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LETTERS TO EDITOR
Year : 2019  |  Volume : 67  |  Issue : 1  |  Page : 287-288

Hemiplegic cluster headache: A case report and review of the literature


Department of Neurology, Smt. B. K. Shah Medical Institute and Research Centre, Sumandeep Vidyapeeth, Vadodara, Gujarat, India

Date of Web Publication7-Mar-2019

Correspondence Address:
Dr. Sanjay Prakash
Department of Neurology, Smt. B. K. Shah Medical Institute and Research Centre, Sumandeep Vidyapeeth, Vadodara - 391760, Gujarat
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.253626

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How to cite this article:
Dave A, Prakash S. Hemiplegic cluster headache: A case report and review of the literature. Neurol India 2019;67:287-8

How to cite this URL:
Dave A, Prakash S. Hemiplegic cluster headache: A case report and review of the literature. Neurol India [serial online] 2019 [cited 2019 May 27];67:287-8. Available from: http://www.neurologyindia.com/text.asp?2019/67/1/287/253626




Sir,

A 41-year-old male patient had a 4-year history of severe excruciating episodic headaches on the left side. The duration of headache attacks was 30–90 min. Most episodes were accompanied by ipsilateral lacrimation, conjunctival injection, and rhinorrhea. Restlessness was also present during attacks. The patient fulfilled the International Classification of Headache Disorder, third edition (ICHD-3) criteria for episodic cluster headache (CH). The cluster period lasted for 3–4 months, with a remission phase of 6–8 months. The frequency of headaches during the cluster period was about four to five attacks in a week. Approximately one-third of the attacks were accompanied by left-sided hemiparesis. Hemiparesis used to start during or immediately after the onset of headache. The duration of hemiparesis was 2–24 h. The patient was investigated on many occasions for the possible cerebrovascular accident and other causes. However, no secondary pathology for headache or hemiparesis was ever noted. He was never diagnosed with CH before being seen at our neurology clinic. The patient was put on lithium as a preventive measure and oxygen inhalation for acute attacks. The patient responded to lithium (300 mg twice daily). We followed the patients for more than 4 years. In each cluster period, we started lithium. The frequency of headaches reduced to less than one attack in a month in a cluster period. Even the frequency of associated hemiparesis reduced greatly (just one attack of hemiparesis in one cluster period). Oxygen inhalation had positive effect on both headache and hemiparesis. The average duration of hemiparesis reduced significantly (less than 60 min) with oxygen inhalation.

Hemiplegia as an aura is frequently associated with migraine. CH is rarely associated with hemiparesis as a preceding or accompanying feature. [Table 1] summarizes all cases of hemiplegia cluster (a total of seven cases).[1],[2],[3],[4] Three patients had ipsilateral hemiplegia. Another three patients noted hemiplegia on the side opposite to the headache attacks. One patient had crossed hemiplegia (ipsilateral facial and contralateral limb weakness). Hemiplegia occurred only with headache attacks in all patients. None of the patient reported hemiplegia preceding the headache. Isolated hemiplegia (without headache) was also not noted in any patient. Most of the hemiplegia attacks subsided in a few hours (less than 12 h), except case 3 of Siow et al.,[1] where it persisted in a mild form for 1 week. Aphasia was the most common accompanying feature (three patients) with hemiplegia. Other accompanying features were dysarthria (n = 1), facial numbness (n = 1), visual loss (n = 1), and myoclonic jerk (n = 1).
Table 1: Cases of hemiplegia cluster headache in the literature

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Previous case reports are almost silent over the treatment aspects of hemiplegia. Our patient showed response to lithium in both headache attack and hemiplegia attacks. Oxygen inhalation also had positive effect in hemiparesis (average duration of hemiplegia reduced markedly with oxygen inhalation).

The pathophysiology of hemiparesis associated with CH has not been explored in the literature. The hemiplegia associated with migraine is probably related to motor cortical excitation because of cortical spreading depression. Very recently, cortical hyperexcitability has been demonstrated in patients with CH.[5] Therefore, a pathophysiology similar to hemiplegic migraine can be speculated for hemiplegic cluster.

This report reveals the unique presentation of CH having hemiparesis as an associated symptom, which is commonly seen with migraine.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Siow HC Young WB, Peres MF, Rozen TD, Silberstein SD. Hemiplegic cluster headache. Headache 2002;42:136-9.  Back to cited text no. 1
    
2.
Langedijk M, van der Naalt J, Luijckx GJ, De Keyser J. Cluster-like headache aura status. Headache 2005;45:80-1.  Back to cited text no. 2
    
3.
Yang JW, Park SG, Jung IH, Sung YH, Park KH, Lee YB, Shin DJ, et al. A case of cluster headache accompanied by myoclonus and hemiparesis. J Clin Neurol 2012;8:83-6.  Back to cited text no. 3
    
4.
Fantini J, Sartori A, Granato A, Manganotti P. The reappearance of hemiplegic cluster headaches: A case report and review of the literature. Clin Neurol Neurosurg 2017;158:12-4.  Back to cited text no. 4
    
5.
Cosentino G, Brighina F, Brancato S, Valentino F, Indovino S, Fierro B, et al. Transcranial magnetic stimulation reveals cortical hyperexcitability in episodic cluster headache. J Pain 2015;16:53-9.  Back to cited text no. 5
    



 
 
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