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全州范围内新生儿普遍听力筛查的预计成本效益。

Projected cost-effectiveness of statewide universal newborn hearing screening.

作者信息

Keren Ron, Helfand Mark, Homer Charles, McPhillips Heather, Lieu Tracy A

机构信息

Department of Medicine, Children's Hospital, Boston, Massachusetts, USA.

出版信息

Pediatrics. 2002 Nov;110(5):855-64. doi: 10.1542/peds.110.5.855.

Abstract

OBJECTIVES

Early identification of hearing impairment may improve language outcomes and subsequent school and occupational performance of the deaf. Universal newborn hearing screening (UNHS), currently mandated by 32 states, can reduce the median age of identification of hearing impairment from 12 to 18 months to 6 months or less. However, because false-negative tests must be minimized, the prevalence of congenital deafness is low, and screening tests are imperfect, UNHS results in many false-positive results and has a low positive predictive value (PPV). The objective of this study was to evaluate UNHS and selective screening in terms of both short- and long-term benefits, harms, and financial costs and to identify steps in the screening process that could be improved to increase cost-effectiveness.

METHODS

The cost-effectiveness analysis, conducted from the societal perspective, compared the projected outcomes of 1) no newborn hearing screening, 2) selective newborn hearing screening, and 3) UNHS for a hypothetical state birth cohort of 80 000 infants. Probability and cost estimates for the decision model were obtained from published studies, expert opinion, and national and state sources. The main outcomes were incremental cost per infant whose deafness was diagnosed by 6 months, which included only the cost of screening and diagnostic evaluation; and incremental cost per deaf child with normal language, which also included the costs of medical care, education and assistive devices, and lost productivity over the lifetime of the deaf individual.

RESULTS

Selective screening identified 62 of the 128 deaf infants in the birth cohort, referred 0.18% of all infants for diagnostic evaluation, and had a PPV of 43%. UNHS identified 116 of the 128 deaf infants, referred 1.6% of all infants, and had a PPV of 8.8%. Our model simulated real-world conditions in which some infants whose deafness is identified at screening do not receive a definitive diagnosis of being deaf before 6 months; and a portion of deaf and hard-of-hearing infants who 1) have false-negative screening test results, 2) are not screened, or 3) fail the hearing screen but are not immediately followed up with diagnostic evaluation nonetheless receive a diagnosis by 6 months of age. In the absence of newborn hearing screening, approximately 30 deaf infants were identified by 6 months of age by passive detection alone at a cost of $69 000. The selective screening protocol, when compared with no newborn hearing screening, resulted in an additional 36 infants whose deafness was diagnosed by 6 months at an additional cost of approximately $600 000, yielding an incremental cost-effectiveness of approximately $16 000 per additional infant whose deafness was diagnosed by 6 months. Compared with selective screening, the UNHS protocol resulted in 33 additional infants whose deafness was diagnosed by 6 months of age at an additional cost of approximately $1.5 million, yielding an incremental cost-effectiveness of approximately $44 000 per additional infant whose deafness was diagnosed by 6 months of age. Increasing the rate of follow-up to diagnostic evaluation from the base-case estimate of 77% to 100% decreased the incremental cost of UNHS to $38 000 per additional infant whose deafness was diagnosed by 6 months. Under the base-case assumptions about lifetime savings that result from normal language with early intervention, UNHS resulted in normal language achievement for more deaf children and was cost saving in the long term compared with both selective screening and no screening.

CONCLUSIONS

The short-term cost-effectiveness of UNHS is comparable to the cost per case diagnosed of other newborn screening programs and could be improved by increasing the rate of follow-up to diagnostic evaluation after positive screening test results. If early identification results in improved language abilities, lower educational and vocational costs, and increased lifetime productivity, then UNHS has the potential for long-term cost savings compared with selective hearing screening and no screening. To understand the actual long-term economic effects of UNHS, better evidence is needed regarding the impact of early intervention on language outcomes and subsequent changes in educational costs and lifetime productivity.

摘要

目的

早期识别听力障碍可改善聋儿的语言发育结果以及随后的学业和职业表现。目前有32个州规定进行新生儿听力普遍筛查(UNHS),这可将听力障碍的识别中位年龄从12至18个月降至6个月或更小。然而,由于必须尽量减少假阴性检测,先天性耳聋的患病率较低,且筛查检测并不完美,UNHS导致许多假阳性结果,且阳性预测值(PPV)较低。本研究的目的是从短期和长期的益处、危害及财务成本方面评估UNHS和选择性筛查,并确定筛查过程中可改进的步骤以提高成本效益。

方法

从社会角度进行成本效益分析,比较了以下三种情况对一个假设的80000名婴儿的州出生队列的预测结果:1)不进行新生儿听力筛查;2)选择性新生儿听力筛查;3)UNHS。决策模型的概率和成本估计来自已发表的研究、专家意见以及国家和州的资料来源。主要结果包括在6个月时被诊断为耳聋的每名婴儿的增量成本,这仅包括筛查和诊断评估的成本;以及语言正常的每名聋儿的增量成本,这还包括医疗护理、教育和辅助设备的成本以及聋人一生中的生产力损失。

结果

选择性筛查在出生队列的128名聋儿中识别出62名,将所有婴儿中的0.18%转诊进行诊断评估,PPV为43%。UNHS在128名聋儿中识别出116名,将所有婴儿中的1.6%转诊,PPV为8.8%。我们的模型模拟了现实世界的情况,即一些在筛查时被识别为耳聋的婴儿在6个月前未得到耳聋的确切诊断;以及一部分聋儿和听力障碍婴儿,他们1)筛查试验结果为假阴性,2)未接受筛查,或3)听力筛查未通过但未立即接受诊断评估,尽管如此在6个月大时仍得到诊断。在没有新生儿听力筛查的情况下,仅通过被动检测在6个月大时大约识别出30名聋儿,成本为69000美元。与不进行新生儿听力筛查相比,选择性筛查方案在6个月时多诊断出另外36名耳聋婴儿,额外成本约为600000美元,每多一名在6个月时被诊断为耳聋的婴儿,增量成本效益约为16000美元。与选择性筛查相比,UNHS方案在6个月大时多诊断出33名耳聋婴儿,额外成本约为150万美元,每多一名在6个月大时被诊断为耳聋的婴儿,增量成本效益约为44000美元。将诊断评估的随访率从基线估计的77%提高到100%,可将UNHS的增量成本降至每多一名在6个月时被诊断为耳聋的婴儿38000美元。在关于早期干预导致正常语言带来终身节省的基线假设下,与选择性筛查和不筛查相比,UNHS使更多聋儿实现正常语言发育,且从长期来看节省成本。

结论

UNHS的短期成本效益与其他新生儿筛查项目每例诊断成本相当,可通过提高阳性筛查试验结果后诊断评估的随访率来改善。如果早期识别能改善语言能力、降低教育和职业成本并提高终身生产力,那么与选择性听力筛查和不筛查相比,UNHS有可能实现长期成本节省。为了解UNHS的实际长期经济影响,需要更好的证据来证明早期干预对语言结果的影响以及随后教育成本和终身生产力的变化。

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