Advertisement

Journal of Assisted Reproduction and Genetics

, Volume 36, Issue 9, pp 1787–1791 | Cite as

Conducting reproductive research during a new childhood cancer diagnosis: ethical considerations and impact on participants

  • Leena NahataEmail author
  • Taylor L. Morgan
  • Keagan G. Lipak
  • Olivia E. Clark
  • Nicholas D. Yeager
  • Sarah H. O’Brien
  • Stacy Whiteside
  • Anthony N. Audino
  • Cynthia A. Gerhardt
  • Gwendolyn P. Quinn
Fertility Preservation
  • 43 Downloads

Abstract

Purpose

Research among adults shows benefits and low perceived burden of engaging in behavioral research. However, questions remain regarding the ethics of conducting behavioral research in pediatric populations during sensitive situations, including during a new life-threatening diagnosis or at end-of-life. We examined reactions to participating in a behavioral reproductive research study among male adolescents newly diagnosed with cancer and their parents, as a step towards optimizing fertility preservation utilization in a population where future infertility is common.

Methods

Pediatric literature regarding the ethics of behavioral research was reviewed. In our pilot, forty-four participants (19 mothers, 11 fathers, 14 male adolescents newly diagnosed with cancer) from 20 families completed demographic questionnaires and a fertility preservation decision tool developed by the study team. Qualitative interviews exploring the impact of study participation were subsequently conducted. Verbatim transcripts were coded for thematic content using the constant comparison method.

Results

Literature review showed positive reactions to research participation among youth/caregivers. In our pilot study, 89% (n = 17) of mothers, 64% (n = 7) of fathers, and 71% (n = 10) of adolescents reported at least one benefit of participating. Eleven percent (n = 2) of mothers, 36% (n = 4) of fathers, and 29% (n = 4) of adolescents said they were not affected; none of the participants reported a negative effect.

Conclusion

Consistent with prior literature, our study suggests behavioral reproductive research prior to cancer treatment can offer direct benefits to participants and society, without increasing burden. These findings will inform future interventions to improve long-term psychosocial and reproductive outcomes for youth with cancer.

Keywords

Cancer Fertility Ethics Participant burden 

Notes

Acknowledgments

The authors would like to thank the families who participated in this study.

Funding information

This research was financially supported by the Clinical and Translational Intramural Funding Program at the Abigail Wexner Research Institute.

