孤獨(dú)癥兒童潛在早期識(shí)別標(biāo)志——發(fā)聲異常及原因探析*

劉 敏 胡 洋 劉巧云

孤獨(dú)癥兒童潛在早期識(shí)別標(biāo)志——發(fā)聲異常及原因探析*

劉 敏1,2胡 洋1劉巧云1,2

(1華東師范大學(xué)教育學(xué)部康復(fù)科學(xué)系,2華東師范大學(xué)附屬婦幼保健院, 上海 200062)

孤獨(dú)癥譜系障礙(Autism Spectrum Disorder, ASD)兒童的早期識(shí)別及早期干預(yù)對(duì)其發(fā)育結(jié)果至關(guān)重要。發(fā)聲是指兒童學(xué)會(huì)說話之前發(fā)出的聲音。已有研究表明, 非類言語(yǔ)和類言語(yǔ)發(fā)聲異常是ASD兒童2歲前的潛在識(shí)別標(biāo)志。ASD兒童發(fā)聲異常的理論解釋主要有動(dòng)機(jī)導(dǎo)向、神經(jīng)運(yùn)動(dòng)導(dǎo)向、感知覺導(dǎo)向和社會(huì)反饋導(dǎo)向理論。未來研究可考慮探討發(fā)聲異常作為ASD兒童獨(dú)特的早期識(shí)別標(biāo)志的可能性, 加強(qiáng)哭聲在ASD兒童早期篩查中的研究, 探索基于最具預(yù)測(cè)性聲學(xué)參數(shù)集的自動(dòng)學(xué)習(xí)分類模型, 探究?jī)?nèi)在動(dòng)機(jī)與社會(huì)動(dòng)機(jī)對(duì)ASD兒童類言語(yǔ)發(fā)聲的影響和進(jìn)一步探究ASD兒童類言語(yǔ)發(fā)聲異常的神經(jīng)機(jī)制。從而為ASD兒童的早期識(shí)別及早期干預(yù)提供更加客觀的理論依據(jù)。

孤獨(dú)癥譜系障礙, 潛在早期識(shí)別標(biāo)志, 發(fā)聲異常, 原因探析

1 引言

孤獨(dú)癥譜系障礙(Autism Spectrum Disorder, ASD)是一種神經(jīng)發(fā)育性疾病, 有兩個(gè)核心癥狀：一是, 社會(huì)溝通和社會(huì)互動(dòng)存在持續(xù)性的缺陷; 二是, 表現(xiàn)出受限的和重復(fù)的行為模式、興趣或活動(dòng)(American Psychiatric Association, 2013)。ASD有兩種主要的發(fā)病模式：通常在1歲前發(fā)病的早發(fā)型(early onset)和2歲左右發(fā)病的倒退型(regressive onset) (Boterberg et al., 2019)。倒退型ASD占比大約為30%, 主要以已習(xí)得的語(yǔ)言或社交技能的倒退或喪失為標(biāo)志(Tan et al., 2021)。最新的數(shù)據(jù)顯示全球ASD發(fā)病率略低于1% (Lord et al., 2020), 我國(guó)ASD發(fā)病率為1/142(Zhou et al., 2020), 美國(guó)ASD發(fā)病率為1/54(Maenner et al., 2020), 由此可見ASD發(fā)病率較高。雖然目前沒有治療ASD核心癥狀的特效藥(Goel et al., 2018), 但在早期尤其是2歲以前若能及時(shí)發(fā)現(xiàn)和干預(yù), 能有效改善ASD個(gè)體的語(yǔ)言、社交溝通等能力的發(fā)展軌跡并影響長(zhǎng)期結(jié)果(Estes et al., 2015; Frazier et al., 2021; Sigafoos & Waddington, 2016; Wetherby et al., 2018), 甚至“預(yù)防”全面綜合征的出現(xiàn)(Yirmiya & Charman, 2010)。

發(fā)聲(vocalization)是指兒童學(xué)會(huì)說話之前發(fā)出的聲音(Schoen Simmons, 2021)。發(fā)聲包括非類言語(yǔ)發(fā)聲(nonspeech-like vocalizations)和類言語(yǔ)發(fā)聲(speech-like vocalizations) (Oller, 2000)。非類言語(yǔ)發(fā)聲是與生物功能有聯(lián)系的發(fā)聲, 包括有固定信號(hào)作用的聲音(fixed signals)和植物性聲音(vegetative sounds) (Garrido et al., 2017; Nathani et al., 2006); 類言語(yǔ)發(fā)聲是指類似言語(yǔ)的聲音, 包括不同的類型并沿著一個(gè)連續(xù)體發(fā)展, 在1歲左右變得更像言語(yǔ)(Nathani et al., 2006; Oller, 2000)。ASD兒童的發(fā)聲特征通常采用更具體的指標(biāo)進(jìn)行分析, 其中非類言語(yǔ)發(fā)聲常用分析指標(biāo)為頻次、比例、基頻、停頓長(zhǎng)度、持續(xù)時(shí)間等(e.g., Esposito et al., 2013; Plumb & Wetherby, 2013), 類言語(yǔ)發(fā)聲常用分析指標(biāo)為發(fā)聲復(fù)雜性和溝通性發(fā)聲(e.g., Mcdaniel et al., 2020; Woynaroski et al., 2016)。發(fā)聲的主要類型及常用分析指標(biāo)見表1。

