Collet-Sicard syndrome associated with probable intracranial vasculitic arteriopathy: a case report.
- 1Institute of Neurology, National Hospital of Sri Lanka, Colombo, Sri Lanka. [email protected].
- 2Institute of Neurology, National Hospital of Sri Lanka, Colombo, Sri Lanka.
Abstract
Background
Collet-Sicard syndrome is unilateral palsy of the glossopharyngeal, vagus, accessory, and hypoglossal nerves. It is most commonly caused by neoplastic, traumatic, or vascular lesions at the skull base. To our knowledge, its association with intracranial vasculitis has not been reported previously.Case presentation
A 35-year-old Sri Lankan woman presented with a 3-month history of dysphagia, dysphonia, and nasal dysarthria. She had had a left-sided cerebral infarction 1 year earlier. Examination showed right-sided cranial nerves IX, X, XI, and XII palsies, consistent with Collet-Sicard syndrome. Contrast-enhanced computed tomography and magnetic resonance imaging of the skull base were normal, with no evidence of jugular foramen pathology, skull-base osteomyelitis, venous thrombosis, or basal meningitis. Cerebrospinal fluid analysis revealed mild lymphocytic pleocytosis and elevated protein. Extensive investigations excluded malignancy, infection, sarcoidosis, IgG4-related disease, and systemic vasculitis. Review of prior neuroimaging showed a corpus callosal infarction. Because of this unusual stroke location, digital subtraction angiography was performed which demonstrated multifocal intracranial arterial stenoses and luminal irregularities suggestive of possible intracranial vasculitis. In the absence of another explanation, we hypothesised that the lower cranial neuropathies may have resulted from vasculitic involvement of the vasa nervorum. She was treated with corticosteroids, rituximab, azathioprine, and aspirin. Despite treatment, severe bulbar dysfunction persisted, and she later died from aspiration pneumonia.Conclusions
This case highlights a rare association between Collet-Sicard syndrome and intracranial vasculitic arteriopathy. When structural skull-base lesions are absent, vasculitic neuropathy should be considered as a potential cause of multiple lower cranial neuropathies. Corpus callosal infarction, an uncommon stroke pattern, should prompt investigation for unusual vascular aetiologies including central nervous system vasculitis.
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BibTeX
@article{ruwanpathirana2026colletsicard,
title = {{Collet-Sicard syndrome associated with probable intracranial vasculitic arteriopathy: a case report.}},
author = {Pramith Ruwanpathirana and Bimsara Senanayake},
journal = {BMC neurology},
year = {2026},
month = aug,
doi = {10.1186/s12883-026-05247-z},
url = {https://europepmc.org/article/MED/42972523},
note = {CC BY-NC-ND},
}
RIS
TY - JOUR
TI - Collet-Sicard syndrome associated with probable intracranial vasculitic arteriopathy: a case report.
AU - Pramith Ruwanpathirana
AU - Bimsara Senanayake
PY - 2026
DA - 2026/08/05
JO - BMC neurology
DO - 10.1186/s12883-026-05247-z
UR - https://europepmc.org/article/MED/42972523
AB - Background Collet-Sicard syndrome is unilateral palsy of the glossopharyngeal, vagus, accessory, and hypoglossal nerves. It is most commonly caused by neoplastic, traumatic, or vascular lesions at the skull base. To our knowledge, its association with intracranial vasculitis has not been reported previously.Case presentation A 35-year-old Sri Lankan woman presented with a 3-month history of dysphagia, dysphonia, and nasal dysarthria. She had had a left-sided cerebral infarction 1 year earlier. Examination showed right-sided cranial nerves IX, X, XI, and XII palsies, consistent with Collet-Sicard syndrome. Contrast-enhanced computed tomography and magnetic resonance imaging of the skull base were normal, with no evidence of jugular foramen pathology, skull-base osteomyelitis, venous thrombosis, or basal meningitis. Cerebrospinal fluid analysis revealed mild lymphocytic pleocytosis and elevated protein. Extensive investigations excluded malignancy, infection, sarcoidosis, IgG4-related disease, and systemic vasculitis. Review of prior neuroimaging showed a corpus callosal infarction. Because of this unusual stroke location, digital subtraction angiography was performed which demonstrated multifocal intracranial arterial stenoses and luminal irregularities suggestive of possible intracranial vasculitis. In the absence of another explanation, we hypothesised that the lower cranial neuropathies may have resulted from vasculitic involvement of the vasa nervorum. She was treated with corticosteroids, rituximab, azathioprine, and aspirin. Despite treatment, severe bulbar dysfunction persisted, and she later died from aspiration pneumonia.Conclusions This case highlights a rare association between Collet-Sicard syndrome and intracranial vasculitic arteriopathy. When structural skull-base lesions are absent, vasculitic neuropathy should be considered as a potential cause of multiple lower cranial neuropathies. Corpus callosal infarction, an uncommon stroke pattern, should prompt investigation for unusual vascular aetiologies including central nervous system vasculitis.
SN - 1471-2377
ER -