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Geniospasm: A systematic review on natural history, prognosis, and treatment

      Highlights

      • Geniospasm generally improved with age.
      • Complications were uncommon.
      • Complete remission was rare.
      • Males or those with geniospasm in sleep were associated with poor outcome.
      • Botulinum toxin injection was by far the only effective treatment.

      Abstract

      Background

      Geniospasm is a rare and generally benign movement disorder of the chin yet with potentially debilitating complications. Due to its rarity, previous literature was limited to only case reports or series with critical knowledge gap on its natural history, prognosis, and management. We aimed to establish the natural history, prognosis, and treatment for geniospasm.

      Methods

      A systematic review on case reports or series was performed with literature search on PubMed and Google Scholar, from inception through December 2021. The quality of the reports was assessed with low-quality articles excluded for analyses other than demographics.

      Results

      Forty-one articles were included for demographic analysis (n = 489) while forty articles for other analyses (n = 451). There was only slight male preponderance of this disease in our cohort (Male:Female = 1.2:1) with 98.6% having family history of geniospasm, 68.1% having onset below 1 year of age, 91.4% having emotional triggers of the symptoms, 25.7% having geniospasm in sleep, 100% involving mentalis muscles, 4.9% involving muscles adjacent to mentalis, 55.2% having regular paroxysms (≥1/week, but brief in 56.3% of the cases) and only 11.1% having complications. The paroxysms reduced with age, but complete remission only happened in 9.9%. Females (p = 0.010) and those without geniospasm in sleep (p = 0.048) were associated with good outcome of the disease. Geniospasm was usually refractory to treatment except for regular botulinum toxin injections.

      Conclusion

      Generally, geniospasm improved with age but complete remission was rare. Considering our review limitations, our findings should be interpreted with caution. Future studies of higher evidence level are crucial.

      Keywords

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