Diffuse infiltrating retinoblastoma: a multicentre, international, data-sharing study.
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Tomar Ankit Singh, Finger Paul T, Gallie Brenda, Kivelä Tero T, Mallipatna Ashwin, Zhang Chengyue, Zhao Junyang, Wilson Matthew W, Brennan Rachel C, Burges Michala, Berry Jesse L, Jubran Rima, Khetan Vikas, Ganesan Suganeswari, Yarovoy Andrey, Yarovaya Vera, Kotova Elena, Volodin Denis, Yousef Yacoub A, Nummi Kalle, Ushakova Tatiana L, Yugay Olga V, Polyakov Vladimir G, Ramirez-Ortiz Marco A, Esparza-Aguiar Elizabeth, Chantada Guillermo, Schaiquevich Paula, Fandiño Adriana, Yam Jason C, Lau Winnie W, Lam Carol P, Sharwood Phillipa, Moorthy Sonia, Long Quah Boon, Essuman Vera Adobea, Renner Lorna A, Semenova Ekaterina, Catala-Mora Jaume, Correa-Llano Genoveva, Carreras Elisa, AJCC OOTF
Аннотация
METHODS: This international, multicentre, registry-based retrospective case series analysed pooled data from January 2001 to December 2013, including 132 eyes from 132 patients with DIR.
RESULTS: Among 2854 eyes with retinoblastoma, 132 (4.6%; 95% CI, 3.9 to 5.5) had DIR. The median age at diagnosis for DIR patients was 24 months (IQR, 15-33), with no bilateral cases of DIR. The American Joint Committee on Cancer staging showed 4.5% cT2 and 95.5% cT3 categories, with no cT1 or cT4 cases. Clinical features associated with DIR included secondary glaucoma (67%), retinal detachment (38%), diffuse vitreous seeds (37%), anterior segment involvement (24%), vitreous haemorrhage (50%) and hyphema (6%). Primary enucleation was the predominant treatment (81%), while 19% initially received systemic chemotherapy, with 6% requiring subsequent enucleation. The 5-year Kaplan-Meier survival rate for cT3 DIR was 82% (95% CI, 78 to 86), significantly lower than the 94% (95% CI, 93 to 95) for cT3 non-DIR cases (p<0.001). Cox proportional hazards regression returned a higher risk of metastatic death for DIR cT3 compared with non-DIR cT3 (HR, 3.3; 95% CI, 1.8 to 5.9; p<0.001). High-risk pathological features were more frequent in DIR (41% vs 28%, p=0.004). There was no association between DIR and local treatment failure.
CONCLUSIONS: Approximately 1 in 20 patients with retinoblastoma had DIR, which often presented with glaucoma, anterior segment involvement or intraocular bleeding. DIR was more lethal than non-DIR cT3, with enucleation revealing high-risk pathology.