發(fā)聲是早期正常發(fā)展的一個(gè)關(guān)鍵成就。正常發(fā)展(Typical Development, TD)兒童的發(fā)聲在0～1歲之間表現(xiàn)出哭聲和準(zhǔn)共振元音比例逐步下降, 元音和規(guī)范音節(jié)比例逐步上升的趨勢(shì)(Cychosz et al., 2021; Nathani et al., 2006)。發(fā)聲的非典型性發(fā)展是發(fā)育異常的指標(biāo), 對(duì)早期發(fā)聲的深入理解使得神經(jīng)發(fā)育性疾病的早期發(fā)現(xiàn)和鑒別成為可能(Marschik et al., 2017)。研究表明, ASD兒童的發(fā)聲存在異?，F(xiàn)象, 與其核心的非典型溝通癥狀有關(guān)(Gabrielsen et al., 2015), 對(duì)發(fā)聲異常的早期識(shí)別可能有利于改善ASD兒童平均在3～5歲才能被診斷的現(xiàn)狀(van’t Hof et al., 2021)。然而, 關(guān)于ASD發(fā)聲特征已有研究存在分歧。例如, 有研究支持ASD兒童比TD兒童有更多非類言語(yǔ)沮喪聲的結(jié)論(Plumb & Wetherby, 2013), 也有研究發(fā)現(xiàn)

兩類兒童非類言語(yǔ)沮喪聲無差異(Schoen et al., 2011)。有研究表明ASD兒童比TD兒童的類言語(yǔ)發(fā)聲更少(Patten et al., 2014), 而另有研究卻發(fā)現(xiàn)ASD兒童類言語(yǔ)發(fā)聲過度(Werner & Dawson, 2005)。那么, ASD兒童有哪些非典型的發(fā)聲異常？這些發(fā)聲異常作為早期識(shí)別標(biāo)志的可能性如何？

本文將總結(jié)近年來ASD兒童早期發(fā)聲的研究成果, 概括ASD兒童發(fā)聲異常的特征, 分析ASD兒童發(fā)聲異常作為早期識(shí)別標(biāo)志的可能性, 探析ASD兒童發(fā)聲異常的原因, 從而為ASD兒童的早期發(fā)現(xiàn)及早期干預(yù)提供一些新思路。

2 發(fā)聲異常與ASD早期識(shí)別

2.1 非類言語(yǔ)發(fā)聲異常與ASD早期識(shí)別

對(duì)ASD兒童非類言語(yǔ)發(fā)聲特征的研究主要包括兩個(gè)方面：一是, 非類言語(yǔ)發(fā)聲頻次或比例; 二是, 哭聲的基頻、停頓長(zhǎng)度和持續(xù)時(shí)間。雖然, 已有非類言語(yǔ)發(fā)聲頻次、比例等研究結(jié)果不一致(e.g., Plumb & Wetherby, 2013; Schoen et al., 2011)。然而, 關(guān)于非類言語(yǔ)發(fā)聲中哭聲聲學(xué)異常特征的結(jié)論相對(duì)一致, 即ASD兒童的哭聲基頻較高, 停頓長(zhǎng)度和持續(xù)時(shí)間較短(e.g., Bornstein et al., 2016; Esposito et al., 2014; Esposito et al., 2015)。

表1 發(fā)聲的類型、定義及常用分析指標(biāo)

注: 非類言語(yǔ)發(fā)聲分為固定信號(hào)聲音和植物性聲音, 其中植物性聲音是反射性發(fā)聲, 如咳嗽、打嗝、打噴嚏等發(fā)聲, 在發(fā)聲研究中一般不對(duì)植物性聲音進(jìn)行分析, 故此處只包括固定信號(hào)聲音; C: 輔音, consonant; V: 元音, vowel。

