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An Epidemiological and Clinical Study of Ocular Manifestations of Congenitalrubella Syndrome in Omani Children

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An Epidemiological and Clinical Study of Ocular Manifestations of Congenitalrubella Syndrome in Omani Children EPIDEMIOLOGY An Epidemiological and Clinical Study of Ocular Manifestations of Congenital Rubella Syndrome in Omani Children Rajiv Khandekar, MS (Ophth) PGDip Epi; Salah Al Awaidy, PGDip Epi; Anuradha Ganesh, MRCOphth; Shyam Bawikar, MD, MPH Objective: To conduct a follow-up study in patients with testing was possible in 16 children; 4 were bilaterally blind. congenital rubella syndrome (CRS) in Oman and ana- Patients who had undergone eye surgery had signifi- lyze the prevalence of ophthalmic disorders and associ- cantly lower visual acuity, as compared with those who ated systemic problems. had not undergone surgery (relative risk 2.53; 95% con- fidence interval, 1.07-6.13). Among the 11 patients with Methods: This historical prospective cohort study in- CRS with cataract, we found hearing loss, cardiac anoma- cluded review of 32 surviving patients with CRS re- lies, and neuropsychologic anomalies in 7, 4, and 6 chil- ported by the surveillance system in Oman from 1987 dren, respectively. through 2002. All patients underwent a complete oph- thalmic examination that included visual acuity estima- Conclusions: Congenital rubella syndrome has a wide va- tion, refraction and anterior and posterior segment evalu- riety of severe ophthalmic and systemic complications. High ation, and intraocular pressure measurement. Pediatric clinical vigilance for signs of CRS and regular observation and otorhinolaryngologic consultations were also per- of surviving patients with CRS is desirable. In patients with formed. cataract, the functional results of surgery, despite state-of- the-art ophthalmic care, continue to be poor. Because of a Results: The age-adjusted prevalence of CRS in Oman high prevalence of visual, audiologic, and neurologic dis- was 73.2 per million in the Omani population younger abilities, surviving patients with CRS pose a burden on the than 20 years, and the incidence was 0.6 per 1000 live medical and social communities. Emphasis in manage- births. Cataract, retinitis, microphthalmos, and glau- ment ought to be prevention of CRS through effective im- coma were observed in 11, 16, 6, and 4 patients, respec- munization programs. tively. Keratoconus, corneal hydrops, and spontaneous resorption of lens were found in 1 patient each. Vision Arch Ophthalmol. 2004;122:541-545 ONGENITAL RUBELLA SYN- later, and patients with little or no hear- drome (CRS) was first de- ing loss initially may later become deaf. scribed by an ophthal- Therefore, all reported and surviving pa- mologist who linked tients with CRS need to be carefully ob- congenital cataract to served for early detection and manage- German measles infection in mothers dur- ment of new disease manifestations.4 C1 ing pregnancy. Although rubella is a mild The World Health Organization en- disease in adults, when a woman con- courages its member countries to From the Eye and Ear Health Care Programme, Ministry of tracts rubella during pregnancy, there is strengthen their surveillance system for Health (Dr Khandekar); a risk for transplacental transmission of the CRS to achieve global elimination of CRS 5 Department of Disease virus and development of serious compli- by 2010. New cases of CRS have been rare Surveillance and Disease cations in the fetus. The consequences of since the development of an attenuated Control, Ministry of Health rubella infection in utero are the manifes- vaccine in 1969 and the effective imple- (Dr Al Awaidy); Department of tations of CRS.2 Congenital cataract, glau- mentation of immunization programs.6 Ophthalmology, Sultan Qaboos coma, and pigmentary retinopathy are con- However, epidemics of rubella continue to University Hospital sidered cardinal features of the syndrome, occur. De Owens et al7 reported the birth (Dr Ganesh); and Department and a diagnosis of CRS can be made in their of 54 neonates with CRS in Panama in of Disease Surveillance and presence despite lack of laboratory evi- 1986. Lee et al8 reported a resurgence of Disease Control, Ministry of 3 Health (Dr Bawikar), Muscat, dence. CRS in the United States in the 1990s. Oman. The authors have no Patients with CRS exhibit progres- The Sultanate of Oman is a member relevant financial interest in sive disease; patients without cataract or country in the Eastern Mediterranean Re- this article. glaucoma in infancy might manifest them gion of the World Health Organization, (REPRINTED) ARCH OPHTHALMOL / VOL 122, APR 2004 WWW.ARCHOPHTHALMOL.COM 541 ©2004 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/25/2021 cally suspected CRS. The child is immediately referred to a pe- Table 1. Profile of the 32 Patients diatrician for detailed examination and laboratory confirmation. With Congenital Rubella Syndrome in Oman The patient is examined by a senior ophthalmologist, oto- rhinolaryngologic surgeon, cardiologist, neurologist, and en- Patient Characteristic No. of Patients (%) docrinologist. All physicians and staff in the specialist clinic