References

  1. 1.
    Ward E, DeSantis C, Robbins A, Kohler B, Jemal A. Childhood and adolescent cancer statistics, 2014. CA Cancer J Clin. 2014;64(2):83–103.CrossRefGoogle Scholar
  2. 2.
    Howlader N, Noone A, Krapcho M, Garshell J, Neyman N, Altekruse S, et al. SEER Cancer Statistics Review, 1975–2010. [Based on the November 2012 SEER data submission, posted to the SEER web site, April 2013.]. Bethesda: National Cancer Institute; 2013. p. 9.Google Scholar
  3. 3.
    Hudson MM, Ness KK, Gurney JG, Mulrooney DA, Chemaitilly W, Krull KR, et al. Clinical ascertainment of health outcomes among adults treated for childhood cancer. Jama. 2013;309(22):2371–81.  https://doi.org/10.1001/jama.2013.6296.CrossRefGoogle Scholar
  4. 4.
    Chemaitilly W, Cohen LE. Diagnosis of endocrine disease: endocrine late-effects of childhood cancer and its treatments. Eur J Endocrinol. 2017;176(4):R183–203.  https://doi.org/10.1530/EJE-17-0054.CrossRefGoogle Scholar
  5. 5.
    Green DM, Kawashima T, Stovall M, Leisenring W, Sklar CA, Mertens AC, et al. Fertility of male survivors of childhood cancer: a report from the Childhood Cancer Survivor Study. J Clin Oncol. 2010;28(2):332–9.CrossRefGoogle Scholar
  6. 6.
    Brignardello E, Felicetti F, Castiglione A, Chiabotto P, Corrias A, Fagioli F, et al. Endocrine health conditions in adult survivors of childhood cancer: the need for specialized adult-focused follow-up clinics. Eur J Endocrinol. 2013;168(3):465–72.CrossRefGoogle Scholar
  7. 7.
    Lehmann V, Nahata L, Ferrante AC, Hansen-Moore JA, Yeager ND, Klosky JL, Gerhardt CA. Fertility-related perceptions and impact on romantic relationships among adult survivors of childhood cancer. J Adolesc Young Adult Oncol. 2018;7(4):409–414.Google Scholar
  8. 8.
    Stein DM, Victorson DE, Choy JT, Waimey KE, Pearman TP, Smith K, et al. Fertility preservation preferences and perspectives among adult male survivors of pediatric cancer and their parents. J Adolesc Young Adult Oncol. 2014;3(2):75–82.  https://doi.org/10.1089/jayao.2014.0007.CrossRefGoogle Scholar
  9. 9.
    Nilsson J, Jervaeus A, Lampic C, Eriksson LE, Widmark C, Armuand GM, et al. ‘Will I be able to have a baby?’ Results from online focus group discussions with childhood cancer survivors in Sweden. Hum Reprod. 2014;29(12):2704–11.  https://doi.org/10.1093/humrep/deu280.CrossRefGoogle Scholar
  10. 10.
    Ellis SJ, Wakefield CE, McLoone JK, Robertson EG, Cohn RJ. Fertility concerns among child and adolescent cancer survivors and their parents: a qualitative analysis. J Psychosoc Oncol. 2016;34(5):347–62.  https://doi.org/10.1080/07347332.2016.1196806.CrossRefGoogle Scholar
  11. 11.
    Chong AL, Gupta A, Punnett A, Nathan PC. A cross Canada survey of sperm banking practices in pediatric oncology centers. Pediatr Blood Cancer. 2010;55(7):1356–61.  https://doi.org/10.1002/pbc.22705.CrossRefGoogle Scholar
  12. 12.
    Grover NS, Deal AM, Wood WA, Mersereau JE. Young men with cancer experience low referral rates for fertility counseling and sperm banking. J Oncol Pract. 2016;12(5):465–71.  https://doi.org/10.1200/JOP.2015.010579.CrossRefGoogle Scholar
  13. 13.
    Nahata L, Caltabellotta NM, Yeager ND, Lehmann V, Whiteside SL, O’Brien SH, et al. Fertility perspectives and priorities among male adolescents and young adults in cancer survivorship. Pediatr Blood Cancer. 2018;65(7):e27019.  https://doi.org/10.1002/pbc.27019.CrossRefGoogle Scholar
  14. 14.
    Vadaparampil ST, Clayton H, Quinn GP, King LM, Nieder M, Wilson C. Pediatric oncology nurses’ attitudes related to discussing fertility preservation with pediatric cancer patients and their families. J Pediatr Oncol Nurs. 2007;24(5):255–63.  https://doi.org/10.1177/1043454207303878.CrossRefGoogle Scholar