2.1.1 較高的哭聲基頻是ASD潛在早期識(shí)別標(biāo)志

基頻作為最能說明哭聲聲學(xué)特征的參數(shù)(Lester & Gasse, 2020), 在ASD兒童哭聲的研究中被研究者廣泛使用。研究中ASD兒童哭聲基頻樣本主要來自兩方面：一是, 熟悉或陌生自然情境中的非疼痛哭聲; 二是, 自然情境中的疼痛哭聲。研究發(fā)現(xiàn), ASD兒童哭聲基頻高于同齡TD兒童。對(duì)已確診的ASD兒童早期哭聲的家庭視頻分析表明, 5～18個(gè)月的ASD嬰兒非疼痛哭聲基頻高于TD嬰兒(Bornstein et al., 2016; Esposito et al., 2012, 2013; Esposito et al., 2015; Esposito & Venuti, 2010a, 2010b)。15個(gè)月的ASD高風(fēng)險(xiǎn)嬰兒和年齡稍大的36～52個(gè)月ASD幼兒陌生情境中較高的非疼痛哭聲基頻支持上述結(jié)果(Esposito et al., 2014; Ozturk et al., 2018)。前瞻性研究發(fā)現(xiàn), 熟悉的自然情境中6個(gè)月的ASD高風(fēng)險(xiǎn)嬰兒疼痛哭聲基頻高于低風(fēng)險(xiǎn)嬰兒, 兩組嬰兒非疼痛哭聲無顯著差異, 但后來被診斷為ASD的嬰兒的兩類哭聲基頻均最高(Sheinkopf et al., 2012)。以上證據(jù)顯示, 較高的哭聲基頻可能是ASD的早期識(shí)別標(biāo)志, 尤其較高的疼痛哭聲基頻可能是一個(gè)更敏感的指標(biāo)。

2.1.2 較短的哭聲停頓長(zhǎng)度和持續(xù)時(shí)間是ASD潛在早期識(shí)別標(biāo)志

哭聲的停頓長(zhǎng)度和持續(xù)時(shí)間反映了呼吸控制能力(LaGasse et al., 2005), 在ASD嬰兒哭聲研究中探究較多。已有研究顯示, ASD兒童哭聲的停頓長(zhǎng)度和持續(xù)時(shí)間短于同齡TD兒童。研究表明, 12～13個(gè)月的ASD嬰兒哭聲的停頓長(zhǎng)度明顯短于TD嬰兒(Esposito et al., 2013; Esposito et al., 2015; Esposito & Venuti, 2009; Venuti et al., 2012)。前瞻性研究發(fā)現(xiàn), ASD高風(fēng)險(xiǎn)嬰兒在12個(gè)月和15個(gè)月時(shí)哭聲的持續(xù)時(shí)間短于TD嬰兒, 且在2歲或3歲被診斷為ASD的嬰兒的哭聲持續(xù)時(shí)間最短(Esposito et al., 2014; Unwin et al., 2017)。以上證據(jù)表明, 較短的哭聲停頓長(zhǎng)度和持續(xù)時(shí)間可能是ASD的早期識(shí)別標(biāo)志。

2.2 類言語(yǔ)發(fā)聲異常與ASD早期識(shí)別

2.2.1 異常的發(fā)聲復(fù)雜性是ASD潛在早期識(shí)別標(biāo)志

有大量研究探討了ASD兒童的發(fā)聲復(fù)雜性, 雖然有兩項(xiàng)研究發(fā)現(xiàn)ASD兒童或ASD高風(fēng)險(xiǎn)兒童的發(fā)聲復(fù)雜性與TD兒童無顯著差異(Chericoni et al., 2016; Talbott et al., 2016), 但有更多的證據(jù)顯示ASD兒童的類言語(yǔ)發(fā)聲復(fù)雜性表現(xiàn)出異?，F(xiàn)象, 主要表現(xiàn)為類言語(yǔ)發(fā)聲復(fù)雜性低于或高于同齡TD兒童(e.g., Garrido et al., 2017; Heymann et al., 2018; Werner & Dawson, 2005)。

首先, 多數(shù)研究證實(shí)ASD兒童類言語(yǔ)發(fā)聲復(fù)雜性低于同齡TD兒童。前瞻性和回溯性研究表明, 2歲前ASD嬰兒的元音、邊緣音節(jié)和規(guī)范音節(jié)等類言語(yǔ)發(fā)聲頻率或比例顯著低于TD嬰兒(Garrido et al., 2017; Patten et al., 2014; Plumb & Wetherby, 2013), 表現(xiàn)出更少的輔音類型(Schoen et al., 2011; Wetherby et al., 2007)。ASD高風(fēng)險(xiǎn)嬰兒的元音、規(guī)范音節(jié)、復(fù)雜音節(jié)等類言語(yǔ)發(fā)聲頻率或比例(Chenausky et al., 2017; Heymann et al., 2018; Paul et al., 2011)和輔音類型(Paul et al., 2011)均低于低風(fēng)險(xiǎn)嬰兒。此外, 研究顯示類言語(yǔ)發(fā)聲復(fù)雜性對(duì)ASD診斷具有良好的預(yù)測(cè)性。Patten等人(2014)發(fā)現(xiàn)9～12個(gè)月時(shí)規(guī)范音節(jié)占總音節(jié)發(fā)聲比率對(duì)ASD診斷預(yù)測(cè)的準(zhǔn)確率為90%。Paul等人(2011)發(fā)現(xiàn)9個(gè)月和12個(gè)月ASD高風(fēng)險(xiǎn)嬰兒的輔音類型能預(yù)測(cè)24個(gè)月時(shí)ASD癥狀, 準(zhǔn)確率分別為77%和65%。以上證據(jù)表明, 較低的類言語(yǔ)發(fā)聲復(fù)雜性可能是ASD早期識(shí)別標(biāo)志。

