언어치료 [언어발달지연]

Abstract

평가 배경 언어치료는 언어(language) 또는 말(speech)의 이상 진단 시 교정을 위해 언어재활사에 의해 행하여지는 전문작업으로 의사소통을 가능하게 하여 자신을 표현할 수 있고 사회적, 직업적으로 복귀할 수 있도록 도와주는 기술로서, 신의료기술평가제도가 확립되기 전인 2005년 비급여로 등재되었다. 2021년 제7차 의료기술재평가위원회(2021.07.09.)에서는 보건의료 자원의 효율적 사용을 지원하기 위한 근거를 제공하기 위하여, 체계적 문헌고찰을 통해 언어치료의 안전성 및 효과성에 대한 의과학적 근거를 재평가하고, 동 안건에 대하여 권고등급 결정을 수행하는 것으로 심의하였다. 동 안건은 건강보험심사평가원의 행위정의에 따라 ① 뇌졸중, 외상성 뇌손상, 뇌성마비 등의 뇌신경계 질환(이하 ‘뇌신경계질환’), ② 구음장애, 발성장애, 유창성장애, 실어증, 청각장애로 인한 언어장애(이하 ‘발성장애 등’), ③ 언어발달지연(이하 ‘언어발달지연’), ④ 기타 특수장애로 인한 언어장애 환자(이하 ‘기타 특수장애’)의 4개의 카테고리로 분류하여 평가를 진행하였다.본 평가에서는 이 중에서 언어발달지연에 대해 평가하였다.

평가 방법 언어발달지연 환자에서 언어치료의 안전성 및 효과성을 재평가하기 위하여 체계적 문헌고찰을 수행하였다. 모든 평가방법은 평가목적을 고려하여 재활의학과 2인, 신경과 1인, 이비인후과 1인, 정신건강의학과 1인, 소아청소년과 1인, 근거기반의학 1인으로 구성된 언어치료 재평가 소위원회(이하 ‘소위원회’)의 심의를 거쳐 확정하였다. 체계적 문헌고찰의 핵심질문은 ‘언어발달지연 환자에서 언어치료는 임상적으로 안전하고 효과적인가?’이다. 본 핵심질문을 토대로 국외 3개, 국내 5개 데이터베이스에서 검색하였으며, 문헌선정 및 배제기준에 따라 두 명의 검토자가 독립적으로 선별하고 선택하였다. 문헌의 비뚤림위험 평가는 Cochrane의 Risk of Bias를 사용하여 두 명의 검토자가 독립적으로 수행하여 의견합의를 이루었다. 자료추출은 미리 정해놓은 자료추출 양식을 활용하여 두 명의 검토자가 독립적으로 수행하였으며, 의견 불일치가 있을 경우 제3자와 함께 논의하여 합의하였다. 자료합성은 양적 합성이 측정도구 간의 이질성이 높아 불가능할 경우로 판단되어, 질적 검토 방법을 적용하였으며, Grading of Recommendations Assessment, Development and Evaluation (GRADE) 방법을 이용하여 근거 수준을 평가하였다. 2022년 제4차 의료기술재평가위원회(2022.04.15.)에서는 소위원회의 검토 의견을 고려하여 최종 권고등급을 결정하였다.

평가 결과 언어발달지연 환자에서 언어치료의 재평가에 선택된 문헌은 무작위배정 비교임상시험연구 5편으로 연구대상자별로, 언어발달지연 아동 3편, 자폐 스펙트럼 아동 1편, 음운장애 아동 1편 대상이었다. 본 평가는 연구대상자별로 구분하지 않고 통합해서 평가하였다.

안전성 언어발달지연 환자에서 언어치료의 안전성은 소위원회의 논의를 바탕으로 치료 관련 부작용 및 이상반응으로 평가하고자 하였으나, 안전성 결과를 보고한 문헌은 없었다. 소위원회는 통상적으로 수행되는 언어치료의 안전성에 문제가 없다는 의견이었다.

