Evidence map›Paper›PMID 33984553›Full record

ArticleBone2021

Burosumab treatment for fibrous dysplasia.

Anne Gladding, Vivian Szymczuk, Bethany A Auble, Alison M Boyce

Open access · greenAbstract readCase Reports
In one paragraph

Article in Bone, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 28 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
28citing papers in PubMed, 1 pooled it
4.3field-weighted citation impact, top 5% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

28 citing papers in PubMed, 1 synthesis or guideline pooled it, 38 citations in OpenAlex.

  1. Comparison of traditional and new treatments for fibrous dysplasia: a systematic review and meta-analysis.Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA · 2026
    Pooled it
  2. Trial
  3. FGF23 - A hormone produced by bone and has many faces.Reviews in endocrine & metabolic disorders · 2026
    Review
  4. Article
  5. Article
  6. Article
  7. Burosumab treatment for FGF23-related hypophosphatemia in a two-year-old girl with McCune-Albright syndrome.Clinical pediatric endocrinology : case reports and clinical investigations : official journal of the Japanese Society for Pediatric Endocrinology · 2026
    Article
  8. Diagnosis and Management of Hypophosphatemic Disorders.Endocrine practice : official journal of the American College of Endocrinology and the American Association of Clinical Endocrinologists · 2025
    Review
  9. Review
  10. Article
  11. Article
  12. Bone pain in fibrous dysplasia does not rely on aberrant sensory nerve sprouting or neuroma formation.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2025
    Article
  13. Article
  14. Review
  15. Review
  16. Article
  17. Update on the medical management of fibrous dysplasia of the bone.Therapeutic advances in endocrinology and metabolism · 2025
    Review
  18. Review
  19. Review
  20. Metabolic Health and Disease: A Role of Osteokines?Calcified tissue international · 2023
    Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

4 authors at 4 institutions in 1 country.

Anne GladdingChildren's Hospital of Wisconsin, Medical College of Wisconsin, Milwaukee, WI, USA.
Vivian SzymczukMetabolic Bone Disorders Unit, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, MD, USA; National Institute of Child Health and Development, National Institutes of Health, Bethesda, MD, USA.
Bethany A AubleChildren's Hospital of Wisconsin, Medical College of Wisconsin, Milwaukee, WI, USA.
Alison M BoyceMetabolic Bone Disorders Unit, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, MD, USA. Electronic address: alison.boyce@nih.gov.
Children's Hospital of Wisconsin · USMedical College of Wisconsin · USNational Institute of Dental and Craniofacial Research · USNational Institutes of Health · US

Funding

Metabolic Bone Disorders UnitZIADE000758 · NIDCR · NATIONAL INSTITUTE OF DENTAL & CRANIOFACIAL RESEARCH · PI BOYCE, ALISON · 2021 to 2025
$8.9M
Intramural NIH HHS ZIA DE000758
6 · The paper itself

Abstract

backgroundFibrous dysplasia/McCune-Albright syndrome (FD/MAS) is a rare mosaic disorder of Gα CASE DESCRIPTION: A 7-year-old boy with severe FD/MAS presented with persistent hypophosphatemia and skeletal complications despite conventional treatment with oral phosphate and calcitriol. He was started on burosumab and achieved sustained normalization of serum phosphorus and marked improvement in alkaline phosphatase levels. This was accompanied by an encouraging clinical response, including decreased bone pain, improved muscle strength, and improved ambulation. No adverse effects of burosumab therapy were observed.

conclusionsThis is the first reported case of burosumab treatment in a patient with FD/MAS. The encouraging biochemical and clinical response in this patient highlights the need for future studies to explore the safety and efficacy of burosumab in the FD/MAS pediatric population.

Indexed as

Familial Hypophosphatemic RicketsHypophosphatemiaAntibodies, MonoclonalAntibodies, Monoclonal, HumanizedChildFibroblast Growth Factor-23HumansMaleAntibodies, MonoclonalAntibodies, Monoclonal, HumanizedburosumabFGF23 protein, humanFibroblast Growth Factor-23FGF23HypophosphatemiaMcCune-Albright syndromeOsteomalaciaRickets

Identifiers

PMID33984553
PMCPMC8272883
OpenAlexW3163140312

What OpenQuestion holds

Textmetadata
LicenceTDM
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.