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Tolosa-Hunt Syndrome in a Kenyan Patient: Case Report and Review of the Literature

Received: 4 April 2023    Accepted: 11 July 2023    Published: 20 July 2023
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Abstract

Tolosa-Hunt syndrome is a rare cause of painful ophthalmoplegia accompanied by ipsilateral ocular motor palsies, oculosympathetic paralysis and peri-orbital pain around the distribution of the ophthalmic division of the trigeminal nerve. Though the cause is unknown, idiopathic inflammation has been implicated. The pathology can be localized in the cavernous sinus, superior orbital fissure or the apex of the orbital cavity. Localization of the pathology requires a combination of careful history, physical examination and imaging, preferably MRI. We present a 40 year old Kenyan female patient with Tolosa-Hunt syndrome. She presented with left-sided periorbital headache and ptosis. Further examination revealed left-sided trochlear and abducens palsy, and partial left-sided oculomotor palsy. She had allodynia and hyperesthesia on the distribution of the left ophthalmic division of the trigeminal nerve. After an initial normal head CT scan, a magnetic resonance imaging revealed a soft-tissue hyperintensity extending from the left cavernous sinus, superior orbital fissure and apex of the orbit. She responded positively to steroid therapy, with cessation of headaches, correction of ptosis and ophthalmoplegia and an improved quality of life. There was minimal improvement in vision, with persisting pallor of the disc. This case shines the spotlight on a relatively rare disorder; whose diagnosis requires careful history, examination and interpretation of MRI findings.

Published in World Journal of Medical Case Reports (Volume 4, Issue 3)
DOI 10.11648/j.wjmcr.20230403.11
Page(s) 32-34
Creative Commons

This is an Open Access article, distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution and reproduction in any medium or format, provided the original work is properly cited.

Copyright

Copyright © The Author(s), 2024. Published by Science Publishing Group

Keywords

Tolosa-Hunt, Ophthalmoplegia, Ptosi, MRI

References
[1] Murtaza G, Konowitz N, Lu H, Faqah A, Kuruvilla A. An Interesting Case of Tolosa-Hunt Syndrome in a Young Male. J Investig Med High Impact Case Rep. 2017 Jan 1; 5 (1).
[2] Amine MM. Tolosa-Hunt Syndrome - Case Report. SM J Neurol Neurosci. 2018; 4 (1): 1022.
[3] Förderreuther S, Straube A. The criteria of the International Headache Society for Tolosa-Hunt syndrome need to be revised. Neurol 1999; 246: 371–7.
[4] Schatz NJ, Farmer P: Tolosa-Hunt syndrome: the pathology of painful ophthalmoplegia. In: Smith JL, ed. Neuroophthalmology. Symposium of the University of Miami and the Bascom Palmer Eye Institute. Vol VI. St Louis: Mosby, 1972: 102–12.
[5] Inzafari D, Sita D, Marconi GP, et al. The Tolosa-Hunt syndrome: further clinical and pathogenetic considerations based on the study of eight cases. J Neurol 1981; 224: 221–8.
[6] Tolosa E. Periarteritic lesions of the carotid siphon with the clinical features of a carotid infraclinoid aneurysm. J Neurol Neurosurg Psychiatry 1954; 17: 300–2.
[7] Hunt WE, Meagher JN, Le Fever HE, et al. Painful ophthalmoplegia. Its relation to indolent inflammation of the cavernous sinus. Neurology 1961; 11: 56–62.
[8] Aron-Rosa D, Doyon D, Sallamon G, et al. Tolosa-Hunt syndrome. Ann Ophthalmol 1978; 10: 1161–8.
[9] Headache Classification Committee of the International Headache Society (IHS). The International Classification of Headache Disorders, 3rd edition. Cephalalgia. 2018; 38 (1): 1-211.
[10] Arshad A, Nabi S, Panhwar MS, Rahil A. Tolosa-Hunt syndrome: an arcane pathology of cavernous venous sinus. BMJ Case Rep. 2015; 2015.
[11] Zhang X, Zhou Z, Steiner TJ, Zhang W, Liu R, Dong Z, Wang X, Wang R, Yu S. Validation of ICHD-3 beta diagnostic criteria for 13.7 Tolosa-Hunt syndrome: analysis of 77 cases of painful ophthalmoplegia. Cephalalgia. 2014; 34 (8): 624–32.
[12] Colnaghi S, Versino M, Marchioni E, Pichiecchio A, Bastianello S, Cosi V, et al. ICHD-II diagnostic criteria for Tolosa-Hunt syndrome in idiopathic inflammatory syndromes of the orbit and/or the cavernous sinus. Cephalagia. 2008; 28: 577–84.
[13] LaMantia L, Curone M, Rapaport AM, Bussone G. Tolosa-Hunt syndrome: critical literature review based on HIS 2004 criteria. Cephalagia. 2006; 26: 772–81.
[14] Kline LB. The Tolosa-Hunt syndrome. Surv Ophthalmol 1982; 27: 79–95.
[15] Özbenli T, Akpýnar K, Doðru H, Ýncesu L. Tolosa-Hunt syndrome: focus on MRI findings and diagnostic criteria. J Headache Pain. 2013; 14 (Suppl 1): P157.
[16] Kline LB, Hoyt WF. The Tolosa-Hunt syndrome. J Neurol Neurosurg Psychiatry. 2001; 71: 577–82.
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  • APA Style

