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PubMed2006

Measuring parent satisfaction with a neonatal hearing screening program.

The primary aim of the present study was to investigate parent satisfaction with a neonatal hearing screening program through use of a valid and reliable questionnaire developed for this purpose (Parent Satisfaction Questionnaire with Neonatal Hearing Screening Program; PSQ-NHSP). Eighty parents whose children had received hearing screening participated in this study. High levels of satisfaction were reported with more than 90% of parents satisfied with all aspects of the program.The PSQ-NHSP was analyzed for validity and reliability and demonstrated excellent internal consistency reliability (sigma = 0.94) and excellent test-retest reliability (p = 0.97). Content validity of the PSQ-NHSP was partially established by reviewing available literature on parent satisfaction studies in other pediatric health-care service programs. Construct validity of the PSQ-NHSP was indicated by a significant positive relationship between overall satisfaction and the three specific dimensions in the questionnaire. The satisfaction questionnaire was found to be a useful instrument for identifying service shortfalls, and routine use of the PSQ-NHSP in other neonatal hearing screening programs is recommended.

Adult↗

PubMed1998

[Programs of systematic screening in neonatology. Pharmaco-economic aspects].

Systematic neonatal screening offers many advantages for the patients, its family and the community. The Genetic center of the University Hospital of Liège provides neonatal screening for the following diseases: phenylketonuria, congenital hypothyroidy, cystic fibrosis, alpha-1-antitrypsin, adrenal hyperplasia and biotinidase deficiency. On economical grounds, it is clear that the organisation of neonatal screening costs less to the community than the cost of the disease if diagnosis is made too late as to allow an alleviation, or even a total recovery, of the symptomatology.

Acyltransferases↗

PubMed1979

Screening for neonatal hypothyroidism by thyroxine and thyrotrophin radioimmunoassays using dried blood samples on filter paper.

A routine and automatized methodology for thyroxine (T4) and thyrotrophin (TSH) radioimmunoassay (RIA) using dried blood samples on filter paper is described. T4-RIA was performed on one single dot (5 mm diameter equivalent to 4 microliters of serum) while two dots were necessary for TSH-RIA. Reference filter papers were introduced in each assay for quality control. In a preliminary study on 4,155 neonates, samples generally obtained between the 5th--7th day gave a mean 'dot-T4' of 97.95 +/- 36.04 nmol/l and a mean 'dot-TSH' of 10.19 mU/l +/- 8.25, corresponding to 2.47 mU/l of serum. Within an 18-month period (November 1976-April 1978), a total of 16.522 neonates have been screened allowing detection of three cases of congenital hypothyroidism (incidence 1 : 5507), two cases of congenitally low TBG and thirty-three cases of transient hypothyroidism.

Female↗

PubMed1999

[Auditory screening of neonates using an objective method--otoacoustic emissions].

Screening for hearing loss in children in Slovak republic with non-objective methods detects only deaf children, mean age of the diagnosis establishment is even 2.5 years. It is necessary for healthy child development to detect and treat a child with bilateral defect before 1 year of age. Preliminary results of auditory screening in 456 newborns with objective method of otoacoustic emissions (OAE) proved the necessity of otoacoustic screening in all risk newborns. In one of 204 risk newborns serious hearing loss was proved with BERA examination and in next 8 of them it is suspected according to the OAE results and additional tympanometric examination with the A curve. Until now in newborn without risk of hearing loss in history were not detected bilateral hearing loss, only unilateral, not requiring treatment. (Fig. 1, Tab. 2. Ref. 9.)

Female↗

PubMed1982

Health economic analysis of the Swedish neonatal metabolic screening programme. A method of optimizing routines.

A benefit-cost analysis was carried out to optimize the routines for neonatal metabolic screening. The basis of the study was provided by results of the Swedish neonatal screening programme from 1965 to 1979. During this period over one million infants were screened by the Guthrie test for phenylketonuria and galactosaemia, and for limited periods also for tyrosinaemia, homocystinuria and histidinaemia. The benefit-cost ratio was calculated for combinations of different screening tests, recall routines, and varying degrees of coverage. The largest benefit-cost ratio was obtained with combined screening for phenylketonuria and galactosaemia, using a borderline blood phenylalanine level of 0.50 mmol/L in the Guthrie test for phenylketonuria. However, the inaccuracy of this test necessitated the use of a lower blood phenylalanine level of 0.25 mmol/L and the acceptance of a lower benefit-cost ratio. An increase in the present 98% coverage of newborns by the screening programme was found to be an effective means of improving the benefit-cost ratio in the present programme.

