Table 1.
Summary of retrospective cohort studies and case series of patients with encephalitis lethargica and tics
Study | Country | Number | Mean age (range) | Male (%) | Aetiology of encephalitis | Tics | Other neuropsychiatric symptoms | Additional findings | Main limitations |
---|---|---|---|---|---|---|---|---|---|
Calne et al. [8] | UK | 40 | 47–73 | 45 | Encephalitis lethargica | 3 patients developed tics | Parkinsonism reported as very common; 9 patients with psychiatric disturbances (6 aggressive and uncooperative, 1 obsessional, 1 paranoid, 1 depressed); 4 with choreoathetosis | All patients had a history of illness in the 1917 encephalitis pandemic and had been in a hospital for an average of 29 years | Case histories not documented for individual patients; description of tics lacking detail |
Sacks et al. [9] | USA | 25 | N/A | N/A | Encephalitis lethargica | 8 patients developed respiratory and phonic tics (sudden deep breaths, yawns, coughs, giggles, sighing, grunting, moaning) | Severe post-encephalitic syndrome, with the development of respiratory crises and tics after treatment with l-dopa | In some cases (not quantified), the respiratory tics and breathing abnormalities were present before the administration of l-dopa and increased after its use | Lack of detail about the presence of tics before the administration of l-dopa and its effects on individual patients |
Dale et al. [10] | UK | 20 | 2–69 | 55 | Basal ganglia encephalitis, reported as encephalitis lethargica | 2 patients developed tics: a 10-year-old boy had motor tics; a 17-year-old boy had both motor and phonic tics | Hypersomnolence, bradykinesia, mutism, and depression (10-year-old boy); sleep inversion, bradykinesia, stereotypies, mutism, catatonia, obsessive-compulsive disorder, trichotillomania (17-year-old boy) | N/A | Description of tics lacking detail (including data on tic onset) |
Dale et al. [11] | Australia | 17 | 7 (0–15) | 53 | Basal ganglia encephalitis, reported as encephalitis lethargica | 1 patient developed motor tics | Somnolence, lethargy, parkinsonism | Diagnosis based on the presence of somnolence, an akinetic movement disorder, and emotional dysregulation in the background of basal ganglia changes on neuroimaging | Demographic data of individual patients not reported; description of tics lacking detail |