June 2023
Volume 64, Issue 8
Open Access
ARVO Annual Meeting Abstract  |   June 2023
Pilot Evaluation of a Genetic Education Booklet For Families Affected By Retinoblastoma
Author Affiliations & Notes
  • Helen Dimaras
    The Hospital for Sick Children, Toronto, Ontario, Canada
    University of Toronto, Toronto, Ontario, Canada
  • Taylor Irvine
    University of Toronto, Toronto, Ontario, Canada
  • Lucy Njambi
    University of Nairobi, Nairobi, Nairobi, Kenya
  • Festus Njuguna
    Moi Teaching and Referral Hospital, Eldoret, Uasin Gishu, Kenya
  • Kahaki Kimani
    University of Nairobi, Nairobi, Nairobi, Kenya
  • Footnotes
    Commercial Relationships   Helen Dimaras None; Taylor Irvine None; Lucy Njambi None; Festus Njuguna None; Kahaki Kimani None
  • Footnotes
    Support  None
Investigative Ophthalmology & Visual Science June 2023, Vol.64, 1271. doi:
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      Helen Dimaras, Taylor Irvine, Lucy Njambi, Festus Njuguna, Kahaki Kimani; Pilot Evaluation of a Genetic Education Booklet For Families Affected By Retinoblastoma. Invest. Ophthalmol. Vis. Sci. 2023;64(8):1271.

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      © ARVO (1962-2015); The Authors (2016-present)

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Abstract

Purpose : Retinoblastoma is a childhood eye cancer which affects approximately 90 new children per year in Kenya. Our prior research showed that parents and survivors of retinoblastoma in Kenya often hold misconceptions about the disease and, in particular, require more extensive and detailed information about the genetic nature of retinoblastoma. Subsequently, together with patient families, we co-developed a genetic education booklet and user guide about retinoblastoma and its genetic causes and consequences.
The aim of this study was to co-develop, with patient and clinical stakeholders, an evaluation protocol, and use it to pilot test the genetic education booklet in a real world setting.

Methods : Semi-structured interviews were conducted with ophthalmologists and eye ward nurses at Kenyatta National Hospital (KNH) and Moi Teaching and Referral Hospital (MTRH). Through convenience sampling, a focus group was conducted with 6 patient family members at KNH to obtain a patient family-specific perspective on development of an evaluation protocol. The stakeholder feedback was used to develop a draft evaluation protocol. A pilot of the evaluation was completed with 3 patient family members at KNH.

Results : The stakeholder engagement resulted in a mixed-methods evaluation protocol focused on knowledge change and utilization. In the quantitative arm of the protocol, pre- and post-test questionnaires assess baseline knowledge and changes immediately and 6-months after implementation of the booklet. In the qualitative arm, interviews with patient families aim to glean perceived change in knowledge and confidence about retinoblastoma genetics, and how they used or planned to use the information in their decision making. A pilot evaluation was performed with 3 participants. The average improvement in knowledge between pre- and post-test questionnaire was 10%. Participants still had difficulty grasping concepts about RB1 pathogenic variants. Perceived confidence and knowledge reportedly improved for 2/3 participants.

Conclusions : The evaluation protocol produced with stakeholder engagement was feasible to use revealed improvements in high level knowledge about retinoblastoma. However, specific knowledge about RB1 pathogenic variants may require improvements to the booklet. Next steps include revision of the booklet and scale-up of the evaluation.

This abstract was presented at the 2023 ARVO Annual Meeting, held in New Orleans, LA, April 23-27, 2023.

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