Abstract
Tolosa-Hunt syndrome (THS) is an idiopathic, granulomatous inflammation in the cavernous sinus, supraorbital fissure or orbit. Although the pathophysiology of this idiopathic granulomatous inflammatory process is not clear, it has been suggested that cerebrospinal fluid (CSF) analyses could be helpful in understanding the underlying mechanisms. We report two cases of the 3rd Edition of the International Classification of Headache Disorders (ICHD-3)-classified THS who presented with headache and diplopia. After early treatment with intravenous methylprednisolone, the symptoms of two patients were significantly improved. On outpatient follow-up, neither patient had a relapse. It is worth noting that the isoelectric focusing showed identical CSF and serum oligoclonal IgG bands (OBs). Such bands, “mirror pattern bands”, are one of five standardized patterns (i.e., type 1 = absence of bands in serum and CSF; type 2 = presence of OBs in CSF; type 3 = presence of OBs in CSF and additional identical OBs in both serum and CSF, indicative of intrathecal synthesis; type 4 = presence of identical OBs in both serum and CSF, “mirror pattern”; and type 5 = presence of a monoclonal band in serum and CSF). Previous studies have only found OBs in the CSF of THS (type 4). The finding of “mirror pattern bands” is interesting as it reveals an underlying possible systemic immune activation mechanism in THS.
Similar content being viewed by others
Data Availability
Not applicable.
Code Availability
Not applicable.
Abbreviations
- THS:
-
Tolosa-Hunt syndrome
- CSF:
-
Cerebrospinal fluid
- OBs:
-
Oligoclonal IgG bands
- LP:
-
Lumbar puncture
- MRI:
-
Magnetic resonance imaging
- BBB:
-
Blood-brain barrier
- QALB:
-
ALB quotient
References
Headache Classification Committee of the International Headache Society (IHS) The international classification of headache disorders, 3rd edition. Cephalalgia. 2018;38(1):1–211.
Tolosa E. Periarteritic lesions of the carotid siphon with the clinical features of a carotid infraclinoidal aneurysm. J Neurol Neurosurg Psychiatry. 1954;17(4):300–2.
Evers S. Facial pain: overlapping syndromes. Cephalalgia. 2017;37(7):705–13.
Carta S, Ferraro D, Ferrari S, Briani C, Mariotto S. Oligoclonal bands: clinical utility and interpretation cues. Crit Rev Clin Lab Sci. 2022;59(6):391–404.
Yang Q, Lai C, Meng C, Chang Q, Wang J. Clinical and cerebrospinal fluid characteristics in 55 cases of Tolosa-Hunt syndrome: a retrospective analytical study. Eur Neurol. 2022;85(4):265–72.
Wang J, Li L, Zhang Y, Wang P. The interpretation of mirror pattern bands during oligoclonal immunoglobulin isoelectric focusing electrophoresis: a retrospective study. Lab Med. 2022.
Jarholm JA, Faiz KW, Nysted T, Zarnovicky S, Kristoffersen ES. Orbital pain, ophthalmoplegia, and oligoclonal bands in the cerebrospinal fluid: a case report of Tolosa-Hunt syndrome. Headache. 2018;58(5):758–60.
Zeman A, McLean B, Keir G, Luxton R, Sharief M, Thompson E. The significance of serum oligoclonal bands in neurological diseases. J Neurol Neurosurg Psychiatry. 1993;56(1):32–5.
Mayringer I, Timeltaler B, Deisenhammer F. Correlation between the IgG index, oligoclonal bands in CSF, and the diagnosis of demyelinating diseases. Eur J Neurol. 2005;12(7):527–30.
Uher T, Horakova D, Tyblova M, Zeman D, Krasulova E, Mrazova K, et al. Increased albumin quotient (QAlb) in patients after first clinical event suggestive of multiple sclerosis is associated with development of brain atrophy and greater disability 48 months later. Mult Scler. 2016;22(6):770–81.
Funding
This study was supported by a grant from the Key Research and Development Program of Ningxia Province of China (2022BEG03130).
Author information
Authors and Affiliations
Contributions
HJH: idea of reporting the case, drafting the manuscript and the figures. RYZ: collection of data, analysis and interpretation of data. QWM: analysis and interpretation of data. YJ: editing of the manuscript in terms of the relevant information and language. LHN: final review of the manuscript and guidance of the authors.
Corresponding author
Ethics declarations
Ethics Approval
Ethical approval is not required for this study in accordance with local or national guidelines.
Consent to Participate
Regarding cases 1 and 2, written informed consent was obtained from the patient’s next of kin for publication of the details of their medical cases and any accompanying images.
Consent for Publication
Written informed patient consent for publication of this case report has been obtained.
Conflict of Interest
The authors declare no competing interests.
Additional information
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
Springer Nature or its licensor (e.g. a society or other partner) holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.
About this article
Cite this article
He, J., Ren, Y., Qi, W. et al. Headache, Ophthalmoplegia, “Mirror Pattern Bands”: Two Case Reports. SN Compr. Clin. Med. 6, 28 (2024). https://doi.org/10.1007/s42399-024-01657-7
Accepted:
Published:
DOI: https://doi.org/10.1007/s42399-024-01657-7