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비디오투시연하검사를이용한 Gugging swallowing screen 과기타 연하곤란선별검사와의비교 연세대학교대학원 의학과 송원우

비디오투시연하검사를이용한 Gugging swallowing screen 과기타 연하곤란선별검사와의비교 지도교수신지철 이논문을석사학위논문으로제출함 29 년 6 월 연세대학교대학원 의학과 송원우

송원우의석사학위논문을인준함 심사위원 인 심사위원 인 심사위원 인 연세대학교대학원 29 년 6 월

감사의글 본논문의계획과진행, 완성에큰기여를하신재활의학과은사이신신지철교수님께감사를드립니다. 또한김덕용교수님의따끔한훈계가이논문을보다좋은방향으로이끌었고이에깊은감사를드립니다. 신경과허지회교수님께도많은감사를드리고싶습니다. 케이스를모으는데많은도움을준이숙희선생님에게도깊은감사를드립니다. 논문의내용과는상관없지만부모님의따뜻한격려가큰도움이되었습니다. 형님과누님의조언도감사합니다. 무엇보다아내의격려와내조가이논문을집필하는데있어좋은밑거름이되었습니다. 그외에도움을주신모든분께진심으로감사를드립니다. 송원우씀

< 차례 > 국문요약 1 Ⅰ. 서론 3 II. 대상및방법 1. 연구대상 7 2. 연구방법 8 가. 연하곤란선별검사 9 (1) Gugging swallowing screen (GUSS) (2) 3온스물연하검사 (3 ounce water swallowing test) (3) Burke dysphagia screening test (BDST) (4) Standardized swallowing assessment (SSA) 나. 비디오투시연하검사 11 (1) Functional dysphagia scale (FDS) (2) Penetration-aspiration scale (P/A 척도 ) 다. 연하곤란의임상척도 12 (1) ASHA NOMS 연하척도 (2) 연하곤란임상척도 (Clinical dysphagia scale) 라. 자료의분석 13 III. 결과 14 1. 시행한연하곤란선별검사들의분석 2. 비디오투시연하검사와 GUSS의상관분석 3. 연하곤란의임상척도와 GUSS의상관분석 IV. 고찰 19 - i -

V. 결론 27 참고문헌 28 영문요약 32 부록 34 - ii -

그림차례 Fig. 1. Receiver operating characteristic curves for the GUSS as a predictor of aspiration risk in stroke patients. (n=37) 15 Fig. 2. Correlations between Gugging swallowing screen (GUSS) and Functional dysphagia scale (FDS).17 Fig. 3. Correlations between Gugging swallowing screen (GUSS) and Penetration-aspiration scale 17 Fig. 4. Correlations between Gugging swallowing screen and American Speech-Language-Hearing Association National Outcome Measurement System (ASHA NOMS) swallowing scale 18 Fig. 5. Correlations between Gugging swallowing screen (GUSS) and Clinical dysphagia scale (CDS)..18 - iii -

표차례 Table 1. General characteristics of the subjects 8 Table 2. Coordinates of the Receiver operating characteristic curves 15 Table 3. Sensitivity, specificity and predictive values for the screening tests 16 Appendix 1. Gugging Swallowing Screen (GUSS) 34 Appendix 2. the Burke Dysphagia Screening Test (BDST) 35 Appendix 3. Standardized Swallowing Assessment(SSA) 36 Appendix 4. The Functional Dysphagia Scale (FDS) Based on Videofluoroscopic Swallowing Study 37 Appendix 5. 8-Point Penetration-Aspiration Scale 38 Appendix 6. The American Speech-Language Hearing Association National Outcomes Measurements System (ASHA NOMS) Swallowing Scale 39 Appendix 7. Clinical Dysphagia Scale (CDS) 4 - iv -

< 국문요약 > 비디오투시연하검사를이용한 Gugging swallowing screen 과기타 연하곤란선별검사와의비교 뇌졸중후연하곤란은뇌졸중환자에게폐렴, 패혈증, 기도폐색, 사망등의심각한합병증을초래할수있다. 뇌졸중환자의재활과정중연하곤란에의한합병증을예방하기위해서는연하곤란선별검사가도움이될수있어다양한선별검사법이소개되고있다. 이중최근에개발된 Gugging swallowing screen(guss) 은발병후 24시간이내의뇌졸중환자에서타당성이증명된바있고, 타선별검사와같이통과 (PASS) 및탈락 (FAIL) 만을보는것이아니라여러식이를시도하고점수화하여연하곤란의정도를단계화하여볼수있는장점이있다. 본연구에서는 GUSS와함께여러가지연하곤란선별검사를실시하고, 비디오투시연하검사를통해흡인여부등을확인하여연하곤란선별검사로서유용성을확인하고다른연하척도와의상관관계를알아보아연하곤란임상척도로서의가능성을확인하고자한다. 대상및방법. 재활병원에입원한뇌졸중을진단받은후 3개월이상지난 37명의환자를대상으로하였다. 침상에서선별검사인 GUSS, 3 온스물연하검사, Burke dysphagia screening test(bdst), Standardized swallowing assessment(ssa) 를실시하였고, 24시간이내에비디오투시연하검사를실시하여흡인여부를알아보고, 이를기준으로각각의선별검사의민감도와특이도등을측정하였고, 흡인이있는군과없는군에서의각선별검사의통과-탈락양상을 chi-square test를통해분석하였다. 비디오투시연하검사를통해얻은기능적연하곤란척도 (Functional dysphagia scale, FDS) 및 Penetration-aspiration scale(p/a 척도 ) - 1 -

