Benign Paroxysmal Torticollis of Infancy: Has it Outgrown its Definition?

Abstract

Benign Paroxysmal Torticollis of Infancy (BPTI) is considered a migraine variant and a precursor of other migraine symptoms. It is classified as a condition that starts before one year of age with symptoms resolving by the age of five. We describe two unrelated children over the age of five who presented with features of BPTI. We would propose that the definition of the condition is therefore extended beyond infancy, and it is possible, if not likely, given the rarity and obscurity of the condition, that it may be overlooked in children of all ages.Benign Paroxysmal Torticollis of Infancy (BPTI) is a well described if not well recognized [1] condition characterised by episodes of torticollis which occur spontaneously and last between half an hour and a few days, before complete spontaneous resolution. The torticollis can be painful and there may be accompanying symptoms such as vomiting, drowsiness, irritability and ataxia. Neurological examination between episodes is normal and although there may be an association with slow development, BPTI does not appear to have long term sequelae [2]. BPTI is of unknown aetiology but has many features suggestive of a channelopathy. It is usually described as a migraine precursor with a possible genetic link to hemiplegic migraine [3-5], however many patients do not proceed to migraine in later life [6]. Of note, as its name implies, it has only been described in children under the age of five.We describe two children over the age of five, who presented with features of BPTI within a few months of each other to a District General Hospital in Hertfordshire UK. We would propose that the definition of the condition is therefore extended beyond infancy, and it is possible, if not likely, given the rarity and obscurity of the condition, that it may be overlooked in children of all ages.