The neuropathophysiology of neonatal complex motor activity such as bicycling is poorly understood. Pedaling and other motor automatisms like boxing or swimming motions may 1-3 or may not 4 have an electroencephalography (EEG) correlate. Associated disorders include diffuse bilateral cortical injury secondary to hypoxic-ischemic encephalopathy (HIE) or infection 1 ; metabolic disturbances such as branched chain organic aciduria 5 and molybdenum cofactor deficiency 6 ; and drug-induced events. 7,8 Neonatal bicycling in the context of a focal lesion within the deep brain matter has not been previously described. We report on a neonate with a stroke involving the left basal ganglia, presenting with paroxysmal bicycling and boxing movements.
Case ReportA 2-day-old term neonate was transferred to our neonatal intensive care unit (NICU) for suspected seizures. She had been noted to have paroxysmal bicycling movements and repetitive boxing motions, more pronounced on the right side, without associated autonomic changes. Before transfer, she had six episodes, each up to 4 minutes in duration.The patient was born at term after an uneventful pregnancy to a healthy 37-year-old woman by uncomplicated caesarean section for failure to progress. Apgar scores were 9 (1 minute) and 9 (5 minutes). Birth weight was 3,754 g, and head circumference was 34 cm.Neurological exam, including tone and deep tendon reflexes, was normal. Complete blood count, extended electrolytes, glucose, and cerebrospinal fluid analysis were normal. The patient was treated with lorazepam and subsequently phenobarbital for suspected seizure activity, with no effect on frequency or duration of the bicycling events. Video EEG monitoring from day 2 to 3 of life captured five episodes within 20 hours, lasting from 1 to 5 minutes in duration, with no evidence of corresponding epileptiform activity during the spells (Video S1).Brain MRI on day 3 of life revealed a focus of restricted diffusion and signal abnormality involving the left corona radiata, periventricular white matter, posterior aspect of the left caudate head, and the left lentiform nucleus, extending into the posterior limb of the internal capsule. The lesion was surrounding a mildly prominent medullary vein, suggestive of a venous infarct, with no evidence of hemorrhagic component. Additionally, there was subtle diffusion restriction extending into the corticospinal tract suggestive of pre-Wallerian degeneration (Fig. 1). MR angiogram, MR venogram, and MR spectroscopy studies were normal.The complex motor paroxysms spontaneously resolved on day 4 of life, and the neonate was discharged from the hospital on day 14 of life with a normal neurological status.
DiscussionWe report on a neonate with excessive repetitive bicycling and boxing movements in the context of a left subcortical venous stroke involving the caudate head, lentiform nucleus, and corticospinal tract. To the best of our knowledge, this the first report of a perinatal stroke associated with neonatal complex motor paroxysms, with no EEG c...