Variant Creutzfeldt-Jakob disease in UK children after 27 years of active prospective surveillance

Arch Dis Child. 2025 May 16;110(6):450-454. doi: 10.1136/archdischild-2025-328472.

Abstract

Objective: To determine whether any children in the UK had variant Creutzfeldt-Jakob disease (vCJD).

Design: This active prospective epidemiological study used the British Paediatric Surveillance Unit, asking UK paediatricians to notify all childhood cases of progressive intellectual and neurological deterioration (PIND), a group that would include all cases of vCJD. Clinical data were obtained by questionnaire or via a site visit. An independent expert group classified the cases. If vCJD was suspected, referral to the National Creutzfeldt-Jakob Disease Research and Surveillance Unit was recommended.

Results: Between May 1997 and April 2024 (27 years), 5222 children were notified. There were four groups. (1) 2540 were 'not cases'-they did not meet the case definition or there were notification errors. (2) 2367 had a known underlying diagnosis other than vCJD; the group contained more than 220 different diseases. (3) 309 had no diagnosis to explain their deterioration; there was evidence that none of these cases had vCJD. (4) There were six cases of vCJD: two males and four females. They developed symptoms between 1998 and 2000, aged 12-15 years, and the last two died in 2003. Their clinical features were similar to those of adults. Four were classified as definite vCJD and two as probable vCJD.

Conclusions: This study has provided unique data about neurodegenerative diseases in UK children. There is no reliable vCJD screening test; so for 27 years, the PIND study has provided reassurance that childhood vCJD cases were not missed. New vCJD cases with the methionine/valine genotype could appear.

Keywords: Epidemiology; Genetics; Neurology; Paediatrics.

MeSH terms

  • Adolescent
  • Child
  • Child, Preschool
  • Creutzfeldt-Jakob Syndrome* / diagnosis
  • Creutzfeldt-Jakob Syndrome* / epidemiology
  • Disease Notification
  • Female
  • Humans
  • Male
  • Population Surveillance / methods
  • Prospective Studies
  • United Kingdom / epidemiology