AJNR. American journal of neuroradiology · 2026
Observer agreement for a novel visual rating scale of vermian atrophy in childhood ataxia telangiectasia.
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Dineen Robert A, Pszczolkowski Stefan, Wright Jenny, Chiavacci Iacopo, Chattopadhyay Arpita, Cliffe Helen, Gagen Richard, Ilginiene Jurgita, Mitra Dipayan, Nash Laura, Patel Hiten, Pereira Nolette, Russell Ian, Thust Stefanie C, Stivaros Stavros M, Coman David, Avula Shivaram
Аннотация
BACKGROUND AND PURPOSE: Progressive cerebellar neurodegeneration occurs in the inherited disorder ataxia telangiectasia (A-T). Trials of disease-modifying therapies for A-T are in development, prompting the need for MRI metrics reflecting cerebellar neurodegeneration. Volumetric brain MRI analysis provides a gold standard, but implementation can be challenging when image acquisition or quality varies, or in low-resource settings. To address this we developed a 10-point scale for rating vermian atrophy (ROVA) in childhood A-T. In this study we report the observer agreement of ROVA and correlations with vermis and cerebellar volumes.
MATERIALS AND METHODS: Twelve attending-level paediatric neuroradiologists from 3 countries applied ROVA to 60 randomly-ordered midline sagittal T1-weighted images from children and young people (A-T, n=42; without A-T, n=18), blinded to age and disease status, and rated image quality using a 5-point ordinal scale. After 4-weeks observers blindly rated a second set of 30 images (A-T, n=20; without A-T, n=10) randomly selected and re-ordered from the initial set. Inter-observer agreement was calculated using Krippendorf's alpha. Intra-observer agreement was calculated using weighted kappa. Correlation between mode ROVA scores and fractional vermis and cerebellar volumes was calculated. Analyses were repeated after splitting the data into acceptable (n=45) and non-acceptable (n=15) image quality.
RESULTS: Inter-observer agreement (alpha = 0.86, 95%CI, 0.77, 0.91) and intra-observer agreement (median weighted kappa = 0.79, range 0.72, 0.89) exceeded the pre-specified values for satisfactory agreement. Intra-observer agreement was not related to years of specialist practice (Spearman's rho -0.11, p = .74). Inter-and intra-observer agreement increased after removing cases with non-acceptable image quality (alpha = 0.89, 95%CI 0.81, 0.92; median weighted kappa = 0.80, range 0.71, 0.90, respectively). Significant correlation was found between mode ROVA score and both fractional vermis volume (Spearman's rho = 0.82, 95%CI 0.71, 0.89, p<0.001) and fractional cerebellar volume (Spearman's rho = 0.80, 95%CI 0.69, 0.88, p<0.001).
CONCLUSIONS: The ROVA scale demonstrates high observer agreement and significant correlation with fractional vermis and cerebellar volumes when applied to a dataset of mixed image quality by paediatric neuroradiologists. This novel scale may be useful to provide a semi-quantitative measure of cerebellar degeneration in treatment trials for childhood A-T.
MATERIALS AND METHODS: Twelve attending-level paediatric neuroradiologists from 3 countries applied ROVA to 60 randomly-ordered midline sagittal T1-weighted images from children and young people (A-T, n=42; without A-T, n=18), blinded to age and disease status, and rated image quality using a 5-point ordinal scale. After 4-weeks observers blindly rated a second set of 30 images (A-T, n=20; without A-T, n=10) randomly selected and re-ordered from the initial set. Inter-observer agreement was calculated using Krippendorf's alpha. Intra-observer agreement was calculated using weighted kappa. Correlation between mode ROVA scores and fractional vermis and cerebellar volumes was calculated. Analyses were repeated after splitting the data into acceptable (n=45) and non-acceptable (n=15) image quality.
RESULTS: Inter-observer agreement (alpha = 0.86, 95%CI, 0.77, 0.91) and intra-observer agreement (median weighted kappa = 0.79, range 0.72, 0.89) exceeded the pre-specified values for satisfactory agreement. Intra-observer agreement was not related to years of specialist practice (Spearman's rho -0.11, p = .74). Inter-and intra-observer agreement increased after removing cases with non-acceptable image quality (alpha = 0.89, 95%CI 0.81, 0.92; median weighted kappa = 0.80, range 0.71, 0.90, respectively). Significant correlation was found between mode ROVA score and both fractional vermis volume (Spearman's rho = 0.82, 95%CI 0.71, 0.89, p<0.001) and fractional cerebellar volume (Spearman's rho = 0.80, 95%CI 0.69, 0.88, p<0.001).
CONCLUSIONS: The ROVA scale demonstrates high observer agreement and significant correlation with fractional vermis and cerebellar volumes when applied to a dataset of mixed image quality by paediatric neuroradiologists. This novel scale may be useful to provide a semi-quantitative measure of cerebellar degeneration in treatment trials for childhood A-T.