{"id":"https://openalex.org/W4412915469","doi":"https://doi.org/10.1093/jbmrpl/ziaf125","title":"Burosumab treatment for fibroblast growth factor-23-associated hypophosphatemia in an adult patient with severe fibrous dysplasia in McCune\u2013Albright syndrome: case report and review of the literature","display_name":"Burosumab treatment for fibroblast growth factor-23-associated hypophosphatemia in an adult patient with severe fibrous dysplasia in McCune\u2013Albright syndrome: case report and review of the literature","publication_year":2025,"publication_date":"2025-07-25","ids":{"openalex":"https://openalex.org/W4412915469","doi":"https://doi.org/10.1093/jbmrpl/ziaf125","pmid":"https://pubmed.ncbi.nlm.nih.gov/40842905","pmcid":"https://www.ncbi.nlm.nih.gov/pmc/articles/12366781"},"language":"en","primary_location":{"id":"doi:10.1093/jbmrpl/ziaf125","doi":"https://doi.org/10.1093/jbmrpl/ziaf125","is_oa":true,"landing_page_url":"https://doi.org/10.1093/jbmrpl/ziaf125","pdf_url":null,"source":{"id":"https://openalex.org/S4210170492","display_name":"JBMR Plus","issn_l":"2473-4039","issn":["2473-4039"],"is_oa":true,"is_in_doaj":true,"is_core":true,"listed_in":["cwts-core","doaj","dongbi-d","jufo-1","ki-jl-1","norway-1","oarl","poland-20","russia-white-list-2","vabb-shw"],"host_organization":"https://openalex.org/P4310320595","host_organization_name":"Wiley","host_organization_lineage":["https://openalex.org/P4310320595"],"host_organization_lineage_names":["Wiley"],"type":"journal"},"license":"cc-by-nc","license_id":"https://openalex.org/licenses/cc-by-nc","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"JBMR Plus","raw_type":"journal-article"},"volume":null,"type":"article","is_traditional":true,"indexed_in":["crossref","doaj","pubmed"],"open_access":{"is_oa":true,"oa_status":"gold","oa_url":"https://doi.org/10.1093/jbmrpl/ziaf125","any_repository_has_fulltext":true},"authorships":[{"author_position":"first","author":{"id":"https://openalex.org/A5022617714","display_name":"Maria Stelmachowska\u2010Bana\u015b","orcid":"https://orcid.org/0000-0002-2961-3316","observed_orcids":["https://orcid.org/0000-0002-2961-3316"]},"institutions":[],"countries":[],"is_corresponding":true,"raw_author_name":"Maria Stelmachowska-Bana\u015b","raw_affiliation_strings":["Department of Endocrinology, The Centre of Postgraduate Medical Education , 01-813 Warsaw ,","Department of Endocrinology, The Centre of Postgraduate Medical Education, 01-813 Warsaw, Poland"],"raw_orcid":"https://orcid.org/0000-0002-2961-3316","affiliations":[{"raw_affiliation_string":"Department of Endocrinology, The Centre of Postgraduate Medical Education , 01-813 Warsaw ,","institution_ids":[]},{"raw_affiliation_string":"Department of Endocrinology, The Centre of Postgraduate Medical Education, 01-813 Warsaw, Poland","institution_ids":[]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5031487172","display_name":"Karolina Cylke-Falkowska","orcid":"https://orcid.org/0009-0005-8503-0253","observed_orcids":["https://orcid.org/0009-0005-8503-0253"]},"institutions":[],"countries":[],"is_corresponding":false,"raw_author_name":"Karolina Cylke-Falkowska","raw_affiliation_strings":["Department of Endocrinology, The Centre of Postgraduate Medical Education , 01-813 Warsaw ,","Department of Endocrinology, The Centre of Postgraduate Medical Education, 01-813 Warsaw, Poland"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Department of Endocrinology, The Centre of Postgraduate Medical Education , 01-813 Warsaw ,","institution_ids":[]},{"raw_affiliation_string":"Department of Endocrinology, The Centre of Postgraduate Medical