언어치료[발성장애 등]

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 진행하였으며, 동 평가에서는 발성장애 등에 대한 재평가를 수행하였다.

평가 방법 발성장애 등 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 ‘발성장애 등 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 가능한 경우 메타분석을 수행하였고, 불가능한 경우 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 발성장애 등 환자에서 언어치료의 재평가에 선택된 문헌은 무작위배정 비교임상시험 14편으로 연구대상자별로 발성장애 11편, 유창성장애 2편, 청각장애 1편, 구음장애와 실어증 대상 문헌은 0편이었다.

안전성 발성장애 등 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 발성장애 등 환자에서 언어치료의 효과성은 소위원회의 논의를 바탕으로 연구대상자별 언어능력, 음성지표, 심리지표, 삶의 질로 평가하였다.

[발성장애] 발성장애 대상 문헌 11편은 모두 언어치료를 무치료와 비교하였다. 발성장애 대상 선택문헌에서 언어능력 결과를 보고한 문헌은 없었으며, 음성지표는 11편의 문헌에서 모두 보고되었다. 음성장애지수를 보고한 7편의 연구 중 5편을 양적합성한 결과 무치료군에 비해 언어치료군에서 유의한 개선이 확인되었다(mean difference (MD) 7.48; 95% CI 4.38, 10.59; I²=66%). 청지각적 평가를 보고한 7편의 연구 중 5편을 양적합성한 결과 무치료군에 비해 언어치료군에서 유의한 개선이 확인되었다(standardized mean difference (SMD) 1.06; 95% CI 0.24, 1.87; I²=90%). 지터를 보고한 연구에서도 무치료군에 비해 언어치료군에서 유의한 음성개선효과를 확인하였으며(5편, MD 0.76; 95% CI 0.38, 1.14; I²=61%), 시머를 기준으로 합성한 경우도 무치료군에 비해 언어치료군에서 유의한 개선이 확인되었다(5편, SMD 0.66; 95% CI 0.36, 0.95; I²=55%). 언어치료군의 최장 발성 시간도 무치료군에 비해 유의하게 향상되었다(5편, MD 4.17; 95% CI 2.81, 5.53; I²=71%). 다만, 메타분석 결과의 이질성이 높아 해석에 주의가 필요하다. 심리지표는 1편의 문헌에서 보고되었는데, 중재 전후 평균 변화에서 언어치료군과 무치료군 간 유의한 차이는 없었다. 삶의 질은 1편의 문헌에서 보고되었는데, 전반적으로 전후 평균 변화에서 언어치료군과 무치료군 간 유의한 차이는 없는 것으로 나타났다. 근거수준은 비뚤림위험 평가 및 이질성 등을 고려하여 평가하였으며, 모든 결과변수에서 ‘낮음’으로 확인되었다.

[유창성장애] 유창성장애 대상 문헌 2편은 모두 언어치료를 무치료와 비교하였다. 언어능력은 2편 중 1편의 문헌에서 보고하였는데, 중재 직후 언어치료군과 무치료군 간 언어능력에 유의한 차이를 보였다. 심리지표는 1편의 문헌에서 보고되었는데, 중재 직후와 2개월 후 시점에서 모두 언어치료군이 무치료군보다 말하기 불안 정도가 유의하게 개선되었다. 유창성장애 대상 선택문헌에서 음성지표와 삶의 질을 보고한 문헌은 없었다. 근거수준은 비뚤림위험 평가 및 이질성 등을 고려하여 평가하였으며, 모든 결과변수에서 ‘낮음’으로 확인되었다.

[청각장애] 청각장애 대상 문헌 1편은 언어치료를 active control과 비교하였다. 언어능력은 모든 척도에서 중재 1주일 후와 2개월 후에 언어치료군이 active control군보다 유의하게 개선된 것으로 나타났다. 심리지표 및 삶의 질 역시 모든 척도에서 중재 1주일 후와 2개월 후에 언어치료군이 active control군보다 유의하게 개선되었다. 해당 청각장애 대상 선택문헌에서 음성지표는 보고하지 않았다. 근거수준은 비뚤림위험 평가 및 이질성 등을 고려하여 평가하였으며, 모든 결과변수에서 ‘낮음’으로 확인되었다.

