A Case of Sjögren's Syndrome Associated With Trigeminal Neuropathy and Enhancement of the Mandibular Nerve at the Foramen Ovale: A Case Report and a Review of the Differential Diagnosis and Mechanisms of the Disease

Farran, Mohamed Ziad; Kesserwani, Hassan

doi:10.7759/cureus.19463

Cited by 3 publications

(5 citation statements)

References 18 publications

(17 reference statements)

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“…More recently, Farran et al described a case of trigeminal neuropathy associated with Sjogren syndrome, suggesting that an immunopathological process may have affected the trigeminal nerve in this patient. 21 Worse pain outcomes for patients with concomitant autoimmune disease and TN in our study suggest a role for inflammatory processes in the pathogenesis of TN pain. Indeed, a potential role for inflammation in the etiology of TN has recently been investigated in the literature.…”

Section: Discussionmentioning

confidence: 65%

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Preoperative Characteristics and Postoperative Pain Outcomes in Trigeminal Neuralgia With Concomitant Autoimmune Disease

Kalluri¹,

So²,

Ran³

et al. 2023

Neurosurgery

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BACKGROUND AND OBJECTIVES: Although the association between multiple sclerosis and trigeminal neuralgia (TN) is well established, little is known about TN pain characteristics and postoperative pain outcomes after microvascular decompression (MVD) in patients with TN and other autoimmune diseases. In this study, we aim to describe presenting characteristics and postoperative outcomes in patients with concomitant TN and autoimmune disease who underwent an MVD. METHODS: A retrospective review of all patients who underwent an MVD at our institution between 2007 and 2020 was conducted. The presence and type of autoimmune disease were recorded for each patient. Patient demographics, comorbidities, clinical characteristics, postoperative Barrow Neurological Institute (BNI) pain and numbness scores, and recurrence data were compared between groups. RESULTS: Of the 885 patients with TN identified, 32 (3.6%) were found to have concomitant autoimmune disease. Type 2 TN was more common in the autoimmune cohort (P = .01). On multivariate analysis, concomitant autoimmune disease, younger age, and female sex were found to be significantly associated with higher postoperative BNI score (P = .04, <0.001, and <0.001, respectively). In addition, patients with autoimmune disease were more likely to experience significant pain recurrence (P = .009) and had shorter time to recurrence on Kaplan-Meier analysis (P = .047), although this relationship was attenuated on multivariate Cox proportional hazards regression. CONCLUSION: Patients with concomitant TN and autoimmune disease were more likely to have Type 2 TN, had worse postoperative BNI pain scores at the final follow-up after MVD, and were more likely to experience recurrent pain than patients with TN alone. These findings may influence postoperative pain management decisions for these patients and support a possible role for neuroinflammation in TN pain.

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Section: Discussionmentioning

confidence: 65%

Section: Discussionmentioning

confidence: 99%

Preoperative Characteristics and Postoperative Pain Outcomes in Trigeminal Neuralgia With Concomitant Autoimmune Disease

Kalluri¹,

So²,

Ran³

et al. 2023

Neurosurgery

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“…The present patient was diagnosed with Sjögren's syndrome during hospitalization. As trigeminal neuropathy has been reported to be associated with Sjögren's syndrome ( 46 - 48 ), it was possibly involved in this case. The mechanism underlying neuropathy following SARS-CoV-2 mRNA vaccination remains unclear.…”

Section: Discussionmentioning

confidence: 70%

New-onset Immune-mediated Necrotizing Myopathy and Trigeminal Neuropathy after SARS-CoV-2 mRNA Vaccination in a Patient with Rheumatoid Arthritis and Sjögren's Syndrome

Tsuzuki Wada,

Yokota,

Inayoshi

et al. 2023

Intern. Med.

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We present the case of a 42-year-old woman with rheumatoid arthritis and Sjögren's syndrome treated with adalimumab who developed immune-mediated necrotizing myopathy (IMNM) and trigeminal neuropathy after severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) mRNA vaccination. Trigeminal neuralgia and elevated serum creatine kinase levels emerged 12 days post-vaccination, followed by myalgia in the femoral muscles. IMNM was histologically diagnosed. The pathogenesis may involve molecular mimicry between the SARS-CoV-2 spike glycoprotein and autologous tissues triggered by vaccination. This case emphasizes the association between SARS-CoV-2 vaccination, tumor necrosis factor inhibitor, IMNM, and trigeminal neuropathy, as well as the importance of monitoring immune-mediated adverse events following SARS-CoV-2 vaccination in patients with autoimmune disease.

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“…GDNF phosphorylates the rearranged during transfection (RET) tyrosine kinase receptor that triggers downstream signaling pathways that allow malignant cell migration [ 18 ]. Adenoid cystic carcinoma and squamous cell carcinoma are the most common malignancies that can spread perineurally [ 3 ]. Although not the most common, basal cell carcinoma has been described in the literature.…”

Section: Discussionmentioning

confidence: 99%

“…The trigeminal nerve divides into three branches at the Gasserian ganglion in Meckel’s cave [ 3 ]. The three branches consist of the ophthalmic nerve (V1), the maxillary nerve (V2), and the mandibular nerve (V3).…”

Section: Introductionmentioning

confidence: 99%

A Case of Secondary Trigeminal Neuropathy Due to Local Malignant Invasion of the Maxillary and Mandibular Nerves at the Skull Base: A Case Report With Review of Differential Diagnosis

Pasala¹,

Park²,

Kesserwani³

2022

Cureus

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Trigeminal neuropathies (TNp) are a group of well-characterized disorders that involve damage to or infiltration of the trigeminal nerve. The underlying etiology of trigeminal neuropathy can be traumatic, inflammatory, autoimmune, paraneoplastic, malignant, and very rarely infectious. We present a case of trigeminal neuropathy due to local malignant invasion of the mandibular nerve with mandibular nerve enhancement at the foramen ovale and foramen rotundum. In the process, we review various etiologies of trigeminal neuropathy associated with trigeminal nerve involvement at the foramina. We emphasize the importance of a comprehensive evaluation in patients with trigeminal neuropathy, which includes searching for perineural spread or invasion by a local head and neck malignancy, as well as ruling out an inflammatory or autoimmune etiology. Our case also demonstrates that a higher field strength magnet can reveal pathology unseen with a lower field strength magnet.

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A Case of Sjögren's Syndrome Associated With Trigeminal Neuropathy and Enhancement of the Mandibular Nerve at the Foramen Ovale: A Case Report and a Review of the Differential Diagnosis and Mechanisms of the Disease

Cited by 3 publications

References 18 publications

Preoperative Characteristics and Postoperative Pain Outcomes in Trigeminal Neuralgia With Concomitant Autoimmune Disease

Preoperative Characteristics and Postoperative Pain Outcomes in Trigeminal Neuralgia With Concomitant Autoimmune Disease

New-onset Immune-mediated Necrotizing Myopathy and Trigeminal Neuropathy after SARS-CoV-2 mRNA Vaccination in a Patient with Rheumatoid Arthritis and Sjögren's Syndrome

A Case of Secondary Trigeminal Neuropathy Due to Local Malignant Invasion of the Maxillary and Mandibular Nerves at the Skull Base: A Case Report With Review of Differential Diagnosis

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