Amino Acid Metabolism, Inborn Errors↗

PubMed1998

Field sensitivity of targeted neonatal hearing screening using the Nottingham ABR Screener.

OBJECTIVE: A population of children having bilateral moderate to profound hearing impairment was investigated to find out the number who passed (false negatives) and who failed (true positives) a screening test based on the auditory brain stem response (ABR). DESIGN: This study of the ABR is a parallel report to Lutman, Davis, Fortnum, and Wood (1997), where the transient evoked otoacoustic emission was examined on a similar but not identical population of at-risk neonates. Hearing screening was undertaken in at-risk neonates (targeted screening) at seven hospitals in the UK using the Nottingham ABR Screener. During the period from January 1988 to December 1993, a total of 6983 neonates had been tested. Assessment of audiological records from the participating centers ascertained that 201 children born between January 1988 and December 1993 had hearing threshold levels in both ears of 50 dB or more, averaged over the speech frequencies 0.5, 1, 2, and 4 kHz. Of these, 51 had completed the ABR screening test. Examination of the ABR records, which included both machine and visual scoring, indicated whether they had passed or failed the screening test. RESULTS: Forty-six of the 51 hearing impaired babies failed the neonatal ABR screen either on one or both ears (five false negatives), and 42 failed the test on both ears (nine false negatives). This resulted in field sensitivities of 90% and 82%, respectively. Two cases of suspected progressive hearing loss have been included in the numbers of false negative results. The percentage of babies passing the screen on both ears and subsequently having normal hearing (specificity) is typically 93%. CONCLUSIONS: Neonatal hearing screening in an at-risk population using a highly automated ABR test is a viable and effective tool for identification of hearing impairment. Although the field sensitivity of the test is high, it is unable to identify all babies with a criterion level of hearing loss. There are a number of possible explanations as to the origin of false negative results: configuration of the pure-tone audiogram, a progressive hearing loss, acquired sensorineural or conductive loss, retrocochlear deafness, or an incorrect interpretation of the screening test. Passing a neonatal screening test, therefore, does not exclude the possibility of subsequent hearing impairment and highlights the need for further surveillance.

Auditory Threshold↗

PubMed1999

Clinical outcomes of newborn screening for cystic fibrosis.

AIM: To determine how early diagnosis of cystic fibrosis, using neonatal screening, affects long term clinical outcome. METHODS: Fifty seven children with cystic fibrosis born before neonatal screening was introduced (1978 to mid 1981) and a further 60 children born during the first three years of the programme (mid 1981 to 1984), were followed up to the age of 10. The cohorts were compared on measures of clinical outcome, including height, weight, lung function tests, chest x-ray picture and Shwachman score. RESULTS: Age and sex adjusted standard deviation scores (SDS) for height and weight were consistently higher in children screened for cystic fibrosis than in those born before screening. At 10 years of age, average differences in SDS between groups were 0.4 (95% CI -0.1, 0.8) for weight and 0.3 (95% CI -0.1, 0.7) for height. This translates to an average difference of about 2.7 cm in height and 1.7 kg in weight. Mean FEV1 and FVC (as percentage predicted) were significantly higher in the screened cohort at 5 and 10 years of age, with an average difference of 9.4% FEV1 (95% CI 0.8, 17.9) and 8.4% FVC (95% CI 1.8, 15.0) at 10 years. Chest x-ray scores were not different between the groups at any age, but by 10 years screened patients scored an average 5.3 (95% CI 1.2, 9.4) points higher on the Shwachman score. CONCLUSION: Although not a randomised trial, this long term observational study indicates that early treatment made possible by neonatal screening may be important in determining subsequent clinical outcomes for children with cystic fibrosis. For countries contemplating the introduction of neonatal screening for cystic fibrosis, its introduction to some areas in a cluster randomised design will permit validation of studies performed to date.