Sex are aware of mandatory notification of a clinically suspected Male 22 (69) CRS case in Oman. The cases that fulfill the CRS definition are Female 10 (31) evaluated annually for the presence of newer manifestations. Age group, y Patients with CRS who cannot be offered treatment at facili- Ͻ5 2 (6) ties available within Oman are sent abroad for treatment at the 5-9 4 (12) government’s expense. The tertiary child health care units main- 10-14 20 (62) tain details of such cases. The information of all CRS cases re- 15-19 6 (19) ported through the surveillance system and case records at sec- Region ondary and tertiary hospitals were reviewed to ensure complete Muscat 5 (16) enlisting of all CRS cases. Dhofar 9 (28) For the present study, so we could determine their pres- Dhakhiliya 2 (6) ent status, all children with CRS underwent a detailed oph- North Sharqiya 6 (19) thalmic examination that included evaluation of best- South Sharqiya 0 (0) corrected visual acuity, slitlamp examination of the anterior North Batinah 3 (9) segment, measurement of intraocular pressure, and examina- South Batinah 3 (9) tion of the posterior segment by means of indirect ophthal- Dhahira 2 (6) moscopy through dilated pupils. B-scan ultrasonography was Musundam 0 (0) performed for posterior segment evaluation in cases of media Al Wousta 2 (6) opacities. Axial length was evaluated by means of A-scan ul- trasonography. Presence of strabismus was established by per- forming the Hirschberg test. Children older than 6 years were with a high-quality surveillance and disease control sys- tested by means of the Snellen chart. Younger children were tem for communicable diseases9 and aims to eliminate evaluated by means of the Snellen chart and Kolt test. When CRS by 2005.10 In Oman, there was an outbreak of ru- formal visual acuity testing was not possible, counting fingers bella in 1992 and 1993, and many infants born in that or identification of items used on a daily basis was tested at a year exhibited features of CRS. Since 1994, important distance of 1 m. milestones in the control of rubella in Oman have been achieved, notably mass vaccination of children; intro- QUALITY ENSURANCE PROCEDURES duction of measles, mumps, and rubella vaccination in A national seminar was conducted by the Department of Dis- an immunization schedule; and rubella immunization for ease Surveillance and Disease Control to explain the revised all mothers after childbirth. A high-quality surveillance surveillance and reporting system for CRS. Uniform pretested and disease control system for communicable diseases data collection forms were used. Multiple sources were used has been established, and strategies have been reorga- to ensure enrollment of all CRS cases. nized to make surveillance of CRS more sensitive.10 The ocular profile pertaining to congenital cataract DATA MANAGEMENT SYSTEM during the 1992 epidemic of rubella in Oman has been reported.11 However, limited information is available about Data were collected from regional hospitals and computed by the entire spectrum of ocular manifestations of CRS in using a pretested format (Epi Info 6.0; Centers for Disease Con- this cohort. We reviewed the magnitude of CRS and the trol and Prevention, Atlanta, Ga). Predetermined checks en- sured a high standard of data entry. The frequencies, percent- ocular profile in children with CRS in Oman. age, and proportion of different ocular manifestations were METHODS calculated (SPSS 9.0; SPSS Inc, Chicago, Ill), and a univariate method of analysis was adopted for the study. The prevalence This was a historical prospective cohort study. All patients with of CRS in the Omani population younger than 20 years was CRS reported through the national surveillance system from adjusted by using an indirect standardization method, for which 14 January 1, 1987, through December 31, 2002, formed the study the proportion of the global population was used. population. Patients had CRS diagnosed on the basis of crite- ria provided by the World Health Organization.12,13 ETHICAL ISSUES At nearly 165 primary health institutions, physicians ex- amine the newly born and children at the time of their vacci- The permission of national and regional health administrators nation visits to assess the presence of white pupil, nystagmus, was obtained to conduct this study. Patient identity was kept and abnormal eyeball size. Clinically suspected CRS cases are confidential. The results of the study were shared with the re- referred to ophthalmologists for confirmation. A newborn with gional health administrators, and recommendations to further any congenital anomaly is screened by a pediatrician to rule improve the care of patients with CRS were discussed. out toxoplasmosis, other agents, rubella, cytomegalovirus, and herpes simplex infection in the mother and the possibility of RESULTS CRS in the child. On the basis of the criteria set by the World Health Organization, all health institutions (governmental and private clinics) notify the Department of Disease Surveillance Thirty-two patients had clinical manifestations compat- and Disease Control, Muscat, Oman, by fax of any child with ible with CRS; 28 (88%) had ocular manifestations.
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