효과성 언어발달지연 환자에서 언어치료의 효과성은 소위원회의 논의를 바탕으로 언어능력, 적응행동으로 평가하였다. 언어발달지연 대상 문헌 5편 중 2편은 언어치료를 무치료와 비교하였다. 언어능력은 2편의 문헌에서 모두 보고하였다. 1편에서는 언어발달지연 아동 환자를 대상으로 발화 명료도, 어휘의 양 등 여러 지표를 통해 언어능력을 보고하였으며, 보고된 모든 결과지표에 대해 무치료군에 비해 언어치료군에서 유의한 개선을 보였다. 나머지 1편에서는 음운장애 아동 환자를 대상으로 언어치료 수행 직후, 자음정확도, 음운평가, 골드만프리스토 발음검사 지표에서 유의하게 개선되었으나 추적관찰 4개월 이후에 효과가 유지된 지표는 자음정확도 1개밖에 없었다. 평균 발화길이 지표에서는 두 군간 유의한 차이가 없었다. 적응행동 지표는 1편의 문헌에서 Vineland adaptive behavior scales (VABS) 지표를 통해 보고하였으며, 해당 문헌에서 언어치료가 무치료군에 비해 유의하게 적응행동이 개선되었다 (p=0.003). 언어발달지연 대상 문헌 5편 중 3편은 언어치료를 active control (지역사회 서비스 또는 책 읽기 등의 가족/보호자 기반의 활동)과 비교하였다. 언어능력은 3편의 문헌에서 모두 보고되었다. 1편은 자폐 스펙트럼 아동 환자를 대상으로 언어치료 수행 직후 지역사회 서비스와 비교시 초기 사회적 의사소통 능력이 유의하게 개선되었으나, 이외 보고된 다른 결과지표에서는 두 군간 유의한 차이가 없었다. 4개월 추적관찰 시점에는 지역사회 서비스와 비교시 사회적 의사소통 발화 지표 중 보호자와 환자와의 상호작용 영역에서 유의한 개선을 확인하였으나, 이외 보고된 다른 결과지표에서는 두 군간 유의한 차이가 없었다. 1편에서는 언어발달지연 아동 환자를 대상으로 추적관찰 3개월 시점에서 지역사회 서비스와 비교시 취학 전 언어척도 지표 중 수용성 영역, Number of different word (NDW) 및 Peabody 그림 어휘 검사에서 유의한 개선을 보였으나, 이외 보고된 다른 결과지표에서는 두 군간 통계적 유의한 차이를 보이지 않았다. 추적관찰 6개월 시점에는 보고된 모든 결과지표에 대해 두 군간 통계적 유의한 차이가 없었으며, 추적관찰 12개월 시점에는 Number of total utterances (NTU) 지표에서만 유의한 개선을 보였고, 이외 보고된 다른 결과지표에서는 두 군간 통계적 유의한 차이를 보이지 않았다. 나머지 1편에서는 언어발달지연 아동 환자를 대상으로 대기자 명단 환자로 대면으로 조언(face-to-face advice)을 받으면서 책 읽기 등의 가족/보호자 기반의 활동을 한 군 또는 온라인 기계(device)를 이용하여 책 읽기 등의 가족/보호자 기반의 활동을 한 군과 언어치료군을 비교하였다. 조음 및 음운의 진단검사 지표에서는 비교군에 비해 언어치료군에서 유의한 개선을 보였으나, 이외 보고된 취학 전 언어 기초의 임상진단검사, Intelligibility in Context Scale (ICS), 취학 전 단어 및 인지 측정검사 지표들에서 두 군간 통계적 유의한 차이를 보이지 않았다. 해당 언어발달지연 대상 선택문헌에서 적응행동 지표는 보고하지 않았다. 근거수준은 비뚤림 위험 평가 및 이질성 등을 고려하여 평가하였으며, ‘매우 낮음’에서 ‘낮음’으로 확인되었다.

