{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,27]],"date-time":"2026-10-27T20:35:55Z","timestamp":1793133355937,"version":"4.5.0"},"reference-count":50,"publisher":"Elsevier BV","license":[{"start":{"date-parts":[[2027,1,1]],"date-time":"2027-01-01T00:00:00Z","timestamp":1798761600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2027,1,1]],"date-time":"2027-01-01T00:00:00Z","timestamp":1798761600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2026,9,17]],"date-time":"2026-09-17T00:00:00Z","timestamp":1789603200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["clinicalkey.com","clinicalkey.com.au","clinicalkey.es","clinicalkey.fr","clinicalkey.jp","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Bone"],"published-print":{"date-parts":[[2027,1]]},"DOI":"10.1016\/j.bone.2026.118088","type":"journal-article","created":{"date-parts":[[2026,9,16]],"date-time":"2026-09-16T00:08:16Z","timestamp":1789517296000},"page":"118088","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":0,"special_numbering":"C","title":["Skeletal effects of 5-year high-dose cyclical etidronate in patients with pseudoxanthoma elasticum"],"prefix":"10.1016","volume":"214","author":[{"given":"Melanie","family":"Haverkamp","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aart-Jan","family":"van Weert","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wouter","family":"Foppen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stijn","family":"Bunk","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frank L.J.","family":"Visseren","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mirza Faisal","family":"Beg","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karteek","family":"Popuri","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Socrates E.","family":"Papapoulos","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wilko","family":"Spiering","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pim A.","family":"de Jong","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"issue":"1","key":"10.1016\/j.bone.2026.118088_bb0005","doi-asserted-by":"crossref","DOI":"10.1016\/j.annder.2025.103440","article-title":"REACT-PXE: a consensus on diagnosis and future research concerning pseudoxanthoma elasticum (PXE)","volume":"153","author":"Martin","year":"2026","journal-title":"Ann. Dermatol. Venereol."},{"key":"10.1016\/j.bone.2026.118088_bb0010","doi-asserted-by":"crossref","first-page":"1049","DOI":"10.1002\/ajmg.a.33329","article-title":"Proposal for updating the pseudoxanthoma elasticum classification system and a review of the clinical findings","author":"Plomp","year":"2010","journal-title":"Am. J. Med. Genet. A"},{"key":"10.1016\/j.bone.2026.118088_bb0015","doi-asserted-by":"crossref","DOI":"10.1016\/j.atherosclerosis.2024.119051","article-title":"Arterial calcification volume is associated with a higher risk of cardiovascular events in Pseudoxanthoma elasticum","volume":"400","author":"Harmsen","year":"2025","journal-title":"Atherosclerosis"},{"issue":"5","key":"10.1016\/j.bone.2026.118088_bb0020","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0096003","article-title":"The contribution of arterial calcification to peripheral arterial disease in pseudoxanthoma elasticum","volume":"9","author":"Leftheriotis","year":"2014","journal-title":"PLoS One"},{"issue":"1","key":"10.1016\/j.bone.2026.118088_bb0025","doi-asserted-by":"crossref","first-page":"142","DOI":"10.1016\/j.ejvs.2022.08.009","article-title":"Peripheral Interventions in Patients with Pseudoxanthoma Elasticum (PXE)","volume":"65","author":"Verwer","year":"2023","journal-title":"Eur. J. Vasc. Endovasc. Surg."},{"issue":"19","key":"10.1016\/j.bone.2026.118088_bb0030","doi-asserted-by":"crossref","first-page":"16860","DOI":"10.1074\/jbc.M110918200","article-title":"Loss of ATP-dependent transport activity in pseudoxanthoma elasticum-associated mutants of human ABCC6 (MRP6)","volume":"277","author":"Ili\u00e1s","year":"2002","journal-title":"J. Biol. Chem."