{
  "abstract": "Burosumab provides treatment benefits in children with X-linked hypophosphatemia (XLH). However, children with low rickets severity (Rickets Severity Score [RSS] < 2) were excluded from a previous phase 3, multicenter, active comparator-controlled randomized clinical trial. This descriptive interim analysis of the prospective SUNFLOWER (Study of longitUdinal observatioN For patients with X-Linked hypOphosphatemic rickets/osteomalacia in collaboration With Asian partnERs) cohort study included 69 children with XLH aged ≤13 yr at enrollment. The burosumab and non-burosumab groups included 48 and 65 participants, respectively, with some overlap due to switching. Longitudinal changes in height and biochemical parameters (overall and stratified by disease severity [RSS <2/≥2; alkaline phosphatase </≥ upper limit of normal]) were assessed. In the burosumab all-patients analysis (irrespective of treatment duration or missing follow-up data), height Z-score (initiation: −2.04 ± 1.08; 3 yr: −1.58 ± 1.09), serum phosphate Z-score (initiation: −4.07 ± 1.77; 3 yr: −2.30 ± 1.45), 1,25(OH)2D (initiation: 39.79 ± 22.78 pg/mL; 3 yr: 84.35 ± 28.26 pg/mL), and ratio of tubular maximum phosphate reabsorption to glomerular filtration rate Z-score (initiation: −4.51 ± 2.60; 3 yr: −0.93 ± 2.50) tended to be higher at 3 yr following burosumab treatment. Rickets Severity Score (initiation: 2.38 ± 1.44; 3 yr: 0.83 ± 0.53) and alkaline phosphatase Z-score (initiation: 3.11 ± 1.79; 3 yr: 1.71 ± 1.14) tended to be lower at 3 yr. The subgroup analyses by RSS also showed similar results. Height Z-score, phosphate Z-score, and 1,25(OH)2D tended to be higher at 3 yr in both subgroups, while alkaline phosphatase Z-score tended to be lower. In the subgroup analysis by alkaline phosphatase status, height Z-score tended to be higher at 3 yr only in the ≥upper limit of normal subgroup; the other results were comparable. Overall, height, phosphate homeostasis, and bone health may improve in children with XLH (including RSS <2) following burosumab treatment.",
  "abstract_shown": true,
  "affiliations": [],
  "article_type": "research article",
  "arxiv": null,
  "author_total": 11,
  "authors": [
    {
      "name": "Hee Gyung Kang"
    },
    {
      "name": "Toshimi Michigami",
      "orcid": "0000-0003-4069-5508"
    },
    {
      "name": "Noriyuki Namba",
      "orcid": "0000-0002-3803-4500"
    },
    {
      "name": "Nobuaki Ito",
      "orcid": "0000-0002-4071-5372"
    },
    {
      "name": "Takuo Kubota"
    },
    {
      "name": "Osamu Miyazaki"
    },
    {
      "name": "Masanori Kanematsu"
    },
    {
      "name": "Chikako Hayashi",
      "orcid": "0009-0008-2258-7478"
    },
    {
      "name": "Haruka Ishii"
    },
    {
      "name": "Seiji Fukumoto",
      "orcid": "0000-0003-3610-3469"
    },
    {
      "name": "Keiichi Ozono",
      "orcid": "0000-0002-6517-8825"
    }
  ],
  "bibtex": "https://research.draconitic.com/paper/dd71jqben56zx.bib",
  "cited_by": 0,
  "doi": "10.1093/jbmrpl/ziag168",
  "fulltext_url": "https://pmc.ncbi.nlm.nih.gov/articles/PMC13732572/",
  "id": "dd71jqben56zx",
  "issns": [
    "2473-4039"
  ],
  "landing_url": "https://doi.org/10.1093/jbmrpl/ziag168",
  "language": "en",
  "licence": "CC-BY-4.0",
  "licence_label": "CC BY 4.0",
  "licence_source": "article",
  "licence_url": "https://creativecommons.org/licenses/by/4.0/",
  "metadata_licence": null,
  "native_subjects": [
    [
      "heading",
      "AcademicSubjects/MED00010"
    ],
    [
      "heading",
      "AcademicSubjects/MED00160"
    ],
    [
      "heading",
      "AcademicSubjects/MED00250"
    ],
    [
      "heading",
      "Research Article"
    ],
    [
      "openalex",
      "2727|Parathyroid Disorders and Treatments"
    ]
  ],
  "openalex": "W7222047678",
  "pdf_url": "https://academic.oup.com/jbmrplus/advance-article-pdf/doi/10.1093/jbmrpl/ziag168/71971653/ziag168.pdf",
  "pmcid": "PMC13732572",
  "pmid": "42985459",
  "published": "2026-10-15",
  "publisher": "Oxford University Press",
  "ris": "https://research.draconitic.com/paper/dd71jqben56zx.ris",
  "sources": [
    {
      "first_seen": "2026-11-08T12:22:32Z",
      "native_id": "PMC13732572",
      "source": "pmc",
      "url": "https://doi.org/10.1093/jbmrpl/ziag168"
    }
  ],
  "subjects": [
    {
      "field": "medicine",
      "origin": "openalex:2727|Parathyroid Disorders and Treatments",
      "subfield": "general"
    }
  ],
  "title": "Effectiveness of burosumab in children with X-linked hypophosphatemia with low rickets severity: SUNFLOWER analysis",
  "title_text": "Effectiveness of burosumab in children with X-linked hypophosphatemia with low rickets severity: SUNFLOWER analysis",
  "type": "peer-reviewed",
  "updated_at": "2026-11-08T12:22:32Z",
  "url": "https://research.draconitic.com/paper/dd71jqben56zx",
  "venue": "JBMR Plus",
  "versions": []
}