Anti-Bacterial Agents↗

PubMed1997

Newborn screening for inborn errors of metabolism: a systematic review.

OBJECTIVES. To establish a database of literature and other evidence on neonatal screening programmes and technologies for inborn errors of metabolism. To undertake a systematic review of the data as a basis for evaluation of newborn screening for inborn errors of metabolism. To prepare an objective summary of the evidence on the appropriateness and need for various existing and possible neonatal screening programmes for inborn errors of metabolism in relation to the natural history of these diseases. To identify gaps in existing knowledge and make recommendations for required primary research. To make recommendations for the future development and organisation of neonatal screening for inborn errors of metabolism in the UK. HOW THE RESEARCH WAS CONDUCTED. There were three parts to the research. A systematic review of the literature on inborn errors of metabolism, neonatal screening programmes, new technologies for screening and economic factors. Inclusion and exclusion criteria were applied, and a working database of relevant papers was established. All selected papers were read by two or three experts and were critically appraised using a standard format. Seven criteria for a screening programme, based on the principles formulated by Wilson and Jungner (WHO, 1968), were used to summarise the evidence. These were as follows. Clinically and biochemically well-defined disorder. Known incidence in populations relevant to the UK. Disorder associated with significant morbidity or mortality. Effective treatment available. Period before onset during which intervention improves outcome. Ethical, safe, simple and robust screening test. Cost-effectiveness of screening. A questionnaire which was sent to all newborn screening laboratories in the UK. Site visits to assess new methodologies for newborn screening. The classical definition of an inborn error of metabolism was used (i.e., a monogenic disease resulting in deficient activity in a single enzyme in a pathway of intermediary metabolism). RESEARCH FINDINGS. INBORN ERRORS OF METABOLISM. Phenylketonuria (PKU) (incidence 1:12,000) fulfilled all the screening criteria and could be used as the 'gold standard' against which to review other disorders despite significant variation in methodologies, sample collection and timing of screening and inadequacies in the infrastructure for notification and continued care of identified patients. Of the many disorders of organic acid and fatty acid metabolism, a case can only be made for the introduction of newborn screening for glutaric aciduria type 1 (GA1; estimated incidence 1:40,000) and medium-chain acyl CoA dehydrogenase (MCAD) deficiency (estimated incidence 1:8000-1:15,000). Therapeutic advances for GA1 offer prevention of neurological damage but further investigation is required into the costs and benefits of screening for this disorder. MCAD deficiency is simply and cheaply treatable, preventing possible early death and neurological handicap. Neonatal screening for these diseases is dependent upon the introduction of tandem mass spectrometry (tandem MS). This screening could however also simultaneously detect some other commonly-encountered disorders of organic acid metabolism with a collective incidence of 1:15,000.(ABSTRACT TRUNCATED)

Cost-Benefit Analysis↗

PubMedDate not supplied

Neonatal hearing screening with transient evoked otoacoustic emissions: a learning curve.

The present paper reports on the implementation of a neonatal hearing screening programme in a private hospital in Belgium. A maternity-based neonatal hearing screening project with transient evoked otoacoustic emissions (TEOAEs) was started in 1993. The cost of the test was not covered by the public health insurance, so the parents had to pay the full cost for screening their child (approximately 30 Euro). Since 1993 the programme strategies have been changed on several occasions to improve the quality and efficacy. A retrospective analysis was performed on: (1) the test pass rate; (2) the coverage; and (3) the number of children who become 'Lost to follow-up' after failing the initial test. The data show a steady learning curve with a time course of several years. They also demonstrate that it is worthwhile and feasible to run a high-quality screening programme in a private establishment.

Belgium↗

PubMed1982

Pitfalls in screening for neonatal hypothyroidism. Report of the New England Regional Screening Program and the New England Congenital Hypothyroidism Collaborative.

