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Effects of extraocular muscle surgery on 15 patients with oculo-cutaneous albinism (OCA) and infantile nystagmus syndrome (INS).
Am J Ophthalmol. 2004 Dec; 138(6):978-87.AJ

Abstract

PURPOSE

The purpose of this report is to characterize the clinical and electrophysiological effects of extraocular muscle surgery in 15 patients with oculo-cutaneous albinism (OCA) and infantile nystagmus syndrome (INS). Our hypothesis is that surgery on the extraocular muscles of patients with OCA and INS changes their nystagmus and their visual function.

DESIGN

Interventional, prospective, cohort, noncomparative case series.

METHODS

All 15 patients had surgery on all four virgin horizontal recti; three for strabismus alone, three for nystagmus alone, five for an eccentric gaze null zone alone, and four for an eccentric gaze null zone plus strabismus. All patients have been followed for at least six months. All 15 patients had the subjective outcome measure of pre- and postoperative binocular best optically corrected acuity (BBOCA). Objective outcome measures included anomalous head posture (AHP) in nine patients, eye movement recording measures of expanded nystagmus acuity function (NAFX) in 10 patients, null zone position (NUZP) and null zone width (NUZW) in 10 patients, and foveation time (FOV) in nine patients.

RESULTS

The results are summarized as follows; BBOCA increased 0.1 LogMar or greater in 14 of 15 patients. In those operated on for an AHP with or without associated strabismus the AHP improved significantly (P < .01 for all). The NAFX, NUZP, NUZW, and FOV measured from eye movement recordings showed persistent, significant increases in all patients (P < .01 for all).

CONCLUSIONS

This report adds to the evidence that surgery on the extraocular muscles in patients with INS has independent neurologic and visual results.

Authors+Show Affiliations

Department of Ophthalmology, Children's Hospital of Pittsburgh, the Pittsburgh Eye and Ear Institute, Pittsburgh, Pennsylvania 15213, USA. Richard.hertle@chp.eduNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Journal Article

Language

eng

PubMed ID

15629289

Citation

Hertle, Richard W., et al. "Effects of Extraocular Muscle Surgery On 15 Patients With Oculo-cutaneous Albinism (OCA) and Infantile Nystagmus Syndrome (INS)." American Journal of Ophthalmology, vol. 138, no. 6, 2004, pp. 978-87.
Hertle RW, Anninger W, Yang D, et al. Effects of extraocular muscle surgery on 15 patients with oculo-cutaneous albinism (OCA) and infantile nystagmus syndrome (INS). Am J Ophthalmol. 2004;138(6):978-87.
Hertle, R. W., Anninger, W., Yang, D., Shatnawi, R., & Hill, V. M. (2004). Effects of extraocular muscle surgery on 15 patients with oculo-cutaneous albinism (OCA) and infantile nystagmus syndrome (INS). American Journal of Ophthalmology, 138(6), 978-87.
Hertle RW, et al. Effects of Extraocular Muscle Surgery On 15 Patients With Oculo-cutaneous Albinism (OCA) and Infantile Nystagmus Syndrome (INS). Am J Ophthalmol. 2004;138(6):978-87. PubMed PMID: 15629289.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Effects of extraocular muscle surgery on 15 patients with oculo-cutaneous albinism (OCA) and infantile nystagmus syndrome (INS). AU - Hertle,Richard W, AU - Anninger,William, AU - Yang,Dongsheng, AU - Shatnawi,Raed, AU - Hill,Vanessa M, PY - 2004/07/13/accepted PY - 2005/1/5/pubmed PY - 2005/1/19/medline PY - 2005/1/5/entrez SP - 978 EP - 87 JF - American journal of ophthalmology JO - Am J Ophthalmol VL - 138 IS - 6 N2 - PURPOSE: The purpose of this report is to characterize the clinical and electrophysiological effects of extraocular muscle surgery in 15 patients with oculo-cutaneous albinism (OCA) and infantile nystagmus syndrome (INS). Our hypothesis is that surgery on the extraocular muscles of patients with OCA and INS changes their nystagmus and their visual function. DESIGN: Interventional, prospective, cohort, noncomparative case series. METHODS: All 15 patients had surgery on all four virgin horizontal recti; three for strabismus alone, three for nystagmus alone, five for an eccentric gaze null zone alone, and four for an eccentric gaze null zone plus strabismus. All patients have been followed for at least six months. All 15 patients had the subjective outcome measure of pre- and postoperative binocular best optically corrected acuity (BBOCA). Objective outcome measures included anomalous head posture (AHP) in nine patients, eye movement recording measures of expanded nystagmus acuity function (NAFX) in 10 patients, null zone position (NUZP) and null zone width (NUZW) in 10 patients, and foveation time (FOV) in nine patients. RESULTS: The results are summarized as follows; BBOCA increased 0.1 LogMar or greater in 14 of 15 patients. In those operated on for an AHP with or without associated strabismus the AHP improved significantly (P < .01 for all). The NAFX, NUZP, NUZW, and FOV measured from eye movement recordings showed persistent, significant increases in all patients (P < .01 for all). CONCLUSIONS: This report adds to the evidence that surgery on the extraocular muscles in patients with INS has independent neurologic and visual results. SN - 0002-9394 UR - https://www.unboundmedicine.com/medline/citation/15629289/Effects_of_extraocular_muscle_surgery_on_15_patients_with_oculo_cutaneous_albinism__OCA__and_infantile_nystagmus_syndrome__INS__ L2 - https://linkinghub.elsevier.com/retrieve/pii/S0002-9394(04)00906-7 DB - PRIME DP - Unbound Medicine ER -