First estimates of the potential cost and cost saving of protecting childhood hearing from damage caused by congenital CMV infection

Background Congenital cytomegalovirus (cCMV) is an important cause of childhood deafness, which is modifiable if diagnosed within the first month of life. Targeted screening of infants who do not pass their newborn hearing screening tests in England is a feasible approach to identify and treat cases to improve hearing outcome. Aims To conduct a cost analysis of targeted screening and subsequent treatment for cCMV-related sensorineural hearing loss (SNHL) in an, otherwise, asymptomatic infant, from the perspective of the UK National Health Service (NHS). Methods Using data from the newborn hearing screening programme (NHSP) in England and a recent study of targeted screening for cCMV using salivary swabs within the NHSP, we estimate the cost (in UK pounds (£)) to the NHS. The cost of screening (time, swabs and PCR), assessing, treating and following up cases is calculated. The cost per case of preventing hearing deterioration secondary to cCMV with targeted screening is calculated. Results The cost of identifying, assessing and treating a case of cCMV-related SNHL through targeted cCMV screening is estimated to be £6683. The cost of improving hearing outcome for an infant with cCMV-related SNHL through targeted screening and treatment is estimated at £14 202. Conclusions The costs of targeted screening for cCMV using salivary swabs integrated within NHSP resulted in an estimate of cost per case that compares favourably with other screening programmes. This could be used in future studies to estimate the full economic value in terms of incremental costs and incremental health benefits.

[1]  Karl Claxton,et al.  Unrelated Future Costs and Unrelated Future Benefits: Reflections on NICE Guide to the Methods of Technology Appraisal. , 2016, Health economics.

[2]  R. DeBiasi,et al.  Valganciclovir for symptomatic congenital cytomegalovirus disease. , 2015, The New England journal of medicine.

[3]  P. Griffiths,et al.  Feasibility and acceptability of targeted screening for congenital CMV-related hearing loss , 2014, Archives of Disease in Childhood: Fetal and Neonatal Edition.

[4]  F. Dekker,et al.  The apparent paradox of maternal seropositivity as a risk factor for congenital cytomegalovirus infection: a population‐based prediction model , 2013, Reviews in medical virology.

[5]  M. Cannon,et al.  Efficient Linking of Birth Certificate and Newborn Screening Databases for Laboratory Investigation of Congenital Cytomegalovirus Infection and Preterm Birth: Florida, 2008 , 2012, Maternal and Child Health Journal.

[6]  M. Sharland,et al.  Evidence based management guidelines for the detection and treatment of congenital CMV. , 2011, Early human development.

[7]  D. Bernstein,et al.  Saliva polymerase-chain-reaction assay for cytomegalovirus screening in newborns. , 2011, The New England journal of medicine.

[8]  C. Peckham,et al.  Surveillance of congenital cytomegalovirus in the UK and Ireland , 2011, Archives of Disease in Childhood: Fetal and Neonatal Edition.

[9]  S. Soong,et al.  Neurodevelopmental outcomes following ganciclovir therapy in symptomatic congenital cytomegalovirus infections involving the central nervous system. , 2009, Journal of clinical virology : the official publication of the Pan American Society for Clinical Virology.

[10]  M. Schleiss,et al.  Detection of Cytomegalovirus DNA in Dried Blood Spots of Minnesota Infants Who Do Not Pass Newborn Hearing Screening , 2009, The Pediatric infectious disease journal.

[11]  A. Poblano,et al.  Cognitive performance of school children with unilateral sensorineural hearing loss. , 2009, Archives of medical research.

[12]  P. Sánchez,et al.  Newborn Hearing Screening and Detection of Congenital Cytomegalovirus Infection , 2008, Pediatrics.

[13]  S. Grosse,et al.  New estimates of the prevalence of neurological and sensory sequelae and mortality associated with congenital cytomegalovirus infection , 2007, Reviews in medical virology.

