BPSU study - Sydenham's chorea

The first ever prospective surveillance of Sydenham's chorea in the UK and Ireland commenced in November 2018 and will run for two years. This study is being undertaken in collaboration with the Exeter Medical School and the Child and Adolescent Psychiatry Surveillance System. The study aims to examine the service related incidence of SC, characterise clinical presentation and outcomes and report on current practice in investigation and management.

Lead investigator

Dr Tamsin Newlove-Delgado

Exeter Medical School
College House
St Luke's Campus
Exeter EX2 2LU

Email: t.newlove-delgado@exeter.ac.uk

About the study

Sydenham's chorea (SC) is a neurological disorder of childhood resulting from infection via Group A beta-haemolytic streptococcus (GABHS), the bacterium that causes rheumatic fever. SC is characterised by rapid, irregular, and aimless involuntary movements of the arms and legs, trunk and facial muscles.

Some children will have a sore throat several weeks before the symptoms begin, but the disorder can also strike up to 6 months after the fever or infection has cleared. Symptoms can appear gradually or all at once, and also may include uncoordinated movements, muscular weakness, stumbling and falling, slurred speech, difficulty concentrating and writing and emotional instability.

SC is a potentially severe illness with widespread impact which can affect functioning at home and at school. At the peak of their illness, children may become entirely dependent on their families. Reports suggest that the disorder is often not recognised promptly by professionals, perhaps due to a widespread belief that it is no longer seen in developed countries, but little is known about the current pattern of the disorder.

This study will help to fill this gap in scientific knowledge, and will be the first such study performed on this condition in the UK and ROI. It will raise awareness of the condition and report on current clinical practice in terms of how such cases are investigated and managed. Data on outcomes will also help clinicians give the most accurate information to families about what to expect.

You can download the protocol card, including references, below.

Case definition

According to the Jones criteria for acute rheumatic fever, Sydenham’s chorea is defined as “purposeless, involuntary, nonstereotypical movements of the trunk or extremities, often associated with muscle weakness and emotional lability”. SC is typically of acute or subacute onset, meaning that chorea reaches a peak within days or weeks rather than months. The Jones criteria include the differential diagnoses which must be excluded in order to confirm a diagnosis of Sydenham’s chorea – these are listed on the notification form.

Chorea is frequently a clinical diagnosis. It is important to note that laboratory confirmation of streptococcal infection provides supportive evidence of SC, but absence of such evidence does not preclude clinical confirmation.

Cases may be either:

  • suspected: cases presenting with chorea with acute or subacute onset, but where no diagnosis of SC has yet been made
  • confirmed clinically: cases where a new diagnosis of SC has been made, with chorea presenting with acute or subacute onset, and lack of clinical or laboratory evidence of an alternative cause as defined by the Jones criteria.

Reporting instructions

Please report children and young people aged 0-16 presenting for the first time to you during the reporting period with a first episode of suspected or confirmed SC (i.e. with no prior diagnosis of SC).

Duration

November 2018 to November 2020 (25 months of surveillance).

Follow up until November 2022 (a 12-month and 24-month follow-up).

How will the information be collected?

Medical doctors across the UK and ROI will fill in a questionnaire for any child up to and including 16 years of age who presents with suspected or confirmed SC.

The questionnaire will ask details about the case of SC and its child service related incidence, its presentation, management and outcomes. This information will be sent to the study team in Exeter who will look at the findings. The results will be then presented in medical journals and on various websites for parents and families.

How will they use the information?

By collecting patient information it is hoped that the investigators will increase our understanding about SC among children in the UK and ROI. BPSU researchers do not contact families or children who have been reported.

Royal Devon and Exeter NHS Foundation Trust (RD&ENFT) is the sponsor for this study based in the UK. They will use information from case medical records to undertake this study and will act as the data controller for this study. This means that they are responsible for looking after patient information and using it properly. Identifiable clinical information including age in months (taken from date of birth), sex and ethnicity are retained securely five years as a paper record and then electronically for 20 years, by RD&ENFT.

Rights to access, change or move one's patient information are limited, as the investigator needs to manage patient information in specific ways in order for the research to be reliable and accurate. Individuals can withdraw from the study, but any clinical information already obtained by the investigators will be analysed. To safeguard patient rights information will be anonymised and kept in confidence.

More information on how data is collected and held is available by contacting Dr Tamsin Newlove-Delgada, principal investigator at the contact details above.

Funding

The study is supported by the Paul Polani Award from the RCPCH and British Academy of Childhood Disability, and by the Sydenham’s Chorea Association.

Approval

This study has been approved by London Bloomsbury REC (REC reference: 18/LO/0760) and has been granted Section 251 HRA-CAG permission (CAG Reference: 18/CAG/0086). This study has been granted Public Benefit and Privacy Panel for Health and Social Care (PBPP) approval in Scotland (PBPP reference: 1819-0077).

Support group

Sydenham's Chorea Association