This report reviews problems encountered during the first five years of operation of the New England Regional Screening Program. Human error was responsible for either overlooked or delayed diagnosis and treatment in 14/159 infants with hypothyroidism. This group consisted of three infants who were never tested, six infants whose diagnosis was missed because of errors during the processing of the blood specimens, and five additional children in whom the diagnosis of hypothyroidism could not be made at birth because the concentrations of thyroid-stimulating hormone were not elevated. Many of the problems, most of which occurred during the first two years of the study, have been eliminated by improved communication and monitoring. This study emphasizes the potential vulnerability of all screening programs and underscores the need for physicians to continue to exercise their best clinical judgement regardless of the circumstances.

Congenital Hypothyroidism↗

PubMed2003

False-negative primary neonatal thyroid screening: the need for clinical vigilance and secondary screening.

The screening of newborn babies for congenital hypothyroidism has changed the natural history of this abnormality. We describe here a case of a female patient with congenital hypothyroidism that was missed by primary neonatal thyroid screening (using thyroid-stimulating hormone) at two days of age; it was detected only after the development and persistence of jaundice during the first three weeks of life. A normal neonatal screening result does not preclude the development of hypothyroidism later in infancy. Clinical vigilance must be maintained by practitioners. A second screening between two and six weeks of age may be useful in order to detect the few cases missed at first screening.

False Negative Reactions↗

PubMed2005

Neonatal hearing screening with transient evoked otoacoustic emissions in Western Saudi Arabia.

OBJECTIVE: To study the incidence of congenital hearing impairment in the Saudi population and to evaluate the need of establishing a Saudi universal neonatal hearing screening program based on transient evoked otoacoustic emission. METHODS: A total of 11986 well non-high-risk neonates were screened by transient evoked otoacoustic emission over period of 8 years from September 1996 to February 2004. The universal hearing screening was consequently done in a daily base before discharge from nursery. Those who failed the initial screening were followed up diagnostically until hearing loss was confirmed or excluded. RESULTS: From the total number of 11,986 neonates (41.4% male and 58.6% females) examined in this study 10943 (91.3%) neonates passed the first screening step while 1043(8.7%) neonates failed. From the 1043 neonates examined in the second screening step in the 5th day of life, 300 (29%) neonates failed. At the age of 5 months, all the 300 infants that failed the second screening step underwent a comprehensive audiological assessment to confirm the existence of hearing loss. The 278 infants that passed the assessment were considered as normal; while 22 failed and were confirmed to have congenital hearing loss. Of these 22 infants, 2 had unilateral sensorineural hearing loss, and the remaining 20 had bilateral sensorineural hearing loss. The incidence of sensorineural hearing loss was estimated to be 0.18% while the incidence of bilateral sensorineural hearing loss was 0.17%. No significant difference between males and females was found. The average age at confirmation of congenital hearing loss was 5.5 months. CONCLUSION: The incidence of congenital hearing loss in the western region of Saudi Arabia is relatively high compared with international figures. Hearing screening for all neonates using transient evoked otoacoustic emission should be part of the standard medical care in Saudi Arabia.

Audiometry, Evoked Response↗

PubMed1992

The association of thyroid dyshormonogenesis and deafness (Pendred syndrome): experience of the Victorian Neonatal Thyroid Screening Programme.

Between 1977 and 1989, the Victorian Neonatal Thyroid Screening Programme detected five subjects with thyroid dyshormonogenesis and sensorineural deafness. These patients have been diagnosed as having Pendred syndrome. In two of the children, thyroid function tests which were initially abnormal at birth returned to normal spontaneously without treatment. However, hypothyroidism subsequently recurred and the children required thyroxine therapy. These two children could have been mistakenly diagnosed as having transient hypothyroidism. The detection of five patients with Pendred syndrome illustrates the importance of audiological assessment in all babies with thyroid dyshormonogenesis in whom there is increased uptake of isotope on thyroid scanning. In our experience, hearing loss in patients with Pendred syndrome may be progressive over time, so that repeated audiological assessments are necessary.

Deafness↗

PubMed2004

Feasibility of implementing a universal neonatal hearing screening programme using distortion product otoacoustic emission detection at a university hospital in Hong Kong.