其次, 少量倒退型ASD兒童類言語(yǔ)發(fā)聲復(fù)雜性高于同齡TD兒童, 出現(xiàn)過度發(fā)聲現(xiàn)象?；厮菪匝芯堪l(fā)現(xiàn), 15名12個(gè)月大的倒退型ASD嬰兒復(fù)雜呀呀語(yǔ)頻率高于TD嬰兒, 而另外21名早發(fā)型ASD嬰兒復(fù)雜呀呀語(yǔ)頻率低于TD嬰兒。進(jìn)一步分析發(fā)現(xiàn)46%倒退型ASD嬰兒的復(fù)雜呀呀語(yǔ)頻率高于或等于TD嬰兒的平均值, 只有6%早發(fā)型ASD嬰兒表現(xiàn)出這種現(xiàn)象(Werner & Dawson, 2005)。這項(xiàng)研究還發(fā)現(xiàn), 隨著年齡的增長(zhǎng), 倒退型ASD兒童的復(fù)雜呀呀語(yǔ)逐步退化, 在24個(gè)月時(shí)他們的復(fù)雜呀呀語(yǔ)與早發(fā)型ASD嬰兒一樣, 都顯著落后于TD嬰兒。最近一項(xiàng)前瞻性研究發(fā)現(xiàn), 9個(gè)月大ASD高風(fēng)險(xiǎn)嬰兒比低風(fēng)險(xiǎn)嬰兒有更多的類言語(yǔ)發(fā)聲,這一結(jié)果主要體現(xiàn)在12名ASD高風(fēng)險(xiǎn)嬰兒的過度發(fā)聲(Swanson et al., 2018)。以上結(jié)果表明, 較高的發(fā)聲復(fù)雜性可能是倒退型ASD兒童的早期識(shí)別標(biāo)志。

2.2.2 較少的溝通性發(fā)聲是ASD潛在早期識(shí)別標(biāo)志

溝通性發(fā)聲能夠評(píng)價(jià)ASD兒童發(fā)聲的社會(huì)性(Mcdaniel et al., 2020)。研究表明ASD兒童溝通性發(fā)聲少于同齡TD兒童, 2歲前ASD嬰兒有意圖的發(fā)聲溝通行為明顯少于TD嬰兒(Bacon et al., 2018; Dow et al., 2020; Dow et al., 2017; Plumb & Wetherby, 2013; Shumway & Wetherby, 2009; Wetherby et al., 2004)。ASD高風(fēng)險(xiǎn)嬰兒研究也支持上述結(jié)果, ASD高風(fēng)險(xiǎn)嬰兒產(chǎn)生更少的社交導(dǎo)向發(fā)聲, 隨后被確診的ASD嬰兒的溝通性發(fā)聲能力最低(Garrido et al., 2017; Sacrey et al., 2021; Winder et al., 2013), 且過度發(fā)聲的ASD高風(fēng)險(xiǎn)嬰兒的社交呀呀語(yǔ)也低于低風(fēng)險(xiǎn)嬰兒(Swanson et al., 2018)。以上證據(jù)表明, 溝通性發(fā)聲可能是ASD的早期識(shí)別標(biāo)志，其與發(fā)聲復(fù)雜性的相互作用可能是預(yù)測(cè)ASD的更有效標(biāo)志。

3 ASD兒童發(fā)聲異常的原因探析

綜合已有研究結(jié)果, 發(fā)現(xiàn)ASD兒童非類言語(yǔ)哭聲基頻較高, 停頓長(zhǎng)度和持續(xù)時(shí)間較短; 類言語(yǔ)發(fā)聲復(fù)雜性較低或較高, 溝通性發(fā)聲較少。這些發(fā)聲異常不僅可能是ASD早期識(shí)別標(biāo)志, 也在一定程度上反映了ASD兒童社會(huì)溝通和社會(huì)互動(dòng)方面的核心缺陷。以下結(jié)合動(dòng)機(jī)導(dǎo)向、神經(jīng)運(yùn)動(dòng)導(dǎo)向、感知覺導(dǎo)向和社會(huì)反饋導(dǎo)向等理論對(duì)現(xiàn)有結(jié)論進(jìn)行分析與解釋。

