Case Report

Cluster-tic syndrome

Andrzej Klimek

CEPHALALGIA

Klimek A. Clustertic syndrome. Cephalalgia 1987;7:161—162. Oslo. ISSN 0333—1024

A case of cluster—tic syndrome is presented. A 51-year-old man developed pain attacks corresponding tothe second branch of the trigeminal nerve. After treatment with 1200 mg carbamazepine daily, the attacksdisappeared. Full remission was achieved, and carbamazepine therapy was continued. Pain attacks of aquite different character then appeared; their clinical picture corresponded to cluster headache. Aftertreatment with cyproheptadine, the cluster headache attacks ceased, but 2 days later, before thediscontinuation of the treatment, the attacks of trigeminal neuralgia reappeared. Treatment withcarbamazepine was started again, and there was remission of the trigeminal neuralgia after several weeks .“Cluster headache, trigeminal neuralgia

Andrzej Klimek, Klinika Neurologii, Akademia Medyczna w. £odzi, 90—153 Lódz, ul. Kopcinskiego, Poland;Accepted 20 January 1987

Neuralgia of the trigeminal nerve (tic douloureux (T)) and cluster headache (CH) are distinct nosologicentities. A coexistence of symptoms indicative of both illnesses was mentioned early (for example,Hornabrook (1) and Lance & Anthony (2)), but Green & Apfelbaum (3) in 1978 were the first to apply thename "cluster-tic syndrome". At present, the number of cases described has reached 19. In the past year,under the title cluster—tic syndrome, two different teams have described nine cases (4,5). Neither thenosology nor the pathogenesis is as yet definitely established. The following case shows some pecularities ascompared with those previously described.

Case report

A 51-year-old man, an engineer, started to complain of paroxysmal pains in the area innervated by the IIndbranch of the right trigeminal nerve. He had never had similar symptoms. The patient had very severe attacksof jerking pain. The attacks occurred 60 or more times daily, always lasting about 1 min. The paroxysms weretriggered by brushing the teeth, eating, or shaving. One week after the onset of his illness, the patient wasreferred by a stomatologist, who had excluded dental origin of the symptoms. The neurologic andophthalmologic examinations and electroencephalography and roentgenography of the skull did not showany abnormalities. Computed tomography scanning was not performed. A diagnosis of T was made, andcarbamazepine, 200 mg three times daily, was prescribed. The treatment reduced the number of painattacks to less than a half—the patient now had 20—30 paroxysms per day, and they were less severe. Twoweeks later the dose of carbamazepine was increased to 1200 mg daily, and after a few days the attacksceased. Full remission was achieved, and carbamazepine therapy was continued. Pain attacks of a quitedifferent character then appeared. The localization of the pain was the same as the previous one, but theattacks lasted for up to 30 min, and were usually accompanied by redness and tearing of the right eye,ipsilateral rhinorrhea, and sometimes by flushing on the right side of the face. The attacks usually occurredthree times per 24 h, and also during nighttime and sleep. The pain could not be triggered by theabove-mentioned maneuvres. A diagnosis of CH was

established. Carbamazepine was withdrawn, and treatment with cyproheptadine, 4 mg three times daily, wasstarted. The CH attacks ceased (and never reappeared) 1 week after the introduction of cyproheptadine. Twodays later, before the discontinuation of the treatment, the attacks of T reappeared. Cyproheptadine waswithdrawn, and carbamazepine was readministered in a dose of 1200 mg daily. Remission of the Tparoxysms was not achieved until several weeks later.

Discussion

In the case described there was an alternate appearance of neuralgic and cluster headache cycles. One kindof attack appeared after the other, and each kind of headache attack stopped during the application of theappropriate drug. There was a clear predominance of neuralgic elements over cluster headache.

Cases of cluster—tic syndrome should probably not be classified as variants of CH, as was suggested byDiamond et al. (6) and Solomon et al. (4). The situation more closely resembles the coexistence of migraineand CH.

Recently, attention has been drawn to the neuropeptides and their possible role in the pathogenesis ofvascular headaches (7, 8). The role of substance P and somatostatin in the pathomechanism of pain hasbeen discussed (9—11). Further studies of neuropeptides may make it possible to elucidate the origin of thisheadache.

Coexistence of T and CH is probably more frequent than is generally believed, and T may not be thesimple clinical entity that it is generally considered.

References

1. Hornabrook RW. Migrainous neuralgia. NZ Med J 1964;63:774—9

2. Lance JW, Anthony M. Migrainous neuralgia or cluster headache? J Neurol Sci 1971;13:401—14

3. Green M, Apfelbaum RI. Cluster-tic syndrome. Headache 1978;18:112

4. Solomon S, Apfelbaum RI, Guglielmo KM. The cluster—tic syndrome and its surgical therapy.Cephalalgia 1985;5:83—9

5. Watson P, Evans R. Cluster—tic syndrome. Headache 1985;25:123—26

6. Diamond S, Freitag FG, Cohen JS. Cluster headache with trigeminal neuralgia. Postgrad Med1984;75:165—72

7. Moskowitz MA, Reinhard JF, Romero J, Melamed F, Pettibone DJ. Neurotransmitters and fifth cranialnerve: is there a relation to headache phase of migraine. Lancet 1979;2:88—4

8. Moskowitz MA. The neurobiology of vascular head pain. Ann Neurol 1984;16:156—68

9. Hardebo JE. The involvement of trigeminal substance P neurons in cluster headache. Headache1984;24:294—304

10. Sicuteri F, Fanciullacci M, Geppetti P, Renzi D, Caleri D, Spillantini MG. Substance P mechanism incluster headache: Evaluation in plasma and cerebrospinal fluid. Cephalalgia 1985;5:143—9

11. Sicuteri F, Geppetti P, Marabini S, Lembeck F. Pain relief by somatostatin in attack of clusterheadache. Pain 1984;18:359—65