와임상척도인 ASHA NOMS 연하척도 (American Speech-Language-Hearing Association National Outcome Measurement System swallowing scale) 및연하곤란임상척도 (clinical dysphagia scale, CDS) 를구하여 GUSS와의상관관계를살펴보았다. 결과. GUSS는비디오투시연하검사에서의흡인여부를기준으로했을때민감도 9.9% 특이도 69.2% 였고, 3온스물연하검사는민감도와특이도가 81.8%, 76.9% 였으며, BDST의경우 9.9%, 61.5%, 그리고, SSA의경우 9.9%, 61.5% 였다. 또한각선별검사의통과-탈락양상은흡인이있는군과없는군간에모두의미있는차이를보였다 (p<.1). GUSS 점수는비디오투시연하검사로부터구한 FDS점수와의미있는상관 관계를보였고 (r=-.594, p<.1), P/A 척도와 상관계수 -.747 (p<.1) 로유의한상관관계를보였고, 임상척도인 ASHA NOMS 연하척도 (r=.432 p<.1), CDS (r=-.899, p<.1) 와도유의한상관관계를보였다. 결론. 뇌졸중재활환자를대상으로한본연구에서 GUSS는다른연하곤란선별검사와비교했을때민감도와특이도에서다른선별검사와동등한양상을보여주었고, 비디오투시연하검사와임상적인연하곤란척도와도의미있는상관관계를보여 GUSS는부가적으로연하곤란정도를반영하는임상척도로의이용가능성을확인할수있었다. 핵심되는말 : dysphagia, swallowing screen, videofluoroscopic swallowing study - 2 -

비디오투시연하검사를이용한 Gugging swallowing screen 과 기타연하곤란선별검사와의비교 < 지도교수신지철 > 연세대학교대학원의학과 송원우 I. 서론 뇌졸중후연하곤란의발생빈도는연구마다차이가있으나 3% 에서 51% 에서나타나는것으로보고되고있다. 1, 2 연하곤란의증상중 에흡인에국한했을때는 2% 정도에서발생하는것으로보고된바 있다. 3 뇌졸중후의연하곤란은영양결핍이나탈수를발생시킬수있 을뿐만아니라흡인에의해폐렴, 패혈증, 기도폐색, 사망등의심 1, 4, 5 각한합병증을초래할수있다. 뇌졸중 이후에발생하는연하곤란 은대개한달내에스스로회복되나 6, 6 개월이후에도 11% 의환자에서 지속되는것으로알려져있다. 2 뇌졸중환자에서연하곤란양상을일 찍발견하고, 음식농도조절이나보상적인연하기술등의사용을하 게되면연하곤란과연관된합병증의발생이감소하는것으로알려져 있다. 7 이와같이뇌졸중환자의재활에있어연하곤란, 특히흡인의 판별은매우중요한문제이며, 뇌졸중환자가급성기치료를마치고, - 3 -

재활병원으로전원되는시점에도연하곤란에대해제대로평가되지않은경우가드물지않기때문에재활의학과에서도효율적인연하곤란선별검사가필요한실정이다. 여러가지연하곤란을위한선별검사들이개발되어임상에서사용되고있는데, 3온스물연하검사 (3-ounce water swallow test) 8, Standardized swallowing assessment( 이하 SSA) 9, Burke dysphagia screening test( 이하 BDST) 1 등이널리사용되고 있다. 3온스물연하검사의경우, 물에대한흡인만을확인하는검사로, 연하곤란을일으킬수있는다른여러요소들을간과하는면이있다. BDST의경우, 가장널리쓰이는검사의하나로병력과식사시간등의항목이추가되어 3온스물연하검사를보완하는면이있으나널리 쓰이는것에비해타당성의검증이제대로안된면이있다. 11 SSA 의 경우환자의기본적인상태를먼저확인한후, 물연하검사를단계적으로하는등 GUSS와비슷한면이있다. 하지만 SSA의경우민감도 (47~68%) 와특이도 (67~86%) 가연구에따라큰편차를보인다. 12 Gugging Swallowing Screen( 이하 GUSS) 은 Trapl 등 13 이개발한뇌졸중환자를위한연하곤란선별검사로사용하기편하면서도, 점수화되어있어단계화된평가치를보여줄수있기때문에, 대부분의 통과 (PASS) 와 탈락 (FAIL) 으로평가하는연하곤란선별검사에비해보 - 4 -

다많은임상적인정보를줄수있다. Trapl 등 13 은 GUSS를뇌졸중발생후 24시간이내의환자를대상으로광섬유내시경연하검사 (Fiberoptic Endoscopic Evaluation of Swallowing) 에서의흡인여부를기준으로 GUSS의절단점을구하고, 이에따른민감도와특이도를구하였고, 검사자간신뢰도등을보여주면서 GUSS가급성기의뇌졸중환자의흡인의선별검사에타당함을보여준바있다. 비디오투시연하검사는해부학적, 기능적문제를방사선투시를통해시각화하여직접확인할수있고, 흡인의여부를신뢰성높게평가할수있어뇌졸중후연하곤란의평가에광섬유내시경연하검사와함께표준적인검사로여겨지고있으며널리사용되고있다. 14 하지만, 비디오투시연하검사는방사선실에환자가가서검사를해야하고, 방사선투시검사장비가필요하며, 검사할때마다방사선에환자가노출되는등의단점이있다. 이와같은이유로모든뇌졸중환자를비디오투시연하검사를시행하는데에는어려움이있는데, 연하곤란선별검사는뇌졸중환자에서비디오투시연하검사나광섬유내시경연하검사의실시여부의결정에도도움을줄수있다. 연하곤란선별검사는대부분제한적인항목만을검사하는것으로, 대부분통과 (PASS) 와탈락 (FAIL) 으로평가하게되는데비해, 임상척도는보다포괄적으로연하기능을평가하고, 이를정량적으로보여줄 - 5 -