[14]  J. Smedley National Institute of Health and Clinical Excellence www.nice.org.uk , 2006 .

[15]  L. J. Marin,et al.  Is saliva as reliable as urine for detection of cytomegalovirus DNA for neonatal screening of congenital CMV infection? , 2006, Journal of clinical virology : the official publication of the Pan American Society for Clinical Virology.

[16]  S. Petrou,et al.  The Economic Costs of Congenital Bilateral Permanent Childhood Hearing Impairment , 2006, Pediatrics.

[17]  J. Bamford,et al.  An analysis of the costs of implementing the National Newborn Hearing Screening Programme in England , 2006, Journal of medical screening.

[18]  J. Niparko,et al.  Effects of Cytomegalovirus (CMV) Related Deafness on Pediatric Cochlear Implant Outcomes , 2005, Otolaryngology--head and neck surgery : official journal of American Academy of Otolaryngology-Head and Neck Surgery.

[19]  R. Pass Congenital cytomegalovirus infection and hearing loss. , 2005, Herpes : the journal of the IHMF.

[20]  E. Endreffy,et al.  Incidence and outcome of congenital cytomegalovirus infection in selected groups of preterm and full-term neonates under intensive care. , 2004, In vivo.

[21]  T. Nikolopoulos,et al.  Cochlear Implantation in Children Deafened by Cytomegalovirus: Speech Perception and Speech Intelligibility Outcomes , 2004, Otology & neurotology : official publication of the American Otological Society, American Neurotology Society [and] European Academy of Otology and Neurotology.

[22]  S. Soong,et al.  Effect of ganciclovir therapy on hearing in symptomatic congenital cytomegalovirus disease involving the central nervous system: a randomized, controlled trial. , 2003, The Journal of pediatrics.

[23]  A. Q. Summerfield,et al.  Health-service costs of pediatric cochlear implantation: multi-center analysis. , 2003, International journal of pediatric otorhinolaryngology.

[24]  M. Barbi,et al.  A wider role for congenital cytomegalovirus infection in sensorineural hearing loss , 2003, The Pediatric infectious disease journal.

[25]  W. Britt,et al.  Longitudinal investigation of hearing disorders in children with congenital cytomegalovirus. , 2000, Journal of the American Academy of Audiology.

[26]  R. Parker,et al.  Children with Minimal Sensorineural Hearing Loss: Prevalence, Educational Performance, and Functional Status , 1998, Ear and hearing.

[27]  W. Britt,et al.  Progressive and fluctuating sensorineural hearing loss in children with asymptomatic congenital cytomegalovirus infection. , 1997, The Journal of pediatrics.

[28]  G. Demmler,et al.  Surveillance for congenital cytomegalovirus disease: a report from the National Congenital Cytomegalovirus Disease Registry. , 1995, Clinical infectious diseases : an official publication of the Infectious Diseases Society of America.

[29]  C. Baboonian,et al.  Congenital and maternal cytomegalovirus infections in a London population , 1991, British journal of obstetrics and gynaecology.

[30]  C. Peckham,et al.  CYTOMEGALOVIRUS INFECTION IN PREGNANCY: PRELIMINARY FINDINGS FROM A PROSPECTIVE STUDY , 1983, The Lancet.

[31]  J. Stockman,et al.  Dried Blood Spot Real-time Polymerase Chain Reaction Assays to Screen Newborns for Congenital Cytomegalovirus Infection , 2011 .

[32]  A. Culyer,et al.  Newborn screening for inborn errors of metabolism: a systematic review. , 1997, Health technology assessment.

[33]  T. Sheldon,et al.  Home parenteral nutrition: a systematic review. , 1997, Health technology assessment.

[34]  F. Sassi,et al.  Cost Effectiveness and Resource Allocation Open Access the Cost-effectiveness of Neonatal Screening for Cystic Fibrosis: an Analysis of Alternative Scenarios Using a Decision Model , 2022 .