Feasibility of whole‐body MRI for cancer screening in children and young people with ataxia telangiectasia: A mixed methods cross‐sectional study

• Published in: Cancer medicine (Malden, MA) Vol. 13; no. 14; pp. e70049 - n/a
• Main Authors: Neves, Renata, Panek, Rafal, Clarkson, Katie, Panagioti, Ouliana, Fernandez, Natasha Schneider, Wilne, Sophie, Suri, Mohnish, Whitehouse, William P., Jagani, Sumit, Dandapani, Madhumita, Glazebrook, Cris, Dineen, Robert A.
• Format: Journal Article
• Language: English
• Published: United States John Wiley & Sons, Inc 01.07.2024; Wiley
• Subjects: Adolescent; Adult; Ataxia; Ataxia telangiectasia; Ataxia Telangiectasia - diagnostic imaging; Brain research; Cancer; cancer predisposition; Cancer screening; Child; Child, Preschool; Children; Children & youth; Cross-Sectional Studies; Data analysis; Early Detection of Cancer - methods; Early Detection of Cancer - psychology; Families & family life; Feasibility; Feasibility Studies; Female; Humans; Informed consent; Interviews; Ionizing radiation; Kinases; Magnetic resonance imaging; Magnetic Resonance Imaging - methods; Male; Medical diagnosis; Medical imaging; Medical research; Medical screening; mixed methods research; Mutation; Neoplasms - diagnostic imaging; Neoplasms - psychology; Neuroimaging; Parents & parenting; Pediatrics; Proteins; psychosocial impact; Whole Body Imaging - methods; whole‐body MRI; Young Adult; mixed methods research; ataxia‐telangiectasia; cancer predisposition; psychosocial impact; cancer screening; whole‐body MRI
• ISSN: 2045-7634; 2045-7634
• DOI: 10.1002/cam4.70049

Background/Objectives Ataxia telangiectasia (A‐T) is an inherited multisystem disorder with increased sensitivity to ionising radiation and elevated cancer risk. Although other cancer predisposition syndromes have established cancer screening protocols, evidence‐based guidelines for cancer screening in A‐T are lacking. This study sought to assess feasibility of a cancer screening protocol based on whole‐body MRI (WB‐MRI) in children and young people with A‐T. Design/Methods Children and young people with A‐T were invited to undergo a one‐off non‐sedated 3‐Tesla WB‐MRI. Completion rate of WB‐MRI was recorded and diagnostic image quality assessed by two experienced radiologists, with pre‐specified success thresholds for scan completion of >50% participants and image quality between acceptable to excellent in 65% participants. Positive imaging findings were classified according to the ONCO‐RADS system. Post‐participation interviews were performed with recruited families to assess the experience of participating and feelings about waiting for, and communication of, the findings of the scan. Results Forty‐six children and young people with A‐T were identified, of which 36 were eligible to participate, 18 were recruited and 16 underwent WB‐MRI. Nineteen parents participated in interviews. Fifteen participants (83%) completed the full WB‐MRI scan protocol. The pre‐specified image quality criterion was achieved with diagnostic images obtained in at least 93% of each MRI sequence. Non‐malignant scan findings were present in 4 (25%) participants. Six themes were identified from the interviews: (1) anxiety is a familiar feeling, (2) the process of MRI scanning is challenging for some children and families, (3) preparation is essential to reduce stress, (4) WB‐MRI provides the reassurance about the physical health that families need, (5) WB‐MRI experience turned out to be a positive experience and (6) WB‐MRI allows families to be proactive. Conclusion This study shows that WB‐MRI for cancer screening is feasible and well‐accepted by children and young people with A‐T and their families. Ataxia telangiectasia (A‐T) is an inherited multisystem disorder with increased sensitivity to ionising radiation and elevated cancer risk. Despite this, evidence‐based guidelines for cancer screening in A‐T are lacking. This study sought to assess the feasibility of a cancer screening protocol based on whole‐body MRI (WB‐MRI) in children and young people with A‐T. Our results suggest that WB‐MRI for cancer screening in A‐T is feasible and well‐accepted by children and young people with A‐T and their families.