Cause-Specific Mortality in Long-Term Survivors of Retinoblastoma

Yu, Chao; Tucker, Margaret A.; Abramson, David H.; Furukawa, Kyoji; Seddon, Johanna M.; Stovall, Marilyn; Fraumeni, Joseph F.; Kleinerman, Ruth A.

doi:10.1093/jnci/djp046

Cited by 132 publications

(106 citation statements)

References 29 publications

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“…Survivors of non-hereditary Rb are at much lower risk of a subsequent primary cancer, similar to the risk in the general population [8]. The risk for sarcomas in hereditary patients has been attributed to genetic susceptibility and past treatment with radiation [9].…”

Section: Resultsmentioning

confidence: 90%

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Ewing’s Sarcoma as Second Malignancy after Bilateral Retinoblastoma: A Case Report and Literature Review

2012

J Blood Lymph

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Background: Children diagnosed with the hereditary form of retinoblastoma (Rb) have excellent survival, but face an increased risk of bone and soft tissue sarcomas. This predisposition to sarcomas has been attributed to genetic susceptibility due to inactivation of the RB1 gene as well as past radiotherapy for Rb. Objectives:To report the case of a child with hereditary form of bilateral RB, who developed Ewing's sarcoma of the right femur 10 years after the Enucleation of the both eyes. Methods:The child was diagnosed as a case of bilateral RB at the age of 3 months. He was fully investigated and found to have locally advanced RB. Enucleation was done. The patient received chemotherapy and radiotherapy. At 14 years, he was investigated for a small swelling in his right lower leg. After extensive investigations, it was reported as Ewing's sarcoma. He is treated with chemotherapy.Results: This report confirms that patients suffering from RB are at an increased risk of developing Second Malignant Neoplasms (SMNs). Conclusions:This case confirms the increased risk of a SMN in children with hereditary RB. These children need a very close follow-up for the early diagnosis of SMNs.

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Section: Resultsmentioning

confidence: 90%

“…The majority of bone sarcomas occurred within the radiation field in the head region, but up to 40% was diagnosed outside the treatment field, primarily in the lower legs [9].…”

Section: Resultsmentioning

confidence: 99%

Ewing’s Sarcoma as Second Malignancy after Bilateral Retinoblastoma: A Case Report and Literature Review

2012

J Blood Lymph

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“…In our data example, further composite endpoints were disease-free survival and overall survival. Also overall survival can be regarded as composite, because one can distinguish between cancer-related mortality and noncancer mortality (27,28). If the composite time-to-event endpoint is all-encompassing in the sense that every patient will at some point in time experience it (although potentially after study closure), standard survival analyses using Kaplan-Meier curves and Cox' regression analyses for the all-events hazard are adequate.…”

Section: Discussionmentioning

confidence: 99%

Competing Risks and Multistate Models

Schmoor

Schumacher

Finke

et al. 2013

Clinical Cancer Research

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Complex clinical endpoints are present in studies in cancer. Especially in studies on hematopoietic stemcell transplantation (HSCT), various risks exist after HSCT. Patients can experience acute and chronic graft versus host disease (GVHD) or need to undergo immunosuppressive therapy (IST), a relapse can occur, or patients can die after relapse or without former relapse (nonrelapse mortality, NRM). Sometimes, endpoints can be reasonably combined in a composite endpoint, as, for example, relapse and NRM are combined into disease-free survival (DFS). In this case, standard survival techniques, as Kaplan-Meier estimation of the DFS probability, can be applied.Often, interest focuses on endpoints for which competing risks are present, as, for example, GVHD, with death without prior GVHD as competing risk. This results in a competing risks model, a special case of a multistate model. A more complex multistate model is required when the effects of events occurring in the course of the study on further disease process shall be investigated, as, for example, the effect of GVHD on relapse and NRM. Another endpoint of interest is time under IST. As patients usually experience multiple episodes of IST, thus switching back and forth between "IST" and "no IST" during follow-up, the multistate model used for analysis must be adapted for this event structure.The aim of this nontechnical report is to explain use and interpretation of Cox-type regression models suitable for the different situations in a randomized trial on the effects of anti-T-cell globulin as GVHD prophylaxis.

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“…The incidence of secondary malignancies in germinal retinoblastoma survivors has been the topic of study in many reports, (Abramson et al, 2001;Eng et al, 1993;Fletcher et al, 2004;Kleinerman et al, 2005;MacCarthy et al, 2009;Marees et al, 2008;Moll et al, 1997;Wong et al, 1997;& Yu et al, 2009) but the variance of sample size and study design in these reports has made interpretation of the cumulative incidence difficult. Using reported cumulative risk rates from sizeable studies with appreciable longterm follow-up, incidence rates are approximately 0.5% to 1% per year.…”

Section: Epidemiologymentioning

confidence: 99%

“…Historically, male retinoblastoma survivors were reported to have a higher incidence of third malignancies, primarily because females had an increased overall mortality rate from second malignancies (Abramson et al, 2001;Eng et al, 1993). More recent studies, however, have no longer identified an increase in female mortality from second malignancies (Marees et al, 2009;Yu et al, 2009). Abramson et al (Abramson et al, 2001) reported a predictable pattern for third, fourth, and fifth malignancy development based on location of the second tumor.…”

Section: Malignantmentioning

confidence: 99%