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Efficacy of antiresorptive agents in fibrous dysplasia and McCune Albright syndrome, a systematic review and meta-analysis

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Abstract

Fibrous dysplasia (FD) is a rare skeletal disorder in which normal bone is replaced by a fibro-osseous tissue, resulting in possible deformities and fractures. The aim of this systematic review and meta-analysis was to synthesize the available evidence on the use of antiresorptive drugs in FD in terms of changes in bone turnover markers (BTMs), bone mineral density (BMD), and reducing pain. Three databases were searched in October 2022, with an update in July 2023. Of the 1037 studies identified, 21 were retained after eligibility assessment. A random-effects model was used to calculate global effect size and the corresponding standard error. Pamidronate and Denosumab were the most reported drugs in a total of 374 patients assessed. The initiation of treatments was accompanied by an average reduction of 40.5% [CI95% -51.6, -29.3] in the bone resorption parameters, and 22.0% [CI95% -31.9, -12.1] in the parameters of bone formation after 6–12 months. BMD was increased in both FD lesions and in the unaffected skeleton. Pain was reduced by 32.7% [CI95% -52.7, -12.6] after 6–12 months of treatment, and by 44.5% [CI95% -65.3, -23.6] after a mean 41.2 months of follow-up. The variation in pain was highly correlated to variation in bone resorption (R2 = 0.08, p < 0.0001) and formation parameters (R2 = 0.17, p < 0.0001). This study supports the overall efficacy of antiresorptive therapies in terms of reducing bone remodeling, improving bone density, and pain in FD.

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Data availability

The datasets generated and analysed during the current study are available from the corresponding author on reasonable request.

Abbreviations

99Tc-MDP:

99Tc-methylene diphosphonate

ALP:

Alkaline phosphatase

BMD:

Bone mineral density

BMSCs:

Bone marrow stromal cells

BPI:

Brief pain inventory

BPs:

Bisphosphonates

BTMs:

Bone turnover markers

cAMP:

Cyclic adenosine monophosphate (cAMP)

CI:

Confidence interval

CT:

Computed tomography

CTX:

Serum C-telopeptides of type I collagen

d:

Day

Dmab:

Denosumab

DPD:

Deoxypyridinoline

DXA:

Dual-energy x-ray absorptiometry

FD:

Fibrous dysplasia

FDG:

[18F]-fluorodeoxyglucose

Gsα:

Stimulatory α-subunit of the G-protein

i.v.:

Intravenous

Il-6:

Interleukin-6

IQR:

Interquartile range

MAS:

McCune Albright syndrome

MFD:

Monostotic FD

NGF:

Nerve growth factor

NIH:

National Institutes of Health

NMD:

Normalized mean difference

NRS:

Numeric rating scale

NSAIDs:

Non-steroidal anti-inflammatory drugs

NTX:

Urinary N-telopeptides of type I collagen

OC:

Octeocalcin

ONJ:

Osteonecrosis of the jaw

P1NP:

Procollagen I N-propeptide

PDE4:

Phosphodiesterase type 4

PET:

Positron emission tomography

PFD:

Polyostotic FD

PRISMA:

Preferred Reporting Items for Systematic reviews and Meta-Analysis

PTHrP:

Parathyroid hormone-related protein

RCTs:

Randomized control trials

s.c.:

Subcutaneous

SBS:

Skeletal burden score

SD:

Standard deviation

SE:

Standard error

SS:

Suramin sodium

TrkA:

Tyrosine kinase A

TRPA1:

Transient receptor potential ankyrin 1

VAS:

Visual analog scale

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This research was supported by the Nantes University Hospital, the Région Pays de la Loire – France (PULSAR), the Association française des chirurgiens de la face (AFCF), and the Fondation les Gueules Cassées (2018-51). The sponsors had no role in the study design, collection, analysis, interpretation and writing of the manuscript.

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Authors and Affiliations

  1. Faculty of Dental Medicine and Oral Health Sciences, McGill University, Montreal, QC, H3A 1G1, Canada

    Hélios Bertin, Mahmoud S. Moussa & Svetlana Komarova

  2. Nantes Université, CHU Nantes, Service de chirurgie maxillo-faciale et stomatologie, F-44000, Nantes, France

    Hélios Bertin

  3. Nantes Université, UnivAngers, CHU Nantes, INSERM, CNRS, CRCI2NA, F-44000, Nantes, France

    Hélios Bertin

  4. Shriners Hospitals for Children - Canada, Montreal, QC, H4A 0A9, Canada

    Mahmoud S. Moussa & Svetlana Komarova

Authors
  1. Hélios Bertin
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  2. Mahmoud S. Moussa
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  3. Svetlana Komarova
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Contributions

All authors contributed to the study conception and design. Idea for the article: Hélios Bertin; literature search: Hélios Bertin and Mahmoud Moussa; data analysis: Hélios Bertin; supervision: Svetlana Komarova; writing – original draft preparation: Hélios Bertin; writin – review and editing: Hélios Bertin, Mahmoud Moussa, and Svetlana Komarova.

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Correspondence to Hélios Bertin.

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Bertin, H., Moussa, M.S. & Komarova, S. Efficacy of antiresorptive agents in fibrous dysplasia and McCune Albright syndrome, a systematic review and meta-analysis. Rev Endocr Metab Disord 24, 1103–1119 (2023). https://doi.org/10.1007/s11154-023-09832-2

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  • DOI: https://doi.org/10.1007/s11154-023-09832-2

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