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Cranial neuropathies
Original research
Medical treatment of SUNCT and SUNA: a prospective open-label study including single-arm meta-analysis
  1. Giorgio Lambru1,
  2. Anker Stubberud2,3,
  3. Khadija Rantell4,
  4. Susie Lagrata1,5,
  5. Erling Tronvik2,3,
  6. Manjit Singh Matharu1,5
  1. 1 Headache and Facial Pain Group, UCL Queen Square Institute of Neurology, London, UK
  2. 2 Department of Neuromedicine and Movement Sciences, Norwegian University of Science and Technology, Trondheim, Norway
  3. 3 Department of Neurology, St. Olavs Hospital, Trondheim, Norway
  4. 4 Education Unit, UCL Queen Square Institute of Neurology, London, UK
  5. 5 Headache and Facial Pain Group, The National Hospital for Neurology and Neurosurgery, London, UK
  1. Correspondence to Dr Manjit Singh Matharu, Headache and Facial Pain Group, UCL Institute of Neurology, London WC1N 3BG, UK; manjit.matharu{at}ucl.ac.uk

Abstract

Introduction The management of short-lasting unilateral neuralgiform headache attacks with conjunctival injection and tearing (SUNCT) and short-lasting unilateral neuralgiform headache attacks with cranial autonomic symptoms (SUNA) remains challenging in view of the paucity of data and evidence-based treatment recommendations are missing.

Methods In this single-centre, non-randomised, prospective open-label study, we evaluated and compared the efficacy of oral and parenteral treatments for SUNCT and SUNA in a real-world setting. Additionally, single-arm meta-analyses of the available reports of SUNCT and SUNA treatments were conducted.

Results The study cohort comprised 161 patients. Most patients responded to lamotrigine (56%), followed by oxcarbazepine (46%), duloxetine (30%), carbamazepine (26%), topiramate (25%), pregabalin and gabapentin (10%). Mexiletine and lacosamide were effective in a meaningful proportion of patients but poorly tolerated. Intravenous lidocaine given for 7–10 days led to improvement in 90% of patients, whereas only 27% of patients responded to a greater occipital nerve block. No statistically significant differences in responders were observed between SUNCT and SUNA. In the meta-analysis of the pooled data, topiramate was found to be significantly more effective in SUNCT than SUNA patients. However, a higher proportion of SUNA than SUNCT was considered refractory to medications at the time of the topiramate trial, possibly explaining this isolated difference.

Conclusions We propose a treatment algorithm for SUNCT and SUNA for clinical practice. The response to sodium channel blockers indicates a therapeutic overlap with trigeminal neuralgia, suggesting that sodium channels dysfunction may be a key pathophysiological hallmark in these disorders. Furthermore, the therapeutic similarities between SUNCT and SUNA further support the hypothesis that these conditions are variants of the same disorder.

http://creativecommons.org/licenses/by-nc/4.0/

This is an open access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited, appropriate credit is given, any changes made indicated, and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/.

https://doi.org/10.1136/jnnp-2020-323999

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    Footnotes

    • Twitter @manjit_matharu

    • Contributors GL: study concept, collection, analysis, and interpretation of data, drafting and revision of the manuscript. AS: analysis and interpretation of data, drafting and revision of the manuscript. KR: statistical analysis and interpretation of data, revision of the manuscript. SL: collection and quality assurance of data, revision of the manuscript. ET: study concept, interpretation of data and manuscript revision. MSM: study concept, recruitment of subjects, interpretation of data and manuscript revision.

    • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

    • Competing interests GL has received speaker honoraria, funding for travel and has received honoraria for participation in advisory boards sponsored by Allergan, Novartis, Eli Lilly and TEVA. He has received speaker honoraria, funding for travel from electroCore, Nevro Corp. and Autonomic Technologies. AS is cofounder of Nordic Brain Tech, a company developing a non-pharmacological biofeedback treatment for migraine and holds a pending patent application relating to the company’s product. KR has nothing to declare. SL has received payment for attending advisory meetings and development of presentation from Allergan Novartis, Eli Lilly and TEVA. ET: is a cofounder and shareholder of Nordic Brain Tech AS and Palion Medical AS. He serves on advisory board for Eli Lilly and Novartis and TEVA. He has received speaker honoraria from Eli Lilly, Novartis, Allergan and TEVA. MSM serves on the advisory board for Abbott, Allergan, Eli Lilly, Medtronic, Novartis, TEVA; has received payment for the development of educational presentations from Allergan, electroCore, Eli Lilly, Medtronic, Novartis, and TEVA; and, has received research grants from Abbott, electroCore and Medtronic.

    • Patient consent for publication Not required.

    • Ethics approval The study was approved by Northwick Park Hospital Research Ethics Committee, London, UK (REC no:11/LO/1709).

    • Provenance and peer review Not commissioned; externally peer reviewed.

    • Data availability statement Data are available upon reasonable request. The data that support the findings of this study are available upon reasonable request to the corresponding author.

    • Supplemental material This content has been supplied by the author(s). It has not been vetted by BMJ Publishing Group Limited (BMJ) and may not have been peer-reviewed. Any opinions or recommendations discussed are solely those of the author(s) and are not endorsed by BMJ. BMJ disclaims all liability and responsibility arising from any reliance placed on the content. Where the content includes any translated material, BMJ does not warrant the accuracy and reliability of the translations (including but not limited to local regulations, clinical guidelines, terminology, drug names and drug dosages), and is not responsible for any error and/or omissions arising from translation and adaptation or otherwise.