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1. Refractive Growth of the Crystalline Lens in the Infant Aphakia Treatment Study

Author

Lambert, Scott R., DuBois, Lindreth, Nizam, Azhar, Long, Qi, Lynn, Michael J., Bridgman, Betsy, Celano, Marianne, Cleveland, Julia, Cotsonis, George, Drews-Botsch, Carey, Freret, Nana, Lu, Lu, Swanson, Seegar, Tutu-Gxashe, Thandeka, Hartmann, E. Eugenie, Carrigan, Anna K., Edwards, Clara, Busettini, Claudio, Hayley, Samuel, Lewis, Eleanor, Joost Felius, Alicia Kindred, Buckley, Edward G., Plager, David A., Wilson, M. Edward, Drews-Botsch, Carolyn, Everett, Donald F., Felius, Joost, Bozic, Margaret, Holleschau, Ann, Russell, Buddy, Ward, Michael, Bradham, Carol, Vanderveen, Deborah K., Mansfield, Theresa A., Miller, Kathryn Bisceglia, Winter, Tamar, Christiansen, Stephen P., Bothun, Erick D., Jedlicka, Jason, Winters, Patricia, Lang, Jacob, Anderson, Jill S., Traboulsi, Elias I., Crowe, Susan, Cimino, Heather Hasley, Orge, Faruk, Kwiatkowski, Megin, Colon, Beth, Meador, Angela, Yen, Kimberly G., Castanes, Maria, Sanchez, Alma, York, Shirley, Malone, Stacy, Olfson, Margaret, Romany, Gihan, Wheeler, David T., Stout, Ann U., Rauch, Paula, Beaudet, Kimberly, Berg, Pam, Wilson, Lorri, Hutchinson, Amy K., Dubois, Lindreth, Robb, Rachel, Shainberg, Marla J., Freedman, Sharon F., Duncan, Lois, Phillips, B.W., Petrowski, John T., Jones, Sarah, Morrison, David, Owings, Sandy, Biernacki, Ron, Franklin, Christine, Ruark, Scott, Neely, Daniel E., Whitaker, Michele, Bates, Donna, Donaldson, Dana, Kruger, Stacey, Tibi, Charlotte, Vega, Susan, Weakley, David R., Stager, David R., Jr., Dias, Clare, Sager, Debra L., Brantley, Todd, Miller, Bonnie, Lutz, Eva, Davis, Lisa, Hardy, Robert, Birch, Eileen, Cheng, Ken, Hertle, Richard, Kollman, Craig, Yeargin-Allsopp, Marshalyn, McDowell, Cyd, Beck, Allen, McClatchey, Thaddeus S., Morrison, David G., Kruger, Stacey J., Wilson, Lorri B., and McClatchey, Scott K.

Published
2022
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2. Refractive Growth of the Crystalline Lens in the Infant Aphakia Treatment Study

