Re: Metastatic deaths in retinoblastoma patients treated with intraarterial chemotherapy (ophthalmic artery chemosurgery) worldwide

Re: Metastatic deaths in retinoblastoma patients treated with intraarterial chemotherapy... Soliman et al. Int J Retin Vitr (2018) 4:19 International Journal https://doi.org/10.1186/s40942-018-0124-0 of Retina and Vitreous Open Access LE T TER TO THE EDITOR Re: Metastatic deaths in retinoblastoma patients treated with intraarterial chemotherapy (ophthalmic artery chemosurgery) worldwide 1,2* 1 1 3 Sameh E. Soliman , Helen Dimaras , Brenda Gallie and Furqan Shaikh Dear Editor, beam radiation therapy. In developing countries where stem cell transplant is not available, metastatic retino- We read with interest the recent Commentary by blastoma is largely incurable [3]. The Commentary does Abramson and colleagues, entitled “Metastatic deaths in not mention the rate of metastases in 1139 patients and retinoblastoma patients treated with intraarterial chemo- focuses on the single outcome of metastatic deaths. therapy (ophthalmic artery chemosurgery) worldwide” Death is, of course, the most important outcome in any [1]. The authors retrospectively collected data on all trial, but is not the only one of importance. patients treated with intra-arterial chemotherapy (IAC) Both metastases and deaths occur after some time from between May 2006 and November 2016 from six retino- therapy, and under-detection of delayed events affects the blastoma centers, in order to determine the rate of meta- validity of cancer studies. This is especially true for ret - static death in patients who received IAC. The six centers rospective studies from tertiary or elite referral centers, collectively treated 1177 eyes of 1139 patients. Three which often receive international or distant patients for patients died from metastatic retinoblastoma, all from a treatment, who then return home and are not longitudi- single center in Argentina, and all associated with refusal nally followed and might have died unreported [4]. Sta- of enucleation or poor follow-up. The authors concluded tistical methods, such as Kaplan–Meier analysis where that the rate of metastatic death after IAC conducted at patients are censored at date last seen can adjust for this centers with expertise is low, at <  1%. The authors are differential follow-up effect. Was the follow-up duration to be congratulated for their efforts in assembling such and quality sufficient to detect most events? How do the a large pooled database of patients with a rare disease, centers retrieve data from lost follow-up patients? The and for determining this reassuring result. However, Commentary does not provide any time-to-event analy- we would like to point out some shortcomings of their sis, nor even a simple median follow-up time. publication. The duration of follow-up is crucial and longer follow- An important goal of treating retinoblastoma is to pre- up often reveals additional events. The Commentary vent tumor spread or metastasis which is a concern with itself provides a noteworthy example of this very point. It IAC as a treatment for retinoblastoma as it provide good states that “a year ago our centers in New York, Philadel- local control without systemic control [2]. Treatment of phia, Argentina and Switzerland reported on 634 cases metastases is an immense burden with the morbidity and with only one metastatic death.” [5] In the Commen- lifelong consequences of intensive chemotherapy, autol- tary, they report three metastatic deaths from the center ogous stem cell transplantation, and possibly external in Argentina, suggesting that two additional deaths occurred in 18 months. The IAC literature has been reported across several *Correspondence: samehelsayedsoliman@yahoo.com scores of publications, each with a different subset of The Department of Ophthalmology and Vision Sciences, The Hospital patients, different measurements, and different outcomes for Sick Children, 555 University Avenue, Room 7265, Toronto, ON M5G [5–17]. This multiplicative piecemeal reporting creates 1X8, Canada Full list of author information is available at the end of the article © The Author(s) 2018. This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creat iveco mmons .org/licen ses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creat iveco mmons .org/ publi cdoma in/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Soliman et al. Int J Retin Vitr (2018) 4:19 Page 2 of 2 2. Yousef YA, Soliman SE, Astudillo PP, Durairaj P, Dimaras H, Chan HS, et al. shortcomings in the scientific evidence and inconsist - Intra-arterial chemotherapy for retinoblastoma: a systematic review. ency of numbers presented. A representative example is JAMA Ophthalmol. 