Education, 01-813 Warsaw, Poland","institution_ids":[]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5082503502","display_name":"Wojciech Zgliczy\u0144ski","orcid":"https://orcid.org/0000-0002-7283-4755","observed_orcids":["https://orcid.org/0000-0002-7283-4755"]},"institutions":[],"countries":[],"is_corresponding":false,"raw_author_name":"Wojciech Zgliczy\u0144ski","raw_affiliation_strings":["Department of Endocrinology, The Centre of Postgraduate Medical Education , 01-813 Warsaw ,","Department of Endocrinology, The Centre of Postgraduate Medical Education, 01-813 Warsaw, Poland"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Department of Endocrinology, The Centre of Postgraduate Medical Education , 01-813 Warsaw ,","institution_ids":[]},{"raw_affiliation_string":"Department of Endocrinology, The Centre of Postgraduate Medical Education, 01-813 Warsaw, Poland","institution_ids":[]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5013126573","display_name":"John Paul Bilezikian","orcid":"https://orcid.org/0000-0002-1570-2617","observed_orcids":["https://orcid.org/0000-0002-1570-2617"]},"institutions":[{"id":"https://openalex.org/I78577930","display_name":"Columbia University","ror":"https://ror.org/00hj8s172","country_code":"US","type":"education","lineage":["https://openalex.org/I78577930"]}],"countries":["US"],"is_corresponding":false,"raw_author_name":"John P Bilezikian","raw_affiliation_strings":["Division of Endocrinology, Vagelos College of Physicians and Surgeons, Columbia University , New York, NY 10032 ,","Division of Endocrinology, Vagelos College of Physicians and Surgeons, Columbia University, New York, NY 10032, United States"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Division of Endocrinology, Vagelos College of Physicians and Surgeons, Columbia University , New York, NY 10032 ,","institution_ids":["https://openalex.org/I78577930"]},{"raw_affiliation_string":"Division of Endocrinology, Vagelos College of Physicians and Surgeons, Columbia University, New York, NY 10032, United States","institution_ids":["https://openalex.org/I78577930"]}]},{"author_position":"last","author":{"id":"https://openalex.org/A5045554018","display_name":"Waldemar Misiorowski","orcid":"https://orcid.org/0000-0003-2852-8010","observed_orcids":["https://orcid.org/0000-0003-2852-8010"]},"institutions":[],"countries":[],"is_corresponding":false,"raw_author_name":"Waldemar Misiorowski","raw_affiliation_strings":["Department of Endocrinology, The Centre of Postgraduate Medical Education , 01-813 Warsaw ,","Department of Endocrinology, The Centre of Postgraduate Medical Education, 01-813 Warsaw, Poland"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Department of Endocrinology, The Centre of Postgraduate Medical Education , 01-813 Warsaw ,","institution_ids":[]},{"raw_affiliation_string":"Department of Endocrinology, The Centre of Postgraduate Medical Education, 01-813 Warsaw, Poland","institution_ids":[]}]}],"institutions":[],"countries_distinct_count":1,"institutions_distinct_count":1,"corresponding_author_ids":["https://openalex.org/A5022617714"],"corresponding_institution_ids":[],"apc_list":{"value":2888,"currency":"USD","value_usd":2888},"apc_paid":{"value":2888,"currency":"USD","value_usd":2888},"fwci":3.3028,"has_fulltext":true,"cited_by_count":6,"citation_normalized_percentile":{"value":0.91214585,"is_in_top_1_percent":false,"is_in_top_10_percent":true},"cited_by_percentile_year":{"min":89,"max":98},"biblio":{"volume":"9","issue":"9","first_page":"ziaf125","last_page":"ziaf125"},"is_retracted":false,"is_paratext":false,"is_xpac":false,"primary_topic":{"id":"https://openalex.org/T10516","display_name":"Parathyroid