OBJECTIVE: To assess the feasibility of implementing a universal neonatal hearing screening programme using distortion product otoacoustic emission detection at a major teaching hospital in Hong Kong. DESIGN: Descriptive study and questionnaire. SETTING. Teaching hospital, Hong Kong. METHODS: A total of 1064 infants, together with their mothers, were successfully recruited for the study. The participation rate was 99.3%. A three-stage hearing screening protocol using distortion product otoacoustic emission detection was adopted. Each of the participating infants was screened on three separate occasions (day 1-4, day 5-14, and day 21-30 after birth), irrespective of the test results. A questionnaire was administered to 364 randomly selected mothers to determine whether as consumers of the hearing screening service, mothers would find screening desirable. RESULTS: Results of the screening demonstrated an incidence of permanent bilateral hearing loss (>or=40 dB in the better ear) of 0.28%. The results also showed that 3.5% of the screened infants were referred for subsequent diagnostic audiological assessment, including those suspected with unilateral as well as bilateral hearing loss. Data obtained were comparable to other reported results obtained using multi-stage screening protocols. Taking both the false positive rate and the default rate into consideration, the most appropriate time for screening in this hospital setting appeared to be around day 5 to 14 when infants returned to the hospital's day centre as out-patients for routine medical follow-up. Since this day centre service is not generally provided by all maternity hospitals in Hong Kong, an alternative time for screening would be around day 21 to 30 when infants could return as out-patients solely for the hearing test. The results of the questionnaire suggested that most mothers thought a neonatal hearing screening would be desirable (91.35%). The majority (81.70%) indicated a preference for screening either within a few days of birth at the maternity ward prior to discharge from the hospital, or between 5 and 30 days when returning to the hospital as an out-patient. CONCLUSION: It was concluded that a universal neonatal hearing screening programme could be readily implemented in a maternity hospital setting in Hong Kong.

Clinical Protocols↗

PubMed2003

[Sensitivity of distortion product otoacoustic emissions and auditory brain-stem response in neonatal hearing screening, a comparative study].

OBJECTIVE: To investigate the sensitivity of distortion product otoacoustic emissions (DPOAE) and auditory brain-stem response (ABR) in neonatal hearing screening. METHODS: DPOAE and ABR were conducted on 99 neonates (198 ears) at risk of hearing impairment as initial screening tests. One month later rescreening tests were conducted: DPOAE was conducted again on those ears failing this test during the initial screening test and ABR was conducted again on the ears failing this test during the initial screening test Those ears failing the DPOAE and ABR tests during the rescreening tests were examined with electric otoscope, diagnostic ABR, etc. to get confirmed diagnosis. RESULTS: 54 ears (27%) failed the DPOAE test during the initial screening and 14 out of the 54 ears (14/198, 7%) still failed the DPOAE test during the rescreening. 26 ears (12%) failed the ABR test during the initial screening and 9 out of the 16 ears (9/198, 4.5%) still failed this test during the rescreening. Two babies were confirmed as with severe sensorineural hearing loss, one unilateral and one bilateral. The sensitivity was 97% for DPOAE and 99% for ABR. CONCLUSION: ABR is a little bit more sensitive then DPOAE in neonatal hearing screening. Two-stage testing procedure increases the sensitivity of neonatal hearing screening.

Evoked Potentials, Auditory, Brain Stem↗

PubMedDate not supplied

[Neonatal hearing screening].

Auditory deprivation in early life significantly effects speech, language, cognition and learning development. The importance of neonatal hearing screening in order to detect and manage children with hearing impairment during the critical period before three years of age can't be neglected. The neonatal hearing screening program at Veterans General Hospital, Taipei, under a cooperation of audiologists, otologists and pediatricians, identified 1885 neonates for hearing loss from September 1990 to August 1991. There were two study groups, a high risk group of hearing impairment and a non-high risk group. We performed hearing screening and followed-up for the failure at initial test using methods including behavioral observation audiometry, auditory brainstem response, tympanogram, pneumatic otoscopy and questionnaire. Approximately 2.7% of neonates were classified as high risk for hearing loss, the incidence of hearing loss was 8% to 14% in this group. The incidence of hearing loss in non-high risk group was 0.11%. If both groups were contained that the incidence of hearing loss was 0.32% to 0.48%, 0.11% with a sensorineural hearing loss, 0.21% to 0.37% with a conductive hearing loss. Both behavioral audiometry (89% false negative rate) and questionnaire (50% return rate) were not effective for screening purposes in this study. Our results also indicated that the most advantage of using a high risk register lay in the fact that only 2.7% of a total population needed to be screened to identify approximately 67% to 78% of hearing-impaired babies. This saved both time and money.

