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R.W. Hertle, L.F. Dell'Osso, E.J. Fitzgibbon, D. Yang, D. Thompson, S.D. Mellow; Horizontal Rectus Tenotomy in the Treatment of Congenital Nystagmus: Results of a Phase 1 Study in Ten Patients . Invest. Ophthalmol. Vis. Sci. 2003;44(13):4248.
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Purpose: The purpose of this study was to determine, in 10 selected adult patients with CN, the effectiveness of Horizontal Rectus Tenotomy with reattachment (The "Dell'Osso" Procedure) in decreasing their nystagmus. Methods: Subjects of this study included 10 adult patients with varied associated sensory defects and waveform subtypes of CN (including (a)periodic alternating nystagmus-APAN) and no other treatment options. Using standard surgical techniques simple tenotomy of all 4 horizontal recti with reattachment at the original insertion was accomplished. Standard search coil recordings were done pre-op and 1,6, 24 and 52 weeks post-op. The primary outcome measure was the "expanded nystagmus acuity function" (NAFX), obtained in masked fashion from motility recordings. Secondary outcomes included: nystagmus amplitude, breadth or creation of null zones, pre- and post-operative masked measure of visual acuity (ETDRS) and the National Eye Institute-Visual Function Questionnaire (NEI-VFQ). Results: Under binocular conditions all but 1 patient had persistent, significant postoperative increases in the NAFX of their fixing (preferred) eye. Average foveation times increased in all non-APAN cases. ETDRS binocular visual acuity increased in 5 patients and was unaffected in 5, while the NEI-VFQ showed an improvement in vision specific mental health in 9 of 10 patients reflecting the decrease in their nystagmus after tenotomy. There were no adverse events. In 3 of 5 patients with APAN the tenotomy also changed the nystagmus cycle. Conclusions: In adult CN patients, tenotomy resulted in significant improvments in their nystagmus and subjective visual functions. The lack of consistent postoperative changes in ETDRS visual acuity may reflect the complex relationship between visual function, associated sensory system deficits and the nystagmus present in visually mature individuals.
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