2016;134(6):584–91. in the Commentary. The authors state that their six cent - 3. Dunkel IJ, Chan HS, Jubran R, Chantada GL, Goldman S, Chintagumpala M, et al. High-dose chemotherapy with autologous hematopoietic ers treated 1177 eyes of 1139 patients, representing all stem cell rescue for stage 4B retinoblastoma. Pediatr Blood Cancer. patients treated with IAC in the included time period. 2010;55(1):149–52. By mathematical necessity, this would mean that no 4. White A, Gallie BL. Life before eye: implications for the whole child and family of attempted eye salvage for unilateral and severe bilateral retino- more 38 patients were treated bilaterally. However, the blastoma. 46th World Congress of the International Society of Paediatric authors from one center have separately published [16] Oncology. 2014. the results of 60 patients treated with IAC bilaterally in 5. Abramson DH, Shields CL, Munier FL, Chantada GL. Treatment of ret- inoblastoma in 2015: agreement and disagreement. JAMA Ophthalmol. 120 eyes from within the same period. 2015;133(11):1341–7. We close by stating that these points should not be 6. Abramson DH, Dunkel IJ, Brodie SE, Kim JW, Gobin YP. A phase I/II study seen as controversial, provocative, or adversarial. There is of direct intraarterial (ophthalmic artery) chemotherapy with mel- phalan for intraocular retinoblastoma initial results. Ophthalmology. really no controversy in stating that studies should have 2008;115(8):1398–404. important outcomes, adequate follow-up, accurate detec- 7. Abramson DH, Dunkel IJ, Brodie SE, Marr B, Gobin YP. Bilateral superse- tion, and consistent information. These are bedrock prin - lective ophthalmic artery chemotherapy for bilateral retinoblastoma: tandem therapy. Arch Ophthalmol. 2010;128(3):370–2. ciples and required standards of all clinical studies. 8. Abramson DH, Dunkel IJ, Brodie SE, Marr B, Gobin YP. Superselective oph- thalmic artery chemotherapy as primary treatment for retinoblastoma Authors’ contributions (chemosurgery). Ophthalmology. 2010;117(8):1623–9. Concept: SS, HD, BG, FS; Design: SS, HD, BG; FS, Revision: SS, HD, BG, FS. All 9. Gobin YP, Dunkel IJ, Marr BP, Brodie SE, Abramson DH. Intra-arterial authors read and approved the final manuscript. chemotherapy for the management of retinoblastoma: four-year experi- ence. Arch Ophthalmol. 2011;129(6):732–7. Author details 10. Abramson DH, Marr BP, Brodie SE, Dunkel I, Palioura S, Gobin YP. Ophthal- The Department of Ophthalmology and Vision Sciences, The Hospital for Sick mic artery chemosurgery for less advanced intraocular retinoblastoma: Children, 555 University Avenue, Room 7265, Toronto, ON M5G 1X8, Canada. five year review. PLoS ONE. 2012;7(4):e34120. Ophthalmology Department, Faculty of Medicine, University of Alexandria, 11. Abramson DH, Marr BP, Dunkel IJ, Brodie S, Zabor EC, Driscoll SJ, et al. Alexandria, Egypt. The Division of Hematology/Oncology, Department Intra-arterial chemotherapy for retinoblastoma in eyes with vitreous and/ of Pediatrics, The Hospital for Sick Children, Toronto, ON, Canada. or subretinal seeding: 2-year results. Br J Ophthalmol. 2012;96(4):499–502. 12. Marr BP, Brodie SE, Dunkel IJ, Gobin YP, Abramson DH. Three-drug Competing interests intra-arterial chemotherapy using simultaneous carboplatin, topotecan The authors declare that they have no competing interests. and melphalan for intraocular retinoblastoma: preliminary results. Br J Ophthalmol. 