수있다. GUSS는연하곤란선별검사이나점수화된검사호서점수에따라현재연하곤란의정도를반영하고있으며, 반고형식, 액체식, 고형식등, 여러가지식이를시도하여점수가채점된다. 각식이에대한연하곤란의양상을다각적으로측정하고이를점수화하는 GUSS의특성상연하곤란임상척도로서의사용도가능할것으로생각된다. 만약비디오투시연하건사의척도나다른연하곤란임상척도들과상관성이높다면 GUSS의임상척도로서의사용가능성을더높여줄것으로생각된다. 본연구에서는 GUSS와다른연하곤란선별검사를재활병원에입원한뇌졸중환자에실시하여민감도와특이도등을비교하여 GUSS의기존선별검사와의차이점및선별검사로의타당성을알아보고, GUSS의점수와연하곤란의임상적척도와비디오투시연하검사로측정한기능적연하곤란척도 (Functional dysphagia scale, 이하 FDS) 점수와의상관관계를확인하여 GUSS의임상척도로의이용가능성을확인하고자하였다. - 6 -

II. 대상및방법 1. 연구대상 28년 11월에서 29년 4월사이에재활병원에입원하여비디오투시연하검사가의뢰된뇌졸중발병 3개월이상지난환자를대상으로하였다. 뇌자기공명영상이나뇌전산화단층촬영술을통해뇌졸중으로확진된환자만을포함시켰고, 선별검사및비디오투시연하검사를시행할수없을정도로인지기능이저하된환자, 즉 1단계구두명령을따를수없는환자나검사를거부하는환자는연구대상에서제외하였다. 대상환자는총 37명이었고평균나이는 6.3세로남자는 29명여자는 8명이었다. 뇌경색 23명, 뇌출혈 14명이었으며, 그중뇌간에병변이있는환자는 6명이었다. 환자식이상태는일반식이 13명, 연하곤란식이 19명, 관급식이 5명이었다. 총 37명중 6명 (16.2%) 은기도삽관상태였다 (Table 1). - 7 -

Table 1. General characteristics of the subjects Characteristics Value Mean Age (years) 6.3 ±11.2 (34-83) Median Time since Stroke 6.85 (3.1-17.2) (months) Gender (n) Male 29 Female 8 Etiology of stroke (n) Hemorrhage 14 Infarction 23 Feeding route (n) Normal diet 13 Dysphagia diet 19 Tube diet 5 Tracheostomy status (n) Yes 6 No 31 2. 연구방법대상환자들에게침상에서동일한날에연하곤란선별검사인 GUSS와 3온스물연하검사, BDST, SSA를실시하였다. 그외에연하곤란의임상적척도인연하곤란임상척도 (Clinical dysphagia scale), ASHA NOMS 연하척도를연하곤란선별검사를시행한검사와다른검사자가실시하였다. 선별검사및임상척도를시행전후 24시간이내에다른재활의학과의사가비디오투시연하검사를실시하였고, 이검사를기반으로흡인의여부및 FDS, P/A 척도를측정하였다. 이때연하곤란선별검사와임상척도검사자및비디오투시연하검사를실시하는검사자간에는서로결과를알지못했다. - 8 -

가. 연하곤란선별검사 (1) Gugging swallowing screen (GUSS) GUSS는간접연하검사와직접연하검사의두단계로구성되어있고, 간접연하검사는환자본인의타액을잘삼키고, 검사를수행할만큼의식이있는지, 스스로기침을할수있는지확인하는절차이다. 직접연하검사는반고형식, 액체식, 고형식순으로 3가지식이형태의세부단계가있으며, 각식이형태에따라흡인의징후가있는지확인하는절차이다. 간접및직접연하검사의각세부단계에서문제가있는경우, 그세부단계에서검사를멈추고채점을하게된다. 각세부단계는 5점척도로구성되는데, 점수가낮을수록연하곤란이심함을의미한다. 흡인의징후인삼킴의지연, 기침, 침흘리기, 음성의변화 4가지를점수화하여합산하게된다. 각세부단계를순서대로시행하면서한세부단계에서문제가있으면그다음단계로넘어가지않고, 그세부단계까지의점수로합산하게된다. 각세부항목을시행하면서간접검사와직접검사의 3가지세부검사의점수를모두더하면 2점이되는데, 어떤세부항목에도이상이없는경우 2점이되고연하곤란이심할수록점수가줄어들게된다 (Appendix 1). 이전논문에서절단점을 14점으로정하였으며, 14점이하에서는선별검사상탈락 (FAIL) 이고, 흡인의가능성이높음을의미하며, 14점초과의경우통과 (PASS) 에해당한다. - 9 -

(2) 3온스물연하검사 (3 ounce water swallowing test) 15 대상자에게 3온스의물을컵에담아주고멈추지않고마시라고지시한다. 물을마시는중에혹은다마신후 1분이내에반사성기침을하거나물을마신후젖은음성이관찰되면비정상으로채점된다. 15 (3) Burke dysphagia screening test (BDST) 1 BDST는 7가지독립된항목으로구성되는데, 항목 1~3은주로환자의연하곤란과관련된병력에관계되고, 항목 4~7은직접적인관찰에의해측정된다. 7가지항목중하나이상의항목에해당되면비정상으로채점된다. 이때항목 4는 3온스의물을마시는것으로 3온스물연하검사와거의동일하다 (Appendix 2). (4) Standardized swallowing assessment (SSA) 16 SSA는 3단계로구성된다. 1단계에서는환자가선별검사자체를받을수있는신체적상태인지확인하는두가지질문이며, 환자가지시에따를수있고, 목을가누며앉을수있어야한다. 이조건들을만족하지못하면검사를중단한다. 2단계에서는환자의기능을평가하는것인데, 환자가기침을할수있는지, 타액을조절할수있는지, 아래위입술을빨수있는지, 호흡을잘하는지등을확인하는것이다. 이들중하나라도할수없으면검사를중단한다. 3단계에서는물연하검사를실시하는데, 물 1티스푼을주고기침이나, 질식, 무호흡, - 1 -

