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Genetic Causes of Inflammatory Bone Disease
Book chapter

Genetic Causes of Inflammatory Bone Disease

James Verbsky and Polly J Ferguson
Textbook of Autoinflammation, pp.455-470
Springer International Publishing
02/14/2019
DOI: 10.1007/978-3-319-98605-0_25

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Abstract

This chapter focuses on monogenic autoinflammatory disorders that affect bone. The presence of sterile bone inflammation may be accompanied by inflammation of the skin and intestinal tract. The pathophysiology varies by syndrome and includes dysregulation of the IL-1 pathway or aberrant intracellular signaling defects leading to activation of innate immune cells including osteoclasts. These are rare disorders with variable outcomes. IL-1 inhibitors have been used successfully to decrease inflammation in Majeed syndrome, deficiency of the interleukin receptor antagonist and for non-osseous manifestations of neonatal onset multisystem inflammatory disease. For other disorders such as cherubism, treatment remains challenging. Recognition of additional monogenic autoinflammatory is likely as this is a very new field of investigation.
Interleukin-1 Cherubism SH3BP2 NLRP3 Pstpip2 Neonatal onset multisystem inflammatory disease (NOMID) Schnitzler syndrome Primary hypertrophic osteoarthropathy Deficiency of the interleukin-1 receptor antagonist (DIRA) LPIN2 Majeed syndrome

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