Disorders and Treatments","score":0.9990000128746033,"subfield":{"id":"https://openalex.org/subfields/2727","display_name":"Nephrology"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},"topics":[{"id":"https://openalex.org/T10516","display_name":"Parathyroid Disorders and Treatments","score":0.9990000128746033,"subfield":{"id":"https://openalex.org/subfields/2727","display_name":"Nephrology"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},{"id":"https://openalex.org/T10975","display_name":"Bone Tumor Diagnosis and Treatments","score":0.00019999999494757503,"subfield":{"id":"https://openalex.org/subfields/2732","display_name":"Orthopedics and Sports Medicine"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},{"id":"https://openalex.org/T13453","display_name":"Alkaline Phosphatase Research Studies","score":9.999999747378752e-05,"subfield":{"id":"https://openalex.org/subfields/2712","display_name":"Endocrinology, Diabetes and Metabolism"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}}],"keywords":[{"id":"https://openalex.org/keywords/burosumab","display_name":"burosumab","score":0.9959999918937683},{"id":"https://openalex.org/keywords/fibrous-dysplasia","display_name":"fibrous dysplasia","score":0.878000020980835},{"id":"https://openalex.org/keywords/mccune-albright-syndrome","display_name":"McCune-Albright syndrome","score":0.9900000095367432},{"id":"https://openalex.org/keywords/fgf23","display_name":"fibroblast growth factor 23","score":0.9390000104904175},{"id":"https://openalex.org/keywords/hypophosphatemia","display_name":"hypophosphatemia","score":0.9570000171661377},{"id":"https://openalex.org/keywords/monoclonal-antibody-therapy","display_name":"monoclonal antibody therapy","score":0.8199999928474426},{"id":"https://openalex.org/keywords/calcium-and-phosphorus-metabolism","display_name":"calcium and phosphorus metabolism","score":0.7160000205039978},{"id":"https://openalex.org/keywords/alkaline-phosphatase","display_name":"alkaline phosphatase","score":0.8939999938011169},{"id":"https://openalex.org/keywords/bone-pain","display_name":"bone pain","score":0.718999981880188},{"id":"https://openalex.org/keywords/adult-patients","display_name":"adult patients","score":0.45399999618530273}],"concepts":[{"id":"https://openalex.org/C2777004107","wikidata":"https://www.wikidata.org/wiki/Q727008","display_name":"McCune\u2013Albright syndrome","level":4,"score":0.8645006418228149},{"id":"https://openalex.org/C2781362533","wikidata":"https://www.wikidata.org/wiki/Q1410864","display_name":"Fibrous dysplasia","level":2,"score":0.8576087951660156},{"id":"https://openalex.org/C71924100","wikidata":"https://www.wikidata.org/wiki/Q11190","display_name":"Medicine","level":0,"score":0.6286901831626892},{"id":"https://openalex.org/C187212893","wikidata":"https://www.wikidata.org/wiki/Q123028","display_name":"Pediatrics","level":1,"score":0.38413798809051514},{"id":"https://openalex.org/C61434518","wikidata":"https://www.wikidata.org/wiki/Q932510","display_name":"General surgery","level":1,"score":0.33001917600631714},{"id":"https://openalex.org/C126322002","wikidata":"https://www.wikidata.org/wiki/Q11180","display_name":"Internal medicine","level":1,"score":0.2534937858581543},{"id":"https://openalex.org/C141071460","wikidata":"https://www.wikidata.org/wiki/Q40821","display_name":"Surgery","level":1,"score":0.21209877729415894},{"id":"https://openalex.org/C2778367240","wikidata":"https://www.wikidata.org/wiki/Q224513","display_name":"Precocious