Surveillance of rare diseases: A public health evaluation of the British Paediatric Surveillance Unit

Journal of Public Health (United Kingdom)

Volumn 34, Issue 2, 2012, Pages 279-286

  Knowles, Rachel L ^a   Friend, Helen ^b   Lynn, Richard ^b   Mitchell, Simon ^c   Michie, Colin ^d   Ihekweazu, Chikwe ^e  

^a UNIVERSITY COLLEGE LONDON   (United Kingdom)

^b ROYAL COLLEGE OF PAEDIATRICS AND CHILD HEALTH   (United Kingdom)

^c ST MARY S HOSPITAL   (United Kingdom)

^d EALING HOSPITAL NHS TRUST   (United Kingdom)

^e PUBLIC HEALTH ENGLAND   (United Kingdom)

Author keywords

child health; public health evaluation; rare disease; surveillance

Indexed keywords

ARTICLE; FEASIBILITY STUDY; FEMALE; HEALTH CARE QUALITY; HEALTH SURVEY; HUMAN; INCIDENCE; INFECTION CONTROL; INTERNET; MALE; PEDIATRICS; PRESCHOOL CHILD; QUESTIONNAIRE; RARE DISEASE; STATISTICS; UNITED KINGDOM;

CHILD, PRESCHOOL; DISEASE NOTIFICATION; FEASIBILITY STUDIES; FEMALE; GREAT BRITAIN; HUMANS; INCIDENCE; INTERNET; MALE; PEDIATRICS; POPULATION SURVEILLANCE; PROGRAM EVALUATION; QUESTIONNAIRES; RARE DISEASES;

EID: 84856103201     PISSN: 17413842     EISSN: 17413850     Source Type: Journal    
DOI: 10.1093/pubmed/fdr058     Document Type: Article

References (40)