Author

McClatchey, Thaddeus S., primary, Lambert, Scott R., additional, Morrison, David G., additional, Kruger, Stacey J., additional, Wilson, Lorri B., additional, McClatchey, Scott K., additional, DuBois, Lindreth, additional, Nizam, Azhar, additional, Long, Qi, additional, Lynn, Michael J., additional, Bridgman, Betsy, additional, Celano, Marianne, additional, Cleveland, Julia, additional, Cotsonis, George, additional, Drews-Botsch, Carey, additional, Freret, Nana, additional, Lu, Lu, additional, Swanson, Seegar, additional, Tutu-Gxashe, Thandeka, additional, Hartmann, E. Eugenie, additional, Carrigan, Anna K., additional, Edwards, Clara, additional, Busettini, Claudio, additional, Hayley, Samuel, additional, Lewis, Eleanor, additional, Joost Felius, Alicia Kindred, additional, Buckley, Edward G., additional, Plager, David A., additional, Wilson, M. Edward, additional, Drews-Botsch, Carolyn, additional, Everett, Donald F., additional, Felius, Joost, additional, Bozic, Margaret, additional, Holleschau, Ann, additional, Russell, Buddy, additional, Ward, Michael, additional, Bradham, Carol, additional, Vanderveen, Deborah K., additional, Mansfield, Theresa A., additional, Miller, Kathryn Bisceglia, additional, Winter, Tamar, additional, Christiansen, Stephen P., additional, Bothun, Erick D., additional, Jedlicka, Jason, additional, Winters, Patricia, additional, Lang, Jacob, additional, Anderson, Jill S., additional, Traboulsi, Elias I., additional, Crowe, Susan, additional, Cimino, Heather Hasley, additional, Orge, Faruk, additional, Kwiatkowski, Megin, additional, Colon, Beth, additional, Meador, Angela, additional, Yen, Kimberly G., additional, Castanes, Maria, additional, Sanchez, Alma, additional, York, Shirley, additional, Malone, Stacy, additional, Olfson, Margaret, additional, Romany, Gihan, additional, Wheeler, David T., additional, Stout, Ann U., additional, Rauch, Paula, additional, Beaudet, Kimberly, additional, Berg, Pam, additional, Wilson, Lorri, additional, Hutchinson, Amy K., additional, Dubois, Lindreth, additional, Robb, Rachel, additional, Shainberg, Marla J., additional, Freedman, Sharon F., additional, Duncan, Lois, additional, Phillips, B.W., additional, Petrowski, John T., additional, Jones, Sarah, additional, Morrison, David, additional, Owings, Sandy, additional, Biernacki, Ron, additional, Franklin, Christine, additional, Ruark, Scott, additional, Neely, Daniel E., additional, Whitaker, Michele, additional, Bates, Donna, additional, Donaldson, Dana, additional, Kruger, Stacey, additional, Tibi, Charlotte, additional, Vega, Susan, additional, Weakley, David R., additional, Stager, David R., additional, Dias, Clare, additional, Sager, Debra L., additional, Brantley, Todd, additional, Miller, Bonnie, additional, Lutz, Eva, additional, Davis, Lisa, additional, Hardy, Robert, additional, Birch, Eileen, additional, Cheng, Ken, additional, Hertle, Richard, additional, Kollman, Craig, additional, Yeargin-Allsopp, Marshalyn, additional, McDowell, Cyd, additional, and Beck, Allen, additional

Published
2022
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3. Topical lambda-cyhalothrin in reducing eye oscillations in a canine model of infantile nystagmus syndrome

Author

Hertle, Richard, Dell'Osso, Louis, Jacobs, Jonathan, Yang, Dongsheng, Dumire, Jeffery, and Evano-Chapman, Michelle

Subjects
Insecticides -- Usage -- Health aspects, Ophthalmic research, Nystagmus -- Care and treatment, Health
Abstract

Byline: Richard. Hertle, Louis. Dell'Osso, Jonathan. Jacobs, Dongsheng. Yang, Jeffery. Dumire, Michelle. Evano-Chapman Purpose: To determine the ocular and systemic safety of using topical Lambda-Cyhalothrin (LCL) in a canine model [...]

Published
2020

4. Genotypic and Phenotypic Spectrum of Foveal Hypoplasia:A Multicenter Study

Author

Kuht, Helen J., Maconachie, Gail D.E., Han, Jinu, Kessel, Line, van Genderen, Maria M., McLean, Rebecca J., Hisaund, Michael, Tu, Zhanhan, Hertle, Richard W., Grønskov, Karen, Bai, Dayong, Wei, Aihua, Li, Wei, Jiao, Yonghong, Smirnov, Vasily, Choi, Jae Hwan, Tobin, Martin D., Sheth, Viral, Purohit, Ravi, Dawar, Basu, Girach, Ayesha, Strul, Sasha, May, Laura, Chen, Fred K., Heath Jeffery, Rachael C., Aamir, Abdullah, Sano, Ronaldo, Jin, Jing, Brooks, Brian P., Kohl, Susanne, Arveiler, Benoit, Montoliu, Lluis, Engle, Elizabeth C., Proudlock, Frank A., Nishad, Garima, Pani, Prateek, Varma, Girish, Gottlob, Irene, Thomas, Mervyn G., Kuht, Helen J., Maconachie, Gail D.E., Han, Jinu, Kessel, Line, van Genderen, Maria M., McLean, Rebecca J., Hisaund, Michael, Tu, Zhanhan, Hertle, Richard W., Grønskov, Karen, Bai, Dayong, Wei, Aihua, Li, Wei, Jiao, Yonghong, Smirnov, Vasily, Choi, Jae Hwan, Tobin, Martin D., Sheth, Viral, Purohit, Ravi, Dawar, Basu, Girach, Ayesha, Strul, Sasha, May, Laura, Chen, Fred K., Heath Jeffery, Rachael C., Aamir, Abdullah, Sano, Ronaldo, Jin, Jing, Brooks, Brian P., Kohl, Susanne, Arveiler, Benoit, Montoliu, Lluis, Engle, Elizabeth C., Proudlock, Frank A., Nishad, Garima, Pani, Prateek, Varma, Girish, Gottlob, Irene, and Thomas, Mervyn G.

