Abstract
There is a paucity of evidence to guide the management of otitis media with effusion (OME), which is a common problem causing significant hearing impairment in children with cleft palate. The insertion of grommets is currently being used to correct hearing impairment and prevent complications of unmanaged OME, but there is ongoing discussion about whether the benefits of grommets outweigh the costs and risks. A decision-tree model was developed to assess the surgical insertion of grommets with two non-surgical alternatives (hearing-aids and do-nothing strategies) in cleft palate children with persistent bilateral OME. The model assumed a 2-year time horizon and a UK National Health Service perspective. Outcomes were valued using quality-adjusted life-years (QALYs) estimated by linking utility values with potential hearing gains measured in decibels. Multiple data sources were used, including reviews of the clinical effectiveness, resource use and utility literature, and supplemented with expert opinion. Uncertainty in the model parameters was assessed using probabilistic sensitivity analysis. Expected value of perfect information analysis was used to calculate the potential value of future research. The results from the probabilistic sensitivity analysis indicated that the grommets strategy was associated with an incremental cost-effectiveness ratio of £9,065 per QALY gained compared with the do-nothing strategy, and the hearing-aids strategy was extended dominated by the grommets strategy. The population expected value of perfect information was £5,194,030 at a willingness to pay threshold of £20,000 per QALY, implying that future research could be potentially worthwhile. This study found some evidence that the insertion of grommets to manage cleft palate children with bilateral OME is likely to be cost-effective, but further research is required to inform this treatment choice.
Similar content being viewed by others
Notes
Data from the CRANE Database showed there were 800 children born with CP in England, Wales and Northern Ireland in 2012 [http://www.craniofacialsociety.org.uk/downloads/CRANE%20Annual%20Report%202013%20(Final).pdf]. Of which, 720 (90 % [2, 4–6] of 800) were assumed to have OME.
Armstrong [15] first described the use of grommets in 1954, and since then, use of grommets to restore hearing to normal has been increased. Given the historical longevity of the technology revealed in the literature, it seemed reasonable to assume that grommets will be used for at least another 10 years before a new technology comes along and replaces grommet technology.
As recommended by the NICE reference case [25].
References
Møller, P.: Hearing, middle ear pressure and otopathology in a cleft palate population. Acta Otolaryngol. 92, 521–528 (1981)
Sheahan, P., Miller, I., Sheahan, J.N., Earley, M.J., Blayney, A.W.: Incidence and outcome of middle ear disease in cleft lip and/or cleft palate. Int. J. Pediatr. Otorhinolaryngol. 67, 785–793 (2003)
Ponduri, S., Bradley, R., Ellis, P.E., Brookes, S.T., Sandy, J.R., Ness, A.R.: The management of otitis media with early routine insertion of grommets in children with cleft palate—a systematic review. Cleft Palate Craniofac. J. 46, 30–38 (2009)
Stool, S.E., Randall, P.: Unexpected ear disease in infants with cleft palate. Cleft Palate J. 4, 99–103 (1967)
Doyle, W.J., Cantekin, E.I., Bluestone, C.D.: Eustachian tube function in cleft palate children. Ann. Otol. Rhinol. Laryngol. Suppl. 89, 34–40 (1980)
Grant, H.R., Quiney, R.E., Mercer, D.M., Lodge, S.: Cleft palate and glue ear. Arch. Dis. Child. 63, 176–179 (1988)
National Collaborating Centre for Women’s and Children’s Health: Surgical management of otitis media with effusion in children. NICE clinical guideline 60. National Institute for Health and Clinical Excellence, London (2008)
Maw, R., Wilks, J., Harvey, I., Peters, T.J., Golding, J.: Early surgery compared with watchful waiting for glue ear and effect on language development in preschool children: a randomised trial. Lancet 353, 960–963 (1999)
Maheshwar, A.A., Milling, M.A.P., Kumar, M., Clayton, M.I., Thomas, A.: Use of hearing aids in the management of children with cleft palate. Int. J. Pediatr. Otorhinolaryngol. 66, 55–62 (2002)