3.1 動(dòng)機(jī)導(dǎo)向理論

ASD兒童對(duì)特殊興趣的強(qiáng)內(nèi)在動(dòng)機(jī)在一定程度上解釋了他們類言語(yǔ)發(fā)聲復(fù)雜性異常的問題。內(nèi)在動(dòng)機(jī)(intrinsic motivation)是指?jìng)€(gè)體自發(fā)的好奇心和興趣、主動(dòng)練習(xí)技能和學(xué)習(xí)知識(shí)的傾向, 受多巴胺能系統(tǒng)的支持, 與注意力控制和自我參照認(rèn)知的神經(jīng)網(wǎng)絡(luò)相關(guān)(di Domenico & Ryan, 2017)。計(jì)算機(jī)建模研究證明, 內(nèi)在動(dòng)機(jī)是嬰兒類言語(yǔ)發(fā)聲發(fā)展的一般機(jī)制(Acevedo-Valle et al., 2018; Moulin-Frier et al., 2014), 其關(guān)鍵思想是自我生成和目標(biāo)選擇。內(nèi)在動(dòng)機(jī)讓嬰兒自主構(gòu)建自己的發(fā)聲試驗(yàn)和制定自己的學(xué)習(xí)計(jì)劃, 解釋了嬰兒從最初忽略周圍語(yǔ)音環(huán)境的自我探索發(fā)聲到受成人語(yǔ)音影響的模仿發(fā)聲的自動(dòng)轉(zhuǎn)變, 使嬰兒的發(fā)聲從最初的非類言語(yǔ)發(fā)聲過渡到不清晰的準(zhǔn)元音再到清晰的規(guī)范音節(jié)。內(nèi)在動(dòng)機(jī)對(duì)TD嬰兒類言語(yǔ)發(fā)聲的作用也在行為研究中得到了驗(yàn)證, TD嬰兒的類言語(yǔ)發(fā)聲大多是在獨(dú)自探索的游戲中產(chǎn)生的(Oller et al., 2019)。當(dāng)成人尋求與嬰兒社會(huì)互動(dòng)時(shí), 嬰兒大多數(shù)發(fā)聲(約60%)似乎不是針對(duì)成人的; 當(dāng)成人與嬰兒在一起但沒有試圖參與嬰兒的活動(dòng)時(shí), 這種探索性發(fā)聲的優(yōu)勢(shì)更強(qiáng)(約80%) (Long et al., 2020)。研究發(fā)現(xiàn)ASD個(gè)體比正常人群有更多刻板地從事特殊興趣的內(nèi)在動(dòng)機(jī)(Grove et al., 2016), 且這一現(xiàn)象在2歲前的ASD嬰兒中也很明顯(Bacon et al., 2018)。ASD嬰兒可能更喜歡重復(fù)玩弄音節(jié), 而TD嬰兒更傾向于探索語(yǔ)音的細(xì)微差別(Long et al., 2021)。那么可以推測(cè), ASD嬰兒對(duì)特殊興趣的強(qiáng)內(nèi)在動(dòng)機(jī)可能影響了他們類言語(yǔ)發(fā)聲復(fù)雜性尤其是輔音多樣性的習(xí)得, 同時(shí)也可能導(dǎo)致倒退型ASD兒童類言語(yǔ)發(fā)聲復(fù)雜性過高。值得注意的是, 目前較缺乏內(nèi)在動(dòng)機(jī)對(duì)ASD兒童類言語(yǔ)發(fā)聲影響的實(shí)證研究。近期Long等人(2021)嘗試探究?jī)?nèi)在動(dòng)機(jī)對(duì)規(guī)范音節(jié)的影響, 結(jié)果顯示內(nèi)在動(dòng)機(jī)影響了不同ASD風(fēng)險(xiǎn)嬰兒規(guī)范音節(jié)的發(fā)展過程。未來對(duì)已確診的ASD嬰兒進(jìn)行探究, 能夠提供內(nèi)在動(dòng)機(jī)影響類言語(yǔ)發(fā)聲的新證據(jù)。

