Involvement of Multiple Trigeminal Nerve Branches in IgG4-Related Orbital Disease

Elkhamary, Sahar; Cruz, Antônio Augusto Velasco e; Zotin, Maria Clara Zanon; Cintra, Murilo Bicudo; Akaishi, Patrícia; Galindo‐Ferreiro, Alicia; Alkatan, Hind M.; Chahud, Fernando

doi:10.1097/iop.0000000000001733

Cited by 8 publications

(9 citation statements)

References 20 publications

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“…In the present case, separate involvement of perineural optic nerves without perineural spreading and branches of the trigeminal nerves was reported. Perineural spreading between the maxillary nerve and infraorbital nerve was noted in the present case, but not in previous reports [17,20]. No previous IgG4-RON cases mentioned the bilateral maxillary nerves with perineural involvement of infraorbital nerves, which makes the present case exceptional.…”

Section: Discussioncontrasting

confidence: 84%

“…Two studies have reported that the infraorbital nerve was enlarged in approximately 30-38.5% of their IgG4-ROD cases [3,10]. The frontal nerves, supraorbital nerves, infraorbital nerves, lesser palatine nerves, and superior alveolar nerves were the involved sites of the trigeminal nerves in these patients [3,17]. Lesion of the trigeminal nerve branch was particularly unique, and 64% of trigeminal nerve branch lesions observed in Sogabe et al's study were bilateral [3].…”

Section: Discussionmentioning

confidence: 85%

“…Of the cranial nerves affected in patients with IgG4-RD, the bilateral maxillary nerves are rarely involved [17]. A case of an IgG4-related cerebral pseudotumor with perineural spreading along all branches of the trigeminal nerves causing right compressive optic neuropathy has been reported [20].…”

Section: Discussionmentioning

confidence: 99%

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Immunoglobulin G4-Related Orbital Disease with Bilateral Optic Perineuritis and Maxillary Nerves Involvement: A Case Report

Hung

2020

Ophthalmol Ther

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Immunoglobulin G4-related optic neuropathy caused by optic perineuritis is a rare complication of immunoglobulin G4-related disease (IgG4-RD). Herein, we report a 38-year-old Asian man with history of sinusitis who presented with painless blurred vision and proptosis for over 6 months. Examination with the Hertel exophthalmometer revealed 21.5 mm on both eyes. Magnetic resonance imaging revealed a doughnut sign encircling the right optic nerve, bilateral tram-track signs on both optic nerves, enlarged bilateral maxillary nerves with perineural spreading to the infraorbital nerves, hypertrophy of extraocular muscles, and pansinusitis. Visual evoked potentials displayed bilateral delayed P100 latency, indicating bilateral optic neuropathy. Biopsy with functional endoscopic sinus surgery demonstrated diffuse dense lymphoplasmacytic infiltrate and fibrosis. IgG4-positive plasma cells exceeded 50 cells per high-power field while the overall IgG4/IgG ratio was above 40%. Serological studies unveiled extremely high serum concentrations of IgG4 (2650 mg/dL), and the calculated serum IgG4/IgG ratio was 100%. These comprehensive features supported the diagnosis of IgG4-RD with bilateral optic perineuritis, branches of trigeminal nerve involvement, and pansinusitis. The visual acuity improved slightly following the initiation of treatment with corticosteroids, but it became worse again during the tapering course. Following another course of corticosteroids followed by subsequent immunosuppressant treatment with azathioprine, vision in both eyes ultimately improved during the 2-year follow-up period.

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Section: Discussioncontrasting

confidence: 84%

Section: Discussionmentioning

confidence: 85%

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Immunoglobulin G4-Related Orbital Disease with Bilateral Optic Perineuritis and Maxillary Nerves Involvement: A Case Report

Hung

2020

Ophthalmol Ther

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“…The radiological studies of the patients were reviewed by three experienced neuroradiologists blinded to patient history, symptoms, and histopathologic data, using a digital imaging and communications in Medicine (DICOM) viewer system (Horos, https://horosproject.org/) and Enterprise Imaging Agfa HealthCare. The diagnosis of trigeminal perineural disease was based on the classical findings of nerve enlargement or enhancement, obliteration of the fat planes around the nerves and their foramina, and enlargement and/or erosion of foramina, canals and fissures [19][20][21].…”

