Evidence map›Paper›PMID 41020249›Full record

ArticleFrontiers in medicine2025

Case Report: A fatal case of CNS actinomycosis mimicking Tolosa-Hunt syndrome: Is it time to reevaluate diagnostic criteria?

Natalie Bristol, Courtney Venegas, Isha Snehal, Mathias Maisenbacher, Ryan Higgins, Thomas Auen, Catherine Cichon, Richard Hankins, Christie Barnes, Sahara Cathcart and 4 more

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PubMed Full text at PMC Publisher
In one paragraph

Article in Frontiers in medicine, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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3 · Its place in the literature

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4 · The record

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5 · Who and what money

Authors and funding

14 authors.

Natalie BristolDepartment of Internal Medicine, University of Nebraska Medical Center, Omaha, NE, United States.
Courtney VenegasDepartment of Neurology, University of Nebraska Medical Center, Omaha, NE, United States.
Isha SnehalNeurology, Mayo Clinic Minnesota, Rochester, MN, United States.
Mathias MaisenbacherDepartment of Neurology, University of Nebraska Medical Center, Omaha, NE, United States.
Ryan HigginsDepartment of Otolaryngology, University of Nebraska Medical Center, Omaha, NE, United States.
Thomas AuenDepartment of Pathology, Microbiology and Immunology, University of Nebraska Medical Center, Omaha, NE, United States.
Catherine CichonDepartment of Internal Medicine, University of Nebraska Medical Center, Omaha, NE, United States.
Richard HankinsDepartment of Internal Medicine, University of Nebraska Medical Center, Omaha, NE, United States.
Christie BarnesDepartment of Otolaryngology, University of Nebraska Medical Center, Omaha, NE, United States.
Sahara CathcartDepartment of Pathology, Microbiology and Immunology, University of Nebraska Medical Center, Omaha, NE, United States.
Ana Yuil-ValdesDepartment of Pathology, Microbiology and Immunology, University of Nebraska Medical Center, Omaha, NE, United States.
Jie ChenDepartment of Pathology, Microbiology and Immunology, University of Nebraska Medical Center, Omaha, NE, United States.
Cindy SchmidtDepartment of Pathology, Microbiology and Immunology, University of Nebraska Medical Center, Omaha, NE, United States.
Subin MathewDepartment of Neurology, University of Nebraska Medical Center, Omaha, NE, United States.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Background: Tolosa-Hunt Syndrome (THS) is a rare diagnosis characterized by headaches, painful ophthalmoplegia, and granulomatous inflammation of the cavernous sinus. It is a diagnosis of exclusion but can mimic many other conditions, including central nervous system (CNS) actinomycosis. This systematic review evaluated the frequency of CNS actinomycosis initially diagnosed as THS and evaluated lapses in the current diagnostic criteria which were related to poor outcomes. Case report: We present a case of a 43-year-old man who was diagnosed with THS 10 weeks prior to his final hospital presentation. Previous infectious work up was negative. He returned with altered mental status and was found to have leptomeningeal enhancement, vasculitis, and acute infarcts. He decompensated while on broad-spectrum antimicrobials. The patient was ultimately found to have CNS actinomycosis when next generation sequencing identified Results: A total of 344 records were analyzed for title/abstract review and ultimately revealed Eight cases of CNS actinomycosis initially diagnosed as THS. 62.5% of cases had dental history prior to symptom onset. Cerebral spinal fluid cultures were obtained in 75% percent of cases, and all were negative for Actinomyces. 88% of cases were diagnosed by histopathology evaluation and 25% of cases were fatal. Conclusion: With the low reported prevalence of CNS actinomycosis, the inadequacy of current testing for Actinomyces, and the significant overlap of symptoms with THS, it is essential that clinicians are aware of CNS actinomycosis as a potential infectious etiology if early and potentially curative treatment is to be provided. The current definition of THS may encourage harmful practices and should be revised.

Indexed as

central nervous system (CNS) actinomyces infectionheadachesmagnetic resonance imaging (MRI)non-specific granulomatous inflammation of the cavernous sinuspainful ophthalmoplegiaserologic next-generation sequencing (NGS)soft tissue mass involving the cavernous sinus

Identifiers

PMID41020249
PMCPMC12462049

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