{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,1]],"date-time":"2026-09-01T04:47:33Z","timestamp":1788238053712,"version":"build-2803163510"},"reference-count":21,"publisher":"Japanese Society for Pediatric Endocrinology","issue":"1","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Clin Pediatr Endocrinol"],"published-print":{"date-parts":[[2026]]},"DOI":"10.1297\/cpe.2025-0063","type":"journal-article","created":{"date-parts":[[2025,10,4]],"date-time":"2025-10-04T22:07:57Z","timestamp":1759615677000},"page":"110-116","source":"Crossref","is-referenced-by-count":2,"title":["Burosumab treatment for FGF23-related hypophosphatemia in a two-year-old girl with McCune-Albright syndrome"],"prefix":"10.1297","volume":"35","author":[{"given":"Tomoki","family":"Saito","sequence":"first","affiliation":[{"name":"Department of Endocrinology and Metabolism, Hyogo Prefectural Kobe Children\u2019s Hospital, Kobe, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kana","family":"Hiromoto","sequence":"additional","affiliation":[{"name":"Department of Genome Medical Center, Hyogo Prefectural Kobe Children\u2019s Hospital, Kobe, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Naoya","family":"Morisada","sequence":"additional","affiliation":[{"name":"Department of Genome Medical Center, Hyogo Prefectural Kobe Children\u2019s Hospital, Kobe, Japan"},{"name":"Department of Clinical Genetics, Hyogo Prefectural Kobe Children\u2019s Hospital, Kobe, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ryosuke","family":"Sakata","sequence":"additional","affiliation":[{"name":"Department of Orthopedic Surgery, Hyogo Prefectural Kobe Children\u2019s Hospital, Kobe, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tomohiro","family":"Ishii","sequence":"additional","affiliation":[{"name":"Department of Endocrinology and Metabolism, Tokyo Metropolitan Children\u2019s Medical Center, Tokyo, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tomonobu","family":"Hasegawa","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Keio University School of Medicine, Tokyo, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Keiichi","family":"Ozono","sequence":"additional","affiliation":[{"name":"Center for Promoting Treatment of Intractable Diseases, ISEIKAI International General Hospital, Osaka, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kayo","family":"Ozaki","sequence":"additional","affiliation":[{"name":"Department of Endocrinology and Metabolism, Hyogo Prefectural Kobe Children\u2019s Hospital, Kobe, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"125","reference":[{"key":"1","doi-asserted-by":"crossref","unstructured":"1. Tufano, M, Ciofi, D, Amendolea, A, Stagi, S. Auxological and endocrinological features in children with McCune Albright Syndrome: A review. Front Endocrinol (Lausanne) 2020;11: 522.","DOI":"10.3389\/fendo.2020.00522"},{"key":"2","unstructured":"2. Szymczuk, V, Florenzano, P, de Castro, LF, Collins, MT, Boyce, AM. Fibrous Dysplasia \/ McCune-Albright Syndrome. In: Adam MP, Feldman J, Mirzaa GM, Pagon RA, Wallace SE, Amemiya A, editors. GeneReviews\u00ae [Internet]. Seattle (WA): University of Washington, Seattle;  1993 [cited 2024 Dec 15]. Available from: http:\/\/www.ncbi.nlm.nih.gov\/books\/NBK274564\/."},{"key":"3","doi-asserted-by":"crossref","unstructured":"3. Boyce, AM, Bhattacharyya, N, Collins, MT. Fibrous dysplasia and fibroblast growth factor-23 regulation. Curr Osteoporos Rep 2013;11: 65\u201371.","DOI":"10.1007\/s11914-013-0144-5"},{"key":"4","doi-asserted-by":"crossref","unstructured":"4. Riminucci, M, Collins, MT, Fedarko, NS, Cherman, N, Corsi, A, White, KE, et al. FGF-23 in fibrous dysplasia of bone and its relationship to renal phosphate wasting. J Clin Invest 2003;112: 683\u201392.","DOI":"10.1172\/JCI200318399"},{"key":"5","doi-asserted-by":"crossref","unstructured":"5. Leet, AI, Chebli, C, Kushner, H, Chen, CC, Kelly, MH, Brillante, BA, et al. Fracture incidence in polyostotic fibrous dysplasia and the McCune-Albright syndrome. J Bone Miner Res 2004;19: 571\u20137.","DOI":"10.1359\/JBMR.0301262"},{"key":"6","doi-asserted-by":"crossref","unstructured":"6. Carpenter, TO, Imel, EA, Holm, IA, Jan de Beur, SM, Insogna, KL. A clinician\u2019s guide to X-linked hypophosphatemia. J Bone Miner Res 2011;26: 1381\u20138.","DOI":"10.1002\/jbmr.340"},{"key":"7","doi-asserted-by":"crossref","unstructured":"7. Florenzano, P, Pan, KS, Brown, SM, Paul, SM, Kushner, H, Guthrie, LC, et al. Age-related changes and effects of bisphosphonates on bone turnover and disease progression in fibrous dysplasia of bone. J Bone Miner Res 2019;34: 653\u201360.","DOI":"10.1002\/jbmr.3649"},{"key":"8","doi-asserted-by":"crossref","unstructured":"8. Boyce, AM, Kelly, MH, Brillante, BA, Kushner, H, Wientroub, S, Riminucci, M, et al. A randomized, double blind, placebo-controlled trial of alendronate