{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,5]],"date-time":"2026-05-05T02:48:54Z","timestamp":1777949334783,"version":"3.51.4"},"reference-count":19,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2026,3,24]],"date-time":"2026-03-24T00:00:00Z","timestamp":1774310400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2026,5,4]],"date-time":"2026-05-04T00:00:00Z","timestamp":1777852800000},"content-version":"vor","delay-in-days":41,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/501100001734","name":"Copenhagen University","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100001734","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["J Med Case Reports"],"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Hypophosphatasia is a rare, inherited metabolic bone disease characterized by impaired activity of tissue-nonspecific alkaline phosphatase leading to pathological mineralization of hard tissue, predisposing to poorly healing fractures. Limited access to enzyme replacement therapy makes the treatment of adults with hypophosphatasia challenging. A few case reports have described a beneficial effect of teriparatide on fracture healing in hypophosphatasia. However, this is the first case of a male patient with pediatric-onset hypophosphatasia, not previously exposed to antiresorptive therapy, treated with teriparatide for bilateral femoral pseudofractures.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case presentation<\/jats:title>\n                    <jats:p>\n                      We report a 59-old white man with pediatric-onset hypophosphatasia who developed bilateral femoral pseudofractures that were successfully treated with teriparatide. At the age of 54\u00a0years, our patient complained about reduced physical activity and pain in both hips and femoral bones. X-ray and later computed tomography scan revealed bilateral femoral pseudofractures. Dual energy X-ray absorptiometry showed normal bone mineral density. Not previously exposed to antiresorptive treatment, our patient was treated with conventional doses of teriparatide (Forsteo\n                      <jats:sup>\u00ae<\/jats:sup>\n                      , 20\u00a0\u00b5g\/day, subcutaneous) for 18\u00a0months. Markers of bone formation and bone resorption increased twofold at 3 months of treatment. At 6 months of treatment with teriparatide normal levels of alkaline phosphatase as well as pyridoxal 5\u2032-phosphate were measured. Early signs of fracture healing were observed at 6 months, and after 18\u00a0months of teriparatide treatment, the complete healing of both femoral pseudofractures was proven by X-ray and a computed tomography scan.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Treatment with teriparatide induces beneficial clinical, biochemical, and radiological responses and may provide an efficient treatment option for adults with hypophosphatasia and pseudofractures or poor healing fractures.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1186\/s13256-026-05903-5","type":"journal-article","created":{"date-parts":[[2026,3,24]],"date-time":"2026-03-24T14:04:48Z","timestamp":1774361088000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Bilateral healing of pseudofractures in hypophosphatasia with teriparatide: a case report"],"prefix":"10.1186","volume":"20","author":[{"ORCID":"https:\/\/orcid.org\/0009-0007-0927-6694","authenticated-orcid":false,"given":"Elaf","family":"Al-Saoudi","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nicola","family":"Hepp","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jens-Erik Beck","family":"Jensen","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2026,3,24]]},"reference":[{"issue":"4","key":"5903_CR1","doi-asserted-by":"publisher","first-page":"233","DOI":"10.1038\/nrendo.2016.14","volume":"12","author":"MP Whyte","year":"2016","unstructured":"Whyte MP. Hypophosphatasia\u2014Aetiology, nosology, pathogenesis, diagnosis and treatment. Nat Rev Endocrinol. 2016;12(4):233\u201346.","journal-title":"Nat Rev Endocrinol"},{"key":"5903_CR2","doi-asserted-by":"publisher","first-page":"15","DOI":"10.1016\/j.bone.2017.02.011","volume":"102","author":"MP Whyte","year":"2017","unstructured":"Whyte MP. Hypophosphatasia: an overview for 2017. Bone. 2017;102:15\u201325.","journal-title":"Bone"},{"issue":"35","key":"5903_CR3","doi-asserted-by":"publisher","first-page":"24168","DOI":"10.1074\/jbc.M114.576843","volume":"289","author":"J Silvent","year":"2014","unstructured":"Silvent J, Gasse B, Mornet E, Sire JY. Molecular evolution of the tissue-nonspecific alkaline phosphatase allows prediction and validation of missense mutations responsible for hypophosphatasia. J Biol Chem. 2014;289(35):24168\u201379.","journal-title":"J Biol Chem"},{"issue":"6","key":"5903_CR4","doi-asserted-by":"publisher","first-page":"723","DOI":"10.1007\/s00774-017-0888-6","volume":"36","author":"A Taillandier","year":"2018","unstructured":"Taillandier A, Domingues C, Dufour A, Debiais F, Guggenbuhl P, Roux C, et al. Genetic analysis of adults heterozygous for ALPL mutations. J Bone Miner Metab. 