Background/Purpose: To report a case of crystalline retinopathy following high-dose tamoxifen use in a pediatric patient. Methods: Observational case report. Results: A 6-year-old boy with history of more than 80-g cumulative tamoxifen use over 25 months for the treatment of atypical teratoid/rhabdoid tumor of the posterior fossa presented with a 4-month history of blurred vision. Fundus examination demonstrated multiple superficial foveal refractile opacities in each eye, and spectral optical coherence tomography revealed numerous punctate hyperreflective deposits located within the inner retina. These findings were suggestive of tamoxifen retinopathy. Conclusion: To our knowledge, this is the first report of multimodal retinal imaging of tamoxifen retinopathy in a pediatric patient. Given the risk of permanent vision loss, ophthalmic baseline screening and monitoring should be considered for children receiving tamoxifen.

Introduction We describe a novel colopathy associated with pentosan polysulfate (PPS) use and measure the strength of the drug-disease association. Methods Two-part investigation. In the cohort study of individuals with a history of prior long-term PPS use, case histories were obtained and gastrointestinal disease course was followed with review of endoscopy records and histopathology specimens. Findings were summarized with descriptive statistics. In the cross-sectional study of individuals with interstitial cystitis, drug exposure and medical histories were obtained for patients seen at a single clinical center. Strength of association between PPS use and diagnoses of inflammatory bowel disease (IBD) and/or irritable bowel syndrome (IBS) was measured with multivariate logistic regression. Results In the cohort study of 13 participants, median PPS exposure was 2.04 kg (0.99-2.54). Eleven (84.6%) developed symptoms suggestive of IBD and/or IBS after initiation of PPS therapy. Of the 10 participants whose endoscopic and histopathologic findings we reviewed, six had abnormal-appearing colonic mucosa on endoscopy and all 10 had abnormal mucosal changes on histology. Clinical and histologic improvement was observed after PPS cessation. In the cross-sectional study of 219 subjects with interstitial cystitis, PPS use was a statistically significant predictor of both the IBD [adjusted odds ratio=3.3 (95% confidence interval, 1.2-8.8, p=0.02)] and the composite IBD+IBS [adjusted odds ratio=3.3 (95% confidence interval, 1.5-7.3, p=0.002)] outcomes. Discussion We describe a strong association between PPS use and a clinical diagnosis of IBD and/or IBS. Histopathologic findings suggest a novel drug-associated colopathy, with some subjects requiring colectomy for dysplasia.

Aim Despite the increasing trend to utilize radiation therapies for the treatment of hepatocellular carcinoma (HCC), there is limited analysis comparing treatment outcomes of various radiation-based therapies including selective internal radiation therapy (SIRT) to stereotactic body radiation therapy (SBRT) and proton beam therapy (PBT). This study aims to examine the heterogeneity of designs and outcomes in existing radiation therapy studies for the treatment of HCC. Methods A systematic review was performed to compare the radiation therapies for HCC including SBRT, PBT, and yttrium-90 (Y90) SIRT by searching through PubMed, EMBASE, Medline, and Cochrane library databases. The main outcomes analyzed were overall survival rates, median overall survival, and progression-free survival with additional analysis of baseline patient characteristics, including Barcelona Clinic Liver Cancer stage, Child–Pugh class, and tumor size. Results Eighty-seven studies comprising 7,462 patients were included. The pooled 1-year overall survival rates for the Y90 SIRT, SBRT, and PBT groups were 57.8, 83.1, and 78.7%, respectively. The rates of Barcelona Clinic Liver Cancer A, B, and C patients were 22.9, 32.2, and 42.5% in Y90 SIRT; 52.5, 20.2, and 25.1% in SBRT; and 36.3, 33.4, and 28.2% in PBT groups, respectively. The baseline intergroup differences were statistically significant (p < 0.0001). The median tumor sizes were 5.8, 3.1, and 4.5 cm in the Y90 SIRT, SBRT, and PBT groups, respectively. Conclusion In the management of HCC, there is substantial heterogeneity in studies investigating Y90 SIRT, SBRT, and PBT, with Y90 SIRT patients having worse Barcelona Clinic Liver Cancer and tumor burden at baseline, which likely contributes to the observed lower survival outcome.

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