Abstract

Purpose: To characterize the genotypic and phenotypic spectrum of foveal hypoplasia (FH). Design: Multicenter, observational study. Participants: A total of 907 patients with a confirmed molecular diagnosis of albinism, PAX6, SLC38A8, FRMD7, AHR, or achromatopsia from 12 centers in 9 countries (n = 523) or extracted from publicly available datasets from previously reported literature (n = 384). Methods: Individuals with a confirmed molecular diagnosis and availability of foveal OCT scans were identified from 12 centers or from the literature between January 2011 and March 2021. A genetic diagnosis was confirmed by sequence analysis. Grading of FH was derived from OCT scans. Main Outcome Measures: Grade of FH, presence or absence of photoreceptor specialization (PRS+ vs. PRS–), molecular diagnosis, and visual acuity (VA). Results: The most common genetic etiology for typical FH in our cohort was albinism (67.5%), followed by PAX6 (21.8%), SLC38A8 (6.8%), and FRMD7 (3.5%) variants. AHR variants were rare (0.4%). Atypical FH was seen in 67.4% of achromatopsia cases. Atypical FH in achromatopsia had significantly worse VA than typical FH (P < 0.0001). There was a significant difference in the spectrum of FH grades based on the molecular diagnosis (chi-square = 60.4, P < 0.0001). All SLC38A8 cases were PRS– (P = 0.003), whereas all FRMD7 cases were PRS+ (P < 0.0001). Analysis of albinism subtypes revealed a significant difference in the grade of FH (chi-square = 31.4, P < 0.0001) and VA (P = 0.0003) between oculocutaneous albinism (OCA) compared with ocular albinism (OA) and Hermansky–Pudlak syndrome (HPS). Ocular albinism and HPS demonstrated higher grades of FH and worse VA than OCA. There was a significant difference (P < 0.0001) in VA between FRMD7 variants compared with other diagnoses associated with FH. Conclusions: We characterized the phenotypic and genotypic spectrum of FH. Atypical FH is associated with a worse prognosis than all other forms

Published
2022

5. Genotypic and Phenotypic Spectrum of Foveal Hypoplasia: A Multicenter Study

Author

MS Oogheelkunde, Child Health, Kuht, Helen J., Maconachie, Gail D.E., Han, Jinu, Kessel, Line, van Genderen, Maria M., McLean, Rebecca J., Hisaund, Michael, Tu, Zhanhan, Hertle, Richard W., Gronskov, Karen, Bai, Dayong, Wei, Aihua, Li, Wei, Jiao, Yonghong, Smirnov, Vasily, Choi, Jae Hwan, Tobin, Martin D., Sheth, Viral, Purohit, Ravi, Dawar, Basu, Girach, Ayesha, Strul, Sasha, May, Laura, Chen, Fred K., Heath Jeffery, Rachael C., Aamir, Abdullah, Sano, Ronaldo, Jin, Jing, Brooks, Brian P., Kohl, Susanne, Arveiler, Benoit, Montoliu, Lluis, Engle, Elizabeth C., Proudlock, Frank A., Nishad, Garima, Pani, Prateek, Varma, Girish, Gottlob, Irene, Thomas, Mervyn G., MS Oogheelkunde, Child Health, Kuht, Helen J., Maconachie, Gail D.E., Han, Jinu, Kessel, Line, van Genderen, Maria M., McLean, Rebecca J., Hisaund, Michael, Tu, Zhanhan, Hertle, Richard W., Gronskov, Karen, Bai, Dayong, Wei, Aihua, Li, Wei, Jiao, Yonghong, Smirnov, Vasily, Choi, Jae Hwan, Tobin, Martin D., Sheth, Viral, Purohit, Ravi, Dawar, Basu, Girach, Ayesha, Strul, Sasha, May, Laura, Chen, Fred K., Heath Jeffery, Rachael C., Aamir, Abdullah, Sano, Ronaldo, Jin, Jing, Brooks, Brian P., Kohl, Susanne, Arveiler, Benoit, Montoliu, Lluis, Engle, Elizabeth C., Proudlock, Frank A., Nishad, Garima, Pani, Prateek, Varma, Girish, Gottlob, Irene, and Thomas, Mervyn G.

