Pain in fibrous dysplasia: Identifying nociceptive mechanisms in a preclinical model

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AudienciaPúblico en generales_ES
CoberturaMéxicoes_ES
Fecha de ingreso2026-10-05T16:33:47Z
Fecha de publicación2025-01-01
ResumenPain is a common symptom of fibrous dysplasia (FD), a rare mosaic disorder characterized by fibro-osseous lesions in the bone. Despite the prevalence of pain in FD patients, there is little knowledge about the nociceptive mechanisms and few efficacious treatments. As such, understanding FD pain is essential for patient care. The overall aim of this study was to identify nocifensive behaviors and potential underlying mechanisms in a transgenic mouse model of FD, previously shown to display high face and translational validity. Significant nocifensive behaviors were observed in FD mice (male and female), compared to control mice in the burrowing, grid hanging, home cage activity, and wheel running assays. These changes corresponded to lesion development, as visualized by X-ray imaging. Behavioral deficits improved when analgesics were administered, indicating a nociceptive origin. Tibias and femurs from FD mice demonstrated characteristic FD lesions and the presence of mono-and multi-nucleated CD68+ cells, calcitonin gene-related peptide sensory nerve fibers, and vascularization. Lumbar dorsal root ganglia from male FD mice displayed increased staining for activating transcription factor-3 and tyrosine hydroxylase neurons. No difference was observed in the spinal cords between the FD and control groups for glial cell presence and neuropeptide expression. Bone marrow stromal cells were obtained from FD and control mice and cultured in vitro. FD cells developed an increased concentration of inflammatory cytokines (IL-6, tumor necrosis factor-Alpha), chemokines (monocyte chemoattractant protein, keratinocyte chemoattractant/human growth-regulated oncogene), and nerve growth factor as compared to controls. Taken together, this study demonstrated for the first time that nociceptive mechanisms such as axonal growth in FD lesions, nerve injury, and inflammation may contribute to FD pain, and it provides a foundation for conducting further studies of pain-and disease-modifying therapeutics for FD patients.es_ES
Doihttps://doi.org/10.1093/jbmr/zjaf039es_ES
URIhttps://riuat.uat.edu.mx/handle/123456789/5565
Idiomaenes_ES
EditorialOxford University Presses_ES
RelaciónJournal of Bone and Mineral Researches_ES
URL relacionadohttps://doi.org/10.1093/jbmr/zjaf039es_ES
DerechosAcceso abierto (Metadatos de producción científica)es_ES
Licenciahttp://purl.org/coar/access_right/c_abf2es_ES
FuenteJournal of Bone and Mineral Research
Palabra clavebone paines_ES
Palabra clavefibrous dysplasiaes_ES
Palabra clavein vivoes_ES
Palabra clavemechanismes_ES
Palabra clavepaines_ES
TítuloPain in fibrous dysplasia: Identifying nociceptive mechanisms in a preclinical modeles_ES
TipoArtículoes_ES
ArbitradoHa sido Arbitradoes_ES
AutorHopkins, Chelsea
AutorDe Castro, Luis Fernandez
AutorBenthin, Julie
AutorDiaz-Delcastillo, Marta
AutorManjappa, Pravallika
AutorBoyce, Alison
AutorMendoza, Ruth Elena Martinez
AutorMora, Juan Antonio Vazquez
AutorLopez-Delgado, Giovanni Emmanuel
AutorGomez, Lizeth Yazmin Ponce
AutorMohamed, Khaled Elhady
AutorLinley, John E
AutorCollins, Michael T
AutorJimenez-Andrade, Juan Miguel
AutorHeegaard, Anne-Marie
AutorHopkins, Chelseaes_ES
AutorDe Castro, Luis Fernandezes_ES
AutorBenthin, Juliees_ES
AutorDiaz-Delcastillo, Martaes_ES
AutorManjappa, Pravallikaes_ES
AutorBoyce, Alisones_ES
AutorMendoza, Ruth Elena Martinezes_ES
AutorMora, Juan Antonio Vazquezes_ES
AutorLopez-Delgado, Giovanni Emmanueles_ES
AutorGomez, Lizeth Yazmin Poncees_ES
AutorMohamed, Khaled Elhadyes_ES
AutorLinley, John Ees_ES
AutorCollins, Michael Tes_ES
AutorJimenez-Andrade, Juan Migueles_ES
AutorHeegaard, Anne-Mariees_ES
InstituciónUniversidad Autónoma de Tamaulipas
InstituciónUniversidad Autónoma de Tamaulipases_ES
Número7es_ES
Rango de páginas891-903es_ES
URL relacionadahttps://doi.org/10.1093/jbmr/zjaf039
Tipo de artículoIndexado
Tipo de artículoIndexadoes_ES
Volumen40es_ES

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