Sinonasal immunoglobin G4—related disease: case report and review

Wilson, Caleb; Brownlee, Benjamin P.; Rassi, Edward T. El; McKinney, Kibwei A.

doi:10.1002/ccr3.5095

Cited by 4 publications

(15 citation statements)

References 30 publications

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“…In patients with a history of CRSwNP, symptoms of facial swelling and nasal obstruction may be mistaken for recalcitrant disease. Here, we demonstrate that these symptoms resulted from concurrent IgG4‐RD and CRSwNP as was also the case in one other report 16 …”

Section: Discussionsupporting

confidence: 87%

“…While autoimmune diseases tend to have a female sex predisposition, there is a stark contrast of male predominance seen in IgG4‐RD, with male to female ratios ranging from 1.6 to 4:1 11,14,15 . However, when the disease process is localized to the head and neck, the sex distribution is roughly equal 13,16,17 . We saw a near equal male‐to‐female ratio in our cohort (4:3).…”

Section: Discussionmentioning

confidence: 54%

“…For our patients the duration of symptoms was quite extensive, up to 50 years in one patient, before a diagnosis was made. Although other IgG4‐RD forms involve multiple organs, sinonasal involvement is typically isolated to sinonasal cavity as was the case in our cohort 1,7,16 . Patients may have allergic features, however, presenting symptoms are often related to the destructive nature of the disease 11,17 .…”

Section: Discussionmentioning

confidence: 61%

“…IgG4‐RD of the sinonasal cavity has non‐specific imaging manifestations that vary considerably on both CT and MRI 21 . Bony and tissue destruction on imaging raises concern for malignancy, which must be ruled out histologically 16 . Our CT findings were non‐descriptive of the etiology of disease.…”

Section: Discussionmentioning

confidence: 85%

“…Furthermore, the presence of epithelioid cell granulomas and prominent neutrophilic infiltrate generally exclude the diagnosis of IgG4‐RD 22 . A variety of cut‐off points have been used to define the density of infiltrate ranging from >10 to >50 IgG4+ plasma cell per high powered field 22,23 or immunohistochemical staining showing IgG4+/IgG+ plasma cell ratio >40–50% 11,16,22 . In the present study, a plasma cell ratio >40% was used to define lymphoplasmacytic infiltration.…”

Section: Discussionmentioning

confidence: 96%

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Sinonasal IgG4‐related sclerosing disease: A rare entity and challenging diagnosis

Hess

Lobo

Leon

et al. 2022

Laryngoscope Investig Oto

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Objectives To describe the rare presentation, imaging and histological findings, and treatments in patients with IgG4‐related disease (IgG4‐RD) and diagnostic pitfalls and difficulties. Methods Cases of sinonasal IgG4‐RD were retrieved, and clinicopathological features were reviewed. Results Seven cases of sinonasal IgG4‐RD were identified over an 11‐year period, including four males and three females, with an age range of 19–66 years (median 58 years). Patients presented with symptoms related to the mass effect of the lesions or the destructive nature of the disease including fullness, swelling, obstruction, and pain. Serum IgG and IgG4 levels, IgG/IgG4 ratios, storiform fibrosis, obliterative phlebitis, and plasma cell infiltration were seen in varying proportions. Bony erosion and tissue inflammation were present in some cases. Conclusion Sinonasal IgG4‐RD is exceedingly rare among other IgG4‐RD and varied in its clinical presentation thus posing as a clinically difficult disease to diagnosis. Proper clinical, pathological, and immunohistopathological analysis is required for accurate diagnosis. Such disease should be considered in all cases of similar presentation to those in this study. Level of Evidence: 4.

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Section: Discussionsupporting

confidence: 87%

Section: Discussionmentioning

confidence: 54%

Section: Discussionmentioning

confidence: 61%

Section: Discussionmentioning

confidence: 85%

Section: Discussionmentioning

confidence: 96%

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Sinonasal IgG4‐related sclerosing disease: A rare entity and challenging diagnosis

Hess

Lobo

Leon

et al. 2022

Laryngoscope Investig Oto

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Ear, Nose, and Throat Manifestations of Vasculitis and Other Systemic Autoimmune Diseases

Wasserman¹,

Chari²,

Gray³

et al. 2023

Rheumatic Disease Clinics of North America

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IgG4 related disease masquerading as a sinonasal mass: a case report and review of literature

Krishnan,

Rangachari,

Katti

et al. 2024

Int J Otorhinolaryngol Head Neck Surg

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IgG4-related disease (IgG4-RD) is a relatively new autoimmune fibroinflammatory disease. Most seen to affect the pancreas and biliary tract, IgG4-RD of the nose and paranasal sinuses is very rare. Known to have tumour-like clinical presentations, they can present with varying levels of dysfunction of the organs they affect. In the paranasal sinuses, it can present with nasal block, discharge, bleeds, and sometimes, with symptoms that arise as a consequence of bony erosions and subsequent extension of the disease. We present a case of a 52-year-old lady, who presented with a painful swelling over the malar region with discoloration of the overlying skin. Radiological evaluation helps to check for bony erosions and soft tissue infiltration. Histopathology with immunohistochemistry helps to confirm the diagnosis of IgG4-RD. Most patients benefit from a long course of oral steroids and regular follow-ups, that help to prevent recurrence and ensure a good quality of life for the patient.

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Sinonasal immunoglobin G4—related disease: case report and review

Abstract: Immunoglobin G4-related disease (IgG4-RD) is a chronic fibro-inflammatory condition that presents as a single or multiple tumefactive lesions affecting virtually any organ system. Here we report a case of recurrent sinonasal IgG4-RD and review the literature of this evolving entity.

Cited by 4 publications

References 30 publications

Sinonasal IgG4‐related sclerosing disease: A rare entity and challenging diagnosis

Sinonasal IgG4‐related sclerosing disease: A rare entity and challenging diagnosis

Ear, Nose, and Throat Manifestations of Vasculitis and Other Systemic Autoimmune Diseases

IgG4 related disease masquerading as a sinonasal mass: a case report and review of literature

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