Publication: The Prevalence of Retinal Disease and Associated CNS Disease in Young Patients with Incontinentia Pigmenti
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2022-01-01
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24686530
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2-s2.0-85133884434
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Mahidol University
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Ophthalmology Retina. (2022)
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Ian D. Danford, Brittni A. Scruggs, Antonio Capone, Michael T. Trese, Kim A. Drenser, Aristomenis Thanos, Eric Nudleman, Atchara Amphornphruet, Boontip Tipsuriyaporn, G. Baker Hubbard, Anna Ells, C. Armitage Harper, Jessica Goldstein, Charles Calvo, Chris Wallace-Carrete, Duncan Berry, Emmanuel Chang, Lisa Leishman, Michael Shapiro, Michael Blair, Mikel Mikhail, Carol L. Shields, Rachel Schwendeman, Yoshihiro Yonekawa, Mrinali P. Gupta, Anton Orlin, Supalert Prakhunhungsit, Shizuo Mukai, Audina Berrocal, M. Elizabeth Hartnett, J. Peter Campbell (2022). The Prevalence of Retinal Disease and Associated CNS Disease in Young Patients with Incontinentia Pigmenti. Retrieved from: https://repository.li.mahidol.ac.th/handle/123456789/74927.
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The Prevalence of Retinal Disease and Associated CNS Disease in Young Patients with Incontinentia Pigmenti
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Author(s)
Ian D. Danford
Brittni A. Scruggs
Antonio Capone
Michael T. Trese
Kim A. Drenser
Aristomenis Thanos
Eric Nudleman
Atchara Amphornphruet
Boontip Tipsuriyaporn
G. Baker Hubbard
Anna Ells
C. Armitage Harper
Jessica Goldstein
Charles Calvo
Chris Wallace-Carrete
Duncan Berry
Emmanuel Chang
Lisa Leishman
Michael Shapiro
Michael Blair
Mikel Mikhail
Carol L. Shields
Rachel Schwendeman
Yoshihiro Yonekawa
Mrinali P. Gupta
Anton Orlin
Supalert Prakhunhungsit
Shizuo Mukai
Audina Berrocal
M. Elizabeth Hartnett
J. Peter Campbell
Brittni A. Scruggs
Antonio Capone
Michael T. Trese
Kim A. Drenser
Aristomenis Thanos
Eric Nudleman
Atchara Amphornphruet
Boontip Tipsuriyaporn
G. Baker Hubbard
Anna Ells
C. Armitage Harper
Jessica Goldstein
Charles Calvo
Chris Wallace-Carrete
Duncan Berry
Emmanuel Chang
Lisa Leishman
Michael Shapiro
Michael Blair
Mikel Mikhail
Carol L. Shields
Rachel Schwendeman
Yoshihiro Yonekawa
Mrinali P. Gupta
Anton Orlin
Supalert Prakhunhungsit
Shizuo Mukai
Audina Berrocal
M. Elizabeth Hartnett
J. Peter Campbell
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Siriraj Hospital
Calgary Retina Consultants
Emory Eye Center
Devers Eye Institute
Oregon Health & Science University
Bascom Palmer Eye Institute
Queen’s University
Thomas Jefferson University
University of Utah Health
Queen Sirikit National Institute of Child Health
Harvard University
Associated Retinal Consultants
Weill Cornell Medicine
Mayo Clinic
Shiley Eye Institute
Ltd
Austin Retina Associates
Nose and Throat Associates
Retina Associates of Orange County
Retina and Vitreous of Texas
Retina Consultants of Nevada
Calgary Retina Consultants
Emory Eye Center
Devers Eye Institute
Oregon Health & Science University
Bascom Palmer Eye Institute
Queen’s University
Thomas Jefferson University
University of Utah Health
Queen Sirikit National Institute of Child Health
Harvard University
Associated Retinal Consultants
Weill Cornell Medicine
Mayo Clinic
Shiley Eye Institute
Ltd
Austin Retina Associates
Nose and Throat Associates
Retina Associates of Orange County
Retina and Vitreous of Texas
Retina Consultants of Nevada
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Abstract
Purpose: To evaluate the prevalence of retinal disease on fluorescein angiography (FA) in patients with incontinentia pigmenti (IP) and to compare the severity of retinal disease in those with and without known CNS disease. Design: Multi-institutional consecutive retrospective case series. Subjects: New patients with a diagnosis of IP were seen at the Casey Eye Institute, Oregon Health and Science University, Moran Eye Center, University of Utah, Wills Eye Hospital, or Bascom Palmer Eye Institute, University of Miami from December 2011 to September 2018. Methods: Detailed ophthalmoscopic examination and FA were recommended for all new patients and performed on every patient who had parental consent. Ophthalmoscopic findings and FA images were graded for severity by 2 masked graders on a 3-point scale: 0 = no disease, 1 = vascular abnormalities without leakage, 2 = leakage or neovascularization, and 3 = retinal detachment. The presence of known CNS disease was documented. Additional cases were obtained from a pediatric retina listserv for examples of phenotypic variation. Main Outcome Measures: The proportion of eyes noted to have disease on ophthalmoscopy compared with FA and the severity of retinal disease in those with and without known CNS disease. Results: Retinal pathology was detected in 18 of 35 patients (51%) by indirect ophthalmoscopy and 26 of 35 patients (74%) by FA (P = 0.048) in a predominantly pediatric population (median age, 9 months). Ten patients (29%) had known CNS disease at the time of the eye examination. A Wilcoxon rank-sum test indicated that the retinal severity scores for patients with CNS disease (median, 2) were significantly higher than the retinal severity scores for patients without CNS disease (median, 1), z = -2.12, P = 0.034. Conclusions: Retinal disease is present in the majority of patients with IP, and ophthalmoscopic examination is less sensitive than FA for detection of disease. There may be a correlation between the severity of retinal and CNS disease.