2012;96(10):1300–3. Availability of data and materials 13. Francis JH, Gobin YP, Dunkel IJ, Marr BP, Brodie SE, Jonna G, et al. Carbo- Not applicable. platin +/− topotecan ophthalmic artery chemosurgery for intraocular retinoblastoma. PLoS ONE. 2013;8(8):e72441. Consent for publication 14. Abramson DH, Fabius AW, Issa R, Francis JH, Marr BP, Dunkel IJ, et al. Not applicable. Advanced unilateral retinoblastoma: the impact of ophthalmic artery chemosurgery on enucleation rate and patient survival at MSKCC. PLoS Ethics approval and consent to participate ONE. 2015;10(12):e0145436. Not applicable. 15. Francis JH, Abramson DH, Gobin YP, Marr BP, Tendler I, Brodie SE, et al. Efficacy and toxicity of second-course ophthalmic artery chemosurgery Funding for retinoblastoma. Ophthalmology. 2015;122(5):1016–22. Not applicable. 16. Abramson DH, Marr BP, Francis JH, Dunkel IJ, Fabius AW, Brodie SE, et al. Simultaneous bilateral ophthalmic artery chemosurgery for bilateral Publisher’s Note retinoblastoma ( Tandem Therapy). PLoS ONE. 2016;11(6):e0156806. Springer Nature remains neutral with regard to jurisdictional claims in pub- 17. Abramson DH, Daniels AB, Marr BP, Francis JH, Brodie SE, Dunkel IJ, et al. lished maps and institutional affiliations. Intra-arterial chemotherapy (ophthalmic artery chemosurgery) for group D retinoblastoma. PLoS ONE. 2016;11(1):e0146582. Received: 14 January 2018 Accepted: 21 May 2018 Ready to submit your research ? Choose BMC and benefit from: References fast, convenient online submission 1. Abramson DH, Shields CL, Jabbour P, Teixeira LF, Fonseca JRF, Marques thorough peer review by experienced researchers in your field MCP, et al. Metastatic deaths in retinoblastoma patients treated with intraarterial chemotherapy (ophthalmic artery chemosurgery) worldwide. rapid publication on acceptance Int J Retina Vitreous. 2017;3:40. support for research data, including large and complex data types • gold Open Access which fosters wider collaboration and increased citations maximum visibility for your research: over 100M website views per year At BMC, research is always in progress. Learn more biomedcentral.com/submissions http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png International Journal of Retina and Vitreous Springer Journals

Re: Metastatic deaths in retinoblastoma patients treated with intraarterial chemotherapy (ophthalmic artery chemosurgery) worldwide

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Medicine & Public Health; Ophthalmology
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Abstract

Soliman et al. Int J Retin Vitr (2018) 4:19 International Journal https://doi.org/10.1186/s40942-018-0124-0 of Retina and Vitreous Open Access LE T TER TO THE EDITOR Re: Metastatic deaths in retinoblastoma patients treated with intraarterial chemotherapy (ophthalmic artery chemosurgery) worldwide 1,2* 1 1 3 Sameh E. Soliman , Helen Dimaras , Brenda Gallie and Furqan Shaikh Dear Editor, beam radiation therapy. In developing countries where stem cell transplant is not available, metastatic retino- We read with interest the recent Commentary by blastoma is largely incurable [3]. The Commentary does Abramson and colleagues, entitled “Metastatic deaths in not mention the rate of metastases in 1139 patients and retinoblastoma patients treated with intraarterial chemo- focuses on the single outcome of metastatic deaths. therapy (ophthalmic artery chemosurgery) worldwide” Death is, of course, the most important outcome in any [1]. The authors retrospectively collected data on all trial, but is not the only one of importance. patients treated with intra-arterial chemotherapy (IAC) Both metastases and deaths occur after some time from between May 2006 and November 2016 from six retino- therapy, and under-detection of delayed events affects the blastoma centers, in order to determine the rate of meta- validity of cancer studies. This is especially true for ret - static death in patients who received IAC. The six centers rospective studies from tertiary or elite referral centers, collectively treated 1177 eyes of 1139 patients. Three which often receive international or distant patients for patients died from metastatic retinoblastoma, all from a treatment, who then return home and are not longitudi- single center in Argentina, and all associated with refusal nally followed and might have died unreported [4]. Sta- of enucleation or poor follow-up. The