결론 및 제언 언어치료 재평가 소위원회는 현재 평가 결과에 근거하여 다음과 같이 제언하였다. 발성장애 등 환자를 대상으로 언어치료는 치료 관련 부작용 및 이상반응에 대한 우려가 낮은 안전한 기술이라고 평가하였다. 발성장애 등 대상으로 언어치료의 효과성은 대상자별로 다음과 같이 평가하였다. 구음장애 대상으로는 선택된 문헌이 없어 언어치료의 효과를 판단할 수 없었다. 발성장애 대상으로는 무치료와 비교하여 언어치료로 인한 유의한 음성지표 개선이 확인되어 언어치료가 효과적이라고 평가하였다. 유창성장애 대상으로는 무치료와 비교하여 언어치료로 인한 유의한 언어능력과 심리지표 개선을 확인할 수 있었으나 선택문헌이 2편이며 연구대상자가 소규모인 점을 고려하여 언어치료의 효과에 대한 결론을 내리기 어렵다는 의견이었다. 뇌졸중 외 원인으로 인한 실어증 대상으로는 선택된 문헌이 없어 언어치료의 효과를 판단할 수 없었다. 청각장애 대상으로는 active control과 비교하여 언어치료로 인한 유의한 언어능력 개선을 확인할 수 있었으나 선택문헌이 1편이며 연구대상자가 소규모인 점을 고려하여 언어치료의 효과에 대한 결론을 내리기 어렵다는 의견이었다. 2022년 제4차 의료기술재평가위원회(2022.04.15.)에서는 소위원회 검토 결과에 근거하여 다음과 같이 심의하였다. 언어치료는 모든 적응증에서 치료 관련 부작용 및 이상반응에 대한 우려가 낮은 안전한 기술로 심의하였다. 구음장애 환자에서는 선택된 문헌이 없어 언어치료의 효과를 판단할 수 없었다. 이에 의료기술재평가위원회는 구음장애 환자를 대상으로 언어치료를 ‘불충분’으로 심의하였다(권고등급: 불충분). 발성장애 환자에서는 무치료와 비교하여 언어치료로 인한 유의한 음성지표 개선을 확인하여 언어치료가 효과적이라고 판단하였다. 이에 의료기술재평가위원회는 발성장애 환자를 대상으로 언어치료를 ‘권고함’으로 심의하였다(권고등급: 권고함). 유창성장애 환자에서는 무치료와 비교하여 언어치료로 인한 유의한 언어능력과 심리지표 개선을 확인할 수 있었다. 이에 의료기술재평가위원회는 유창성장애 환자를 대상으로 언어치료를 ‘조건부 권고함’으로 심의하였다(권고등급: 조건부 권고함). 뇌졸중 외 원인으로 인한 실어증 환자에서는 선택된 문헌이 없어 언어치료의 효과를 판단할 수 없었다. 이에 의료기술재평가위원회는 뇌졸중 외 원인으로 인한 실어증 환자를 대상으로 언어치료를 ‘불충분’으로 심의하였다(권고등급: 불충분). 청각장애 환자에서는 active control과 비교하여 언어치료로 인한 유의한 언어능력 개선을 확인할 수 있었으나 선택문헌이 1편이며 연구대상자가 소규모인 점을 고려하여 언어치료의 효과에 대한 결론을 내리기 어렵다는 의견이었다. 이에 의료기술재평가위원회는 청각장애 환자를 대상으로 언어치료를 ‘불충분’으로 심의하였다(권고등급: 불충분).

주요어 언어치료, 발성장애, 유창성장애, 청각장애, 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 dysphonia, etc.

Methods In order to perform reassessment of the safety and effectiveness of SLT in patients with dysphonia, etc., 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 dysphonia, etc.?” 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. For data extraction, a predefined format was used and it was also conducted 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, when quantitative synthesis was possible, meta-analysis was performed and otherwise, qualitative review was performed. 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 selected literature for the reassessment of SLT in patients with dysphonia, etc. was 14 articles on randomized controlled trials. According to the patients participated in each study, there were 11 articles on dysphonia, 2 articles on stuttering, 1 article on hearing impairment, and no article on dysarthria and aphasia.

Safety We aimed to perform safety assessment of SLT in patients with dysphonia, etc. 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 in patients with dysphonia, etc. was assessed by language ability, voice parameters, psychological measures, and quality of life for different conditions of patients based on the discussion of the Sub-committee.

[Dysphonia] All of the 11 articles on dysphonia compared the outcomes of SLT with those of no-treatment (control). There was no literature that reported the outcome of language ability in the selected articles for dysphonia, and voice parameters were reported in all of the 11 articles. As a result of quantitative synthesis (meta-analysis) in 5 of the 7 studies reporting the Voice Handicap Index, a significant improvement was confirmed in the SLT group compared to the control group (mean difference (MD) 7.48; 95% CI 4.38, 10.59; I²=66%). As a result of quantitative synthesis of 5 out of 7 studies reporting auditory-perceptual evaluation, significant improvement was confirmed in the SLT group compared to the control group (standardized mean difference (SMD) 1.06; 95% CI 0.24, 1.87; I²=90%). In studies that reported jitter, an effect of significant improvement was confirmed in the SLT group compared to the control group (5 articles, MD 0.76; 95% CI 0.38, 1.14; I²=61%). Also, when results on shimmer were synthesized, significant improvement was confirmed in the SLT group compared to the control group (5 articles, SMD 0.66; 95% CI 0.36, 0.95; I²=55%). The maximum phonation time of the SLT group also showed a significant improvement compared to the control group (5 articles, MD 4.17; 95% CI 2.81, 5.53; I²=71%). However, due to the high heterogeneity of the meta-analysis results, caution is required in the interpretation of the results Psychological measures were reported in one article, and there was no significant difference between the SLT group and the control group in terms of the mean score change before and after the intervention. Quality of life was reported in one article. Overall, there was no significant difference between the Speech and Language TherapySLT group and the control group in terms of the mean score change before and after the intervention. The level of evidence was evaluated considering the risk of bias assessment and heterogeneity, and it was confirmed as 'Low' in all outcome measures.