Published
2022

6. Visual electrodiagnostics and eye movement recording - World Society of Pediatric Ophthalmology and Strabismus (WSPOS) consensus statement

Author

Pompe, Manca, Liasis, Alki, and Hertle, Richard

Subjects
Electrodiagnosis -- Usage -- Analysis, Eye movements -- Analysis, Practice guidelines (Medicine) -- Research, Health
Abstract

Byline: Manca. Pompe, Alki. Liasis, Richard. HertleVisual electrodiagnostics and eye movement recording are important additional clinical tools in evaluation, diagnosing and management of ophthalmic and neurological disorders. Due to their [...]

Published
2019

7. Clinical and electrophysiological results of eye muscle surgery in 17 patients with downbeat nystagmus

Author

Hertle, Richard and Ahmad, Ashraf

Subjects
Eye muscle surgery -- Usage, Nystagmus -- Care and treatment -- Research, Electrophysiology -- Analysis, Health
Abstract

Byline: Richard. Hertle, Ashraf. AhmadPurpose: To test the hypothesis that eye muscle surgery in treatment of patients with acquired downbeat nystagmus results in improvement measures of visual and ocular motor [...]

Published
2019

15. Eye muscle surgery for infantile nystagmus syndrome in the first two years of life

Author

Hertle,Richard, Felius,Joost, Yang,Dongsheng, Kaufman,Matthew, Hertle,Richard, Felius,Joost, Yang,Dongsheng, and Kaufman,Matthew

Abstract

Richard W Hertle1, Joost Felius2, Dongsheng Yang1, Matthew Kaufman11The UPMC and Children’s Hospital of Pittsburgh Eye Centers and The Laboratory of Visual and Ocular Motor Physiology Pittsburgh, PA, USA; 2The Retina Foundation of the Southwest, Department of Ophthalmology, University of Texas Southwestern Medical Center, Dallas, TX, USAPurpose: To report visual and elctrophysioloigcal effects of eye muscle surgery in young patients with infantile nystagmus syndrome (INS).Methods: Prospective, interventional case cohort of 19 patients aged under 24 months who were operated on for combinations of strabismus, an anomalous head posture, and nystagmus. All patients were followed at least nine months. Outcome measures, part of an institutionally approved study, included Teller acuity, head position, strabismic deviation, and eye movement recordings, from which waveform types and a nystagmus optimal foveation fraction (NOFF). Computerized parametric and nonparametric statistical analysis of data were perfomed using standard software on both individual and group data.Results: Age averaged 17.7 months (13.1-month follow-up). Thirteen (68%) patients had associated optic nerve or retinal disease. 42% had amblyopia, 68% had refractive errors. Group means in binocular Teller acuity (P < 0.05), strabismic deviation (P < 0.05), head posture (P < 0.001), and the NOFF measures (P < 0.01) from eye movement recordings improved in all patients. There was a change in null zone waveforms to more favorable jerk types. There were no reoperations or surgical complications.Conclusions: Surgery on the extraocular muscles in patients aged less than two years with INS results in improvements in multiple aspects of ocular motor and visual function.Keywords: infantile nystagmus, anomalous head posture, eye muscle surgery

Published
2009

23. Albinism: Particular Attention to the Ocular Motor System.

Author

Hertle, Richard W.

Subjects
*ALBINISM, *BINOCULAR vision disorders, *NYSTAGMUS, *OCULAR hypotony, *VISUAL evoked potentials, *PATIENTS, *THERAPEUTICS
Abstract

The purpose of this report is to summarize an understanding of the ocular motor system in patients with albinism. Other than the association of vertical eccentric gaze null positions and asymmetric, (a) periodic alternating nystagmus in a large percentage of patients, the ocular motor system in human albinism does not contain unique pathology, rather has "typical" types of infantile ocular oscillations and binocular disorders. Both the ocular motor and afferent visual system are affected to varying degrees in patients with albinism, thus, combined treatment of both systems will maximize visual function. [ABSTRACT FROM AUTHOR]

Published
2013
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25. Achromatopsia: New oculomotor characteristics of rare inherited disease: Tinted spectacles, lenses, low-vision aids, atropine drops, and patching options.

Author

Hertle, Richard W.

Subjects
*COLOR blindness, *CONTRAST media, *EYE abnormalities, *VISUAL acuity, *EYE examination
Abstract

The article discusses the rare inherited disease called achromatopsia, its diagnosis and possible treatments. Also cited are the adverse effects of achromatopsia in children like photophobia, intense blepharospasm, and infantile nystagmus syndrome (INS), and the special testing solutions like color vision, optical coherence tomography (OCT), and genetic testing.