社會(huì)動(dòng)機(jī)缺陷可能是ASD兒童溝通性發(fā)聲較少的原因之一。社會(huì)動(dòng)機(jī)(social motivation)是一套心理傾向和生物機(jī)制, 偏向于個(gè)人優(yōu)先適應(yīng)社會(huì)世界, 在社會(huì)互動(dòng)中尋求和享受快樂, 努力培養(yǎng)和維持社會(huì)紐帶(Chevallier et al., 2012)。Chevallier等人(2012)在總結(jié)已有證據(jù)基礎(chǔ)上認(rèn)為, 社會(huì)動(dòng)機(jī)機(jī)制的破壞可能構(gòu)成了ASD的主要缺陷。ASD社會(huì)動(dòng)機(jī)缺陷與社會(huì)性獎(jiǎng)勵(lì)腦區(qū)功能失調(diào)有關(guān), 主要涉及尾狀核和前扣帶回功能降低, 中腦腹側(cè)被蓋區(qū)和伏隔核之間的功能連接性異常(Clements et al., 2018; Supekar et al., 2018)。ASD兒童對(duì)面孔關(guān)注減少, 更少參與到親子互動(dòng)中和發(fā)起共同注意互動(dòng)行為也為社會(huì)動(dòng)機(jī)缺陷假說提供了有力證據(jù)(Adamson et al., 2019; Guillon et al., 2016)。在社會(huì)動(dòng)機(jī)缺陷模型下, ASD兒童很少主動(dòng)采用類言語(yǔ)發(fā)聲的形式主動(dòng)與他人溝通。此外, 兒童的類言語(yǔ)發(fā)聲發(fā)展涉及兒童與成人雙主體的互動(dòng)(Elmlinger et al., 2019; Oller et al., 2016), ASD兒童的社會(huì)動(dòng)機(jī)缺陷使得他們難以與成人形成良好的社會(huì)互動(dòng)。那么, 社會(huì)動(dòng)機(jī)也可能影響ASD兒童類言語(yǔ)發(fā)聲復(fù)雜性的發(fā)展。然而, 目前關(guān)于社會(huì)動(dòng)機(jī)對(duì)ASD兒童類言語(yǔ)發(fā)聲發(fā)展影響的實(shí)證研究極其有限, 未來還需要開展該領(lǐng)域的研究。

3.2 神經(jīng)運(yùn)動(dòng)導(dǎo)向理論

類言語(yǔ)發(fā)聲神經(jīng)運(yùn)動(dòng)方面的不成熟或紊亂對(duì)ASD兒童類言語(yǔ)發(fā)聲復(fù)雜性可能有特定影響。類言語(yǔ)發(fā)聲神經(jīng)運(yùn)動(dòng)機(jī)制可以從言語(yǔ)的神經(jīng)運(yùn)動(dòng)方面進(jìn)行分析, 因?yàn)轭愌哉Z(yǔ)發(fā)聲或準(zhǔn)言語(yǔ)(quasispeech)具備言語(yǔ)的語(yǔ)音結(jié)構(gòu)但不帶有言語(yǔ)的意義(Kent, 2015), 且有大量研究采用無意義的元音或規(guī)范音節(jié)作為材料對(duì)言語(yǔ)產(chǎn)生的神經(jīng)系統(tǒng)進(jìn)行研究(Kumar et al., 2016)。位于額葉初級(jí)運(yùn)動(dòng)皮質(zhì)中的喉運(yùn)動(dòng)皮質(zhì)(Laryngeal Motor Cortex, LMC)參與控制言語(yǔ)運(yùn)動(dòng)(Eichert et al., 2020)。對(duì)元音發(fā)聲的功能核磁共振研究表明, 初級(jí)運(yùn)動(dòng)皮質(zhì)中喉部和下頜部肌肉控制區(qū)域有重疊, 研究者認(rèn)為這種皮質(zhì)間的重疊可能支持了喉部發(fā)聲和下頜振蕩之間的耦合, 從而產(chǎn)生音節(jié)結(jié)構(gòu)(Brown et al., 2021)。此外, 額葉皮質(zhì)的認(rèn)知功能也參與控制發(fā)聲(Hage, 2018)。然而, ASD兒童在2歲前前額葉和其它皮質(zhì)過度生長(zhǎng)(Bonnet-Brilhault et al., 2018; Hazlett et al., 2017; Hazlett et al., 2011; Kaushik & Zarbalis, 2016), 這可能會(huì)影響類言語(yǔ)發(fā)聲運(yùn)動(dòng)的控制, 從而導(dǎo)致類言語(yǔ)發(fā)聲復(fù)雜性異常。不過, 目前ASD兒童如何控制發(fā)聲器官以協(xié)調(diào)產(chǎn)生類言語(yǔ)發(fā)聲的神經(jīng)運(yùn)動(dòng)機(jī)制尚不清晰, 未來還需開展此方面的研究。