},{"issue":"2","key":"10.1016\/j.bone.2026.118088_bb0035","doi-asserted-by":"crossref","first-page":"223","DOI":"10.1038\/76102","article-title":"Mutations in a gene encoding an ABC transporter cause pseudoxanthoma elasticum","volume":"25","author":"Le Saux","year":"2000","journal-title":"Nat. Genet."},{"issue":"2","key":"10.1016\/j.bone.2026.118088_bb0040","doi-asserted-by":"crossref","first-page":"228","DOI":"10.1038\/76109","article-title":"Mutations in ABCC6 cause pseudoxanthoma elasticum","volume":"25","author":"Bergen","year":"2000","journal-title":"Nat. Genet."},{"issue":"11","key":"10.1016\/j.bone.2026.118088_bb0045","doi-asserted-by":"crossref","first-page":"6001","DOI":"10.1073\/pnas.100041297","article-title":"Pseudoxanthoma elasticum: mutations in the MRP6 gene encoding a transmembrane ATP-binding cassette (ABC) transporter","volume":"97","author":"Ringpfeil","year":"2000","journal-title":"Proc. Natl. Acad. Sci. USA"},{"issue":"9","key":"10.1016\/j.bone.2026.118088_bb0050","doi-asserted-by":"crossref","first-page":"1985","DOI":"10.1161\/ATVBAHA.114.304017","article-title":"ABCC6-mediated ATP secretion by the liver is the main source of the mineralization inhibitor inorganic pyrophosphate in the systemic circulation-brief report","volume":"34","author":"Jansen","year":"2014","journal-title":"Arterioscler. Thromb. Vasc. Biol."},{"issue":"3","key":"10.1016\/j.bone.2026.118088_bb0055","doi-asserted-by":"crossref","DOI":"10.3390\/jcm12031047","article-title":"Plasma level of pyrophosphate is low in pseudoxanthoma elasticum owing to mutations in the ABCC6 gene, but it does not correlate with ABCC6 genotype","volume":"12","author":"Koz\u00e1k","year":"2023","journal-title":"J. Clin. Med."},{"issue":"1","key":"10.1016\/j.bone.2026.118088_bb0060","doi-asserted-by":"crossref","first-page":"431","DOI":"10.1186\/s13063-025-09064-6","article-title":"The TEMP-PREVENT trial: a study protocol for a randomized, double-blind, placebo-controlled clinical trial of etidronate for treatment in young adult patients with Pseudoxanthoma elasticum","volume":"26","author":"Harmsen","year":"2025","journal-title":"Trials"},{"issue":"1","key":"10.1016\/j.bone.2026.118088_bb0065","doi-asserted-by":"crossref","first-page":"30","DOI":"10.1186\/s13063-024-08666-w","article-title":"The PROPHECI trial: a phase II, double-blind, placebo-controlled, randomized clinical trial for the treatment of pseudoxanthoma elasticum with oral pyrophosphate","volume":"26","author":"Clotaire","year":"2025","journal-title":"Trials"},{"key":"10.1016\/j.bone.2026.118088_bb0070","series-title":"A Study of DS-1211b in Individuals With PseudoXanthoma Elasticum [Internet]","author":"Daiichi Sankyo","year":"2024"},{"key":"10.1016\/j.bone.2026.118088_bb0075","series-title":"ADAPT study: long-term safety study of INZ-701 in patients with ENPP1 deficiency and ABCC6 deficiency [Internet]","author":"Inozyme Pharma","year":"2024"},{"issue":"1","key":"10.1016\/j.bone.2026.118088_bb0080","doi-asserted-by":"crossref","first-page":"263","DOI":"10.1016\/j.jaad.2019.02.055","article-title":"Magnesium supplementation in the treatment of pseudoxanthoma elasticum: a randomized trial","volume":"81","author":"Rose","year":"2019","journal-title":"J. Am. Acad. Dermatol."},{"issue":"2","key":"10.1016\/j.bone.2026.118088_bb0085","doi-asserted-by":"crossref","first-page":"216","DOI":"10.1016\/j.ajpath.2018.09.014","article-title":"Pseudoxanthoma elasticum as a paradigm of heritable ectopic mineralization disorders: pathomechanisms and treatment development","volume":"189","author":"Li","year":"2019","journal-title":"Am. J. Pathol."},{"issue":"5","key":"10.1016\/j.bone.2026.118088_bb0090","doi-asserted-by":"crossref","first-page":"683","DOI":"10.2106\/00004623-197658050-00019","article-title":"Disodium etidronate in the prevention of postoperative recurrence of heterotopic ossification in spinal-cord injury patients","volume":"58","author":"Stover","year":"1976","journal-title":"J. Bone Joint Surg. Am."