  15. 15.
    Vadaparampil S, Quinn G, King L, Wilson C, Nieder M. Barriers to fertility preservation among pediatric oncologists. Patient Educ Couns. 2008;72(3):402–10.  https://doi.org/10.1016/j.pec.2008.05.013.CrossRefGoogle Scholar
  16. 16.
    Cohen CB. Ethical issues regarding fertility preservation in adolescents and children. Pediatr Blood Cancer. 2009;53(2):249–53.  https://doi.org/10.1002/pbc.21996.CrossRefGoogle Scholar
  17. 17.
    Wiener L, Battles H, Zadeh S, Pao M. Assessing the experience of medically ill youth participating in psychological research: benefit, burden, or both? IRB. 2015;37(6):1.Google Scholar
  18. 18.
    Klosky JL, Simmons JL, Russell KM, Foster RH, Sabbatini GM, Canavera KE, et al. Fertility as a priority among at-risk adolescent males newly diagnosed with cancer and their parents. Support Care Cancer. 2015;23(2):333–41.  https://doi.org/10.1007/s00520-014-2366-1.CrossRefGoogle Scholar
  19. 19.
    Quinn GP, Knapp C, Murphy D, Sawczyn K, Sender L. Congruence of reproductive concerns among adolescents with cancer and parents: pilot testing an adapted instrument. Pediatrics. 2012;129(4):e930–6.  https://doi.org/10.1542/peds.2011-2568.CrossRefGoogle Scholar
  20. 20.
    Saito K, Suzuki K, Iwasaki A, Yumura Y, Kubota Y. Sperm cryopreservation before cancer chemotherapy helps in the emotional battle against cancer. Cancer. 2005;104(3):521–4.  https://doi.org/10.1002/cncr.21185.CrossRefGoogle Scholar
  21. 21.
    Hohmann C, Borgmann-Staudt A, Rendtorff R, Reinmuth S, Holzhausen S, Willich SN, et al. Patient counselling on the risk of infertility and its impact on childhood cancer survivors: results from a national survey. J Psychosoc Oncol. 2011;29(3):274–85.  https://doi.org/10.1080/07347332.2011.563344.CrossRefGoogle Scholar
  22. 22.
    Quinn GP, Murphy D, Knapp C, Stearsman DK, Bradley-Klug KL, Sawczyn K, et al. Who decides? Decision making and fertility preservation in teens with cancer: a review of the literature. J Adolesc Health. 2011;49(4):337–46.  https://doi.org/10.1016/j.jadohealth.2011.01.005.CrossRefGoogle Scholar
  23. 23.
    Hagenas I, Jorgensen N, Rechnitzer C, Sommer P, Holm M, Schmiegelow K, et al. Clinical and biochemical correlates of successful semen collection for cryopreservation from 12-18-year-old patients: a single-center study of 86 adolescents. Hum Reprod. 2010;25(8):2031–8.  https://doi.org/10.1093/humrep/deq147.CrossRefGoogle Scholar
  24. 24.
    Nahata L, Cohen LE, Lehmann LE, Yu RN. Semen analysis in adolescent cancer patients prior to bone marrow transplantation: when is it too late for fertility preservation? Pediatr Blood Cancer. 2013;60(1):129–32.  https://doi.org/10.1002/pbc.24172.CrossRefGoogle Scholar
  25. 25.
    Oktay K, Harvey BE, Partridge AH, Quinn GP, Reinecke J, Taylor HS et al. Fertility preservation in patients with cancer: ASCO Clinical Practice Guideline Update. J Clin Oncol Off J Am Soc Clin Oncol. 2018;JCO2018781914.  https://doi.org/10.1200/JCO.2018.78.1914.
  26. 26.
    Klosky JL, Wang F, Russell KM, Zhang H, Flynn JS, Huang L et al. Prevalence and predictors of sperm banking in adolescents newly diagnosed with cancer: examination of adolescent, parent, and provider factors influencing fertility preservation outcomes. J Clin Oncol Off J Am Soc Clin Oncol. 2017;JCO2016704767.  https://doi.org/10.1200/JCO.2016.70.4767.
  27. 27.
    Klosky JL, Flynn JS, Lehmann V, Russell KM, Wang F, Hardin RN, et al. Parental influences on sperm banking attempts among adolescent males newly diagnosed with cancer. Fertil Steril. 2017;108(6):1043–9.  https://doi.org/10.1016/j.fertnstert.2017.08.039.CrossRefGoogle Scholar