젖은목소리등의변화가없는지살펴보고문제가없으면두번째, 세번째티스푼을시도한다. 그래도문제가없다면, 물반컵을마시게한후살펴보게된다. 이과정중하나라도문제가있다면비정상으로측정된다 (Appendix 3). 나. 비디오투시연하검사검사를위해 Logeman의프로토콜을일부변형하여검사하였다. 17 희석된바륨 2mL를두번마시게하고, 희석된바륨 5mL를추가로마시게하면서측면상을방사선투시검사 (Winscope 6, TOSHIBA co., JAPAN) 를통해확인하였다. 점도와재질이표준화된요구르트, 죽, 밥등과같은식이에바륨을섞어동일한검사를반복하여시행하였다. 디지털비디오로녹화된영상을보면서환자의연하상태, 특히흡인의여부를평가하였다. 비디오투시연하검사를시행하는중에한가지의식이에서도성대이하로음식이넘어가는것으로관찰되면흡인이있는것으로확인하였다. (1) 기능적연하곤란척도 (Functional dysphagia scale, FDS) 비디오투시연하검사에기록된환자의연하곤란상태를객관적으로정량화하기위해 Han 등이개발한 FDS를이용하였다 (Appendix 4). 18 Han 등의연구에서타당도가입증된이후, 연하곤란의치료의성과를추적관찰하기위한도구로서쓰이고있다. 19, 2 FDS는비디오투시연하 - 11 -

검사에의해관찰된연하의구강단계의소견과인두단계의소견을항목별로점수화하여흡인에대한상대적인영향정도를고려하여만들어진점수체계로서 1점이만점이며점수가적을수록연하곤란이심하고, 흡인의위험이높음을의미한다. (2) 8 point penetration- aspiration scale (P/A 척도 ) P/A 척도는비디오투시연하검사를통해음식물과성대와의위치관계, 음식물이다시나오는지, 그대로머물러있거나들어가는지등 에따라 8 단계로나눈척도이다. 21 여러연하곤란연구에서비디오투 시연하검사결과의기술에널리사용되고있고, 신뢰도가입증된척 도이다 (Appendix 5). 22 다. 연하곤란의임상척도연하곤란의선별검사와달리현재환자의연하곤란정도를임상적으로파악하여이를점수화하여나타내는척도들이다. (1) ASHA NOMS 연하척도 (American Speech-Language-Hearing Association National Outcome Measurement System swallowing scale) 23 미국언어청각협회 (American Speech-Language-Hearing Association) 에서개발된연하척도로 7단계로구성되어비경구식이의유무, 독립적인식이가가능한지여부, 식이를위한보상적인기술의사용, 식이의제한등, 환자가가지고있는연하곤란의양상을경중에따라단계별로 - 12 -

나타낸척도이다. 7단계일때정상식이를의미하며단계가낮아질수록연하곤란의상태가심함을의미한다 (Appendix 6). (2) 연하곤란임상척도 (Clinical dysphagia scale, CDS) 24 연하곤란과관계된임상소견의각항목에비디오투시연하검사상흡인여부와연관하여상대적가중치를부여하여만든척도이다. 1점이만점으로점수가높을수록연하곤란이심함을의미하게된다 (Appendix 7). 라. 자료의분석연하곤란의선별검사인 GUSS, 3온스물연하검사, BDST, SSA의민감도와특이도를구하였다. 이때보편적인표준검사로는비디오투시연하검사의흡인여부를기준으로하였다. 특히, GUSS의경우절단점을구하기위해 ROC 곡선을사용하였고, 이에따른민감도와특이도를구하였다. 각선별검사는통과-탈락양상을흡인이있는군과없는군으로나누어 chi-square test를실시하였다. GUSS와비디오투시연하검사와의연관성을보기위해 GUSS와 FDS를 Pearson 상관분석을시행하였다. GUSS와임상적척도간의연관성을보기위해 GUSS와 CDS는 Pearson 상관분석을실시하였고, GUSS와 ASHA NOMS 연하척도, P/A 척도사이에는 Spearman's rho 분석을사용하였다. 통계프로그램은 SPSS version 15.을이용하였다. - 13 -

III. 결과 1. 연하곤란선별검사상비교비디오투시연하검사의흡인여부를기준으로 3온스물연하검사의경우민감도 81.8%, 특이도 76.9% 이었고, BDST의경우민감도 9.9%, 특이도 61.5% 이었고, SSA는민감도 9.9%, 특이도 61.5% 이었다 (Table 3). GUSS는민감도 9.9% 특이도 69.2% 이었고, ROC 곡선에서곡선하면적은.92이었다 (Fig. 1). 본연구에구한 ROC 곡선의각절단점에서의좌표는 Table 2와같다. GUSS를포함한각선별검사의통과-탈락양상은흡인군과무흡인군간에의미있는차이를보였다 (p<.1, Table 3). - 14 -

1..8 Sensitivity.6.4.2...2.4.6 1 - Specificity.8 1. Fig. 1. Receiver operating characteristic curves for the GUSS as a predictor of aspiration risk in stroke patients. (n=37) Table 2. Coordinates of the Receiver operating characteristic curves GUSS Sensitivity 1 - Specificity 3... 6.5.636. 11..727. 13.5.818. 16.5.99.38 19.5.99.5 21. 1. 1. *GUSS : Gugging swallowing screen - 15 -

Table 3. Sensitivity, specificity and predictive values for the screening tests Videofluoroscopic swallowing study aspiration no aspiration 3oz water test PASS 2 2 NPV=9.9% FAIL 9 6 PPV=6.% sensitivity=81.8% specificity=76.9% *p=.1 Burke dysphagia screening test PASS 1 16 NPV=94.1% FAIL 1 1 PPV=5.% sensitivity=9.9% specificity=61.5% *p=.3 Standardized swallowing assessment PASS 1 16 NPV=94.1% FAIL 1 1 PPV=5.% sensitivity=9.9% specificity=61.5% *p=.3 Gugging swallowing screen > 14 1 18 NPV=94.7% 14 1 8 PPV=55.5% sensitivity=9.9% specificity=69.2% *p=.1 NPV: negative predictive value PPV: positive predictive value *p : p-value of Pearson s chi-square test - 16 -