},{"issue":"6","key":"10.1016\/j.bone.2026.118088_bb0095","doi-asserted-by":"crossref","first-page":"926","DOI":"10.3109\/17453677408989703","article-title":"Effect of a diphosphonate on para-articular ossification after total hip replacement","volume":"45","author":"Bijvoet","year":"1974","journal-title":"Acta Orthop. Scand."},{"issue":"1","key":"10.1016\/j.bone.2026.118088_bb0100","doi-asserted-by":"crossref","first-page":"2","DOI":"10.1016\/j.bone.2011.04.022","article-title":"Bisphosphonates: the first 40 years","volume":"49","author":"Russell","year":"2011","journal-title":"Bone"},{"issue":"56","key":"10.1016\/j.bone.2026.118088_bb0105","doi-asserted-by":"crossref","first-page":"30721","DOI":"10.18632\/oncotarget.10738","article-title":"Etidronate prevents, but does not reverse, ectopic mineralization in a mouse model of pseudoxanthoma elasticum (Abcc6\u2212\/\u2212)","volume":"9","author":"Li","year":"2018","journal-title":"Oncotarget"},{"issue":"7","key":"10.1016\/j.bone.2026.118088_bb0110","doi-asserted-by":"crossref","first-page":"1082","DOI":"10.1080\/15384101.2015.1007809","article-title":"The effects of bisphosphonates on ectopic soft tissue mineralization caused by mutations in the ABCC6 gene","volume":"14","author":"Li","year":"2015","journal-title":"Cell Cycle"},{"key":"10.1016\/j.bone.2026.118088_bb0115","series-title":"Atherosclerosis","first-page":"37","article-title":"Etidronate halts systemic arterial calcification in Pseudoxanthoma elasticum","volume":"292","author":"Bartstra","year":"2020"},{"issue":"10","key":"10.1016\/j.bone.2026.118088_bb0120","doi-asserted-by":"crossref","first-page":"1117","DOI":"10.1016\/j.jacc.2017.12.062","article-title":"Etidronate for prevention of ectopic mineralization in patients with Pseudoxanthoma elasticum","volume":"71","author":"Kranenburg","year":"2018","journal-title":"J. Am. Coll. Cardiol."},{"issue":"16","key":"10.1016\/j.bone.2026.118088_bb0125","doi-asserted-by":"crossref","first-page":"4612","DOI":"10.3390\/jcm13164612","article-title":"Cyclical etidronate reduces the progression of arterial calcifications in patients with Pseudoxanthoma elasticum: a 6-year prospective observational study","volume":"13","author":"Harmsen","year":"2024","journal-title":"J. Clin. Med."},{"issue":"4","key":"10.1016\/j.bone.2026.118088_bb0130","doi-asserted-by":"crossref","first-page":"1555","DOI":"10.1210\/jc.2009-1947","article-title":"Long-term use of bisphosphonates in osteoporosis","volume":"95","author":"Watts","year":"2010","journal-title":"J. Clin. Endocrinol. Metab."},{"issue":"6","key":"10.1016\/j.bone.2026.118088_bb0135","doi-asserted-by":"crossref","first-page":"468","DOI":"10.1016\/S0002-9343(97)00278-7","article-title":"Cyclical etidronate in the treatment of postmenopausal osteoporosis: efficacy and safety after seven years of treatment","volume":"103","author":"Miller","year":"1997","journal-title":"Am. J. Med."},{"issue":"2","key":"10.1016\/j.bone.2026.118088_bb0140","doi-asserted-by":"crossref","first-page":"131","DOI":"10.1007\/s00439-008-0592-7","article-title":"Strengthening the reporting of genetic association studies (STREGA): an extension of the STROBE statement","volume":"125","author":"Little","year":"2009","journal-title":"Hum. Genet."},{"issue":"7","key":"10.1016\/j.bone.2026.118088_bb0145","doi-asserted-by":"crossref","first-page":"2074","DOI":"10.1007\/s00330-014-3584-0","article-title":"Opportunistic screening for osteoporosis on routine computed tomography? An external validation study","volume":"25","author":"Buckens","year":"2015","journal-title":"Eur. Radiol."},{"issue":"8","key":"10.1016\/j.bone.2026.118088_bb0150","doi-asserted-by":"crossref","first-page":"588","DOI":"10.7326\/0003-4819-158-8-201304160-00003","article-title":"Opportunistic screening for osteoporosis using abdominal computed tomography scans obtained for other indications","volume":"158","author":"Pickhardt","year":"2013","journal-title":"Ann. Intern. Med."