Background: Spasmodic torticollis refers to involuntary dystonic movements of the head that may be associated with certain body and neck positions. The pathophysiology of dystonia is not fully known.
Case Report: We report a case of torticollis that was unusual in the sense that it occurred only when the patient was lying on one side.
Discussion: The cause of the cervical dystonia in this patient was unclear and the positional nature of the movements was unusual.
As a rule, symptoms of dystonia diminish or disappear in a recumbent position and are exacerbated when upright1, 2 and with some movements.3 Evidence of this clinical pattern may be traced back to the earliest observations of the disorder. Thus, in 1911, Oppenheim4 described hyperkinesias in four patients; these were not apparent in the supine position. Similar features were observed in two siblings described by Bernstein et al. in 1912.5 In 1914, Beling6 described a patient whose generalized dystonic movements ceased when lying in bed. Here we report a case of a female patient whose dystonic “yes–yes” head jerking was eventually seen only when lying on her side. The case was unusual for that reason.
The patient, a woman born in 1941, was a typist by profession. Her involuntary movements had begun in 1970 during pregnancy, and treatment at St. Petersburg Mechnikov Medical University Clinic resulted in prolonged remission. Her past medical history was unremarkable, and there was no neuroleptic exposure. She had had seven pregnancies and two deliveries. There was no family history of involuntary movements.
An exacerbation occurred in 1996 in the setting of an acute respiratory infection. Brain magnetic resonance imaging at that time was normal. She was hospitalized in the Neurologic Department of St. Petersburg Academy of postgraduate education. She complained of involuntary backward jerking of her head that worsened in certain positions.
Here we report a case of a female patient whose dystonic “yes–yes” head jerking was visualized only when lying on her side. The case is unusual for that reason. Indeed, most patients experience amelioration of dystonia when lying on their back and/or on one side. By contrast, Jahanshahi7 reported a small percentage of cervical dystonia patients who noted worsening when lying on one side. To our knowledge, though, the pattern observed in our patient has not been documented by videotape previously.Video 1.Examination of the patient with positional torticollisSegment 1. The patient is examined when lying on her side and then on her back. Head jerking disappears when she is lying on her back.
1 Funding: None.
2 Conflict of Interest: The authors report no conflict of interest.
Professor Elan D. Louis from the G.H. Sergievsky Center, Columbia University, New York, United States of America, provided critical feedback on the manuscript.
Bouchard, M and Furtado, S (2011). Speech-induced cervical dystonia.. Can J Neurol Sci 38: 929–930. [PubMed]
Beling, C (1914). Case of dystonia musculorum deformance.. J Nerv Ment Dis 3: 148–153, DOI: https://doi.org/10.1097/00005053-191403000-00003