puberty","level":3,"score":0.04778960347175598},{"id":"https://openalex.org/C71315377","wikidata":"https://www.wikidata.org/wiki/Q11364","display_name":"Hormone","level":2,"score":0.0}],"mesh":[],"locations_count":3,"locations":[{"id":"doi:10.1093/jbmrpl/ziaf125","doi":"https://doi.org/10.1093/jbmrpl/ziaf125","is_oa":true,"landing_page_url":"https://doi.org/10.1093/jbmrpl/ziaf125","pdf_url":null,"source":{"id":"https://openalex.org/S4210170492","display_name":"JBMR Plus","issn_l":"2473-4039","issn":["2473-4039"],"is_oa":true,"is_in_doaj":true,"is_core":true,"listed_in":["cwts-core","doaj","dongbi-d","jufo-1","ki-jl-1","norway-1","oarl","poland-20","russia-white-list-2","vabb-shw"],"host_organization":"https://openalex.org/P4310320595","host_organization_name":"Wiley","host_organization_lineage":["https://openalex.org/P4310320595"],"host_organization_lineage_names":["Wiley"],"type":"journal"},"license":"cc-by-nc","license_id":"https://openalex.org/licenses/cc-by-nc","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"JBMR Plus","raw_type":"journal-article"},{"id":"pmh:oai:pubmedcentral.nih.gov:12366781","doi":null,"is_oa":true,"landing_page_url":"https://pmc.ncbi.nlm.nih.gov/articles/PMC12366781/","pdf_url":"https://pmc.ncbi.nlm.nih.gov/articles/PMC12366781/pdf/","source":{"id":"https://openalex.org/S2764455111","display_name":"PubMed Central","issn_l":null,"issn":null,"is_oa":true,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I1299303238","host_organization_name":"National Institutes of Health","host_organization_lineage":["https://openalex.org/I1299303238"],"host_organization_lineage_names":[],"type":"repository"},"license":"cc-by-nc","license_id":"https://openalex.org/licenses/cc-by-nc","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"JBMR Plus","raw_type":"Text"},{"id":"pmid:40842905","doi":null,"is_oa":false,"landing_page_url":"https://pubmed.ncbi.nlm.nih.gov/40842905","pdf_url":null,"source":{"id":"https://openalex.org/S4306525036","display_name":"PubMed","issn_l":null,"issn":null,"is_oa":false,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I1299303238","host_organization_name":"National Institutes of Health","host_organization_lineage":["https://openalex.org/I1299303238"],"host_organization_lineage_names":[],"type":"repository"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"JBMR plus","raw_type":"Case Reports"}],"best_oa_location":{"id":"doi:10.1093/jbmrpl/ziaf125","doi":"https://doi.org/10.1093/jbmrpl/ziaf125","is_oa":true,"landing_page_url":"https://doi.org/10.1093/jbmrpl/ziaf125","pdf_url":null,"source":{"id":"https://openalex.org/S4210170492","display_name":"JBMR Plus","issn_l":"2473-4039","issn":["2473-4039"],"is_oa":true,"is_in_doaj":true,"is_core":true,"listed_in":["cwts-core","doaj","dongbi-d","jufo-1","ki-jl-1","norway-1","oarl","poland-20","russia-white-list-2","vabb-shw"],"host_organization":"https://openalex.org/P4310320595","host_organization_name":"Wiley","host_organization_lineage":["https://openalex.org/P4310320595"],"host_organization_lineage_names":["Wiley"],"type":"journal"},"license":"cc-by-nc","license_id":"https://openalex.org/licenses/cc-by-nc","version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"JBMR Plus","raw_type":"journal-article"},"sustainable_development_goals":[{"display_name":"Good health and well-being","score":0.9940000176429749,"id":"https://metadata.un.org/sdg/3"}],"study_designs":[{"display_name":"Case