},{"issue":"10","key":"10.1016\/j.bone.2026.118088_bb0155","doi-asserted-by":"crossref","first-page":"2009","DOI":"10.1515\/cclm-2012-0531","article-title":"IDS iSYS automated intact procollagen-1-N-terminus pro-peptide assay: method evaluation and reference intervals in adults and children","volume":"51","author":"Morovat","year":"2013","journal-title":"Clin. Chem. Lab. Med."},{"issue":"2","key":"10.1016\/j.bone.2026.118088_bb0160","doi-asserted-by":"crossref","first-page":"399","DOI":"10.1016\/j.bone.2013.09.010","article-title":"Reference intervals for serum concentrations of three bone turnover markers for men and women","volume":"57","author":"Michelsen","year":"2013","journal-title":"Bone"},{"issue":"2","key":"10.1016\/j.bone.2026.118088_bb0165","doi-asserted-by":"crossref","first-page":"271","DOI":"10.1016\/j.bone.2013.04.003","article-title":"Age-related reference intervals for bone turnover markers from an Australian reference population","volume":"55","author":"Jenkins","year":"2013","journal-title":"Bone"},{"issue":"4844","key":"10.1016\/j.bone.2026.118088_bb0170","doi-asserted-by":"crossref","first-page":"911","DOI":"10.1038\/195911a0","article-title":"Mechanism of calcification: inhibitory role of pyrophosphate","volume":"195","author":"Fleisch","year":"1962","journal-title":"Nature"},{"issue":"7625","key":"10.1016\/j.bone.2026.118088_bb0175","doi-asserted-by":"crossref","first-page":"845","DOI":"10.1016\/S0140-6736(69)92293-4","article-title":"Diphosphonates in the treatment of myositis ossificans","volume":"294","author":"Bassett","year":"1969","journal-title":"Lancet"},{"issue":"1","key":"10.1016\/j.bone.2026.118088_bb0180","first-page":"2","article-title":"Historical perspectives on the clinical development of bisphosphonates in the treatment of bone diseases","volume":"7","author":"Francis","year":"2007","journal-title":"J. Musculoskelet. Neuronal Interact."},{"key":"10.1016\/j.bone.2026.118088_bb0185","doi-asserted-by":"crossref","DOI":"10.1016\/j.bone.2020.115222","article-title":"History of etidronate","volume":"134","author":"Watts","year":"2020","journal-title":"Bone"},{"issue":"18","key":"10.1016\/j.bone.2026.118088_bb0190","doi-asserted-by":"crossref","first-page":"1265","DOI":"10.1056\/NEJM199005033221803","article-title":"Effect of intermittent cyclical etidronate therapy on bone mass and fracture rate in women with postmenopausal osteoporosis","volume":"322","author":"Storm","year":"1990","journal-title":"N. Engl. J. Med."},{"issue":"4","key":"10.1016\/j.bone.2026.118088_bb0195","article-title":"Etidronate for the primary and secondary prevention of osteoporotic fractures in postmenopausal women","volume":"2024","author":"Wells","year":"2024","journal-title":"Cochrane Database Syst. Rev."},{"issue":"11","key":"10.1016\/j.bone.2026.118088_bb0200","doi-asserted-by":"crossref","DOI":"10.1210\/clinem\/dgae023","article-title":"Bisphosphonate use and risk of atypical femoral fractures: a danish case-cohort study with blinded radiographic review","volume":"109","author":"Bauer","year":"2024","journal-title":"J. Clin. Endocrinol. Metab."},{"issue":"1","key":"10.1016\/j.bone.2026.118088_bb0205","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1002\/jbmr.1998","article-title":"Atypical subtrochanteric and diaphyseal femoral fractures: second report of a task force of the American Society for Bone and Mineral Research","volume":"29","author":"Shane","year":"2014","journal-title":"J. Bone Miner. Res."},{"issue":"18","key":"10.1016\/j.bone.2026.118088_bb0210","doi-asserted-by":"crossref","first-page":"1728","DOI":"10.1056\/NEJMoa1010650","article-title":"Bisphosphonate use and atypical fractures of the femoral shaft","volume":"364","author":"Schilcher","year":"2011","journal-title":"N. Engl. J. Med."