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Peer-reviewedCC BY-NCBone reports

Early-onset medication-related osteonecrosis of the jaw in adolescents with Duchenne muscular dystrophy receiving long-term antiresorptive combined with glucocorticoid therapy: Two case reports

  1. 1Sainte-Justine University Hospital Center, Montreal, Canada

Abstract

Background

Youth with Duchenne muscular dystrophy (DMD) are at high risk of osteoporosis due to progressive muscle weakness and chronic glucocorticoid (GC) therapy. Current guidelines recommend routine vertebral fracture screening leading to earlier antiresorptive initiation. Medication-related osteonecrosis of the jaw (MRONJ) is a well-established complication of antiresorptives in adults but is exceedingly rare in youth, with no reported cases in children or adolescents with DMD.Methods

We describe two adolescents with DMD who developed MRONJ following dental extraction during antiresorptive therapy for GC-induced osteoporosis.Results

Case 1: A 15-year-old on long-term deflazacort began zoledronic acid at age 11.8 years for a vertebral fracture. MRONJ developed at age 15 post dental extraction, seven months after their last infusion. Case 2: An 18-year-old receiving bisphosphonates from age 8 years, was switched to denosumab at age 18 years. MRONJ occurred six months later, following tooth extraction. In both cases, antiresorptives were suspended and conservative management led to complete healing of MRONJ. Antiresorptives were later resumed for pain and fracture prevention, with no recurrence after one year. Review of more than 1000 pediatric patients treated with bisphosphonates or denosumab identified no prior reports of MRONJ in children or adolescents with DMD.Conclusions

To our knowledge, these are the two youngest reported cases of MRONJ in patients with DMD. They highlight a potential vulnerability related to prolonged GC exposure combined with long-term antiresorptive therapy. Although pediatric MRONJ risk remains extremely low, clinicians should be aware of this complication, particularly when invasive dental procedures are planned.

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BibTeX
@article{rio2026earlyonset,
  title = {{Early-onset medication-related osteonecrosis of the jaw in adolescents with Duchenne muscular dystrophy receiving long-term antiresorptive combined with glucocorticoid therapy: Two case reports}},
  author = {Agathe Rio and Patricia Olivier and Coralie Leblicq and Nathalie Alos and Melissa Fiscaletti},
  journal = {Bone reports},
  year = {2026},
  month = oct,
  doi = {10.1016/j.bonr.2026.101960},
  url = {https://europepmc.org/article/PMC/PMC13717085},
  note = {CC BY-NC},
}
RIS
TY  - JOUR
TI  - Early-onset medication-related osteonecrosis of the jaw in adolescents with Duchenne muscular dystrophy receiving long-term antiresorptive combined with glucocorticoid therapy: Two case reports
AU  - Agathe Rio
AU  - Patricia Olivier
AU  - Coralie Leblicq
AU  - Nathalie Alos
AU  - Melissa Fiscaletti
PY  - 2026
DA  - 2026/10/19
JO  - Bone reports
DO  - 10.1016/j.bonr.2026.101960
UR  - https://europepmc.org/article/PMC/PMC13717085
AB  - Background  Youth with Duchenne muscular dystrophy (DMD) are at high risk of osteoporosis due to progressive muscle weakness and chronic glucocorticoid (GC) therapy. Current guidelines recommend routine vertebral fracture screening leading to earlier antiresorptive initiation. Medication-related osteonecrosis of the jaw (MRONJ) is a well-established complication of antiresorptives in adults but is exceedingly rare in youth, with no reported cases in children or adolescents with DMD.Methods  We describe two adolescents with DMD who developed MRONJ following dental extraction during antiresorptive therapy for GC-induced osteoporosis.Results  Case 1: A 15-year-old on long-term deflazacort began zoledronic acid at age 11.8 years for a vertebral fracture. MRONJ developed at age 15 post dental extraction, seven months after their last infusion. Case 2: An 18-year-old receiving bisphosphonates from age 8 years, was switched to denosumab at age 18 years. MRONJ occurred six months later, following tooth extraction. In both cases, antiresorptives were suspended and conservative management led to complete healing of MRONJ. Antiresorptives were later resumed for pain and fracture prevention, with no recurrence after one year. Review of more than 1000 pediatric patients treated with bisphosphonates or denosumab identified no prior reports of MRONJ in children or adolescents with DMD.Conclusions  To our knowledge, these are the two youngest reported cases of MRONJ in patients with DMD. They highlight a potential vulnerability related to prolonged GC exposure combined with long-term antiresorptive therapy. Although pediatric MRONJ risk remains extremely low, clinicians should be aware of this complication, particularly when invasive dental procedures are planned.
SN  - 2352-1872
ER  -