Published
2022

26. Pinealoblastoma Metastatic to the Optic Nerve

Author

Hertle, Richard W. and Robb, Richard M.

Abstract

We report a 10-year-old white girl who developed a pinealoblastoma 2 years before presenting to us with metastatic spread to the left optic nerve. She was treated for the original tumor with irradiation and chemotherapy. She presented to us with decreased vision in the left eye, unilateral disk edema, and diffuse left optic nerve swelling without increased intracranial pressure. Biopsy of the posterior optic nerve sheath was required for diagnosis. Vision was lost in the left eye, but the optic nerve swelling diminished after chemotherapy and additional radiation therapy.

Published
1990

27. Oculographic Analysis of Acute Esotropia Secondary to a Thalamic Hemorrhage

Author

Hertle, Richard W. and Bienfang, Don C.

Abstract

We used electrooculography to study the saccadic velocities, smooth pursuit, and vestibular ocular reflex in a patient with an acute thalamic hemorrhage. Our findings confirmed what others have shown in that there were hypometric saccades contralateral to the lesion, impaired smooth pursuit ipsilaterally, and a preserved vestibular ocular reflex. In addition, we demonstrated an asymmetry with the contralateral eye being more affected. It is also shown that the “convergence” movements seen on attempted upgaze are typical of saccades and not vergence movements. A discussion of possible pathophysiologic mechanisms with review of other studies is presented.

Published
1990

28. Mutations in FRMD7, a newly identified member of the FERM family, cause X-linked idiopathic congenital nystagmus

Author

Tarpey, Patrick, Thomas, Shery, Sarvananthan, Nagini, Mallya, Uma, Lisgo, Steven, Talbot, Chris J., Roberts, Eryl O., Awan, Musarat, Surendran, Mylvaganam, McLean, Rebecca J., Reinecke, Robert D., Langmann, Andrea, Lindner, Susanne, Koch, Martina, Jain, Sunila, Woodruff, Geoffrey, Gale, Richard P., Bastawrous, Andrew, Degg, Chris, Droutsas, Konstantinos, Asproudis, Ioannis, Zubcov, Alina A., Pieh, Christina, Veal, Colin D., Machado, Rajiv D., Backhouse, Oliver C., Baumber, Laura, Constantinescu, Cris S., Brodsky, Michael C., Hunter, David G., Hertle, Richard W., Read, Randy J., Edkins, Sarah, O'Meara, Sarah, Parker, Adrian, Stevens, Claire, Teague, Jon, Wooster, Richard, Futreal, P. Andrew, Trembath, Richard C., Stratton, Michael R., Raymond, F. Lucy, Gottlob, Irene, Tarpey, Patrick, Thomas, Shery, Sarvananthan, Nagini, Mallya, Uma, Lisgo, Steven, Talbot, Chris J., Roberts, Eryl O., Awan, Musarat, Surendran, Mylvaganam, McLean, Rebecca J., Reinecke, Robert D., Langmann, Andrea, Lindner, Susanne, Koch, Martina, Jain, Sunila, Woodruff, Geoffrey, Gale, Richard P., Bastawrous, Andrew, Degg, Chris, Droutsas, Konstantinos, Asproudis, Ioannis, Zubcov, Alina A., Pieh, Christina, Veal, Colin D., Machado, Rajiv D., Backhouse, Oliver C., Baumber, Laura, Constantinescu, Cris S., Brodsky, Michael C., Hunter, David G., Hertle, Richard W., Read, Randy J., Edkins, Sarah, O'Meara, Sarah, Parker, Adrian, Stevens, Claire, Teague, Jon, Wooster, Richard, Futreal, P. Andrew, Trembath, Richard C., Stratton, Michael R., Raymond, F. Lucy, and Gottlob, Irene

Abstract

Idiopathic congenital nystagmus is characterized by involuntary, periodic, predominantly horizontal oscillations of both eyes. We identified 22 mutations in FRMD7 in 26 families with X-linked idiopathic congenital nystagmus. Screening of 42 singleton cases of idiopathic congenital nystagmus (28 male, 14 females) yielded three mutations (7%). We found restricted expression of FRMD7 in human embryonic brain and developing neural retina, suggesting a specific role in the control of eye movement and gaze stability.

29. IOLs safe, effective in kids.

Author

Hertle, Richard W.