3. 비디오투시연하검사와 GUSS 의상관분석 GUSS 와비디오투시연하검사로부터구한 FDS 점수를 Pearson 상 관분석을실시한결과 r= -.594 (p<.1) 의유의한음의상관관계가보 였다 (Fig. 2). GUSS 와 P/A 척도를 Spearman's rho 상관분석결과 r= -.747 (p<.1) 로유의한상관관계가보였다.(Fig. 3.) 6. r= -.594, p<.1 5. 4. FDS 3. 2. 1... 5. 1. GUSS 15. 2. Fig. 2. Correlations between Gugging swallowing screen (GUSS) and Functional dysphagia scale (FDS). 8. r= -.747, p<.1 7. Penetration-Aspiration Scale 6. 5. 4. 3. 2. 1.. 5. 1. GUSS 15. 2. Fig. 3. Correlations between Gugging swallowing screen (GUSS) and Penetration-aspiration scale - 17 -

3. 연하곤란의임상척도와 GUSS의상관분석 GUSS와임상척도인 CDS를상관분석을실시한결과 r=-.899(p<.1) 의음의상관관계가보였고 (Fig. 4), ASHANOMS 연하척도간에상관계수가.432로유의한상관관계가보였다 (p<.1, Fig. 5). 7. r=.432, p<.1 6. ASHA NOMS swallowing scale 5. 4. 3. 2. 1.. 5. 1. GUSS 15. 2. Fig. 4. Correlations between Gugging swallowing screen and American Speech-Language-Hearing Association National Outcome Measurement System (ASHA NOMS) swallowing scale 8. r=-.899, p<.1 6. CDS 4. 2... 5. 1. GUSS 15. 2. Fig. 5. Correlations between Gugging swallowing screen (GUSS) and Clinical dysphagia scale (CDS). - 18 -

IV. 고찰 기도흡인의유무와관계없이연하곤란이있는뇌졸중환자는흡 인성폐렴이생길가능성이연하곤란이없는뇌졸중환자에비해 3 배 높아진다. 25 특히, 기도흡인이있는뇌졸중환자의경우연하곤란이 없는뇌졸중환자에비해흡인성폐렴이생길확률이 11 배정도로높 아진다. 25 그러므로뇌졸중환자에서연하곤란중특히기도흡인을 찾아내는것은흡인성폐렴과같은합병증의발생을줄이기위해매우중요하며, 본연구에서는연하곤란의증상중비디오투시연하검사상흡인을표준검사로연구를진행하였다. 대상환자를뇌졸중발병 3개월이후로한이유는이미기존연구에서뇌졸중발생후급성기에서는 GUSS의타당성이확인된바있으므로, 주로재활병원에입원하는시기인아급성기의뇌졸중환자를대상으로삼고자하였다. 13 본연구에서 GUSS 및 BDST, SSA는흡인을기준으로할때모두 9% 가넘는우수한민감도를가지고있었으나, 3온스물연하검사만이 82% 로상대적으로낮은민감도를보여주었다. 특이도의경우, BDST나 SSA보다 GUSS에서경미하게높은경향을보였고, 3 온스물연하검사가높게나타났다본연구에서 3온스물연하검사만다른세가지선별검사에비해 - 19 -

민감도가상대적으로낮은이유로는다른세검사가액체식의연하를평가하기위한부분으로 3온스물연하검사의검사내용을거의포함하면서다른선별항목들이부가적으로포함되어있기때문에 3온스물연하검사의민감도가상대적으로더낮게나타난것으로보인다. Trapl 등 13 의논문에서는급성뇌졸중환자를대상으로광섬유내시경연하검사에서의흡인여부를기준으로 GUSS를실시한결과절단점 14에서민감도 1%, 특이도 5~69% 였다. 본연구에서는민감도 9.9%, 특이도 69.2% 였는데, 민감도는이전연구에비해좀더낮았으나, 특이도의경우조금높거나비슷하였다. 이전논문에서는흡인의확인을광섬유내시경연하검사로하였고, 본연구에서는비디오투시연하검사를이용하였으므로, 표준검사의차이가있다. 표준검사의차이는민감도와특이도에영향을미칠수있지만, 비디오투시연하검사와광섬유내시경검사어느한쪽이기도흡인을판단하는데우월하다고확실히밝혀지지는않은상황이다. 14 본연구에서 GUSS는 ROC 곡선에서곡선하면적이.92로높게나와상당히우수한선별검사가될수있음을보여주었다 (Fig. 1). 이전논문에서의 GUSS의 ROC 곡선에서도곡선하면적이.77 ~.93이어서본연구와비슷한양상을보여주었다. 이전논문에서제안된 14점은본연구에서산출된 ROC곡선에서도각절단점에따른민감도, 특이 - 2 -

도변화양상을고려해도 ROC곡선의좌측상단에위치하여, 민감도가특이도보다상대적으로높아절단점으로채택하기에적당한값으로생각된다 (Table 2, Fig.1). 3온스물연하검사의경우 DePippo등의연구에서뇌졸중환자를대상으로비디오투시연하검사에서의흡인을표준검사로했을때민 감도 76%, 특이도 59% 를보였다. 15 Suiter 등이뇌졸중, 파킨슨병, 두경 부암, 치매등다양한진단명의환자군이포함된연구에서는광섬유 내시경연하검사를표준검사로했을때민감도 96.5%, 특이도 48.7% 였다. 8 Chong 등의뇌졸중노인환자를대상으로한연구에서는광섬 유내시경연하검사를기준으로했을때, 민감도 79.4%, 특이도 62.5% 였다. 26 본연구에서는각각 81.8%, 76.9% 로나와민감도는비슷했고, 특이도가좀더높게나타났다. 기존연구들에서도민감도나특이도가연구마다차이가있다는점이고려되어야할것으로보인다. BDST의경우, 흡인을기준으로한민감도와특이도를구한연구는없었으나, 뇌졸중에이후발생되는흡인성폐렴을표준으로한연구 에서는민감도 1%, 특이도 1% 를보였다. 16 또다른연구에서는 BDST 에양성인경우, 폐렴과질식등이발생하는상대위험도 (relative risk) 가 7.65 나되는것으로알려져있다. 1 BDST 는흡인을표준검사 로한연구가없어서본연구와직접적인비교는어렵다. 하지만, - 21 -

