Background & Aims
Focal temporal muscle tremor also known as moving ear syndrome is a very uncommon condition. To date no case was reported in children. We report here a case of headache and moving ear with a favorable outcome with treatment, in a 15 year old teenager, and discuss its similarities with the syndrome of painful legs and moving toes.
Methods
This was a case report, at the neurology outpatient consultation of the Dschang Regional Hospital in Cameroon. We obtained a verbal and written consent from the patient and her father.
Results
A 15 year-old female, presented a four month history of continuous unvoluntary jerking of the left ear of progressive onset. Evolution was marked two months later by an exacerbation of these movements, associated with new onset headaches. Headache was left temporal, sometimes a left hemicrania, throbbing, moderate to severe. It was increased with physical effort, and not responding to paracetamol. It was associated with dizziness but no nausea/vomiting, photophobia or phonophobia. Headache occurred on a daily basis, with a mean of five episodes a day, lasting 1 to 5 hours, in temporal relationship with the period of exacerbation of ear movements. Physical examination revealed a continuous rhythmic approximately 1.5 Hz antero-posterior jerk of the left ear. There was left temporal and sub-occipital allodynia. Laboratory work-up and brain MRI were normal. The patient was treated with clonazepam at 1mg bid. Headache and ear tremor progressively resolved after one month of treatment.
Conclusions
Only few cases of moving ear syndrome are reported in the published literature. We report the first case of headache and moving ear in a teenager. Unilateral ear tremor is probably associated to a brainstem generator. Headache could be attributed to the phenomenenon of peripheral, then central sensitization of temporal nociceptors, due to prolonged repeated ear movements. The latency between ear tremor and the onset of headache and localized allodynia are arguments for this hypothesis. Another mechanism could be a central sensitization of the trigemino-cervical complex by the brainstem generator of tremor. This case has some similarities with the syndrome of painful legs and moving toes. In-depth neurophysiological and functional neuroimaging studies are needed to better characterize this syndrome.
References
Jabbour, C. E., Sawaya, R. A., & Zaytoun, G. M. (2021). Auricular Myoclonus: A Case Report and Literature Review. The journal of international advanced otology, 17(6), 581–583. https://doi.org/10.5152/iao.2021.21064
Chaudhuri, K. R., Leigh, P. N., Gibb, W. R., & Pye, I. F. (1996). The moving ear syndrome: a focal dyskinesia. Journal of neurology, neurosurgery, and psychiatry, 60(1), 106. https://doi.org/10.1136/jnnp.60.1.106
Kirk, A., & Heilman, K. M. (1991). Auricular myoclonus. The Canadian journal of neurological sciences. Le journal canadien des sciences neurologiques, 18(4), 503–504. https://doi.org/10.1017/s0317167100032236
Keshavan M. S. (1988). The ear wigglers: tics of the ear in 10 patients. The American journal of psychiatry, 145(11), 1462–1463. https://doi.org/10.1176/ajp.145.11.1462
Presenting Author
Yannick Fogang
Poster Authors
Topics
- Specific Pain Conditions/Pain in Specific Populations: Headache