坐姿發(fā)育延遲可能是ASD兒童類言語(yǔ)發(fā)聲復(fù)雜性較低的一個(gè)重要原因。坐姿為下頜活動(dòng)提供了生物力學(xué)的支持, 兒童類言語(yǔ)發(fā)聲是通過打開和閉合下頜實(shí)現(xiàn)的(Green et al., 2002)。有研究比較了6個(gè)月時(shí)能獨(dú)立坐和不能獨(dú)立坐嬰兒的音節(jié)發(fā)聲, 結(jié)果表明他們的音節(jié)發(fā)聲頻率存在差異, 41%能獨(dú)立坐的嬰兒產(chǎn)生了音節(jié)發(fā)聲, 而只有9%未獨(dú)立坐的嬰兒產(chǎn)生了音節(jié)發(fā)聲(Leezenbaum, 2016)。回溯性研究發(fā)現(xiàn), ASD兒童穩(wěn)定坐立的平均年齡為9個(gè)月, 延遲于TD兒童的7.9個(gè)月(Ucuz & Cicek, 2020)。前瞻性研究支持上述結(jié)論, ASD高風(fēng)險(xiǎn)嬰兒的坐姿與低風(fēng)險(xiǎn)嬰兒不同, 6個(gè)月時(shí)ASD高風(fēng)險(xiǎn)嬰兒獨(dú)立坐立的時(shí)間顯著低于低風(fēng)險(xiǎn)嬰兒(Leezenbaum & Iverson, 2019)。這些研究表明, 坐姿習(xí)得較慢限制了ASD兒童坐立時(shí)探索新發(fā)聲可能性的機(jī)會(huì), 從而影響了規(guī)范音節(jié)的習(xí)得(Iverson, 2018)。

迷走神經(jīng)復(fù)合體損傷與基頻的非典型模式有關(guān), 自主神經(jīng)系統(tǒng)參與呼吸控制(LaGasse et al., 2005), 自主神經(jīng)系統(tǒng)障礙可能會(huì)影響哭聲的時(shí)間模式。人類聲音基頻的改變主要是通過肌肉運(yùn)動(dòng)改變聲帶的物理特性, 如聲帶的長(zhǎng)度、單位長(zhǎng)度的質(zhì)量、緊張度和硬度而實(shí)現(xiàn)的, 而控制聲帶運(yùn)動(dòng)的喉內(nèi)肌群主要由迷走神經(jīng)支配(Dankbaar & Pameijer, 2014)。ASD個(gè)體自主神經(jīng)系統(tǒng)功能存在障礙(Kong et al., 2021), 正常情況下自主神經(jīng)系統(tǒng)中的交感神經(jīng)與迷走神經(jīng)處于動(dòng)態(tài)平衡。然而, ASD兒童表現(xiàn)出交感神經(jīng)張力增強(qiáng)和迷走神經(jīng)張力減弱的現(xiàn)象(Benevides & Lane, 2015), 這可能會(huì)導(dǎo)致支配哭聲的聲帶控制不良(Sheinkopf et al., 2016)與呼吸肌群運(yùn)動(dòng)控制不良, 從而導(dǎo)致哭聲基頻較高、哭聲停頓長(zhǎng)度和持續(xù)時(shí)間較短等異常的哭聲模式。

3.3 感知覺導(dǎo)向理論

ASD兒童的聽處理障礙和對(duì)言語(yǔ)聲音刺激的非典型加工(Filipe et al., 2018; Soskey et al., 2017; Vlaskamp et al., 2017; Yau et al., 2016)可能影響他們的類言語(yǔ)發(fā)聲復(fù)雜性。言語(yǔ)誘發(fā)聽覺腦干反應(yīng)的研究表明, ASD兒童腦干水平語(yǔ)音聽覺加工發(fā)育不成熟(Chen et al., 2019)。ASD兒童對(duì)兒向言語(yǔ)(Infant-Directed Speech, IDS)表現(xiàn)出非典型的聽處理過程, IDS未引起ASD兒童神經(jīng)增強(qiáng)反應(yīng)(Chen et al., 2021)。ASD兒童對(duì)自己名字的弱回應(yīng)也印證了ASD兒童較弱的言語(yǔ)聲音刺激加工能力。研究表明, 9個(gè)月ASD嬰兒更不容易對(duì)自己名字產(chǎn)生反應(yīng), 這一現(xiàn)象一直持續(xù)到24個(gè)月(Miller et al., 2017)和36個(gè)月(Hatch et al., 2021)。ASD高風(fēng)險(xiǎn)嬰兒的研究也發(fā)現(xiàn), 6個(gè)月的ASD高風(fēng)險(xiǎn)嬰兒對(duì)言語(yǔ)聲音刺激的回應(yīng)也低于低風(fēng)險(xiǎn)嬰兒(Paterson et al., 2019)。以上證據(jù)可推測(cè), ASD嬰兒較少受成人照顧者言語(yǔ)的影響, 缺失了類言語(yǔ)發(fā)聲學(xué)習(xí)的機(jī)會(huì), 進(jìn)而影響他們類言語(yǔ)發(fā)聲復(fù)雜性的發(fā)展。近期的研究表明, 聽覺皮層和言語(yǔ)運(yùn)動(dòng)皮層之間存在相互作用, 言語(yǔ)聽覺感知影響言語(yǔ)產(chǎn)生運(yùn)動(dòng)皮層的反應(yīng)(Cheung et al., 2016), 言語(yǔ)運(yùn)動(dòng)規(guī)劃會(huì)影響言語(yǔ)感知皮層的反應(yīng)(Daliri & Max, 2016)。ASD兒童言語(yǔ)聽覺皮層和類言語(yǔ)發(fā)聲運(yùn)動(dòng)皮層之間的相互作用機(jī)制還需要進(jìn)一步研究。