  28. 28.
    Morgan TL, Clark OE, Whiteside S, Audino A, Yeager ND, Klosky J, O’Brien SH, Gerhardt CA, Nahata L. Recruiting families and children newly diagnosed with cancer for behavioral research: important considerations and successful strategies. Psycho-Oncology. 2019.Google Scholar
  29. 29.
    Emanuel EJ, Fairclough DL, Wolfe P, Emanuel LL. Talking with terminally ill patients and their caregivers about death, dying, and bereavement: is it stressful? Is it helpful? Arch Intern Med. 2004;164(18):1999–2004.  https://doi.org/10.1001/archinte.164.18.1999.CrossRefGoogle Scholar
  30. 30.
    Pessin H, Galietta M, Nelson CJ, Brescia R, Rosenfeld B, Breitbart W. Burden and benefit of psychosocial research at the end of life. J Palliat Med. 2008;11(4):627–32.  https://doi.org/10.1089/jpm.2007.9923.CrossRefGoogle Scholar
  31. 31.
    Kassam-Adams N, Newman E. Child and parent reactions to participation in clinical research. Gen Hosp Psychiatry. 2005;27(1):29–35.  https://doi.org/10.1016/j.genhosppsych.2004.08.007.CrossRefGoogle Scholar
  32. 32.
    Wendler D, Abdoler E, Wiener L, Grady C. Views of adolescents and parents on pediatric research without the potential for clinical benefit. Pediatrics. 2012;130(4):692–9.  https://doi.org/10.1542/peds.2012-0068.CrossRefGoogle Scholar
  33. 33.
    Tager J, Battles H, Bedoya SZ, Gerhardt CA, Young-Saleme T, & Wiener, L. Participation in online research examining end-of-life experiences: Is it beneficial, burdensome, or both for caregivers bereaved by childhood cancer?. Submitted for publication. 2018.Google Scholar
  34. 34.
    Butler AE, Hall H, Copnell B. Bereaved parents’ experiences of research participation. BMC Palliative Care. 2018;17(1):122.CrossRefGoogle Scholar
  35. 35.
  36. 36.
    Klosky JL, Lehmann V, Flynn JS, Su Y, Zhang H, Russell KM, et al. Patient factors associated with sperm cryopreservation among at-risk adolescents newly diagnosed with cancer. Cancer. 2018;124(17):3567–75.  https://doi.org/10.1002/cncr.31596.CrossRefGoogle Scholar
  37. 37.
    Cohen J. Statistical power analysis for the behavioral sciences. 2nd ed. Hillsdale: Lawrence Earlbaum Associates; 1988.Google Scholar
  38. 38.
    Kodish E, Eder M, Noll RB, Ruccione K, Lange B, Angiolillo A, et al. Communication of randomization in childhood leukemia trials. Jama. 2004;291(4):470–5.CrossRefGoogle Scholar
  39. 39.
    Smith BM, Duncan FE, Ataman L, Smith K, Quinn GP, Chang RJ, et al. The National Physicians Cooperative: transforming fertility management in the cancer setting and beyond. Future Oncol. 2018;14:3059–72.  https://doi.org/10.2217/fon-2018-0278.CrossRefGoogle Scholar

Copyright information

© Springer Science+Business Media, LLC, part of Springer Nature 2019

Authors and Affiliations

  • Leena Nahata
    • 1
    • 2
    • 3
    Email author
  • Taylor L. Morgan
    • 1
  • Keagan G. Lipak
    • 1
  • Olivia E. Clark
    • 1
  • Nicholas D. Yeager
    • 3
    • 4
  • Sarah H. O’Brien
    • 3
    • 4
    • 5
  • Stacy Whiteside
    • 4
  • Anthony N. Audino
    • 3
    • 4
  • Cynthia A. Gerhardt
    • 1
    • 3
  • Gwendolyn P. Quinn
    • 6
  1. 1.Center for Biobehavioral HealthAbigail Wexner Research InstituteColumbusUSA
  2. 2.Division of EndocrinologyNationwide Children’s HospitalColumbusUSA
  3. 3.The Ohio State University College of MedicineColumbusUSA
  4. 4.Division of Hematology/OncologyNationwide Children’s HospitalColumbusUSA
  5. 5.Center for Innovation and Pediatric PracticeAbigail Wexner Research InstituteColumbusUSA
  6. 6.Department of Obstetrics and GynecologyNew York University School of MedicineNew YorkUSA

Personalised recommendations