1. European Parliament and the Council. Decision no 1295/1999/EC: Adopting a programme of community action on rare diseases within the framework for action in the field of public health (1999 to 2003). Official Journal of the European Communities: European Parliament and the Council 1999:L155/1.
2. Donaldson L. On the state of the public health: Annual report of the Chief Medical Officer 2009. London: Department of Health, 2010.
3. Hall SM, Glickman M. The British paediatric surveillance unit. Arch Dis Child 1988;63(3):344-6.
4. Townsend CL, Willey BA, Cortina-Borja M et al. Antiretroviral therapy and congenital abnormalities in infants born to HIV-infected women in the UK and Ireland, 1990-2007. AIDS 2009;23(4):519-24.
5. Foot B, Stanford M, Rahi J et al. The British Ophthalmological Surveillance Unit: An evaluation of the first 3 years. Eye (Lond) 2003;17(1):9-15.
6. Elliott EJ, Nicoll A, Lynn R et al. Rare disease surveillance: An international perspective. Paediatr Child Health 2001;6(5):251-60.
7. German RR, Lee LM, Horan JM et al. Updated guidelines for evaluating public health surveillance systems: Recommendations from the Guidelines Working Group. MMWR Recomm Rep 2001;50(RR-13):1-35.
8. Gazarian M, Williams K, Elliott E et al. Evaluation of a national surveillance unit. Arch Dis Child 1999;80(1):21-7.
9. Morris A, Ridley GF, Elliott EJ. Australian Paediatric Surveillance Unit: Progress report. J Paediatr Child Health 2002;38(1):8-15.
10. He S, Zurynski YA, Elliott EJ. Evaluation of a national resource to identify and study rare diseases: The Australian Paediatric Surveillance Unit. J Paediatr Child Health 2009;45(9):498-504.
11. Grenier D, Doherty J, Macdonald D et al. Canadian Paediatric Surveillance Program evaluation: An excellent report card. Paediatr Child Health 2004;9(6):379-84.
12. Knowles RL, Smith A, Lynn R et al. Using multiple sources to improve and measure case ascertainment in surveillance studies: 20 years of the British Paediatric Surveillance Unit. J Public Health (Oxf ) 2006;28(2):157-65.
13. Godward S, Dezateux C. Validation of the reporting bases of the orthopaedic and paediatric surveillance schemes. Arch Dis Child 1996;75(3):232-6.
14. Lynn RM, Pebody R, Knowles R. Twenty years of active paediatric surveillance in the the UK and Republic of Ireland. Euro Surveill 2006;11(7):E060720 4.
15. Verity C, Nicoll A. Consent, confidentiality, and the threat to public health surveillance. BMJ 2002;324(7347):1210-3.
16. Verity C, Preece M. Surveillance for rare disorders by the BPSU. British Paediatric Surveillance Unit. Arch Dis Child 2002;87(4):269-71.
17. Nicoll A, Lynn R, Rahi J et al. Public health outputs from the British Paediatric Surveillance Unit and similar clinician-based systems. J R Soc Med 2000;93(11):580-5.
18. Lynn R, Ross E. The British Paediatric Surveillance Unit: The first 20 years. Arch Dis Child 2007;92(9):744-5.
19. Grenier D, Elliott EJ, Zurynski Y et al. Beyond counting cases: Public health impacts of national Paediatric Surveillance Units. Arch Dis Child 2007;92(6):527-33.
20. Grenier D, Lynn R, Zurynski Y., On behalf of all national paediatric surveillance unit investigators. Public health impacts of the International Network of Paediatric Surveillance Units. Paediatr Child Health 2009;14(8):499-500.
21. Tookey PA, Peckham CS. Surveillance of congenital rubella in Great Britain, 1971-96. BMJ 1999;318(7186):769-70.
22. Hurtig AK, Nicoll A, Carne C et al. Syphilis in pregnant women and their children in the United Kingdom: Results from national clinician reporting surveys 1994-7. BMJ 1998;317(7173): 1617-9.
23. Heath PT, Balfour G, Weisner AM et al. Group B streptococcal disease in UK and Irish infants younger than 90 days. Lancet 2004;363(9405):292-4.
24. Tookey P, Peckham CS. Neonatal herpes simplex virus infection in the British Isles. Paediatr Perinat Epidemiol 1996;10(4):432-42.
25. Leonard JV, Dezateux C. Newborn screening for medium chain acyl CoA dehydrogenase deficiency. Arch Dis Child 2009;94(3): 235-8.
26. McNinch AW, Tripp JH. Haemorrhagic disease of the newborn in the British Isles: Two year prospective study. BMJ 1991;303(6810):1105-9.
27. McNinch A, Busfield A, Tripp J. Vitamin K deficiency bleeding in Great Britain and Ireland: British Paediatric Surveillance Unit Surveys, 1993-94 and 2001-02. Arch Dis Child 2007;92(9): 759-66.
28. Hardie RM, Newton LH, Bruce JC et al. The changing clinical pattern of Reye's syndrome 1982-1990. Arch Dis Child 1996;74(5): 400-5.
29. McKiernan PJ, Baker AJ, Lloyd C et al. British paediatric surveillance unit study of biliary atresia: Outcome at 13 years. J Pediatr Gastroenterol Nutr 2009;48(1):78-81.
30. Knight M, Allen D, Murphy M et al. Fetomaternal alloimmune thrombocytopenia in the United Kingdom: A prospective national study of incidence, management and outcomes using obstetric, paediatric and laboratory reporting systems. Arch Dis Child Fetal Neonatal Ed 2008;93:(Suppl. 1)Fa38.
31. Rahi JS, Dezateux C. Capture-recapture analysis of ascertainment by active surveillance in the British Congenital Cataract Study. Invest Ophthalmol Vis Sci 1999;40(1):236-9.
32. Khalid JM, Oerton J, Cortina-Borja M et al. Ethnicity of children with homozygous c.985A.G medium-chain acyl-CoA dehydrogenase deficiency: Findings from screening approximately 1.1 million newborn infants. J Med Screen 2008;15(3):112-7.
33. Verity CM, Nicoll A, Will RG et al. Variant Creutzfeldt-Jakob disease in UK children: A national surveillance study. Lancet 2000;356(9237):1224-7.
34. Gilbert RE, Tookey PA. Perinatal mortality and morbidity among babies delivered in water: Surveillance study and postal survey. BMJ 1999;319(7208):483-7.
35. Milford DV, Taylor CM, Guttridge B et al. Haemolytic uraemic syndromes in the British Isles 1985-8: Association with verocytotoxin producing Escherichia coli. Part 1: Clinical and epidemiological aspects. Arch Dis Child 1990;65(7):716-21.
36. Verity C, Stellitano L, Winstone AM. Memorandum submitted to the Commons Select Committee on Science and Technology (SAGE 06). Scientific Advice and Evidence in Emergencies. London: House of Commons, 2011.
37. Knowles R, Smith A, Lynn R et al. What is the contribution of specialists to the ascertainment of rare childhood conditions through the British Paediatric Surveillance Unit? Arch Dis Childhood 2006;91(Suppl. 1):A87.
38. BPSU. An evaluation of the surveillance system of the British Paediatric Surveillance Unit 2008-09. http://www.bpsu.inopsu. com/about/ Evaluation%20Full%20Report.pdfon (24 May 2011, date last accessed).
39. He S, Zurynski YA, Elliott EJ. What do paediatricians think of the Australian Paediatric Surveillance Unit? J Paediatr Child Health 2010;46(7-8):412-8.
40. Lynn RM, Riding K, McIntosh N. The use of electronic reporting to aid surveillance of ADRs in children: A proof of concept study. Arch Dis Child 2010;95(4):262-5.