2018;36(6):723\u201333.","journal-title":"J Bone Miner Metab"},{"issue":"4","key":"5903_CR5","doi-asserted-by":"publisher","first-page":"309","DOI":"10.1002\/(SICI)1098-1004(200004)15:4<309::AID-HUMU2>3.0.CO;2-C","volume":"15","author":"E Mornet","year":"2000","unstructured":"Mornet E. Hypophosphatasia: the mutations in the tissue-nonspecific alkaline phosphatase gene. Hum Mutat. 2000;15(4):309\u201315.","journal-title":"Hum Mutat"},{"issue":"8","key":"5903_CR6","doi-asserted-by":"publisher","first-page":"1815","DOI":"10.1007\/s00198-018-4552-3","volume":"29","author":"F Genest","year":"2018","unstructured":"Genest F, Seefried L. Subtrochanteric and diaphyseal femoral fractures in hypophosphatasia-not atypical at all. Osteoporos Int. 2018;29(8):1815\u201325.","journal-title":"Osteoporos Int"},{"issue":"2","key":"5903_CR7","doi-asserted-by":"publisher","first-page":"93","DOI":"10.1016\/S2213-8587(18)30307-3","volume":"7","author":"MP Whyte","year":"2019","unstructured":"Whyte MP, Simmons JH, Moseley S, Fujita KP, Bishop N, Salman NJ, et al. Asfotase alfa for infants and young children with hypophosphatasia: 7 year outcomes of a single-arm, open-label, phase 2 extension trial. Lancet Diabetes Endocrinol. 2019;7(2):93\u2013105.","journal-title":"Lancet Diabetes Endocrinol"},{"issue":"5","key":"5903_CR8","doi-asserted-by":"publisher","first-page":"304","DOI":"10.1002\/jbm4.10052","volume":"2","author":"P Klidaras","year":"2018","unstructured":"Klidaras P, Severt J, Aggers D, Payne J, Miller PD, Ing SW. Fracture healing in two adult patients with hypophosphatasia after asfotase alfa therapy. JBMR Plus. 2018;2(5):304\u20137.","journal-title":"JBMR Plus"},{"issue":"3","key":"5903_CR9","first-page":"266","volume":"6","author":"KB Doshi","year":"2009","unstructured":"Doshi KB, Hamrahian AH, Licata AA. Teriparatide treatment in adult hypophosphatasia in a patient exposed to bisphosphonate: a case report. 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Calcif Tissue Int. 2012;90(3):250.","journal-title":"Calcif Tissue Int"},{"issue":"1","key":"5903_CR12","doi-asserted-by":"publisher","first-page":"75","DOI":"10.2169\/internalmedicine.9624-22","volume":"62","author":"H Mizuno","year":"2023","unstructured":"Mizuno H, Sawa N, Sekine A, Inoue N, Oba Y, Ikuma D, et al. A bone histomorphometric analysis of hypophosphatasia-related osteoporosis after teriparatide treatment. Intern Med Tokyo Jpn. 2023;62(1):75\u20139.","journal-title":"Intern Med Tokyo Jpn."},{"issue":"4","key":"5903_CR13","first-page":"584","volume":"21","author":"SA Polyzos","year":"2021","unstructured":"Polyzos SA, Tournis S, Goulas A, Kollia P, Whyte MP. Adult hypophosphatasia treated with reduced frequency of teriparatide dosing. 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Endocr Pract Off J Am Coll Endocrinol Am Assoc Clin Endocrinol. 2008;14(2):204\u20138.","journal-title":"Endocr Pract Off J Am Coll Endocrinol Am Assoc Clin Endocrinol"},{"issue":"3","key":"5903_CR18","doi-asserted-by":"publisher","first-page":"293","DOI":"10.1002\/(SICI)1098-1004(200003)15:3<293::AID-HUMU11>3.0.CO;2-Q","volume":"15","author":"A Taillandier","year":"2000","unstructured":"Taillandier A, Cozien E, Muller F, Merrien Y, Bonnin E, Fribourg C, et al. Fifteen new mutations (-195C>T, L-12X, 298\u20132A>G, T117N, A159T, R229S, 997+2T>A, E274X, A331T, H364R, D389G, 1256delC, R433H, N461I, C472S) in the tissue-nonspecific alkaline phosphatase (TNSALP) gene in patients with hypophosphatasia. Hum Mutat. 2000;15(3):293.","journal-title":"Hum Mutat"},{"issue":"12","key":"5903_CR19","doi-asserted-by":"publisher","first-page":"bvac159","DOI":"10.1210\/jendso\/bvac159","volume":"6","author":"N Khanjee","year":"2022","unstructured":"Khanjee N, Maalouf NM. Romosozumab-aqqg in the treatment of osteoporosis in a patient with hypophosphatasia. 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The research was conducted in accordance with the ethical standards.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethical approval and consent to participate"}},{"value":"Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"Informed consent was obtained from the subjects to publish their data.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Informed consent"}},{"value":"EAS has nothing to disclose. NH received research funding from Alexion and AstraZeneca Rare Disease and lecturer fees from Gedeon Richter and UCB. JEBJ is a board member in Eli Lilly, Amgen, and UCB. JEBJ received funding from Eli Lilly, Novo Nordic, and Amgen and consulting fees from UCB, Giliad, Amgen, GSK, Utsaka, and Abbvie.","order":5,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"235"}}