Report","id":"https://openalex.org/study-designs/case-report"}],"awards":[],"funders":[],"has_content":{"pdf":true,"grobid_xml":true},"content_urls":{"pdf":"https://content.openalex.org/works/W4412915469.pdf","grobid_xml":"https://content.openalex.org/works/W4412915469.grobid-xml"},"referenced_works_count":21,"referenced_works":["https://openalex.org/W1925356513","https://openalex.org/W1965113966","https://openalex.org/W1983840259","https://openalex.org/W2035541415","https://openalex.org/W2054026360","https://openalex.org/W2090490099","https://openalex.org/W2109177164","https://openalex.org/W2138736651","https://openalex.org/W2170351820","https://openalex.org/W2475962588","https://openalex.org/W2809314614","https://openalex.org/W2948888421","https://openalex.org/W2989227751","https://openalex.org/W2995873363","https://openalex.org/W3163140312","https://openalex.org/W3164119144","https://openalex.org/W4307232963","https://openalex.org/W4321482772","https://openalex.org/W4399027859","https://openalex.org/W4402672566","https://openalex.org/W4410729006"],"related_works":["https://openalex.org/W3163140312","https://openalex.org/W7160377685","https://openalex.org/W4402672566","https://openalex.org/W4408292798","https://openalex.org/W4414760453","https://openalex.org/W4396741766","https://openalex.org/W4308133164","https://openalex.org/W4410729006","https://openalex.org/W7204778068","https://openalex.org/W7084594171"],"abstract_inverted_index":{"mutation":[0],"leads":[1],"to":[2],"overproduction":[3],"of":[4,53,85,116,134,160],"fibroblast":[5],"growth":[6],"factor-23":[7],"(FGF23),":[8],"which":[9],"may":[10],"result":[11],"in":[12,40,103,141,163,196],"hypophosphatemia.":[13],"Burosumab,":[14],"a":[15,139],"monoclonal":[16],"antibody":[17],"against":[18],"FGF23,":[19],"is":[20,155],"approved":[21],"for":[22,57],"the":[23,51,54,156,176],"treatment":[24,64,73,162],"X-linked":[25],"hypophosphatemia":[26,59],"and":[27,38,60,69,82,100,113,144,181,187],"tumor-induced":[28],"osteomalacia.":[29],"There":[30],"are":[31,191],"currently":[32],"no":[33],"data":[34],"on":[35,175],"its":[36],"efficacy":[37],"safety":[39,189],"fibrous":[41,168,198],"dysplasia/MAS":[42,199],"patients.":[43,200],"A":[44],"27-yr-old":[45],"male":[46],"with":[47,65,74,167],"MAS":[48],"was":[49],"under":[50],"care":[52],"Endocrinology":[55],"Department":[56],"persistent":[58],"skeletal":[61],"complications":[62],"despite":[63],"oral":[66],"phosphate":[67],"supplements":[68],"calcitriol.":[70],"He":[71],"started":[72],"burosumab":[75,161],"(1":[76],"mg/kg":[77],"s.c.":[78],"every":[79],"4":[80],"wk)":[81],"achieved":[83],"normalization":[84],"calcium-phosphate":[86,179],"metabolism":[87],"(phosphate":[88],"0.83":[89],"mmol/L":[90],"vs":[91,97,110,123],"0.38":[92],"mmol/L;":[93],"PTH":[94],"70.4":[95],"pg/mL":[96],"177":[98],"pg/mL)":[99],"significant":[101],"reduction":[102,140],"alkaline":[104,117],"phosphatase":[105,118],"activity":[106,119],"(ALP":[107],"620":[108],"IU/L":[109,122],"1182":[111],"IU/L)":[112],"bone":[114,142,182],"fraction":[115],"(BALP":[120],"327":[121],"603":[124],"IU/L).":[125],"No":[126,147],"further":[127,188],"fractures":[128],"were":[129,150],"observed":[130],"during":[131,152],"24":[132],"mo":[133],"treatment.":[135,153],"The":[136,170],"patient":[137,166],"reported":[138,151,158],"pain":[143],"improved":[145],"well-being.":[146],"adverse":[148],"effects":[149,174],"This":[154],"first":[157],"case":[159],"an":[164],"adult":[165,197],"dysplasia/MAS.":[169],"therapy":[171],"had":[172],"positive":[173],"patient's":[177],"well-being,":[178],"balance,":[180],"markers.":[183],"However,":[184],"longer":[185],"follow-up":[186],"studies":[190],"needed":[192],"before":[193],"routine":[194],"use":[195]},"counts_by_year":[{"year":2026,"cited_by_count":5},{"year":2025,"cited_by_count":1}],"updated_date":"2026-11-12T12:28:57.059296","created_date":"2025-08-04T00:00:00"}
