Evidence map›Paper›PMID 25033066›Full record

Trial reportThe Journal of clinical endocrinology and metabolism2014

A randomized, double blind, placebo-controlled trial of alendronate treatment for fibrous dysplasia of bone.

Alison M Boyce, Marilyn H Kelly, Beth A Brillante, Harvey Kushner, Shlomo Wientroub, Mara Riminucci, Paolo Bianco, Pamela G Robey, Michael T Collins

Open access · bronzeAbstract readRandomized Controlled Trial
In one paragraph

Trial report in The Journal of clinical endocrinology and metabolism, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 79 papers, 5 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
79citing papers in PubMed, 5 pooled it
6.9field-weighted citation impact, top 3% 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

79 citing papers in PubMed, 5 syntheses or guidelines pooled it, 156 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
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  8. Age-Related Changes and Effects of Bisphosphonates on Bone Turnover and Disease Progression in Fibrous Dysplasia of Bone.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2019
    Trial
  9. Activation of RANK/RANKL/OPG Pathway Is Involved in the Pathophysiology of Fibrous Dysplasia and Associated With Disease Burden.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2019
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  15. 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
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19 more citing papers are in PubMed but not listed here.

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

9 authors at 4 institutions in 3 countries.

Alison M BoyceSkeletal Clinical Studies Unit, Craniofacial and Skeletal Diseases Branch (A.M.B., M.H.K., B.A.B., P.G.R., M.T.C.), National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, Maryland 20892; Division of Endocrinology and Diabetes (A.M.B.), Children's National Health System, Washington, DC 20010; Bone Health Program, Division of Orthopaedics and Sports Medicine (A.M.B.), Children's National Health System, Washington, DC 20010; BioMedical Computer Research Institute (H.K.), Philadelphia, Pennsylvania 19115; Department of Pediatric Orthopedics (S.W.), Dana Children's Hospital, Tel-Aviv Sourasky Medical Center, Sackler Faculty of Medicine, Tel-Aviv University, Israel 64239; Department of Molecular Medicine (M.R., P.B.), La Sapienza Universita di Roma, Rome, Italy 00185.
Marilyn H Kelly
Beth A Brillante
Harvey Kushner
Shlomo Wientroub
Mara Riminucci
Paolo Bianco
Pamela G Robey
Michael T Collins
National Institute of Dental and Craniofacial Research · USNational Institutes of Health · USSapienza University of Rome · ITTel Aviv University · IL

Funding

Skeletal Disorders and Mineral Homeostasis SectionZIADE000649 · NIDCR · NATIONAL INSTITUTE OF DENTAL & CRANIOFACIAL RESEARCH · PI COLLINS, MICHAEL · 2009 to 2025
$32.8M
The role of stem cells in skeletal health and diseaseZIADE000380 · NIDCR · NATIONAL INSTITUTE OF DENTAL & CRANIOFACIAL RESEARCH · PI ROBEY, PAMELA G · 2009 to 2025
$30.6M
Intramural NIH HHSTelethon GGP09227
6 · The paper itself

Abstract

contextFibrous dysplasia (FD) is a rare skeletal disorder, resulting in deformity, fracture, functional impairment, and pain. Bisphosphonates have been advocated as a potential treatment.

objectiveTo determine the efficacy of alendronate for treatment of FD.

designTwo-year randomized, double-blind, placebo-controlled trial.

settingClinical research center. PATIENTS: Forty subjects with polyostotic FD (24 adults, 16 children). Subjects were randomized and stratified by age.

interventionsStudy drug was administered over a 24 month period in 6 month cycles (6 months on, 6 months off). Alendronate dosing was stratified: 40 mg daily for subjects >50 kg, 20 mg for 30-50 kg, 10 mg for 20-30 kg.

main outcome measuresPrimary endpoints were bone turnover markers, including serum osteocalcin, and urinary NTX-telopeptides. Secondary endpoints included areal bone mineral density (aBMD), pain, skeletal disease burden score, and functional parameters including the 9-min walk test and manual muscle testing.

resultsClinical data was collected on 35 subjects who completed the study. There was a decline in NTX-telopeptides in the alendronate group (P = .006), but no significant difference in osteocalcin between groups. The alendronate group had an increase in areal BMD in normal bone at the lumbar spine (P = .006), and in predetermined regions of FD (P < .001). There were no significant differences in pain scores, skeletal disease burden scores, or functional parameters between the groups.

conclusionsAlendronate treatment led to a reduction in the bone resorption marker NTX-telopeptides, and improvement in aBMD, but no significant effect on serum osteocalcin, pain, or functional parameters.

Indexed as

AdolescentAdultAlendronateBiomarkersBone DensityBone Density Conservation AgentsBone RemodelingChildCollagen Type IDouble-Blind MethodFemaleFibrous Dysplasia of BoneHumansMaleMiddle AgedOsteocalcinAlendronateBiomarkersBone Density Conservation AgentsCollagen Type Icollagen type I trimeric cross-linked peptideOsteocalcinPeptides

Identifiers

PMID25033066
PMCPMC4223439
OpenAlexW2138736651

What OpenQuestion holds

Textmetadata
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.