Gani, B., Kinshuck, A.J., Sharma, R.: A review of hearing loss in cleft palate patients. Int. J. Otolaryngol. (2012)
Rovers, M.M., Schilder, A.G.M., Zielhuis, G.A., Rosenfeld, R.M.: Otitis media. Lancet 363, 465–473 (2004)
Rosenfeld, R.M., Kay, D.: Natural history of untreated otitis media. Laryngoscope 113, 1645–1657 (2003)
Rovers, M.M., Glasziou, P., Appelman, C.L., Burke, P., McCormick, D.P., Damoiseaux, R.A., Gaboury, I., Little, P., Hoes, A.W.: Antibiotics for acute otitis media: a meta-analysis with individual patient data. Lancet 368, 1429–1435 (2006)
Tierney, S., O’Brien, K., Harman, N.L., Sharma, R.K., Madden, C., Callery, P.: Otitis media with effusion: experiences of children with cleft palate and their parents. Cleft Palate Craniofac. J. (2013)
Armstrong, B.W.: A new treatment for chronic secretory otitis media. AMA Arch. Otolaryngol. 59, 653–654 (1954)
Lous, J., Burton, M.J., Felding, J.U., Ovesen, T., Rovers, M.M., Williamson, I.: Grommets (ventilation tubes) for hearing loss associated with otitis media with effusion in children. Cochrane Database Syst. Rev. CD001801 (2005)
Schilder, A.G.M., Rovers, M.M.: International perspective on management. In: Rosenfeld, R.M., Bluestone, C.D. (eds.) Evidence-Based Otitis Media, pp. 325–332. Hamilton, BC Decker (2003)
Berman, S., Roark, R., Luckey, D.: Theoretical cost effectiveness of management options for children with persisting middle ear effusions. Pediatrics 93, 353–363 (1994)
Gates, G.A.: Cost-effectiveness considerations in otitis media treatment. Otolaryngol. Head Neck Surg. 114, 525–530 (1996)
Hartman, M., Rovers, M.M., Ingels, K., Zielhuis, G.A., Severens, J.L., van der Wilt, G.J.: Economic evaluation of ventilation tubes in otitis media with effusion. Arch. Otolaryngol. Head Neck Surg. 127, 1471–1476 (2001)
Sculpher, M., Drummond, M., Buxton, M.: The iterative use of economic evaluation as part of the process of health technology assessment. J. Health Serv. Res. Policy 2, 26–30 (1997)
Gould, H.J.: Hearing loss and cleft palate: the perspective of time. Cleft Palate J. 27, 36–39 (1990)
Phua, Y.S., Salkeld, L.J., de Chalain, T.M.B.: Middle ear disease in children with cleft palate: protocols for management. Int. J. Pediatr. Otorhinolaryngol. 73, 307–313 (2009)
Liu, L., Sun, Y., Ma, L., Zhao, W., Wu, R.: Effect of ventilation tube insertion on otitis media with effusion in cleft palate children. Zhonghua Er Bi Yan Hou Ke Za Zhi. 39, 216–218 (2004)
National Institute for Clinical Excellence: Guide to the methods of technology appraisal. http://www.nice.org.uk/niceMedia/pdf/TAP_Methods.pdf (2004). Accessed 10 Apr 2013
Barton, P., Bryan, S., Robinson, S.: Modelling in the economic evaluation of health care: selecting the appropriate approach. J. Health Serv. Res. Policy 9, 110–118 (2004)
Kwan, W.M.Y., Abdullah, V.J., Liu, K., van Hasselt, C.A., Tong, M.C.F.: Otitis media with effusion and hearing loss in Chinese children with cleft lip and palate. Cleft Palate Craniofac. J. 48, 684–689 (2011)
Sheahan, P., Blayney, A.W., Sheahan, J.N., Earley, M.J.: Sequelae of otitis media with effusion among children with cleft lip and/or cleft palate. Clin. Otolaryngol. Allied Sci. 27, 494–500 (2002)
Kay, D.J., Nelson, M., Rosenfeld, R.M.: Meta-analysis of tympanostomy tube sequelae. Otolaryngol. Head Neck Surg. 124, 374–380 (2001)
Fior, R., Veljak, C.: Late results and complications of tympanostomy tube insertion for prophylaxis of recurrent purulent otitis media in pediatric age. Int. J. Pediatr. Otorhinolaryngol. 8, 139–146 (1984)
Vlastarakos, P.V., Nikolopoulos, T.P., Korres, S., Tavoulari, E., Tzagaroulakis, A., Ferekidis, E.: Grommets in otitis media with effusion: the most frequent operation in children. But is it associated with significant complications? Eur. J. Pediatr. 166, 385–391 (2007)
Gibb, A.G.: President’s address. Tympanosclerosis. Proc. R. Soc. Med. 69, 155–162 (1976)
Williamson, I.: Otitis media with effusion in children. Clin. Evid. (2011)
NHS Choices: Complications of glue ear. http://www.nhs.uk/Conditions/Glueear/Pages/Complications.aspx. Accessed 10 Jul 2013
Hoffmann, K.K., Thompson, G.K., Burke, B.L., Derkay, C.S.: Anesthetic complications of tympanostomy tube placement in children. Arch. Otolaryngol. Head Neck Surg. 128, 1040–1043 (2002)
