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Prognostic value of magnetic resonance imaging in pediatric-onset primary sclerosing cholangitis
Enni J Vanhanen1, Tiina E Lehtimäki2, Kaija-Leena Kolho3
1Department of Radiology, Helsinki University Hospital and University of Helsinki, P.O. Box 340, Helsinki 00029, Finland.
Insights
Repeatedly assessed ANALI scores, a magnetic resonance imaging (MRI) tool, predict adverse outcomes in pediatric-onset primary sclerosing cholangitis (PSC). Baseline ANALI scores alone showed limited prognostic value in children with PSC.
Area of Science:
- Hepatology
- Pediatric Gastroenterology
- Radiology
Background:
- Prognostic value of MRI-based ANALI scores in pediatric-onset primary sclerosing cholangitis (PSC) is understudied.
- ANALI scores (with and without gadolinium) have predicted disease progression in adults but not yet in children.
- This study evaluates the prognostic relevance of ANALI scores in pediatric PSC.
Purpose of the Study:
- To assess the prognostic relevance of ANALI scores in pediatric-onset PSC.
- To determine if MRI-based ANALI scores can predict disease progression and complications in children with PSC.
Main Methods:
- Retrospective cohort study of 34 pediatric-onset PSC patients from a national registry.
- Two expert radiologists calculated ANALI scores (ANALIGd and ANALINoGd) from baseline and follow-up MRIs.
- Cox proportional hazards regression analyzed the association between longitudinal ANALI scores and clinical endpoints (decompensated cirrhosis, liver transplantation).
Main Results:
- Baseline ANALI scores alone did not predict adverse outcomes in pediatric PSC.
- Higher follow-up ANALINoGd scores (HR 1.59) and ANALIGd scores (HR 3.03) were associated with adverse outcomes.
- Excellent interrater agreement was found for ANALINoGd (0.89) and substantial for ANALIGd (0.73).
Conclusions:
- ANALI scores are associated with adverse outcomes in pediatric-onset PSC when assessed repeatedly during follow-up.
- Baseline ANALI score measurements alone may have limited prognostic value in pediatric PSC patients.
- Longitudinal assessment of ANALI scores is crucial for predicting disease progression in pediatric PSC.
Background:
The prognostic value of magnetic resonance imaging (MRI) in pediatric-onset primary sclerosing cholangitis (PSC) remains poorly studied. The MRI-based ANALI scores with and without gadolinium (ANALIGd and ANALINoGd) have predicted disease progression and complications in adults, but these scores have not been studied in children.
Objectives:
To assess the prognostic relevance of ANALI scores in pediatric-onset PSC.
Design:
This retrospective cohort study was conducted at a single tertiary-care center and included patients with pediatric-onset PSC identified from a national PSC registry.
Methods:
We included 34 patients with pediatric-onset PSC (median age at diagnosis 14 years, interquartile range (IQR) 12.0-16.3; median follow-up time 10.4 years, IQR 7.0-13.2) who had undergone at least two MRIs. Two expert radiologists evaluated the presence of intrahepatic duct dilatation, hepatic dysmorphia, collateral veins, and parenchymal enhancement heterogeneity, which were used to calculate ANALI scores for the baseline and follow-up MRIs. The longitudinal development of ANALI scores was assessed, and the association between scores and clinical endpoints (decompensated cirrhosis, liver transplantation) was examined using Cox proportional hazards regression. Interrater agreement was evaluated for the total scores and the individual score items.
Results:
Disease progression was variable, and baseline ANALI scores alone did not predict adverse outcomes. When follow-up scores were also assessed, higher scores were associated with outcomes (hazard ratio (HR) per 1-point increase in ANALINoGd 1.59, 95% confidence interval (CI) 1.15-2.20; p = 0.005; HR per 1-point increase in ANALIGd 3.03, 95% CI 1.06-8.70, p = 0.039). Interrater agreement was excellent for ANALINoGd (0.89, 95% CI 0.83-0.95) and substantial for ANALIGd (0.73, 95% CI 0.62-0.84).
Conclusion:
ANALI scores were associated with adverse outcomes in pediatric-onset PSC when assessed repeatedly during follow-up; baseline measurements alone may have limited prognostic value in the pediatric population.
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