Disturbed ATP and AMPK homeostasis in an Ank <sub>F377del</sub> mouse model for craniometaphyseal dysplasia
mice. Limitations of systemic administration of SBI-0206965 include its off-target effects on other cell types and the inability to inhibit AMPK only on fusing osteoclasts. Nonetheless, this proof-of-principle study reveals an important role of the ATP-AMPK axis in CMD pathogenesis. Take-home message: Suppression of increased activation of AMPK restores the function of osteoclasts, suggesting that abnormal energy metabolism is an integral component of the disease phenotype in CMD.
Authors
- Ernst Reichenberger (ORCID: https://orcid.org/0000-0003-2229-2481)
- Koen van de Wetering (ORCID: https://orcid.org/0000-0003-1905-2743)
- Ayano Hatori (ORCID: https://orcid.org/0009-0001-1719-8469)
- Shyam Kishor Sah (ORCID: https://orcid.org/0000-0001-9843-0356)
- I‐Ping Chen (ORCID: https://orcid.org/0000-0002-7349-3091)
Institutions
- Thomas Jefferson University (US)
- UConn Health (US)
Publication Details
- Journal
- bioRxiv (Cold Spring Harbor Laboratory)
- Published
- 2026-04-17
- DOI
- https://doi.org/10.64898/2026.04.15.717889
- Primary Topic
- Bone Metabolism and Diseases
- Type
- article
- Field-Weighted Citation Impact
- 0.00
Funders
- National Institutes of Health
- National Institute of Arthritis and Musculoskeletal and Skin Diseases
- National Institute of Dental and Craniofacial Research