Children’s Hospital: Grommets surgery for glue ear—information for patients. Oxford Radcliffe Hospitals NHS Trust. http://www.ouh.nhs.uk/patient-guide/leaflets/files%5C101018grommets.pdf (2012). Accessed 10 Apr 2013
Gunasekera, H., O’Connor, T.E., Vijayasekaran, S., Mar, C.B.D.: Primary care management of otitis media among Australian children. Med. J. Aust. 191 (2009)
Maw, R., Bawden, R.: Spontaneous resolution of severe chronic glue ear in children and the effect of adenoidectomy, tonsillectomy, and insertion of ventilation tubes (grommets). BMJ 306, 756–760 (1993)
Kubba, H.: Quality of life assessment in paediatric otolaryngology. MD thesis, University of Glasgow (2004)
Rosenfeld, R.M., Bhaya, M.H., Bower, C.M., Brookhouser, P.E., Casselbrant, M.L., Chan, K.H., Cunningham, M.J., Derkay, C.S., Gray, S.D., Manning, S.C., Messner, A.H., Smith, R.J.: Impact of tympanostomy tubes on child quality of life. Arch. Otolaryngol. Head Neck Surg. 126, 585–592 (2000)
Paradise, J.L.: Middle ear problems associated with cleft palate. An internationally-oriented review. Cleft Palate J. 12, 17–22 (1975)
Bluestone, C.D.: Eustachian tube obstruction in the infant with cleft palate. Ann. Otol. Rhinol. Laryngol. 80(Suppl 2), 1–30 (1971)
Rawlins, M.D., Culyer, A.J.: National Institute for Clinical Excellence and its value judgments. BMJ 329, 224–227 (2004)
Claxton, K., Ginnelly, L., Sculpher, M., Philips, Z., Palmer, S.: A pilot study on the use of decision theory and value of information analysis as part of the NHS Health Technology Assessment programme. Health Technol. Assess. Winch. Engl. 8, 1–103 (2004)
Schönweiler, R., Lisson, J.A., Schönweiler, B., Eckardt, A., Ptok, M., Tränkmann, J., Hausamen, J.E.: A retrospective study of hearing, speech and language function in children with clefts following palatoplasty and veloplasty procedures at 18–24 months of age. Int. J. Pediatr. Otorhinolaryngol. 50, 205–217 (1999)
Kalcioglu, M.T., Cokkeser, Y., Kizilay, A., Ozturan, O.: Follow-up of 366 ears after tympanostomy tube insertion: why is it draining? Otolaryngol. Head Neck Surg. 128, 560–564 (2003)
Schönweiler, R., Schönweiler, B., Schmelzeisen, R.: Hearing capacity and speech production in 417 children with facial cleft abnormalities. HNO 42, 691–696 (1994)
Paradise, J.L., Bluestone, C.D.: Early treatment of the universal otitis media of infants with cleft palate. Pediatrics 53, 48–54 (1974)
Britten, N., Ukoumunne, O.: The influence of patients’ hopes of receiving a prescription on doctors’ perceptions and the decision to prescribe: a questionnaire survey. BMJ 315, 1506–1510 (1997)
Macfarlane, J., Holmes, W., Macfarlane, R., Britten, N.: Influence of patients’ expectations on antibiotic management of acute lower respiratory tract illness in general practice: questionnaire study. BMJ 315, 1211–1214 (1997)
Russell, C., Black, O., Dutt, D., Ray, A., Devlin, M., Wynne, D.: Are ventilation tubes (grommets) in cleft children truly associated with increased complication rates? Results of a nested case control study of cleft and non-cleft children. Br. J. Oral Maxillofac. Surg. 50, S2–S3 (2012)
Acknowledgments
This study was supported by the National Institute for Health Research Health Technology Assessment Programme at the National Health Service and the Healing Foundation, UK. The Management of Otitis Media with Effusion in Children with Cleft Palate (MOMENT) Study Management Group (Iain Bruce, Peter Callery, Nicola Harman, Syed Mohiuddin, Kevin O’Brien, Katherine Payne, Bill Shaw, Tri Tat, Stephanie Tierney and Paula Williamson). The Healing Foundation Cleft and Craniofacial Clinical Research Centre, University of Manchester, and Paul Jacklin (senior health economist, National Collaborating Centre for Women’s and Children’s Health, UK) are thanked for providing advice and guidance.
Conflict of interest
None.
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Mohiuddin, S., Payne, K., Fenwick, E. et al. A model-based cost-effectiveness analysis of a grommets-led care pathway for children with cleft palate affected by otitis media with effusion. Eur J Health Econ 16, 573–587 (2015). https://doi.org/10.1007/s10198-014-0610-8
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s10198-014-0610-8
Keywords
- Otitis media with effusion
- Cleft palate
- Grommets
- Hearing aids
- Cost-effectiveness
- Expected value of perfect information