결론 및 제언 언어치료 재평가 소위원회는 현재 평가 결과에 근거하여 다음과 같이 제언하였다. 언어치료의 안전성은 치료 관련 부작용 및 이상반응에 대한 우려가 낮아 안전한 기술이라고 평가하였다. 언어치료의 효과성은 무치료에 비해 언어능력 및 적응행동 개선에 도움이 되었으나 Active control과 비교하였을 때는 언어치료의 효과가 일관적이지 않은 것으로 확인하였다. 이와 관련하여 소위원회에서는 선택된 문헌에서의 언어발달지연 환자의 임상적 특징이 매우 다양하며, 언어치료의 효과를 판단하기에는 평가에 포함된 문헌 대부분의 연구대상자가 소규모인 점을 고려하여 언어발달지연 환자에서 언어치료의 효과에 대한 결론을 내리기 어렵다는 의견이었다. 2022년 제4차 의료기술재평가위원회(2022.04.15.)에서는 소위원회 검토 결과에 근거하여 다음과 같이 심의하였다. 언어발달지연 대상의 언어치료는 치료 관련 부작용 및 이상반응에 대한 우려가 낮은 안전한 기술이나 효과성에 있어 active control과 비교시 언어치료의 효과가 일관적이지 않고 선택문헌에서 제시된 환자의 임상적 특징이 다양하고 대부분의 연구대상자가 소규모인 점을 고려하여 의료기술재평가위원회는 언어발달지연 대상의 언어치료를 ‘불충분’으로 심의하였다(권고등급: 불충분).

주요어 언어치료, 언어발달지연, 안전성, 효과성

Background Speech and Language Therapy (SLT) is a professional service performed by speech-language pathologists for corrective treatment of patients diagnosed with language or speech disorders. The technology provides a treatment for difficulties with communication to enable expression of one’s messages or thoughts as well as support for occupational and social rehabilitation. It was listed as a non-benefit item in 2005 before the New Health Technology Assessment system was established At the 7th Health Technology Reassessment Committee (July 09, 2021) in 2021, in order to provide evidence in support of efficient utilization of healthcare resources, medical and scientific evidence for safety and effectiveness of SLT was reassessed through systematic review, and Grade of Recommendation was determined for the technology. According to the definition of reimbursable act by the Health Insurance Review and Assessment Service, health technology reassessment (HTR) was performed based on the four categories as follows: ① cranial nerve diseases such as stroke, traumatic brain injury, and cerebral palsy (hereinafter 'cranial nerve diseases'), ② dysarthria, dysphonia, stuttering, aphasia, and speech disorders due to hearing impairment. (hereinafter 'dysphonia, etc.'), ③ delayed language development (hereinafter 'delayed language development'), and ④ speech disorder patients with other special disabilities (hereinafter 'other special disabilities'). In this report, from the four categories, reassessment was performed for delayed language development.

Methods In order to perform reassessment of the safety and effectiveness of SLT in patients with delayed language development, a systematic review was performed. All assessment methods were finalized after review by the SLT Reassessment Sub-committee (Hereinafter Sub-committee) in consideration of the objectives of the assessment. The Sub-committee consisted of two experts in Rehabilitation Medicine, one in Neurology, one in Otolaryngology, one in Psychiatry, one in Pediatrics and one in Evidence-based Medicine. The key question of systematic review is “Is Speech and Language Therapy clinically safe and effective in patients with delayed language development?” In the process of systematic review, based on the key question, three international and five domestic databases were used for literature search and the articles were selected based on independent screening by two reviewers according to the inclusion/exclusion criteria for the related literature. The Cochrane’s Risk of Bias tool was used for assessment of risk of bias (RoB) of the literature and two reviewers independently carried out the assessment to draw a consensus. Data extraction was performed using a predefined format and independently by two reviewers, and in case of disagreement, the two reviewers held discussions with a third party to draw consensus on the matter. For synthesis of data, since quantitative synthesis was considered not possible due to high heterogeneity between instruments, qualitative review was applied. The level of evidence for the technology was assessed based on the Grading of Recommendations Assessment, Development and Evaluation (GRADE) method. At the 4th Health Technology Reassessment Committee (April 15, 2022) in 2022, the final Grade of Recommendation was determined in consideration of the result of the review by the Sub-committee.

Results The literature selected for the reassessment of SLT for patients with delayed language development was 5 articles on randomized controlled clinical trials, which included 3 articles on children with delayed language development, 1 article on children with autism spectrum disorder, and 1 article on children with phonological disorder. This HTR was conducted in an integrated manner without dividing by the conditions of the patients.

