Objective: We wished to determine the effectiveness of horizontal rectus tenotomy in changing the nystagmus of patients with congenital nystagmus and, secondarily, how their visual function changed.

Design: This was a prospective, noncomparative, interventional case series.

Participants: Ten adult patients with varied associated sensory defects and oculographic subtypes of congenital nystagmus (including asymmetric periodic or aperiodic alternating nystagmus) and no nystagmus treatment options.

Methods: By using standard surgical techniques, simple tenotomy of all four horizontal recti with reattachment at the original insertion was accomplished. Search-coil eye movement recordings and clinical examinations were performed before and 1, 6, 24, and 52 weeks after surgery.

Main outcome measures: The primary outcome measure was the expanded nystagmus acuity function, obtained in "masked" fashion directly from ocular motility recordings. Secondary outcomes included breadth of null zones, preoperative and postoperative masked measures of visual acuity (Early Treatment Diabetic Retinopathy Study [ETDRS] chart), and the National Eye Institute Visual Function Questionnaire (NEI-VFQ-25).

Results: At 1 year after tenotomy and under binocular conditions, 9 of 10 patients had persistent, significant postoperative increases in the expanded nystagmus acuity function of their fixing (preferred) eye; 1 remained high, and 1 was not tested under the same conditions. Average foveation times increased in all 9 fixing (preferred) eyes. Binocular visual acuity measured with the ETDRS chart increased in 5 patients and was unaffected in five, whereas the NEI-VFQ-25 showed an improvement in vision-specific mental health in 9 patients. There were no adverse events. Tenotomy also radically changed the periodicity of one patient's asymmetric periodic or aperiodic alternating nystagmus.

Conclusions: In 9 of 10 adult patients with clinical and oculographic variations in their congenital nystagmus, tenotomy resulted in significant improvements in a nystagmus measure and subjective visual functions.