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Peer-reviewedCC BYBone reports

Automated detection and quantification of bone disorganization in hypophosphatasia before and after asfotase alfa: A case report

  • Simon Zhang1
  • Cat Shore-Lorenti1
  • Cherie Chiang2
  • Frances Milat1
  • Peter R. Ebeling3
  • Roger Zebaze1
  1. 1Monash University School of Clinical Sciences at Monash Health, Clayton, Victoria, Australia
  2. 2Austin Health, Heidelberg, Victoria, Australia
  3. 3Division of Endocrinology, Department of Medicine, Temerty Faculty of Medicine, University of Toronto, Toronto, Canada
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Abstract

Conventional imaging poorly captures bone disorganization, a pathological feature of hypophosphatasia (HPP). This single-patient case report applied ALIGNOGRAM to previously published open-access distal radius radiographs from a 16-year-old with severe HPP before and after six months of asfotase alfa. It detected overt, subtle, and covert lesions and quantified reduced lesion burden post-treatment. Residual disorganization persisted. These findings support preliminary technical feasibility, with independent validation in larger prospective cohorts required to establish diagnostic and treatment-response validity.

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Cite

BibTeXRIS
BibTeX
@article{zhang2026automated,
  title = {{Automated detection and quantification of bone disorganization in hypophosphatasia before and after asfotase alfa: A case report}},
  author = {Simon Zhang and Cat Shore-Lorenti and Cherie Chiang and Frances Milat and Peter R. Ebeling and Roger Zebaze},
  journal = {Bone reports},
  year = {2026},
  month = oct,
  doi = {10.1016/j.bonr.2026.101963},
  url = {https://europepmc.org/article/PMC/PMC13722107},
  note = {CC BY},
}
RIS
TY  - JOUR
TI  - Automated detection and quantification of bone disorganization in hypophosphatasia before and after asfotase alfa: A case report
AU  - Simon Zhang
AU  - Cat Shore-Lorenti
AU  - Cherie Chiang
AU  - Frances Milat
AU  - Peter R. Ebeling
AU  - Roger Zebaze
PY  - 2026
DA  - 2026/10/23
JO  - Bone reports
DO  - 10.1016/j.bonr.2026.101963
UR  - https://europepmc.org/article/PMC/PMC13722107
AB  - Conventional imaging poorly captures bone disorganization, a pathological feature of hypophosphatasia (HPP). This single-patient case report applied ALIGNOGRAM to previously published open-access distal radius radiographs from a 16-year-old with severe HPP before and after six months of asfotase alfa. It detected overt, subtle, and covert lesions and quantified reduced lesion burden post-treatment. Residual disorganization persisted. These findings support preliminary technical feasibility, with independent validation in larger prospective cohorts required to establish diagnostic and treatment-response validity.
SN  - 2352-1872
ER  -