Subjects
*INTRAOCULAR lenses, *PEDIATRIC surgery
Abstract

Reports on the efficacy and safety of the use of intraocular lens (IOL) implantation as a primary method for visual rehabilitation after lensectomy in children. Indications for IOL implantation; Indications for lensectomy; Surgical method; Potential complications; Performing surgical iridectomies.

Published
1996

31. Relationships between versional and vergent quick phases of the involuntary version-vergence nystagmus.

Author

Zhu M, Hertle RW, and Yang D

Subjects
Adult, Humans, Photic Stimulation, Reaction Time physiology, Vision, Binocular physiology, Vision, Monocular physiology, Motion Perception physiology, Nystagmus, Physiologic physiology, Saccades physiology
Abstract

We used ground-plane motion stimuli displayed on a computer monitor positioned below eye level to induce involuntary version-vergence nystagmus (VVN). The VVN was recorded with a search coil system. It was shown that the VVN had both vertical versional and horizontal vergence components. The VVN induced by backward motion (toward subjects) had upward versional and divergence quick phases, whereas those induced by forward motion (away from subjects) had downward and biphasic divergence-convergence quick phases. The versional and vergence components of the VVN quick phases were analyzed. A temporal dissociation of about 20 ms between version velocity peak and convergence velocity peak was revealed, which supported a modified saccade-related vergence burst neuron (SVBN) model. We suggest that the temporal dissociation may be partly because of a lower-level OKN control mechanism. Vergence peak time was dependent on version peak time. Linear relationships between vergence peak velocity and versional saccadic peak velocity were demonstrated, which was in line with the new multiplicative model. Our data support the hypothesis that the vergence system and the saccadic system can act separately but interact with each other whenever their movements occur simultaneously.

Published
2008
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32. Eye movement recordings as an effectiveness indicator of gene therapy in RPE65-deficient canines: implications for the ocular motor system.

Author

Jacobs JB, Dell'Osso LF, Hertle RW, Acland GM, and Bennett J

Subjects
Animals, Blindness genetics, Blindness physiopathology, Blindness therapy, Dog Diseases genetics, Dog Diseases physiopathology, Dogs, Electronystagmography, Eye Movements physiology, Nystagmus, Congenital physiopathology, Nystagmus, Congenital therapy, Retinal Degeneration genetics, Retinal Degeneration physiopathology, Retinal Degeneration therapy, Treatment Outcome, Blindness veterinary, Dog Diseases therapy, Eye Proteins genetics, Genetic Therapy, Nystagmus, Congenital veterinary, Retinal Degeneration veterinary
Abstract

Purpose: To perform ocular motility recordings of infantile nystagmus (IN) in RPE65-deficient canines and determine whether they can be used as a motor indicator of restored retinal function to investigate the effects of gene therapy., Methods: Treated and untreated canines were comfortably suspended in a custom-built sling and encouraged to fixate on distant targets at gaze angles varying between +/-15 degrees horizontally and +/-10 degrees vertically. Ocular motility recordings were made, using two distinct methods-infrared reflection and high-speed video. The resultant recordings from three untreated, four treated, and three pre- and post-treatment dogs were analyzed for using the eXpanded Nystagmus Acuity Function (NAFX), which yields an objective assessment of best potential visual acuity, based on the duration and repeatable accuracy of foveation and centralisation., Results: During fixation, the untreated dogs exhibited large-amplitude, classic IN waveforms, including pendular and jerk in both the horizontal and vertical planes, which prevented them from keeping the targets within the area centralis (the region of highest receptor density, spanning +/-3 degrees horizontally by +/-1.5 degrees vertically, analogous to the fovea). Some untreated dogs also had small-amplitude (0.5-1 degrees), high-frequency (6-9 Hz) oscillations. Under the same conditions, successfully treated canines no longer exhibited clinically detectable IN. Their IN was converted to waveforms with very low amplitudes that yielded higher NAFX values and allowed target images to remain well within the area centralis. Of note, uniocular treatment appeared to damp the IN in both eyes. Behaviorally, the treated dogs were able to successfully navigate through obstacles more easily without inadvertent contact, a task beyond the untreated dogs' ability., Conclusions: Gene therapy that successfully restored retinal function also reduced the accompanying IN to such a great extent that it was not clinically detectable approximately 90% of the time in many of the dogs. IN improvement, as quantified by the NAFX, is an objective motor indicator of visual improvement due to gene therapy.

Published
2006
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