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Longitudinal Studies and Eye-Movement-Based Treatments of Infantile Nystagmus Syndrome: Estimated and Measured Therapeutic Improvements in Three Complex Cases.
J Binocul Vis Ocul Motil. 2018 Oct-Dec; 68(4):122-133.JB

Abstract

INTRODUCTION AND PURPOSE

To demonstrate the utility of using eye-movement data to reveal the diagnostic characteristics of infantile nystagmus syndrome (INS), determine treatment, and both estimate and document therapeutic improvements in three patients with well-developed foveation periods, fairly broad, lateral gaze "nulls," head turns, strabismus, and complex, multiplanar nystagmus.

PATIENTS AND METHODS

Infrared reflection, magnetic search coil, and high-speed digital video systems were used to record the eye movements of INS patients, pre- and post-Kestenbaum null-point correction surgery (horizontal or vertical). Data were analyzed and estimations made, using the eXpanded Nystagmus Acuity Function (NAFX) that is part of the OMtools toolbox for MATLAB.

RESULTS

In all three subjects (S1-S3), both peak NAFX and longest foveation domain (LFD) improved from their pre-Kestenbaum values. S1: 0.700-0.745 (6.4%) and 25-34° (36%), respectively. S2: 0.445-0.633 (42.4%) and >40° to >50° (10%), respectively. S3: 0.250-0.300 (20%) and 13° to ≫18° (see text), respectively.

CONCLUSIONS

S1: Even at the high ends of the pre-therapy NAFX and LFD spectra, INS foveation (and therefore, visual-function) improvements may be adequate to justify nystagmus surgery and provide clinical improvements beneficial to the patient. S2: INS foveation improvements in the vertical plane are equal to those originally estimated using the horizontal data in prior patients. S3: Two apparent NAFX peaks can be converted into a very broad peak by surgery based on the preferred lower peak.

Authors+Show Affiliations

a Daroff-Dell'Osso Ocular Motility Laboratory, Louis Stokes Cleveland Department of Veterans Affairs Medical Center and Department of Neurology, CASE Medical School, Cleveland, Ohio. b Department of Neurology , Case Western Reserve University and University Hospitals Cleveland Medical Center , Cleveland , Ohio.c Department of Ophthalmology , Case Western Reserve University and University Hospitals Cleveland Medical Center , Cleveland , Ohio.a Daroff-Dell'Osso Ocular Motility Laboratory, Louis Stokes Cleveland Department of Veterans Affairs Medical Center and Department of Neurology, CASE Medical School, Cleveland, Ohio. b Department of Neurology , Case Western Reserve University and University Hospitals Cleveland Medical Center , Cleveland , Ohio. d Department of Biomedical Engineering , Case Western Reserve University and University Hospitals Cleveland Medical Center , Cleveland , Ohio.a Daroff-Dell'Osso Ocular Motility Laboratory, Louis Stokes Cleveland Department of Veterans Affairs Medical Center and Department of Neurology, CASE Medical School, Cleveland, Ohio. d Department of Biomedical Engineering , Case Western Reserve University and University Hospitals Cleveland Medical Center , Cleveland , Ohio.

Pub Type(s)

Case Reports
Journal Article

Language

eng

PubMed ID

30332339

Citation

Dell'Osso, Louis F., et al. "Longitudinal Studies and Eye-Movement-Based Treatments of Infantile Nystagmus Syndrome: Estimated and Measured Therapeutic Improvements in Three Complex Cases." Journal of Binocular Vision and Ocular Motility, vol. 68, no. 4, 2018, pp. 122-133.
Dell'Osso LF, Orge FH, Jacobs JB, et al. Longitudinal Studies and Eye-Movement-Based Treatments of Infantile Nystagmus Syndrome: Estimated and Measured Therapeutic Improvements in Three Complex Cases. J Binocul Vis Ocul Motil. 2018;68(4):122-133.
Dell'Osso, L. F., Orge, F. H., Jacobs, J. B., & Wang, Z. I. (2018). Longitudinal Studies and Eye-Movement-Based Treatments of Infantile Nystagmus Syndrome: Estimated and Measured Therapeutic Improvements in Three Complex Cases. Journal of Binocular Vision and Ocular Motility, 68(4), 122-133. https://doi.org/10.1080/2576117X.2018.1522917
Dell'Osso LF, et al. Longitudinal Studies and Eye-Movement-Based Treatments of Infantile Nystagmus Syndrome: Estimated and Measured Therapeutic Improvements in Three Complex Cases. J Binocul Vis Ocul Motil. 2018 Oct-Dec;68(4):122-133. PubMed PMID: 30332339.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Longitudinal Studies and Eye-Movement-Based Treatments of Infantile Nystagmus Syndrome: Estimated and Measured Therapeutic Improvements in Three Complex Cases. AU - Dell'Osso,Louis F, AU - Orge,Faruk H, AU - Jacobs,Jonathan B, AU - Wang,Zhong I, Y1 - 2018/10/17/ PY - 2018/10/18/pubmed PY - 2019/8/10/medline PY - 2018/10/18/entrez KW - Infantile nystagmus syndrome KW - acuity KW - diagnosis KW - eye movements KW - foveation KW - surgery SP - 122 EP - 133 JF - Journal of binocular vision and ocular motility JO - J Binocul Vis Ocul Motil VL - 68 IS - 4 N2 - INTRODUCTION AND PURPOSE: To demonstrate the utility of using eye-movement data to reveal the diagnostic characteristics of infantile nystagmus syndrome (INS), determine treatment, and both estimate and document therapeutic improvements in three patients with well-developed foveation periods, fairly broad, lateral gaze "nulls," head turns, strabismus, and complex, multiplanar nystagmus. PATIENTS AND METHODS: Infrared reflection, magnetic search coil, and high-speed digital video systems were used to record the eye movements of INS patients, pre- and post-Kestenbaum null-point correction surgery (horizontal or vertical). Data were analyzed and estimations made, using the eXpanded Nystagmus Acuity Function (NAFX) that is part of the OMtools toolbox for MATLAB. RESULTS: In all three subjects (S1-S3), both peak NAFX and longest foveation domain (LFD) improved from their pre-Kestenbaum values. S1: 0.700-0.745 (6.4%) and 25-34° (36%), respectively. S2: 0.445-0.633 (42.4%) and >40° to >50° (10%), respectively. S3: 0.250-0.300 (20%) and 13° to ≫18° (see text), respectively. CONCLUSIONS: S1: Even at the high ends of the pre-therapy NAFX and LFD spectra, INS foveation (and therefore, visual-function) improvements may be adequate to justify nystagmus surgery and provide clinical improvements beneficial to the patient. S2: INS foveation improvements in the vertical plane are equal to those originally estimated using the horizontal data in prior patients. S3: Two apparent NAFX peaks can be converted into a very broad peak by surgery based on the preferred lower peak. SN - 2576-117X UR - https://www.unboundmedicine.com/medline/citation/30332339/Longitudinal_Studies_and_Eye_Movement_Based_Treatments_of_Infantile_Nystagmus_Syndrome:_Estimated_and_Measured_Therapeutic_Improvements_in_Three_Complex_Cases_ L2 - http://www.tandfonline.com/doi/full/10.1080/2576117X.2018.1522917 DB - PRIME DP - Unbound Medicine ER -