Section: Imaging Techniquementioning

confidence: 99%

“…In 2011 Katsura et al reported a patient with isolated enlargement of V2 and V3 branches due to an inflammatory process diagnosed as IgG4-RD [9]. Following this report, TGN enlargement, especially of the infraorbital nerve, has been strongly associated with IgG4-RD [10][11][12][13][14][15][16][17][18], Widespread involvement of the TGN is an important feature of IgG4-related disease [19] and less frequently with idiopathic inflammation [20] and lymphoid hyperplasia [16]. Herein we present a new finding of group of patients with orbital disease in whom different TGN divisions were enlarged not only with IgG4-RD, but also with vasculitis and RDD with bilateral perineural enhancement even in unilateral eye disease.…”

Section: Introductionmentioning

confidence: 99%

Enlargement of Multiple Branches of the Trigeminal Nerve in Non-Neoplastic Orbital Diseases: Review of MR Imaging in 14 Cases

SM¹,

MC²,

Cintra³

et al. 2022

JCMI

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Background: Head and neck perineural disease is typically associated with cancer, but it can be associated with non-neoplastic conditions, especially IgG4-Related disease. Purpose: We present the MRI findings of trigeminal nerve involvement in 14 patients with 3 different types of non-neoplastic orbital disease. Material and Methods: A retrospective review of MR images of patients with enlargement of distinct TGN divisions who were evaluated in a neuro-ophthalmology clinic and were subsequently proved to have non-neoplastic orbital infiltrative or inflammatory diseases. Three experienced radiologists reviewed the MRI studies looking for extent and characteristics of perineural disease along the pathway of the 3 TGN divisions. Results: MRI demonstrated enhancement of at least one TGN division in 14 patients with bilateral or unilateral orbital infiltrative non-neoplastic conditions (22 orbits). Six patients had IgG4-RD, 3 RDD, and 5 c-ANCA-associated vasculitis. Overall, 13 (92.8%) patients displayed enlargement of V2, 12 (85.7%) of V1 and 7 (50%) of V3. Simultaneous enlargement of V1 and V2 was detected in 11 (78.6%) patients, and all 3 divisions were simultaneously involved in 6 (42.8%). Other radiological findings included abnormal contrast enhancement and soft tissue thickening in the cavernous sinus, Meckel’s cave, and/or the cisternal segment of the trigeminal nerve. Conclusions: Although trigeminal thickening associated with non-malignant orbital infiltration is commonly considered a sign of IgG4-RD, other conditions such as vasculitis and RDD should be considered in the differential diagnosis of these uncommon cases.

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The “great imitator”: IgG4‐related disease of the oral cavity. Two case reports and scoping review

Azzi,

Magnoli,

Krepysheva

et al. 2024

Head & Neck

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This study aimed to review the lesser‐known intraoral manifestations of immunoglobulin G4‐related disease (IgG4‐RD). In this paper we report an unprecedented case of oral IgG4‐RD mimicking angiolymphoid hyperplasia with eosinophilia (ALHE), and another case presenting as plasma cell gingivitis. We then performed a scoping review of published cases of IgG4‐RD involving the oral cavity. The following data were collected for each case: age, sex, intraoral site(s) involved, clinical appearance, imaging features, serum IgG4 values, histopathology, treatment, and follow‐up duration. Fifty‐one cases of oral IgG4‐RD were published in literature. The hard palate and jaw bones were the two main locations reported, while the histological identification of a IgG4/IgG plasma cells ratio ≥40% was fundamental for diagnosis. Conversely, the pathological features of storiform fibrosis and obliterative phlebitis were not common. Future reports regarding oral IgG4‐RD should report clear adherence to the recognized international diagnostic criteria of the disease.

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Involvement of Multiple Trigeminal Nerve Branches in IgG4-Related Orbital Disease

Cited by 8 publications

References 20 publications

Immunoglobulin G4-Related Orbital Disease with Bilateral Optic Perineuritis and Maxillary Nerves Involvement: A Case Report

Immunoglobulin G4-Related Orbital Disease with Bilateral Optic Perineuritis and Maxillary Nerves Involvement: A Case Report

Enlargement of Multiple Branches of the Trigeminal Nerve in Non-Neoplastic Orbital Diseases: Review of MR Imaging in 14 Cases

The “great imitator”: IgG4‐related disease of the oral cavity. Two case reports and scoping review

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