BDST는임상적으로가장흔히쓰이는선별검사이기때문에본연구에포함시킬필요가있었다. 27 SSA는급성기뇌졸중환자에대한연구에서비디오투시연하검사의흡인을표준검사로했을때치료사의경우민감도 47% 특이도 86% 를보였고, 의사의경우, 민감도 68%, 특이도 67% 를보였다. 2 SSA 는평가자간신뢰도가치료사와의사사이에는 kappa=.24~.48, 의사간에는 kappa=.5로일치도가낮게측정되었다. 본연구에서는상대적으로민감도는높았고, 특이도는비슷한양상을보였다. 이전연구에서도 SSA가검사자간민감도의차이가상당히나는것과평가자간신뢰도가낮은것을고려할때, SSA가검사자에따라결과의편차가상당히있는검사인것으로보인다. 본연구에서는같은환자군을대상으로여러가지선별검사를실시하였기때문에단순히여러다른논문에서산출된민감도와특이도등을통해다른선별검사들을비교하는것에비해검사간의비교라는측면에있어서는의미가있었다. 과거여러연구에서선별검사들을 서로비교하여발표한바있지만 12, 16, 연구형태는체계적분석 (systemic review) 이었고, 동일환자군을대상으로한연구가아니었고, 본연구와같이동일환자군을대상으로여러선별검사를실시하여 비교한경우는없었다. - 22 -

GUSS의경우, 본연구에서 BDST나 SSA와같은수준의민감도를가지면서도상대적으로약간더높은특이도를보여주었다. GUSS가다른선별검사와동등한정도의민감도와특이도를보인다는사실은단지선별검사의목적만으로는다른선별검사를대체하여, 반드시 GUSS를써야할타당성은없는것으로생각된다. 특히검사의편이성과용이성의측면을생각해보면 GUSS의경우, 액체식, 반고형식, 고형식등을모두시도해야할수가있기때문에시간이더많이걸릴수있고, 빵과식품점증제 (food thickener) 등의준비물이필요한데비해, 3온스물연하검사의경우물만있으면검사가가능하고, 걸리는시간도훨씬적다는장점이있다. 민감도의차이에서보이는선별검사로서의우월성에비해검사의편이성은 GUSS가더떨어져, 단지연하곤란선별검사만을목적으로한다면보다간단한선별검사들을사용하는것도고려해볼필요가있겠다. 물론 GUSS의경우, 여러개의세부검사들이단계로이루어져이전의검사를통과하지못하면, 다음검사로넘어가지못하므로기본적인연하가이루어지지않는환자에게무리하게물연하검사를하지않아도되는장점이있다. 즉, 침도제대로삼킬수없는환자의경우에는 3온스물연하검사나 BDST의경우, 물 3온스를먹여봐야하지만, GUSS는그상태에서검사를종료하게되어, 무리한물연하검사를피하게된다. 그러므로경 - 23 -

우에따라서는 3온스물연하검사보다더간단하게시행되는수도있다. 본연구에서는 GUSS를실시할때, 이전의논문과같이고형식으로빵을사용하였다. 우리나라에서는고형식으로빵보다는밥을먹는먹는다는점을고려하면추후연구에서는검사항목을수정하여빵대신밥을포함시키는것도좋을것으로생각된다. GUSS와 FDS의상관분석결과의미있는상관관계를보여주었다 (r=-.594, Fig. 2). 또한, 흡인과기도침투를 8단계로세분화하여기술하는 P/A 척도와의상관관계도의미있었다 (r=-.747, Fig. 3). 일반적으로재활병원에서는뇌졸중환자의연하곤란을확인하기위한검사로비디오투시연하검사가널리사용되고있다. 특히최근에는광섬유내시경연하검사와함께뇌졸중후연하곤란의표준검사의하나로여겨 지고있는상황이어서, 14 GUSS 가비디오투시연하검사를잘반영한다 는점은임상적으로의미가있다. GUSS가비디오투시연하검사의소견과의미있는상관관계를보이는이유로 GUSS가지닌장점인반고형식, 액체식, 고형식등을모두다평가하고있다는포괄적인면을들수있겠다. 하지만, GUSS의시행이비디오투시연하검사와같은표준검사로서의중요성을침해하기는어려운데, 무증상흡인 (silent aspiration) 과 - 24 -

같은경우, 간접적인징후만으로확인하는선별검사로는민감도가낮아발견되지못할가능성이높기때문이다 (27~85%). 28 최근에는물연하검사와같은선별검사와산소포화도검사를함께검사하는경우민감도가 73~98% 로증가된다는연구도있으므로, 26 GUSS와산소포화도측정을함께하는경우, 무증상흡인의선별가능성이향상될수있는지에대한추가연구가필요할것으로보인다. 뇌졸중환자의재활치료과정에서여러가지연하기능의호전을위한치료를실시한다. 임상척도는연하기능의추적관찰시변화의평가및치료효과의판정, 적절한식이의선택등에도움을줄수있는데, 치료효과의확인을위해매번비디오투시연하검사를실시하는것에비해이점이있다. 미국언어청각협회에서개발한 ASHA NOMS 연하척도와같은임상척도도있으나 7단계에불과하고, 항목이제한적이어서실제로사용하기에는제한점이있다. CDS와같은연하곤란임상척도는뇌병변의위치, 흡인병력, 씹기능력, 혀의돌출능력등, 연하곤란과관련된임상양상을중심으로점수화한체계이고, 실제음식물을시도하는방식이아니어서각음식물의형태에따른환자의연하곤란양상이어떤지는알수가없다. GUSS는임상적인척도인 ASHA NOMS 연하척도와 CDS와도의미 - 25 -