Safety We aimed to perform safety assessment of SLT in patients with delayed language development through treatment-related complications and adverse events based on the discussion of the Sub-committee, but there were no articles that reported safety outcomes. The Sub-committee presented a view that there was no problem with the safety in the usual practice of the SLT.

Effectiveness The effectiveness of SLT for patients with delayed language development was assessed by language ability, and adaptive behavior based on the discussion of the Sub-committee. Two out of five articles on delayed language development compared the outcomes of SLT with those of no-treatment (control). Language ability was reported in both of the two articles. In one article, language ability was reported by various indicators such as speech intelligibility and vocabulary size for children with delayed language development, and for all reported outcome measures, the SLT group showed significant improvement compared to the control group. In the other article, there were significant improvement in terms of the Percentage of Consonants Correct, phonological assessment, and Goldman-Fristoe Test of Articulation immediately after SLT for children with phonological disorder, but after 4 months of follow-up, the only outcome measure that retained the effect was Percentage of Consonants Correct. There was no significant difference between the two groups in terms of the mean length of utterance. For reporting of adaptive behavior, the Vineland adaptive behavior scales (VABS) were used as the indicator in one article, and adaptive behavior showed a significant improvement in the SLT group compared to the control group (p=0.003). Three out of five articles for delayed language development compared SLT with active controls (community service or family/guardian-based activities such as reading books). Language ability was reported in all of the three articles. In one article, there was a significant improvement in the social communication skills in early stage when compared with the community service immediately after the intervention of SLT for children with autism spectrum disorder, but there was no significant difference between the two groups in other reported outcome measures. At the time of 4-month follow-up, there was a significant improvement in the interaction between the caregiver and the patient among the social communication indicators when compared with the outcomes of community service, but there was no significant difference between the two groups in other reported outcome measures. One article reported significant improvements in receptivity domain, number of different word (NDW), and Peabody Picture Vocabulary Test among preschool language skills metrics when compared with outcomes of community service at 3 months of follow-up for children with delayed language development. However, there was no statistically significant difference between the two groups in other reported outcome measures. At 6 months of follow-up, there was no statistically significant difference between the two groups for all reported outcome measures, and at 12 months of follow-up, only the number of total utterances (NTU) showed a significant improvement but other reported outcome measures showed no statistically significant difference between the two groups. In the other article, for children with delayed language development, a group that received face-to-face advice and carried out family/caregiver-based activities such as book reading, a group that used an online device to carry out family/caregiver-based activities such as book reading, and a group that underwent SLT were compared. In terms of metrics for articulation and phonological testing, the SLT group showed a significant improvement compared to other comparative groups, but other reported measures such as “clinical evaluation of language fundamentals preschool”, Intelligibility in Context Scale (ICS), and preschool vocabulary and perception testing measures did no show statistically significant differences between the two groups. There was no reporting on indicators of adaptive behavior in the selected literature on delayed language development. The level of evidence was assessed considering the RoB assessment and heterogeneity, and it was confirmed as from ‘Very Low’ to ‘Low.’

Conclusion and Suggestions The SLT Reassessment Sub-committee suggested the following based on the results of the present assessment. SLT was assessed as a safe technology with a low risk of treatment-related complications and adverse events. In terms of the effectiveness of SLT, it was confirmed that the technology contributed to improvements in language ability and adaptive behavior compared to the outcomes of no-treatment group, but when compared with active controls, the effect of SLT showed mixed results. In relation to this inconsistency, the Sub-committee presented a view that the clinical characteristics of patients with delayed language development were widely varied in selected articles, and the number of participants in most of the studies included in the assessment was small to determine the effect of the SLT. The 4th Health Technology Reassessment Committee (April 15, 2022) in 2022 presented a final review as follows based on the results of the review by the Sub-committee. Therefore, although SLT for patients with delayed language development is a safe technology with a low risk of treatment-related complications and adverse events, when comparing its effectiveness with active controls, the effect of SLT was not consistent, and furthermore, considering that the clinical characteristics of patients presented in the selected articles were widely varied, and the number of participants in most of the studies included in the assessment was small, the Health Technology Reassessment Committee determined that the Grade of Recommendation of SLT for patients with delayed language development was “Insufficient” (Grade of Recommendation: Insufficient).

Keywords: SLT, Delayed language development, Safety, Effectiveness