},{"issue":"1","key":"10.1016\/j.bone.2026.118088_bb0215","doi-asserted-by":"crossref","first-page":"15","DOI":"10.1186\/s40360-023-00657-y","article-title":"Medication-induced osteonecrosis of the jaw: a review of cases from the Food and Drug Administration Adverse Event Reporting System (FAERS)","volume":"24","author":"Ahdi","year":"2023","journal-title":"BMC Pharmacol. Toxicol."},{"issue":"9","key":"10.1016\/j.bone.2026.118088_bb0220","doi-asserted-by":"crossref","first-page":"1549","DOI":"10.1248\/bpb.b16-00041","article-title":"A strategy against the osteonecrosis of the jaw associated with nitrogen-containing bisphosphonates (N-BPs): attempts to replace N-BPs with the non-N-BP etidronate","volume":"39","author":"Oizumi","year":"2016","journal-title":"Biol. Pharm. Bull."},{"issue":"6","key":"10.1016\/j.bone.2026.118088_bb0225","doi-asserted-by":"crossref","first-page":"557","DOI":"10.1016\/0002-9343(93)90350-X","article-title":"Four-year study of intermittent cyclic etidronate treatment of postmenopausal osteoporosis: three years of blinded therapy followed by one year of open therapy","volume":"95","author":"Harris","year":"1993","journal-title":"Am. J. Med."},{"issue":"3","key":"10.1016\/j.bone.2026.118088_bb0230","doi-asserted-by":"crossref","first-page":"245","DOI":"10.1177\/1358863X241235669","article-title":"Pilot study to evaluate the safety and effectiveness of etidronate treatment for arterial calcification due to deficiency of CD73 (ACDC)","volume":"29","author":"Ferrante","year":"2024","journal-title":"Vasc. Med."},{"issue":"3","key":"10.1016\/j.bone.2026.118088_bb0235","doi-asserted-by":"crossref","DOI":"10.1093\/jbmrpl\/ziag003","article-title":"High-dose oral pyrophosphate inhibits connective tissue calcification in Abcc6 null mice but may affect bone structure","volume":"10","author":"Rajpar","year":"2026","journal-title":"JBMR Plus."},{"key":"10.1016\/j.bone.2026.118088_bb0240","doi-asserted-by":"crossref","DOI":"10.3389\/fcell.2022.823249","article-title":"Abcc6 null mice-a model for mineralization disorder PXE shows vertebral osteopenia without enhanced intervertebral disc calcification with aging","volume":"10","author":"Boneski","year":"2022","journal-title":"Front. Cell Dev. Biol."},{"issue":"3","key":"10.1016\/j.bone.2026.118088_bb0245","doi-asserted-by":"crossref","first-page":"983","DOI":"10.1007\/s00198-016-3804-3","article-title":"Opportunistic screening for osteoporosis by routine CT in Southern Europe","volume":"28","author":"Alacreu","year":"2017","journal-title":"Osteoporos. Int."},{"key":"10.1016\/j.bone.2026.118088_bb0250","doi-asserted-by":"crossref","DOI":"10.1007\/s00198-026-07863-4","article-title":"Diagnostic performance of lumbar computed tomography Hounsfield unit thresholds for osteoporosis and osteopenia: a systematic review and meta-analysis","author":"Lubbad","year":"2026","journal-title":"Osteoporos. Int."}],"container-title":["Bone"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S8756328226003145?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S8756328226003145?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2026,10,27]],"date-time":"2026-10-27T20:27:38Z","timestamp":1793132858000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S8756328226003145"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2027,1]]},"references-count":50,"alternative-id":["S8756328226003145"],"URL":"https:\/\/doi.org\/10.1016\/j.bone.2026.118088","relation":{},"ISSN":["8756-3282"],"issn-type":[{"value":"8756-3282","type":"print"}],"subject":[],"published":{"date-parts":[[2027,1]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Skeletal effects of 5-year high-dose cyclical etidronate in patients with pseudoxanthoma elasticum","name":"articletitle","label":"Article Title"},{"value":"Bone","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.bone.2026.118088","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2026 The Authors. Published by Elsevier Inc.","name":"copyright","label":"Copyright"}],"article-number":"118088"}}