authors concluded tistical methods, such as Kaplan–Meier analysis where that the rate of metastatic death after IAC conducted at patients are censored at date last seen can adjust for this centers with expertise is low, at <  1%. The authors are differential follow-up effect. Was the follow-up duration to be congratulated for their efforts in assembling such and quality sufficient to detect most events? How do the a large pooled database of patients with a rare disease, centers retrieve data from lost follow-up patients? The and for determining this reassuring result. However, Commentary does not provide any time-to-event analy- we would like to point out some shortcomings of their sis, nor even a simple median follow-up time. publication. The duration of follow-up is crucial and longer follow- An important goal of treating retinoblastoma is to pre- up often reveals additional events. The Commentary vent tumor spread or metastasis which is a concern with itself provides a noteworthy example of this very point. It IAC as a treatment for retinoblastoma as it provide good states that “a year ago our centers in New York, Philadel- local control without systemic control [2]. Treatment of phia, Argentina and Switzerland reported on 634 cases metastases is an immense burden with the morbidity and with only one metastatic death.” [5] In the Commen- lifelong consequences of intensive chemotherapy, autol- tary, they report three metastatic deaths from the center ogous stem cell transplantation, and possibly external in Argentina, suggesting that two additional deaths occurred in 18 months. The IAC literature has been reported across several *Correspondence: samehelsayedsoliman@yahoo.com scores of publications, each with a different subset of The Department of Ophthalmology and Vision Sciences, The Hospital patients, different measurements, and different outcomes for Sick Children, 555 University Avenue, Room 7265, Toronto, ON M5G [5–17]. This multiplicative piecemeal reporting creates 1X8, Canada Full list of author information is available at the end of the article © The Author(s) 2018. This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creat iveco mmons .org/licen ses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creat iveco mmons .org/ publi cdoma in/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Soliman et al. Int J Retin Vitr (2018) 4:19 Page 2 of 2 2. Yousef YA, Soliman SE, Astudillo PP, Durairaj P, Dimaras H, Chan HS, et al. shortcomings in the scientific evidence and inconsist - Intra-arterial chemotherapy for retinoblastoma: a systematic review. ency of numbers presented. A representative example is JAMA Ophthalmol. 2016;134(6):584–91. in the Commentary. The authors state that their six cent - 3. Dunkel IJ, Chan HS, Jubran R, Chantada GL, Goldman S, Chintagumpala M, et al. High-dose chemotherapy with autologous hematopoietic ers treated 1177 eyes of 1139 patients, representing all stem cell rescue for stage 4B retinoblastoma. Pediatr Blood Cancer. patients treated with IAC in the included time period. 2010;55(1):149–52. By mathematical necessity, this would mean that no 4. White A, Gallie BL. Life before eye: implications for the whole child and family of attempted eye salvage for unilateral and severe bilateral retino- more 38 patients were treated bilaterally. However, the blastoma. 46th World Congress of the International Society of Paediatric authors from one center have separately published [16] Oncology. 2014. the results of 60 patients treated with IAC bilaterally in 5. Abramson DH, Shields CL, Munier FL, Chantada GL. Treatment of ret- inoblastoma in 2015: agreement and disagreement. JAMA Ophthalmol. 120 eyes from within the same period. 2015;133(11):1341–7. We close by stating that these points should not be 6. Abramson DH, Dunkel IJ, Brodie SE, Kim JW, Gobin YP. A phase I/II study seen as controversial, provocative, or adversarial. There is of direct intraarterial (ophthalmic artery) chemotherapy with mel- phalan for intraocular retinoblastoma initial results. Ophthalmology. really no controversy in stating that studies should have 2008;115(8):1398–404. important outcomes, adequate follow-up, accurate detec- 7. Abramson DH, Dunkel IJ, Brodie SE, Marr B, Gobin YP. Bilateral superse- tion, and consistent information. These are bedrock prin - lective ophthalmic artery chemotherapy for bilateral retinoblastoma: tandem therapy. Arch Ophthalmol. 