Audiometry, Evoked Response↗

PubMed1998

The costs of early hearing screening in England and Wales.

A survey was carried out in 10 centres in England and Wales to determine the costs of hearing screening in the first year of life. The screens that were studied were targeted neonatal, universal neonatal, and the health visitor distraction test (HVDT) or alternative surveillance. Valid data were available from five centres for targeted neonatal screening (TNS), three for universal neonatal screening (UNS), and nine for the HVDT, although only five of the HVDT screens had valid data for follow up costs. The neonatal costs were consistent across the centres surveyed, whereas those for the HVDT screen varied considerably. The mean service costs for TNS, UNS, and the HVDT at 1994 prices were 5052 Pounds, 13,881 Pounds, and 24,519 Pounds for a standardised district of 1000 live births. Three conclusions seem justified. Firstly, UNS need not be prohibitively expensive as it costs considerably less than HVDT screening. Secondly, TNS appears to be a relatively inexpensive way of improving the age of identification of a proportion of the congenitally hearing impaired. Thirdly, given the published yields for UNS and the HVDT, the results indicate that UNS offers the most cost effective overall approach with alternative systems in place to identify late onset permanent hearing losses.

England↗

PubMed2006

Neonatal hearing screening: evaluation of tone-burst and click-evoked otoacoustic emission test criteria.

OBJECTIVE: Click-evoked otoacoustic emissions (CEOAEs) are widely used in universal neonatal hearing screening programs. A common finding in many such programs is a relatively high false-positive rate. This is often due to infant physiological noise adversely affecting the emission recording, leading to a "refer" screening outcome. In an attempt to reduce false-positive screening outcomes related to the effects of noise on otoacoustic emission response detection, tone-burst-evoked otoacoustic emissions (TBOAEs) were included in a neonatal hearing screening program because TBOAEs may elicit a greater signal-to-noise ratio than CEOAEs. The research project compared the pass/refer rate for a CEOAE-based test using established pass/refer criteria with the pass/refer rate for screening criteria that were based on TBOAE results alone or on combined CEOAE and TEOAE results. DESIGN: Neonates were recruited at the Hong Kong Adventist Hospital, and both CEOAEs and TBOAEs were performed. Six passing criteria were used in this study, based on CEOAEs only; CEOAEs plus 1 kHz TBOAEs; CEOAEs plus 2 kHz TBOAEs; CEOAEs plus 3 kHz TBOAEs; CEOAEs plus 1, 2, and 3 kHz TBOAEs; and TBOAEs only. RESULTS: Data from 298 neonates (546 ears) were obtained. Criteria set 1, using CEOAEs only, demonstrated a pass rate of 79.1%, and 114 ears were referred. Criteria set 2, using CEOAEs together with TBOAEs recorded at 1 kHz, passed 39 more ears than Protocol 1, and the pass rate was 86.3%. Hence, the overall referral rate for total number of screened ears decreased by 7.2 percentage points. Criteria set 3, using CEOAEs together with TBOAEs recorded at 2 kHz, and Criteria set 4, using CEOAEs in conjunction with TBOAEs recorded at 3 kHz, gave pass rates similar to Criteria set 1. Criteria set 5, using TBOAE information at frequencies where CEOAEs were not rated as "pass," raised the pass rate from 79.1 to 87.6%, reducing the overall referral rate by 8.5 percentage points. Criteria set 6, in which neonates were screened with TBOAEs recorded at 1, 2, and 3 kHz, gave a pass rate of 78.4%, similar to results for the CEOAE-only procedure. CONCLUSIONS: Both Criteria sets 2 and 5, which combined CEOAE and TBOAE recordings, gave significantly higher pass rates than Criteria sets 1, 3, 4, and 6. The results suggest that the introduction of combined CEOAE and TBOAE protocols may assist in the reduction of refer outcomes, and hence the false-positive rates, of neonatal hearing screening programs.

False Positive Reactions↗