Increased neutrophil infiltration, IL-1 production and a SAPHO syndrome-like phenotype in PSTPIP2-deficient mice

Hsiu Jung Liao, I. Tsu Chyuan, Chien Sheng Wu, Shu Wha Lin, Kun Hung Chen, Huei-Fang Tsai, Ping Ning Hsu

研究成果: 雜誌貢獻文章同行評審

19 引文 斯高帕斯(Scopus)

摘要

Objective. Proline-serine-threonine-phosphatase-interacting protein 2 (PSTPIP2) is involved in macrophage activation, neutrophil motility and osteoclast differentiation. However, the role of PSTPIP2 in inflammation and autoinflammatory diseases is still not clear. In this study, we generated PSTPIP2 knockout (Pstpip2-/-) mice to investigate its phenotype and role in autoinflammatory diseases. Methods. We constructed a Pstpip2-targeting vector and generated Pstpip2-/- mice. The phenotype and immunopathology of Pstpip2-/- mice were analysed. Results. All Pstpip2-/- mice developed paw swelling, synovitis, hyperostosis and osteitis, resembling SAPHO syndrome, an inflammatory disorder of the bone, skin and joints. Multifocal osteomyelitis was found in inflamed paws, with increased macrophage and marked neutrophil infiltrations in the bone, joint and skin. Profound osteolytic lesions with markedly decreased bone volume density developed in paws and limbs. Neutrophil-attracting chemokines and IL-1β were markedly elevated in inflamed tissues. Conclusion. Our study suggests that PSTPIP2 could play a role in innate immunity and development of autoinflammatory bone disorders, and may be associated with the pathogenesis of human SAPHO syndrome.
原文英語
文章編號keu481
頁(從 - 到)1317-1326
頁數10
期刊Rheumatology
54
發行號7
DOIs
出版狀態已發佈 - 七月 1 2015

ASJC Scopus subject areas

  • 風濕病
  • 藥學(醫學)

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