Sinonasal IgG4-related disease: a rare and emerging entity broadening the differential diagnosis in the sinonasal universe

Kaur, Kanwalpreet; Manchanda, Smita; Chatterjee, Prasun; Kaur, Harpreet; Mishra, Deepika; Verma, Hitesh; Kumar, Rajeev; Sagar, Prem; Jain, Deepali; Bhalla, Ashu Seith

doi:10.1007/s00405-020-06564-w

Cited by 12 publications

(37 citation statements)

References 23 publications

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“…Of the 5 patients who had both surgery and steroid treatment, 1 had recurrent disease (20%) which improved after the addition of rituximab. While one case presented by Kaur et al 24 reported no recurrence with the use of rituximab alone, that was not true of the case presented here. While the patient did have initial improvement with his initial two infusions of rituximab in 2012, his disease recurred a year later and persisted despite a third infusion of rituximab and intermittent oral steroids.…”

Section: Treatmentcontrasting

confidence: 82%

“…Ultimately, 20 references meeting the selection criteria were included. [5][6][7][8][9][10][11][12][13][14][15][16][17][18][19][20][21][22][23][24] Epidemiology, presentations, imaging characteristics, locations of involvement, IgG4 serum and histopathologic levels, treatments rendered, and follow-up of all 31 cases (when available) were extracted and summarized.…”

Section: Methodsmentioning

confidence: 99%

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Sinonasal IgG4-RD: Case Report and Systematic Review

Wilson

Brownlee

Rassi

et al. 2021

Preprint

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Section: Treatmentcontrasting

confidence: 82%

Section: Methodsmentioning

confidence: 99%

Sinonasal IgG4-RD: Case Report and Systematic Review

Wilson

Brownlee

Rassi

et al. 2021

Preprint

View full text Add to dashboard Cite

show abstract

“…Of the 5 patients who had both surgery and steroid treatment, 1 had recurrent disease (20%) which improved after the addition of rituximab. While one case presented by Kaur et al 24 reported no recurrence with the use of rituximab alone, this was not the experience of the patient presented here. While the patient did have initial improvement with his initial two infusions of rituximab in 2012, his disease recurred a year later and persisted despite a third infusion of rituximab and intermittent oral steroids.…”

Section: Treatmentcontrasting

confidence: 55%

“…Ultimately, 20 references meeting the selection criteria were included. [5][6][7][8][9][10][11][12][13][14][15][16][17][18][19][20][21][22][23][24] The epidemiology, presentation, imaging characteristics, locations of involvement, IgG4 serum and histopathologic levels, treatments rendered, and follow-up of all 31 cases (when available) were extracted and summarized.…”

Section: Methodsmentioning

confidence: 99%

Sinonasal immunoglobin G4—related disease: case report and review

Wilson

Brownlee

Rassi

et al. 2021

Clinical Case Reports

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“…Various cut-off values were proposed for different sites on involvement, ranging from >10/HPF in the pancreas and liver to >100/HPF for the lacrimal and salivary glands [14]. However, the cut-off values have not been proposed for the sinonasal region IgG4-RD [15]. The IgG4-RD diagnostic criteria are often more than 30 IgG4 plasma cells in high powered field, and IgG4-positive/IgG-positive plasma cell ratio >40% is considered a more powerful diagnostic parameter than IgG4-positive plasma cell count in establishing the diagnosis [8, 16, 17].…”

Section: Discussionmentioning

confidence: 99%

Immunoglobulin G4-Related Disease Presenting as Temporal Bone Lesion with Facial Nerve Palsy

Polianskis

Ivaška

Dadonienė

et al. 2022

ORL

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Immunoglobulin G4-related disease (IgG4-RD) is a systemic condition which could present as local infiltration of skull base structures. We report a case of IgG4-RD with infiltration of the temporal bone and surrounding structures in a patient with systemic vasculitis on systemic steroids. A 31-year-old woman presented with right-sided facial weakness, headache, and right ear hearing loss. On examination, redness and retraction of the right tympanic membrane and facial paresis (House-Brackman IV) were noted. Computed tomography imaging showed mastoiditis, temporal lobe stroke, and brain abscess. Magnetic resonance imaging (MRI) showed infiltration in the infratemporal fossa, nasopharynx, spreading along the Eustachian tube and perineurally along the branches of CN V and CN VII intracranially, forming a dural based mass in the middle cranial fossa. Intracranial mass compressed the temporal lobe of the brain, causing perifocal brain edema. Endoscopic biopsy of the nasopharynx was chosen as the least invasive method. It showed marked fibrosis of the tissue, dense lymphoplasmacytic infiltrates, and an increased number of IGG4-positive plasma cells. Serum IgG4 levels were below the diagnostic criteria of IgG4-RD, but histological characteristics of IgG4-RD were met. The patient was treated with high-dose oral prednisolone. Resolution of symptoms, including facial nerve paresis, was observed and infiltration in the nasopharynx, infratemporal fossa decreased on subsequent MRI tests. No recurrence was noted on the follow-up of 16 months. The case presented itself as a diagnostic challenge for a multidisciplinary team to differentiate pathology caused by either IgG4-RD, systemic vasculitis, or atypic mastoiditis. MRI and histological reports were essential to establish a correct diagnosis.

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Sinonasal IgG4-related disease: a rare and emerging entity broadening the differential diagnosis in the sinonasal universe

Cited by 12 publications

References 23 publications

Sinonasal IgG4-RD: Case Report and Systematic Review

Sinonasal IgG4-RD: Case Report and Systematic Review

Sinonasal immunoglobin G4—related disease: case report and review

Immunoglobulin G4-Related Disease Presenting as Temporal Bone Lesion with Facial Nerve Palsy

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