있는상관관계를보여주었고, 특히, CDS와는높은상관계수를보여주었다. ASHA NOMS 연하척도의상관계수는비교적낮았는데 (r=.432, Fig. 4), 이는 ASHA NOMS 연하척도의단계의결정에음식의투여경로, 음식의형태등이중요한요소인데, 이러한항목들은연하곤란의상태를직접적으로나타내기보다는임상적판단에의해시행하는사람마다차이가생길수있기때문에 GUSS와같이직접음식을투여하고관찰하는검사와는차이가있었던것으로보인다. 반면 CDS의경우평가하는항목이직접적인연하상태를관찰하는항목이대부분이어서오히려 GUSS와 CDS 사이에는상관계수가상당히높게나온것으로생각된다 (r=-.899, Fig. 5). 본연구의결과를종합해볼때, GUSS가비디오투시연하검사의연하곤란양상을반영한다는점과다른연하곤란임상척도와의의미있는상관관계를함께고려하면, GUSS를연하곤란의임상척도로사용할수있을가능성을고려해볼수있을것으로보인다. 추후 GUSS를임상척도및선별검사로사용하여, 연하곤란의추적관찰및음식의선택등에이용될때, 실제뇌졸중후흡인성폐렴과같은합병증을얼마나예방할수있는지를확인하는연구가진행된다면더욱의미있으리라생각된다. 본연구의제한점으로는대상자의수가많지않다는점에있다. 추 - 26 -

후더많은환자를포함시켜연구를진행한다면보다신뢰도높은결과를얻을수있을것으로생각된다. 검사당검사자가한명이었다는점도제한점이될수있겠다. 다수의검사자가한검사를실시하여평가자간신뢰도 (interrater reliability) 에대한확인이필요할것으로생각된다. V. 결론뇌졸중재활환자를대상으로한본연구에서 GUSS는다른연하곤란선별검사와비교했을때민감도와특이도에서다른선별검사와동등한양상을보여주었고, 비디오투시연하검사와임상적인연하곤란척도와도의미있는상관관계를보여 GUSS는부가적으로연하곤란정도를반영하는임상척도로의이용가능성을확인할수있었다. - 27 -

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Abstract Comparison between Gugging swallowing screen and other dysphagia screening tests using videofluoroscopic swallowing test Wonwoo Song Department of Medicine The Graduate School, Yonsei University (Directed by Professor Ji Cheol Shin) Screening tests for dysphagia after stroke are crucial in order to avoid severe complications such as aspiration pneumonia, sepsis, asphyxia and death. Several screening tests have been introduced to prevent complications arising from dysphagia in stroke patients in their rehabilitation stage. Among them, Gugging swallowing screen (GUSS), a recently developed test, was proven as an effective screening tests for dysphagia of stroke patients in 24 hours after onset. Unlike another dysphagia screening tests, GUSS can express the dysphagic state not only as PASS and FAIL, but also as graded rating. We compared several screening tests and clinical scales including GUSS. At the same time videofluoroscopic swallowing study (VFS) was used to identify aspiration. In this way, we wanted to confirm the effectiveness of GUSS in the view of a screening test and a clinical scale for dysphagia. Subjects and method. Subjects were 37 stroke patients 3 months after onset who were admitted to a rehabilitation hospital. GUSS, 3 oz water test, Burke dysphagia screening test (BDST) and Standardized swallowing assessment (SSA) were carried out at bedside. VFS was performed in 24 hours after other studies were done. Sensitivity and specificity of each screen tools were measured on the basis of aspiration in VFS. Chi square test was employed to - 32 -

compare the result of each screen tools between aspiration group and no aspiration group. Functional dysphagia scale (FDS) and Penetration-aspiration scale (P/A scale) were derived from VFS. Clinical scales such as ASHA NOMS swallowing scale and Clinical dysphagia scale (CDS) were measured to see the correlation between GUSS and another dysphagia scales. Result. GUSS had a sensitivity of 9.9% and a specificity of 69.2% at a cut-off of 14 or below. 3 oz water test had a sensitivity of 81.8% and a specificity of 76.9%. Sensitivity and specificity of BDST were respectively 9.9%, 61.5%. Sensitivity of SSA was 9.9%, specificity 61.5%. The result of each screening tests showed a significant difference between aspiration and no aspiration groups (p<.1). Correlation between GUSS and FDS was significant (r=-.594, p<.1). Correlation between GUSS and P/A scale also showed significance (r=-.747,p<.1). ASHA NOMS swallowing scale and GUSS showed significant correlation (r=-.899, p<.1). CDS and GUSS showed significant correlation as well (r=.432, p<.1) Conclusion. The screening ability of GUSS was equal to other screening tests for dysphagia of stroke patients. Correlations among GUSS and other VFS and clinical scales showed availability of GUSS as a clinical scale for dysphagia. ---------------------------------------------------------------------------------------- Key Words : dysphagia, swallowing screen, videofluoroscopic swallowing study - 33 -

부록 Appendix 1. Gugging Swallowing Screen 13 1. Preliminary Investigation /Indirect Swallowing Test Yes No Vigilance (The patient must be alert for at least for 15 minutes) 1 Cough and/or throat clearing (voluntary cough) 1 (Patient should cough or clear his or her throat twice) Saliva Swallow: Swallowing successful 1 Drooling 1 Voice change (hoarse, gurgly, coated, weak) 1 Sum: (5) 1-4= Investigate further¹ 5= Continue with part 2 2. Direct Swallowing Test In the following order: 1 2 3 SEMISOLID* LIQUID** SOLID *** DEGLUTITION: Swallowing not possible Swallowing delayed (> 2 sec.) (Solid textures > 1 sec.) Swallowing successful COUGH (involuntary): (before, during or after swallowing until 3 minutes later) Yes No DROOLING: Yes No VOICE CHANGE: (listen to the voice before and after swallowing - Patient should speak O ) Yes No SUM: 1 2 1 1 1 2 1 1 1 1 1 (5) (5) (5) 1-4= Investigate further¹ 5= Continue Liquid 1-4= Investigate further¹ 5= Continue Solid 1 2 1 1 1-4= Investigate further¹ 5= Normal SUM: (Indirect Swallowing Test AND Direct Swallowing Test) (2) * First administer ⅓ up to a half teaspoon water with food thickener (pudding-like consistency). If there are no symptoms apply 3 to 5 teaspoons. Assess after the 5th spoonful. ** 3, 5, 1, 2 ml water - if there are no symptoms continue with 5 ml water Assess and stop the investigation when one of the criteria is observed! *** Clinical: dry bread ¹ Use functional investigations such as Videofluoroscopic Evaluation of Swallowing (VFES), Fiberoptic Endoscopic Evaluation of Swallowing (FEES) - 34 -