    Paul Bundi Karau, Winnie Mueni Saumu. (2023). Tolosa-Hunt Syndrome in a Kenyan Patient: Case Report and Review of the Literature. World Journal of Medical Case Reports, 4(3), 32-34. https://doi.org/10.11648/j.wjmcr.20230403.11

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    ACS Style

    Paul Bundi Karau; Winnie Mueni Saumu. Tolosa-Hunt Syndrome in a Kenyan Patient: Case Report and Review of the Literature. World J. Med. Case Rep. 2023, 4(3), 32-34. doi: 10.11648/j.wjmcr.20230403.11

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    AMA Style

    Paul Bundi Karau, Winnie Mueni Saumu. Tolosa-Hunt Syndrome in a Kenyan Patient: Case Report and Review of the Literature. World J Med Case Rep. 2023;4(3):32-34. doi: 10.11648/j.wjmcr.20230403.11

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  • @article{10.11648/j.wjmcr.20230403.11,
      author = {Paul Bundi Karau and Winnie Mueni Saumu},
      title = {Tolosa-Hunt Syndrome in a Kenyan Patient: Case Report and Review of the Literature},
      journal = {World Journal of Medical Case Reports},
      volume = {4},
      number = {3},
      pages = {32-34},
      doi = {10.11648/j.wjmcr.20230403.11},
      url = {https://doi.org/10.11648/j.wjmcr.20230403.11},
      eprint = {https://article.sciencepublishinggroup.com/pdf/10.11648.j.wjmcr.20230403.11},
      abstract = {Tolosa-Hunt syndrome is a rare cause of painful ophthalmoplegia accompanied by ipsilateral ocular motor palsies, oculosympathetic paralysis and peri-orbital pain around the distribution of the ophthalmic division of the trigeminal nerve. Though the cause is unknown, idiopathic inflammation has been implicated. The pathology can be localized in the cavernous sinus, superior orbital fissure or the apex of the orbital cavity. Localization of the pathology requires a combination of careful history, physical examination and imaging, preferably MRI. We present a 40 year old Kenyan female patient with Tolosa-Hunt syndrome. She presented with left-sided periorbital headache and ptosis. Further examination revealed left-sided trochlear and abducens palsy, and partial left-sided oculomotor palsy. She had allodynia and hyperesthesia on the distribution of the left ophthalmic division of the trigeminal nerve. After an initial normal head CT scan, a magnetic resonance imaging revealed a soft-tissue hyperintensity extending from the left cavernous sinus, superior orbital fissure and apex of the orbit. She responded positively to steroid therapy, with cessation of headaches, correction of ptosis and ophthalmoplegia and an improved quality of life. There was minimal improvement in vision, with persisting pallor of the disc. This case shines the spotlight on a relatively rare disorder; whose diagnosis requires careful history, examination and interpretation of MRI findings.},
     year = {2023}
    }
    

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    AB  - Tolosa-Hunt syndrome is a rare cause of painful ophthalmoplegia accompanied by ipsilateral ocular motor palsies, oculosympathetic paralysis and peri-orbital pain around the distribution of the ophthalmic division of the trigeminal nerve. Though the cause is unknown, idiopathic inflammation has been implicated. The pathology can be localized in the cavernous sinus, superior orbital fissure or the apex of the orbital cavity. Localization of the pathology requires a combination of careful history, physical examination and imaging, preferably MRI. We present a 40 year old Kenyan female patient with Tolosa-Hunt syndrome. She presented with left-sided periorbital headache and ptosis. Further examination revealed left-sided trochlear and abducens palsy, and partial left-sided oculomotor palsy. She had allodynia and hyperesthesia on the distribution of the left ophthalmic division of the trigeminal nerve. After an initial normal head CT scan, a magnetic resonance imaging revealed a soft-tissue hyperintensity extending from the left cavernous sinus, superior orbital fissure and apex of the orbit. She responded positively to steroid therapy, with cessation of headaches, correction of ptosis and ophthalmoplegia and an improved quality of life. There was minimal improvement in vision, with persisting pallor of the disc. This case shines the spotlight on a relatively rare disorder; whose diagnosis requires careful history, examination and interpretation of MRI findings.
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Author Information
  • Department of Internal Medicine, School of Medicine and Health Sciences, Kenya Methodist University, Meru, Kenya

  • Chuka County Referral Hospital, Chuka, Kenya

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