Journal of Neurological Surgery Reports (Oct 2015)

Probable Immunoglobulin Subtype—G4-Related Disease in the Head and Neck from Foreign Body Injection: A Case Report

  • Mallory Jayne Highstein,
  • Jonathan Mallen,
  • Tristan Tham,
  • Tara Brennan,
  • Alexandra Boubour,
  • Elena Opher,
  • Vira Wolf,
  • Prabhjyot Singh,
  • Peter Costantino

DOI
https://doi.org/10.1055/s-0035-1564602
Journal volume & issue
Vol. 76, no. 02
pp. e265 – e269

Abstract

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Abstract Introduction Immunoglobulin subtype G4-related disease (IgG4-RD) is a fibroinflammatory disease of unknown etiology, with manifestations involving nearly every organ system. Its association with foreign bodies is not established. Here, we present a novel case of IgG4-RD in response to foreign body injection. Case Description A 58-year-old woman presented with history of persistent left facial pain, xerophthalmia, blurred vision, and trismus. The patient's medical history was significant for left-sided temporomandibular joint (TMJ) reconstruction with silicone injection into the joint. Magnetic resonance imaging revealed a lesion in the left skull base. Biopsies demonstrated the cardinal histopathological features of IgG4-RD. The patient was treated with a tapering dose of prednisolone followed by rituximab, resulting in tumor shrinkage and resolution of her symptoms. Discussion This is the first reported case of IgG4-RD potentially precipitated by a foreign body, in this case injected silicone into the TMJ. The pathogenesis and etiology of IgG4-RD is still not fully elucidated, but allergic and reactive inflammatory reactions have been implicated in the disease process. This case report should raise the idea of reactive foreign bodies as a causative agent for IgG4-RD.

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