treatment for fibrous dysplasia of bone. J Clin Endocrinol Metab 2014;99: 4133\u201340.","DOI":"10.1210\/jc.2014-1371"},{"key":"9","doi-asserted-by":"crossref","unstructured":"9. de Castro, LF, Michel, Z, Pan, K, Taylor, J, Szymczuk, V, Paravastu, S, et al. Safety and efficacy of denosumab for fibrous dysplasia of bone. N Engl J Med 2023;388: 766\u20138.","DOI":"10.1056\/NEJMc2214862"},{"key":"10","doi-asserted-by":"crossref","first-page":"ziaf042","DOI":"10.1093\/jbmrpl\/ziaf042","volume":"9","year":"2025","unstructured":"10. Sakka, SD, Georgakopoulou, D, Doulgeraki, A, Krieg, AH, Anastasopoulos, J, Szinnai, G, et al. Burosumab treatment of a child with McCune-Albright syndrome\/polyostotic fibrous dysplasia: challenges and benefits. JBMR Plus 2025;9: ziaf042.","journal-title":"JBMR Plus","ISSN":"https:\/\/id.crossref.org\/issn\/2473-4039","issn-type":"print"},{"key":"11","doi-asserted-by":"crossref","unstructured":"11. Barbato, A, Vaiasuso, R, Trinati, E, Del Medico, G, Chiti, N, Baroncelli, GI, et al. Use of burosumab in McCune Albright syndrome: case report and review of literature in mosaic disorders with FGF23 overproduction. Front Endocrinol (Lausanne) 2025;16: 1577734.","DOI":"10.3389\/fendo.2025.1577734"},{"key":"12","doi-asserted-by":"crossref","unstructured":"12. Apperley, LJ, Senniappan, S. Burosumab therapy in a paediatric patient with McCune-Albright syndrome: a case report. Horm Res Paediatr 2023;96: 341\u20138.","DOI":"10.1159\/000527129"},{"key":"13","doi-asserted-by":"crossref","unstructured":"13. Gladding, A, Szymczuk, V, Auble, BA, Boyce, AM. Burosumab treatment for fibrous dysplasia. Bone 2021;150: 116004.","DOI":"10.1016\/j.bone.2021.116004"},{"key":"14","doi-asserted-by":"crossref","unstructured":"14. Sawamura, K, Hamajima, T, Kitoh, H. Improvement of fibrous dysplasia after burosumab therapy in a pediatric patient with McCune-Albright syndrome: a case report. JBJS Case Connect 2024;14.","DOI":"10.2106\/JBJS.CC.24.00279"},{"key":"15","doi-asserted-by":"crossref","unstructured":"15. Kagami, R, Sato, T, Ishii, T, Araki, E, Yamashita, Y, Shibata, H, et al. Central precocious puberty in a boy with pseudohypoparathyroidism type Ia due to a novel <b><i>GNAS<\/i><\/b> mutation. Clin Pediatr Endocrinol 2020;29: 89\u201390.","DOI":"10.1297\/cpe.29.89"},{"key":"16","doi-asserted-by":"publisher","unstructured":"16. Imel, EA, Glorieux, FH, Whyte, MP, Munns, CF, Ward, LM, Nilsson, O, et al. Burosumab versus conventional therapy in children with X-linked hypophosphataemia: a randomised, active-controlled, open-label, phase 3 trial. Lancet 2019;393: 2416\u201327.","DOI":"10.1016\/S0140-6736(19)30654-3"},{"key":"17","doi-asserted-by":"crossref","unstructured":"17. Tack, LJW, Tatsi, C, Stratakis, CA, Lodish, MB. Effects of glucocorticoids on bone: what we can learn from pediatric endogenous Cushing\u2019s syndrome. Horm Metab Res 2016;48: 764\u201370.","DOI":"10.1055\/s-0042-117721"},{"key":"18","doi-asserted-by":"crossref","unstructured":"18. Boyce, AM, Glover, M, Kelly, MH, Brillante, BA, Butman, JA, Fitzgibbon, EJ, et al. Optic neuropathy in McCune-Albright syndrome: effects of early diagnosis and treatment of growth hormone excess. J Clin Endocrinol Metab 2013;98: E126\u201334.","DOI":"10.1210\/jc.2012-2111"},{"key":"19","doi-asserted-by":"crossref","unstructured":"19. Collins, MT, Singer, FR, Eugster, E. McCune-Albright syndrome and the extraskeletal manifestations of fibrous dysplasia. Orphanet J Rae Dis 2012; 7(Suppl 1): S4.","DOI":"10.1186\/1750-1172-7-S1-S4"},{"key":"20","doi-asserted-by":"crossref","unstructured":"20. Kelly, MH, Brillante, B, Collins, MT. Pain in fibrous dysplasia of bone: age-related changes and the anatomical distribution of skeletal lesions. Osteoporos Int 2008;19: 57\u201363.","DOI":"10.1007\/s00198-007-0425-x"},{"key":"21","doi-asserted-by":"crossref","unstructured":"21. Gun, ZH, Osamor, C, Taylor, J, Li, X, Szymczuk, V, Boyce, AM. Serum phosphorus as a driver of skeletal morbidity in fibrous dysplasia. J Clin Endocrinol Metab 2024;109: 1334\u201340.","DOI":"10.1210\/clinem\/dgad671"}],"container-title":["Clinical Pediatric Endocrinology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.jstage.jst.go.jp\/article\/cpe\/35\/1\/35_2025-0063\/_pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,1,10]],"date-time":"2026-01-10T04:21:27Z","timestamp":1768018887000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.jstage.jst.go.jp\/article\/cpe\/35\/1\/35_2025-0063\/_article"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026]]},"references-count":21,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2026]]}},"URL":"https:\/\/doi.org\/10.1297\/cpe.2025-0063","relation":{},"ISSN":["0918-5739","1347-7358"],"issn-type":[{"value":"0918-5739","type":"print"},{"value":"1347-7358","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026]]},"article-number":"2025-0063"}}