Telemedicine can be broadly defined as the remote interpretation of a patient for the purpose of screening, diagnosis and monitoring. In ophthalmology it has been used with variable success in adult populations for screening of diabetes and glaucoma.1,2 In addition, variations of traditional physician-guided telemedicine have been employed for home monitoring of macular disease that relies on patient-centric monitoring, specifically the ForeseeHome device-based system and the DigiSight smartphone-based system.3,4 Google and Alcon have recently announced a partnership to commercialize a contact lens-based insulin monitor for patients with diabetes, and a similar system is also being developed for intraocular pressure monitoring in patients with glaucoma.5,6 These approaches have taken a while to gain market penetrance due to resistance from patients and physicians, based upon both technological and financial hurdles. It has been difficult to develop a financial model that makes sense for both the physician and the patient, creating a barrier to wider acceptance, and patients have demonstrated resistance to spending money for preventive care.

Partly, this is a problem of casting too wide a net—if one screens for all diabetic retinopathy, one is likely to find a lot of diabetic retinopathy that does not need intervention, ironically increasing the burden on the same physicians it is intended to be assisting. A notable exception has been for retinopathy of prematurity screening. This application has worked for three simple reasons: 1) the remote screening is highly effective at identifying a treatment intervention time point; 2) the disease is self-limited in that it will either spontaneously resolve or result in retinal detachment and blindness within a 15-week time frame; and, 3) all neonatal intensive care units (NICUs) are required to perform screening in order to maintain accreditation, removing most of the financial uncertainty. These three components give us great insight into greater application of telemedicine in ophthalmology. First, let us review the ROP scenario.

ROP Telemedicine

Right eye. Scattered subretinal blot hemorrhages sparing the fovea in a normal-term infant.

Left eye. Multiple near-confluent white-centered subretinal hemorrhages in the macula, peri-papillary and extramacular locations.

 
ROP screening with wide-angle, fiber optic cameras was demonstrated to be feasible in the early 2000s,7 equivalent to bedside indirect ophthalmoscopy for detecting any ROP in the PHOTO-ROP trial,8 able to detect treatment-warranted ROP (e.g., Type 1 disease: Zone I and Plus OR Zone I and Stage 3 OR Zone I or II, Stage 3, with Plus) in the SUNDROP experience,9 and equivalent to BIO for detecting referral-warranted ROP (similar to Type 1 disease) in the e-ROP trial.10 Basically, remote digital fundus imaging (RDFI) using wide-angle cameras (130 degrees) captured all treatment inflection points for whatever ROP severity was present. This does not mean it identified all disease; in fact, in the SUNDROP experience, it has been shown that oftentimes disease exists that is detected on BIO, not on camera. However, this disease is in Zone III and would not trigger a treatment intervention using current guidelines. This is an important point: Telemedicine screening is designed to monitor for treatment inflection points, not capture of all disease. Noting the success of these endeavors, the Joint Statement Screening guidelines were updated in 2013 to allow for telemedicine screening of ROP,11 and the American Academy of Pediatrics recently issued a joint technical report on telemedicine screening for ROP in 2015 which concluded “… evidence of moderate (levels II and III) quality supports the use of RDFI to identify patients with clinically significant or referral-warranted ROP for ophthalmic evaluation and management.”12 The SUNDROP network, which I founded in 2005 at Stanford University, has the longest experience in provision of telemedicine screening services for ROP in the United States. This is a community outreach project, which provides underserved NICUs with access to quaternary ROP experts. Its success has been predicated in no small measure on the fact that it has been financially sustainable from the beginning because of the NICU requirements. We have developed a partnership with our member NICUs to endeavor to prevent follow-up miscues upon discharge of the patients. Review of medical records indicates that most blindness from ROP occurs because of lack of screening for one reason or another, as opposed to actual treatment failures. This has placed a greater onus on expanding the screening to cover all eligible patients. The success has empowered everyone to maintain a culture of pride about prevention of blindness in this at-risk population.

Universal Screening of Newborns

Pediatric vitreoretinal surgery physicians have been looking to expand upon the success of telemedicine screening for ROP. One potential opportunity is universal screening of newborn infants. In China, Brazil, Hungary and Spain, universal screening has been practiced in single-hospital settings for up to seven years.
The telemedicine experience in pediatric retinal diseases has been successful because it has avoided the pitfalls of casting too wide a net. 
There has been remarkable similarity of findings across diverse populations: 10- to 20-percent incidence of fundus hemorrhages, and 1- to 2-percent incidence of all other pathology.13 This is of great importance for two reasons: 1) the 2-percent incidence of non-hemorrhage pathology meets the 2-percent incidence threshold for a screening program to have socioeconomic benefit if performed on a large scale in a general population; and, 2) depending on socio-economic status, there is a 1.8- to 4-percent rate of unexplained amblyopia in the United States, which may be due to transient phenomena that can temporarily occlude the visual axis (such as retinal, optic nerve and foveal hemorrhages). Fortunately, we will have two- and three-year follow-up on development of amblyopia in children identified with ocular abnormalities in the Newborn Eye Screen Testing (NEST) prospective study at Stanford University School of Medicine in the summers of 2015 and 2016, respectively.

Other strategies that are being employed are assessment of axial length, refraction, optical coherence studies of the macula, and, potentially, intraocular pressure assessment in the near future. While the NEST program is currently being evaluated as a prospective study with longitudinal follow-up with pediatric ophthalmology/retina specialists, it is evident from early data that commercialization and widespread adoption will be offered in the future.

The telemedicine experience in pediatric retinal diseases has been successful because it has avoided the pitfalls of casting too wide a net. Instead, we have identified niche markets with well-defined intervention points that are easily identified using the technology. These markets, whether in ROP or congenital ocular pathology, have a limited timeframe in which therapy is beneficial, but can result in life-long benefit. Therefore, the societal and patient benefit is large from these screening programs. As ophthalmologists, we need to continue to define very narrow ranges of targeted telemedicine screening that will offer immediate relief and benefit, while still maintaining economic feasibility.  REVIEW


Dr. Moshfeghi is an associate professor of ophthalmology at Stanford University School of Medicine where he is the director of the vitreoretinal surgery fellowship program and director of pediatric vitreoretinal surgery as well as director of telemedicine (ophthalmology). He may be reached at dariusm@stanford.edu.



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