2010;128(3):370–2. ciples and required standards of all clinical studies. 8. Abramson DH, Dunkel IJ, Brodie SE, Marr B, Gobin YP. Superselective oph- thalmic artery chemotherapy as primary treatment for retinoblastoma Authors’ contributions (chemosurgery). Ophthalmology. 2010;117(8):1623–9. Concept: SS, HD, BG, FS; Design: SS, HD, BG; FS, Revision: SS, HD, BG, FS. All 9. Gobin YP, Dunkel IJ, Marr BP, Brodie SE, Abramson DH. Intra-arterial authors read and approved the final manuscript. chemotherapy for the management of retinoblastoma: four-year experi- ence. Arch Ophthalmol. 2011;129(6):732–7. Author details 10. Abramson DH, Marr BP, Brodie SE, Dunkel I, Palioura S, Gobin YP. Ophthal- The Department of Ophthalmology and Vision Sciences, The Hospital for Sick mic artery chemosurgery for less advanced intraocular retinoblastoma: Children, 555 University Avenue, Room 7265, Toronto, ON M5G 1X8, Canada. five year review. PLoS ONE. 2012;7(4):e34120. Ophthalmology Department, Faculty of Medicine, University of Alexandria, 11. Abramson DH, Marr BP, Dunkel IJ, Brodie S, Zabor EC, Driscoll SJ, et al. Alexandria, Egypt. The Division of Hematology/Oncology, Department Intra-arterial chemotherapy for retinoblastoma in eyes with vitreous and/ of Pediatrics, The Hospital for Sick Children, Toronto, ON, Canada. or subretinal seeding: 2-year results. Br J Ophthalmol. 2012;96(4):499–502. 12. Marr BP, Brodie SE, Dunkel IJ, Gobin YP, Abramson DH. Three-drug Competing interests intra-arterial chemotherapy using simultaneous carboplatin, topotecan The authors declare that they have no competing interests. and melphalan for intraocular retinoblastoma: preliminary results. Br J Ophthalmol. 2012;96(10):1300–3. Availability of data and materials 13. Francis JH, Gobin YP, Dunkel IJ, Marr BP, Brodie SE, Jonna G, et al. Carbo- Not applicable. platin +/− topotecan ophthalmic artery chemosurgery for intraocular retinoblastoma. PLoS ONE. 2013;8(8):e72441. Consent for publication 14. Abramson DH, Fabius AW, Issa R, Francis JH, Marr BP, Dunkel IJ, et al. Not applicable. Advanced unilateral retinoblastoma: the impact of ophthalmic artery chemosurgery on enucleation rate and patient survival at MSKCC. PLoS Ethics approval and consent to participate ONE. 2015;10(12):e0145436. Not applicable. 15. Francis JH, Abramson DH, Gobin YP, Marr BP, Tendler I, Brodie SE, et al. Efficacy and toxicity of second-course ophthalmic artery chemosurgery Funding for retinoblastoma. Ophthalmology. 2015;122(5):1016–22. Not applicable. 16. Abramson DH, Marr BP, Francis JH, Dunkel IJ, Fabius AW, Brodie SE, et al. Simultaneous bilateral ophthalmic artery chemosurgery for bilateral Publisher’s Note retinoblastoma ( Tandem Therapy). PLoS ONE. 2016;11(6):e0156806. Springer Nature remains neutral with regard to jurisdictional claims in pub- 17. Abramson DH, Daniels AB, Marr BP, Francis JH, Brodie SE, Dunkel IJ, et al. lished maps and institutional affiliations. Intra-arterial chemotherapy (ophthalmic artery chemosurgery) for group D retinoblastoma. PLoS ONE. 2016;11(1):e0146582. Received: 14 January 2018 Accepted: 21 May 2018 Ready to submit your research ? Choose BMC and benefit from: References fast, convenient online submission 1. Abramson DH, Shields CL, Jabbour P, Teixeira LF, Fonseca JRF, Marques thorough peer review by experienced researchers in your field MCP, et al. Metastatic deaths in retinoblastoma patients treated with intraarterial chemotherapy (ophthalmic artery chemosurgery) worldwide. rapid publication on acceptance Int J Retina Vitreous. 2017;3:40. support for research data, including large and complex data types • gold Open Access which fosters wider collaboration and increased citations maximum visibility for your research: over 100M website views per year At BMC, research is always in progress. Learn more biomedcentral.com/submissions

Journal

International Journal of Retina and VitreousSpringer Journals

Published: May 29, 2018

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