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Clinical Note |

Congenital Torticollis and Saccular Dysfunction: A Case Report

Ashley Hallberg, AuD; Robert T. Standring, MD; Syed Ahsan, MD
JAMA Otolaryngol Head Neck Surg. 2013;139(6):639-642. doi:10.1001/jamaoto.2013.3283.
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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.

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Figure 1. An 18-month-old infant demonstrating left lateral tilt and rotation of the head to the right side.

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Figure 2. Asymmetric cervical vestibular evoked myogenic potential (cVEMP) testing generated in response to air conduction 500 Hz tone bursts was reduced in amplitude by 69% on the left (affected) side (A) compared with responses on the right (normal) side (B), indicating left-sided saccular dysfunction (normal symmetry is <40%). p13 and n23 indicate the p13-n23 complex.

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Figure 3. Repeated cervical vestibular evoked myogenic potential (cVEMP) testing after 1 year of vestibular rehabilitation demonstrated minimally asymmetric reduced amplitude (20%) on the left (affected) side, confirming improvement of saccular function. The right side is normal. p13 and n23 indicate the p13-n23 complex.

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