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J Neurol Surg Rep 2015; 76(02): e265-e269
DOI: 10.1055/s-0035-1564602
Case Report
Georg Thieme Verlag KG Stuttgart · New York

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

Mallory Jayne Highstein
1   Hofstra North Shore, LIJ School of Medicine, Hempstead, New York, United States
,
Jonathan Mallen
1   Hofstra North Shore, LIJ School of Medicine, Hempstead, New York, United States
,
Tristan Tham
2   New York Head and Neck Institute, Lenox Hill Hospital, North Shore-LIJ Health System, New York, New York, United States
,
Tara Brennan
2   New York Head and Neck Institute, Lenox Hill Hospital, North Shore-LIJ Health System, New York, New York, United States
,
Alexandra Boubour
2   New York Head and Neck Institute, Lenox Hill Hospital, North Shore-LIJ Health System, New York, New York, United States
,
Elena Opher
3   Lenox Hill Hospital, Department of Pathology, North Shore, LIJ Healthy System, New York, New York, United States
,
Vira Wolf
3   Lenox Hill Hospital, Department of Pathology, North Shore, LIJ Healthy System, New York, New York, United States
,
Prabhjyot Singh
2   New York Head and Neck Institute, Lenox Hill Hospital, North Shore-LIJ Health System, New York, New York, United States
,
Peter Costantino
2   New York Head and Neck Institute, Lenox Hill Hospital, North Shore-LIJ Health System, New York, New York, United States
› Author Affiliations
Further Information

Publication History

28 January 2015

10 August 2015

Publication Date:
09 October 2015 (online)

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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.

Keywords

immunoglobulin subtype - G4-related disease - foreign body injection - head and neck
 

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