Importance: This is the first report, to our knowledge, of a child with torticollis due to saccular dysfunction.
Observation: An 18-month-old infant with torticollis was referred for postural imbalance and observed rotary nystagmus. The infant had undergone physical therapy treatment of left torticollis for nearly 15 months. Cervical vestibular evoked myogenic potentials (cVEMPs) were recorded to assess saccular function and caloric stimulation and positional and rotational testing were performed to evaluate other vestibular receptors. The child demonstrated abnormal cVEMP findings, with a low-amplitude response on the left, which indicated left-sided saccular dysfunction. The patient's rotary-torsional nystagmus suggested positional vertigo secondary to abnormal saccular function.
Conclusions and relevance: This case highlights that saccular dysfunction should be considered when timely resolution of congenital torticollis is not obtained with physical therapy. Early detection of abnormal saccular function in infants and young children with CPT is necessary to ensure appropriate intervention. Further study needs to be done to confirm our findings.