/1H+ antiporter that is almost ubiquitously expressed, although the mutations hit especially the osteoclasts impairing bone resorption. By immunofluorescence, we observed that the mutant ClC7 ...

ba0005ht2 | (1) | ECTS2016

Mice lacking estrogen receptor α in hypothalamic POMC neurons display enhanced estrogenic response on cortical bone mass

Farman Helen , Windahl Sara , Clegg Deborah , Xie Shang Kui , Westberg Lars , Isaksson Hanna , Egecioglu Emil , Schele Erik , Johnsson John Olov , Tuukkanen Juha , Hahner Lisa , Zehr Jordan , Lagerquist Marie , Ohlsson Claes

Estrogens are important regulators of bone mass and exert their physiological effects on bone mainly via estrogen receptor α (ERα). Central ERα has been reported to exert an inhibitory role on bone mass. ERα is widely distributed in the brain with a high expression in the arcuate nucleus (ARC) and the ventral medial nucleus (VMN) in the hypothalamus. Here, we tested the hypothesis that ERα in hypothalamic pro-opiomelanocortin (POMC) neurons, located in...

ba0005ht3 | (1) | ECTS2016

Deletion of protease-activated receptor-2 improves bone and muscle pathology in dystrophin-deficient (mdx) mice

Esfandouni Neda Taghavi , Sanaei Reza , Samuel Chrishan , Pagel Charles , Mackie Eleanor

Duchenne muscular dystrophy (DMD) is associated with osteoporosis, and dystrophic (dystrophin-deficient) mdx mice show reduced bone mass characterised by decreased mineral apposition and elevated bone resorption. To investigate a potential role of the G-protein-coupled receptor protease-activated receptor-2 (PAR2) in the muscle and bone pathology associated with DMD, we established a colony of PAR2-null-mdx mice. Limb and diaphragm muscles, tibiae and ser...

ba0005ht4 | (1) | ECTS2016

Vitamin D supplementation in pregnancy leads to greater bone mass in UK infants born during winter months: the MAVIDOS multicentre, randomised, double-blind, placebo-controlled trial

Cooper Cyrus , Harvey Nicholas , Bishop Nicholas , Kennedy Stephen , Papageorghiou Aris , Schoenmakers Inez , Fraser Robert , Gandhi Saurabh , D'Angelo Stefania , Crozier Sarah , Moon Rebecca , Arden Nigel , Dennison Elaine , Godfrey Keith , Inskip Hazel , Prentice Ann , Mughal Zulf , Eastell Richard , Reid David , Javaid Kassim

Maternal vitamin D status has been positively associated with infant bone mass in observational studies. We therefore evaluated whether 1000 IU/day cholecalciferol during pregnancy would lead to greater offspring bone mass at birth, in a UK, multicentre, randomised, double-blind, placebo-controlled trial (MAVIDOS, ISRCTN82927713).At 12 weeks’ gestation, pregnant women with a serum 25-hydroxyvitamin D [25(OH)D] 25-100 nmol/l were randomised to either...

ba0005ht5 | (1) | ECTS2016

Superior Gains in Bone Mineral Density (BMD) and Estimated Strength at the Hip for Romosozumab Compared With Teriparatide (TPTD) in Women With Postmenopausal Osteoporosis Transitioning From Bisphosphonate Therapy: Results of the Phase 3 Open-label STRUCTURE Study

Langdahl B , Libanati C , Crittenden D B , Bolognese M A , Brown J P , Daizadeh N S , Dokoupilova E , Engelke K , Finkelstein J S , Genant H K , Goemaere S , Hyldstrup L , Jodar-Gimeno E , Keaveny T M , Kendler D , Lakatos P , Maddox J , Malouf J , Massari F E , Molina J F

STRUCTURE was a phase 3, open-label study evaluating the effect of romosozumab or TPTD for 12 months in women with postmenopausal osteoporosis transitioning from bisphosphonate therapy (NCT01796301). This study enrolled women with postmenopausal osteoporosis who had taken an oral bisphosphonate for ≥3 years prior to screening and alendronate in the year prior to screening; had a BMD T-score ≤−2.5 at the total hip (TH), lumbar spine (LS), or femoral neck (FN);...

ba0005ht6 | (1) | ECTS2016

Effect of KRN23, a fully human anti-FGF23 monoclonal antibody, on rickets in children with X-linked hypophosphatemia (XLH): 40-week interim results from a randomized, open-label Phase 2 study

Imel Erik , Carpenter Thomas , Boot Annemieke , Hogler Wolfgang , Linglart Agnes , Padidela Raja , van't Hoff William , Whyte Michael , Agarwal Sunil , Chen Chao-Yin , Skrinar Alison , Martin Javier San , Portale Anthony

In XLH, high circulating FGF23 causes hypophosphatemia, rickets, and short stature. In our Phase 2 study, 52 XLH children (ages 5-12 years, ≥Tanner 2) were randomized to receive KRN23 subcutaneously biweekly (Q2W) or monthly (Q4W). Serum phosphate (Pi) was measured biweekly. KRN23 dose was titrated (maximum 2 mg/kg) targeting age-appropriate serum Pi concentrations.The first 36 subjects had a mean 6.6 years of standard-of-care treatment before wash...