Appendix 2. the Burke Dysphagia Screening Test (BDST) 1 present absent 1. Bilateral stroke 2. Brainstem stroke 3. History of Pneumonia acute stroke phase 4. Coughing associated with feeding or during 3 oz. water swallow test 5. Failure to consume one-half of meals 6. Prolonged time required for feeding. 7. Non oral feeding program in progress. Presence of one or more of these features is scored as failing the Burke Dysphagia Screening Test Results: Pass Fail - 35 -

Appendix 3. Standardized Swallowing Assessment 2 Section One: To ensure patient is physically capable of undertaking screening: 1 Is the patient awake and alert, or responding to speech? 2 Is the patient able to be positioned upright, with some head control? If your answer is No to either of the above questions, go no further and do not screen. Section Two: Checklist: 3 Can the patient cough when asked to? 4 Is the patient able to maintain some control of their saliva? 5 Is the patient able to lick top and bottom lip? 6 Is the patient able to breathe freely? (i.e. no difficulty breathing or problems maintaining SaO2) If answers to questions 3±6 are yes, proceed with screen. If any answer is no, stop and refer to SLT. 7 Does the patient have a `wet' or hoarse-sounding voice? If no, proceed with screen. If yes, stop and refer to SLT. If in doubt, discuss with SLT/medical team. Section Three: Water swallow test: With the patient alert and sitting upright, give first a teaspoonful of water. Problems are identified if no attempts are made to swallow, or water leaks straight out of the mouth, if there is coughing, choking, or breathlessness, or a wet/gurgly voice afterwards. Wait to ensure the water is swallowed. If there are no problems, repeat the process with a second and third teaspoonful. If there is still no problem evident, give half a glass of water. If there is no problem, order a diet to suit the patient's nutritional requirements. Make sure the patient is sitting up to eat and supervise the patient whilst eating the test meal. If there are any concerns, refer to SLT. If there are no concerns, continue and maintain vigilance. Repeat screening if there is any deterioration. - 36 -

Appendix 4. The Functional Dysphagia Scale Based on Videofluoroscopic Study 18 Factor Coded value Score Lip closure Bolus formation Residue in oral cavity Oral transit time Intact Inadequate None Intact Inadequate None None <=1% 1 5% >=5% <=1.5 sec >1.5 sec 5 1 3 6 2 4 6 6 1 6 6 6 Triggering of pharyngeal Normal 1 swallow Delayed Normal 1 12 Laryngeal elevation and Reduced 12 epiglottic closure Nasal penetration Residue in valleculae Residue in pyriform sinuses Coating of pharyngeal wall after swallow None 1% 1 5% 5% None 1% 1 5% 5% None 1% 1 5% 5% No Yes 1. sec 4 8 12 4 8 12 4 8 12 1 12 12 12 1 4 Pharyngeal transit time >1. sec 4 Total 1-37 -

Appendix 5. 8-Point Penetration-Aspiration Scale 21 1. Material does not enter the airway 2. Material enters the airway, remains above the vocal folds, and is ejected from the airway 3. Material enters the airway, remains above the vocal folds, and is not ejected from the airway 4. Material enters the airway, contacts the vocal folds, and is ejected from the airway 5. Material enters the airway, contacts the vocal folds, and is not ejected from the airway 6. Material enters the airway, passes below the vocal folds and is ejected into the larynx or out of the airway 7. Material enters the airway, passes below the vocal folds, and is not ejected from the trachea despite effort 8. Material enters the airway, passes below the vocal folds, and no effort is made to eject - 38 -

Appendix 6. The American Speech-Language Hearing Association National Outcomes Measurements System Swallowing Scale 23 Explanation Level 1 Individual is not able to swallow anything safely by mouth. All nutrition and hydration is received Level 2 through nonoral means (e.g. nasogastric tube, PEG*)). Individual is not able to swallow safely by mouth for nutrition and hydration but may take some Level 3 consistency with consistent maximal cues in therapy only. Alternative method of feeding is required. Alternative method of feeding required as individual takes less than 5% Level 4 of nutrition and hydration by mouth, and/or swallowing is safe with consistent use of moderate cues to use compensatory strategies and/or requires maximum diet restrictions. Level 5 Swallowing is safe but usually requires moderate cues to use compensatory strategies, and/or individual has moderate diet restrictions and/or still requires tube feedings and/or oral supplements. Level 6 Swallow is safe with minimal diet restrictions and/or occasionally requires minimal cueing to use compensatory strategies. May occasionally self cue. All nutrition and hydration needs are met by mouth at mealtime. Level 7 Swallowing is safe and individual eats and drinks independently and may rarely require minimal cueing. Usually self cues when difficulty occurs. May need to avoid specific food items (e.g., popcorn and nuts), or requires additional time (due to dysphagia). Individual's ability to eat independently is not limited by swallow function. Swallowing would be safe and efficient for all consistencies. Compensatory strategies are effectively used when needed. PEG: Percutaneous endoscopic gastrostomy - 39 -

Appendix 7. Clinical Dysphagia Scale 24 Location T-cannula Aspiration Lip sealing Chewing and mastication Tongue protrusion Laryngeal elevation Reflex coughing Non-stem lesion Stem lesion No Yes No Yes Intact Inadequate None Intact Inadequate None Intact Inadequate None Intact Inadequate None No Yes 5 25 1 2 4 4 8 4 8 5 1 3 Total 1-4 -