Cluster-Tic Syndrome

A Cross-sectional Study of Cluster Headache Patients

Leopoldine A. Wilbrink MD; Claudia M. Weller MD; Carlo Cheung MD; Joost Haan MD, PhD; Michel D. Ferrari MD, PhD

Disclosures

Headache. 2013;53(8):1334-1340. 

In This Article

Abstract and Introduction

Abstract

Objective To determine the prevalence and nature of trigeminal neuralgia in a large group of cluster headache patients.

Background Cluster-tic syndrome is a rare headache syndrome in which trigeminal neuralgia and cluster headache co-occur. The existence of cluster-tic syndrome as a separate entity is questioned, and figures on prevalence of simultaneous existence of cluster headache and trigeminal neuralgia are not available.

Methods As part of a nationwide study on headache mechanisms in cluster headache (Leiden University Medical Centre Cluster headache Neuro Analysis programme), we collected clinical data of 244 cluster headache patients using a semistructured telephone interview in a cross-sectional design.

Results In 11 (4.5%) cluster headache patients, attacks fulfilling International Headache Society criteria for trigeminal neuralgia were also present. In all cases, trigeminal neuralgia occurred ipsilateral to cluster headache and in the majority (82%) in the ophthalmic branch. In 8 of these 11 patients (73%), the frequency and time pattern of trigeminal neuralgia seemed to parallel cluster headache and was likely a part of the cluster headache spectrum. In the 3 remaining patients, cluster headache and trigeminal neuralgia were unrelated in time and appeared to occur independently.

Conclusion Trigeminal neuralgia co-occurred in 11/244 (4.5%) of cluster headache patients. In only 3 (1.2%) patients, trigeminal neuralgia seemed to occur independently from cluster headache episodes. Trigeminal neuralgia (-like) attacks in cluster headache patients are most of the time part of the cluster headache spectrum and should then probably not be treated separately. A shared underlying pathophysiological mechanism of cluster headache and trigeminal neuralgia is not supported by this study.

Introduction

The cluster-tic syndrome is characterized by the coexistence of cluster headache and trigeminal neuralgia and has been described in single case reports and small case series only.[1–12] Cluster headache consists of attacks of severe, strictly unilateral, (supra)orbital, or temporal pain lasting 15–180 minutes, accompanied by facial autonomic symptoms and/or restlessness (Table 1).[13–15] Trigeminal neuralgia is characterized by stereotyped, brief (seconds to minutes) attacks of sharp or stabbing pain within the distribution area of one or more branches of the trigeminal nerve, usually the second and third branches which may come spontaneously or after specific triggers (Table 1).[13,16]

Cluster-tic syndrome is described very heterogeneously in the literature, and it is still questioned whether it is to be seen as a separate syndrome. The term "cluster-tic syndrome" was introduced in 1978 in an abstract.[8] From this abstract, it is not clear if the author described 2 different types of attacks in 1 patient, or that these patients suffered from 1 type of attack having features of both cluster headache and trigeminal neuralgia. As a consequence, the term "cluster-tic" has been used to describe both conditions. Mostly, it is used for the co-occurrence of attacks of cluster headache and trigeminal neuralgia within the same patient. Previously, we suggested that trigeminal neuralgia and cluster headache only co-occur by chance.[2] Others, however, have stated that this co-occurrence is not coincidental and that attacks of cluster headache and trigeminal neuralgia are caused by the same underlying pathophysiological mechanism[3,4,6,9] or that short-lasting attacks can be part of the cluster headache spectrum and should not receive a separate diagnosis.[10] Last, it is asserted that the cluster-tic syndrome is a separate entity consisting of 3 types of attacks: trigeminal neuralgia attacks, cluster headache attacks, and mixed attacks. A single lesion affecting the trigeminal sensory pathway with involvement of both myelinated and unmyelinated fibers is hypothesized to be the underlying mechanism of cluster-tic.[1] The headache classification of the International Headache Society (International Classification of Headache Disorders: 2nd Edition [ICHD-2]) mentions cluster-tic syndrome as a note in the cluster headache section. It defines cluster-tic syndrome as the co-occurrence of trigeminal neuralgia and cluster headache and advises that these patients should receive both diagnoses, because these 2 types of attacks each need separate treatments.[13] The objective of our study was to determine the prevalence and nature of trigeminal neuralgia in a large group of cluster headache patients.

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