Publication: Combined hamartoma of the retina and retinal pigment epithelium at pediatric age: surgical versus conservative approach
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Date
2022-10-10
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Abstract
Purpose: To report outcomes of pediatric patients with combined hamartoma of the retina and the retina
pigment epithelium (CHRRPE) followed up conservatively or after pars plana vitrectomy (PPV).
Methods: This retrospective multicenter study included sixty-two eyes of 59 pediatric patients with
CHRRPE from 13 different international centers with an average age of 7.7±4.7 (0.3-17) years at the
time of the diagnosis, and having undergone PPV or followed conservatively. At baseline and each visit,
visual acuity values, optical coherence tomography (OCT) for features and central foveal thickness
(CFT), and tumor location were noted. Lesions were called as zone 1, if it involves the macular and
peripapillary area, and the others were called as zone 2 lesions.
Results: Twenty-one eyes of 20 patients in the intervention group and 41 eyes of 39 patients in the
conservative group were followed for a mean of 36.2±40.4 (6-182) months. BCVA improved in 11
(68.8%) of 16 eyes in the intervention group and 4 (12.9%) of 31 eyes in the conservative group
(p<0.001). The mean CFT decreased from 602.0±164.9 μm to 451.2±184.3 μm in the intervention
group, while it increased from 709.5±344.2 μm to 791.0±452.1 μm in zone 1 eyes of the conservative
group. Posterior location of tumor, irregular configuration of the foveal contour and ellipsoid zone
defect in OCT, subretinal exudate and prominent vascular tortuosity were associated with poor visual
acuity.
Conclusions: Vitreoretinal surgery is safe and effective in improving vision and reducing retinal
distortion in zone 1 CHRRPE in children.
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Keywords
combined hamartoma of the retina and the retina pigment epithelium, pediatric age, pars plana vitrectomy, retina, retinal pigment epithelium, surgery, amblyopia treatment
Citation
Ozdek S., Ucgul A. Y., Hartnett M. E., Akdogan M., Sen P., Bhende M., Besirli C. G., Karacorlu M., Dedania V., Parolini B., et al., "Combined Hamartoma of the Retina and Retinal Pigment Epithelium at Pediatric Age: Surgical versus Conservative Approach.", Retina (Philadelphia, Pa.), 2022