[Stuttering] In both of the two articles on stuttering, outcomes of SLT were compared with those of no-treatment (control). Language ability was reported in one of the two articles, and there was a significant difference in language ability between the SLT group and the control group immediately after the intervention. Psychological measures were reported in one article, and at timepoints of both immediately after the intervention and after 2 months, the SLT group showed a significant improvement in terms of speech-related anxiety than the control group. There was no literature reporting voice parameters and quality of life in the selected literature for stuttering. The level of evidence was evaluated considering the RoB assessment and heterogeneity, and it was confirmed as 'Low' in all outcome measures.

[Hearing impairment] In one article on hearing impairment, outcomes of SLT were compared with those of active control. Language ability was significantly improved in all measures in the SLT group than the active control group after 1 week and 2 months after the intervention. Psychological measures and quality of life were also significantly improved in all measures in the SLT group than the active control group after 1 week and 2 months after the intervention. Voice parameters were not reported in the selected literature for hearing impairment. The level of evidence was assessed considering the RoB assessment and heterogeneity, and it was confirmed as 'Low' in all outcome measures.

Conclusion and Suggestions The SLT Reassessment Sub-committee suggested the following based on the results of the present assessment. For patients with dysphonia, etc., SLT was assessed as a safe technology with a low risk of treatment-related complications and adverse events. For patients with dysphonia, etc., the effectiveness of SLT was assessed for each type of patient as follows. There was no literature selected for patients with dysarthria, so the effectiveness of SLT could not be determined for this condition. For patients with dysphonia, a significant improvement in voice parameters was confirmed due to SLT compared to the no-treatment group; thus, SLT was assessed to be effective for this condition. For patients with stuttering, significant improvements in language ability and psychological measures were confirmed due to SLT compared to the no-treatment group; however, since there were only two articles selected and the number of participants was small, the Sub-committee considered that it was difficult to draw a final conclusion on the effectiveness of SLT for stuttering. For patients with aphasia from causes other than stroke, no articles were selected; thus, it was not possible to determine the effectiveness of SLT for this condition. For patients with hearing impairment, a significant improvement in language ability was confirmed due to SLT compared to the active control; however since there was only one article selected and the number of participants was small, the Sub-committee considered that it was difficult to draw a final conclusion on the effectiveness of SLT for hearing impairment. 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. SLT was considered a safe technology with a low risk of treatment-related complications and adverse events in all indications. For patients with dysarthria, the effectiveness of SLT could not be determined because there was no selected literature. Therefore, the Health Technology Reassessment Committee determined that the Grade of Recommendation for SLT was ‘Insufficient’ for patients with dysarthria (Grade of Recommendation: Insufficient). For patients with dysphonia, compared with no-treatment group, a significant improvement in voice parameters was confirmed following SLT, and therefore, SLT was considered to be effective. Thus, the Health Technology Reassessment Committee determined that the Grade of Recommendation for SLT was ‘Recommended’ for patients with dysphonia (Grade of Recommendation: Recommended). For patients with stuttering, compared with the no-treatment group, significant improvements in language ability and psychological measures were confirmed due to SLT. Accordingly, the Health Technology Reassessment Committee determined that the Grade of Recommendation for SLT was ‘Conditional Recommendation’ for patients with stuttering (Grade of Recommendation: Conditional Recommendation). For patients with aphasia due to causes other than stroke, there was no selected literature; thus, the effectiveness of SLT could not be determined. Therefore, the Health Technology Reassessment Committee determined that the Grade of Recommendation for SLT was ‘Insufficient’ for patients with aphasia due to causes other than stroke (Grade of Recommendation: Insufficient). For patients with hearing impairment, a significant improvement in language ability was confirmed due to SLT compared with the active control, but considering the fact that there was only one literature selected and the number of participants was small, the Sub-committee considered that it was difficult to draw conclusions about the effectiveness of SLT. Accordingly, the Health Technology Reassessment Committee determined that the Grade of Recommendation for SLT was ‘Insufficient’ for patients with hearing impairment (Grade of Recommendation: Insufficient).

Keywords: Speech and Language Therapy, Dysphonia, Stuttering, Hearing Impairment, Safety, Effectiveness