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

被引:2
|
作者
Neves, Renata [1 ,2 ]
Panek, Rafal [3 ,4 ]
Clarkson, Katie [1 ,5 ]
Panagioti, Ouliana [6 ]
Fernandez, Natasha Schneider [7 ]
Wilne, Sophie [8 ]
Suri, Mohnish [4 ,9 ]
Whitehouse, William P. [4 ,10 ]
Jagani, Sumit [11 ]
Dandapani, Madhumita [8 ,12 ]
Glazebrook, Cris [13 ]
Dineen, Robert A. [1 ,2 ,14 ,15 ]
机构
[1] Univ Nottingham, Sch Med, Radiol Sci Mental Hlth & Clin Neurosci, Nottingham, England
[2] Nottingham Univ Hosp NHS Trust, Dept Radiol, Nottingham, England
[3] Nottingham Univ Hosp NHS Trust, Med Phys & Clin Engn, Nottingham, England
[4] Univ Nottingham, Sch Med, Nottingham, England
[5] Loughborough Univ, Sch Sport Exercise & Hlth Sci, Loughborough, England
[6] NIHR Nottingham Clin Res Facil, Nottingham, England
[7] Univ Nottingham, Sch Med, Radiol Sci, Mental Heatlh & Clin Neurosci, Nottingham, England
[8] Nottingham Univ Hosp NHS Trust, Dept Paediat Oncol, Nottingham, England
[9] Nottingham Univ Hosp NHS Trust, Nottingham Clin Genet Serv, Nottingham, England
[10] Nottingham Univ Hosp NHS Trust, Paediat Neurol, Nottingham, England
[11] Nottingham Univ Hosp NHS Trust, Nottingham Childrens Hosp, Dept Radiol, Nottingham, England
[12] Univ Nottingham, Childrens Brain Tumour Res Ctr, Nottingham, England
[13] Univ Nottingham, Inst Mental Hlth, Nottingham, England
[14] NIHR Nottingham Biomed Res Ctr, Nottingham, England
[15] Univ Nottingham, Sir Peter Mansfield Imaging Ctr, Nottingham, England
来源
CANCER MEDICINE | 2024年 / 13卷 / 14期
关键词
ataxia-telangiectasia; cancer predisposition; cancer screening; mixed methods research; psychosocial impact; whole-body MRI; TP53 MUTATION CARRIERS; MISMATCH REPAIR DEFICIENCY; LI-FRAUMENI SYNDROME; SURVEILLANCE RECOMMENDATIONS; IMAGING SURVEILLANCE; INDIVIDUALS; PREDISPOSITION; MORBIDITY; EDUCATION; BENEFIT;
D O I
10.1002/cam4.70049
中图分类号
R73 [肿瘤学];
学科分类号
100214 ;
摘要
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.
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页数:16
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