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To the Editor: Benign paroxysmal torticollis of infancy (BPTI) is a self-limited condition in which the child can have recurrent episodes of head tilt alone or sometimes associated with vomiting, pallor, irritability, ataxia, or drowsiness which could create confusion in the clinician’s mind about the possibility of seizures.
We present a case of a 5-y-old girl who presented with complaints of paroxysmal tilting of the head to one side for 3 mo of age, once in 1–2 mo and each episode lasted up to 2–12 h, the frequency of such episodes decreased as the age progressed. The parents also complained of headaches for 2 y of age associated with vomiting. There was a history of migraine in the father and history of motion sickness in the mother. The examination was essentially normal. Keeping possibility of BPTI, parents were counseled regarding the benign nature and reassured with advice to follow up regularly if symptoms persist.
BPTI is a paroxysmal event of head tilt and these episodes vary from side to side and can last from few minutes to few days. It is one of the important seizure mimics during the infancy period and can lead to the deliberate use of antiseizure medications. Paroxysmal torticollis can be a predictor of migraine or benign paroxysmal vertigo in some [1].
Recently, few genetic mutations like CACNA1A and PRRT2 are associated in patients with paroxysmal torticollis and migraine [1, 2]. It is a benign condition and does not require any drugs for treatment; however, migraine might persist and may require medical attention. Recently, topiramate has shown to be a promising drug in some cases of BPTI [3].
Symptoms of BPTI in the first 2 y of life, family history of migraine, and motion sickness could be a clue towards benign nature of migrainous headaches in a younger child.
References
Giffin NJ, Benton S, Goadsby PJ. Benign paroxysmal torticollis of infancy: four new cases and linkage to CACNA1A mutation. Dev Med Child Neurol. 2002;44:490–3.
Méneret A, Gaudebout C, Riant F, Vidailhet M, Depienne C, Roze E. PRRT2 mutations and paroxysmal disorders. Eur J Neurol. 2013;20:872–8.
Yaghini O, Badihian N, Badihian S. The efficacy of topiramate in benign paroxysmal torticollis of infancy: report of four cases. Pediatrics. 2016;137:e20150868.
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Krishna, D., Saini, L., Choudhary, S. et al. A Curious Case of Migrainous Headache in a 5-Year-Old Girl. Indian J Pediatr 89, 417 (2022). https://doi.org/10.1007/s12098-021-04024-9
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DOI: https://doi.org/10.1007/s12098-021-04024-9