3.4 社會(huì)反饋導(dǎo)向理論

社會(huì)反饋環(huán)路(social feedback loop)理論在一定程度上解釋了ASD兒童類言語(yǔ)發(fā)聲復(fù)雜性較低的現(xiàn)象。該理論強(qiáng)調(diào)社會(huì)環(huán)境中的成人回應(yīng)對(duì)兒童類言語(yǔ)發(fā)聲發(fā)展的作用, 認(rèn)為成人更有可能對(duì)兒童的類言語(yǔ)發(fā)聲做出回應(yīng), 在成人立即回應(yīng)后兒童更有可能產(chǎn)生類言語(yǔ)發(fā)聲(Warlaumont et al., 2014)。Warlaumont等人(2014)使用一種有很好穩(wěn)定性和效度的、主要針對(duì)0～4歲兒童與成人交互發(fā)聲的語(yǔ)言環(huán)境分析(Language ENvironment Analysis, LENA)軟件(Harbison et al., 2018; Jones et al., 2019; Richards et al., 2017; Woynaroski et al., 2017), 對(duì)自然環(huán)境中TD兒童與成人互動(dòng)中的發(fā)聲進(jìn)行了微觀分析并證實(shí)該觀點(diǎn)。研究者將ASD兒童與TD兒童發(fā)聲作對(duì)比, 發(fā)現(xiàn)ASD兒童社會(huì)反饋環(huán)路的有效性降低。主要表現(xiàn)在兩個(gè)方面：一是, ASD兒童發(fā)出較少的類言語(yǔ)發(fā)聲; 二是, 成人對(duì)ASD兒童的回應(yīng)取決于類言語(yǔ)發(fā)聲的程度降低。那么隨著時(shí)間的推移, 社會(huì)反饋環(huán)路迭代次數(shù)減少, 導(dǎo)致ASD兒童類言語(yǔ)發(fā)聲進(jìn)一步減少。

4 研究展望

4.1 探討發(fā)聲異常作為ASD兒童獨(dú)特的早期識(shí)別標(biāo)志的可能性

從上文的綜述可知, 與TD兒童相比, ASD兒童早期發(fā)聲確實(shí)存在異常現(xiàn)象。然而, 這些發(fā)聲特征也可能是其他障礙兒童的特征。例如, 研究發(fā)現(xiàn)9～12個(gè)月的脆性X綜合征嬰兒的規(guī)范音節(jié)少于同齡TD嬰兒(Belardi et al., 2017)。此外, 幾項(xiàng)同時(shí)包含了ASD兒童和發(fā)育遲緩(Developmental Delay, DD)兒童的研究結(jié)論不一致。有研究發(fā)現(xiàn)ASD兒童比DD兒童表現(xiàn)出更少的溝通性發(fā)聲(Lee et al., 2018), 而另有研究卻未發(fā)現(xiàn)兩類兒童在溝通性發(fā)聲方面存在顯著差異(Dow et al., 2020)。關(guān)于非類言語(yǔ)發(fā)聲中的非典型發(fā)聲, Wetherby等人(2004)發(fā)現(xiàn)ASD兒童和DD兒童無差異, 而Lee等人(2018)卻發(fā)現(xiàn)ASD兒童比DD兒童表現(xiàn)出更多的非典型發(fā)聲。因此, 非常有必要開展大規(guī)模的、納入其他障礙類型兒童的前瞻性研究, 以探討發(fā)聲異常作為ASD兒童獨(dú)特的早期識(shí)別標(biāo)志的可能性。

4.2 加強(qiáng)哭聲在ASD兒童早期篩查中的研究

如上文所述, 非典型的哭聲可能是ASD兒童的早期識(shí)別標(biāo)志。然而, 目前缺乏哭聲在ASD兒童早期篩查研究中的實(shí)證數(shù)據(jù)。近期研究也發(fā)現(xiàn), 非典型的哭聲可能影響ASD兒童的預(yù)后(Esposito et al., 2017), 因?yàn)檎疹櫿邔?duì)ASD兒童非典型哭聲難以理解會(huì)影響護(hù)理質(zhì)量(Bornstein et al., 2016)。此外, 成人也能較好地區(qū)分ASD兒童和TD兒童的哭聲。例如, 一項(xiàng)研究發(fā)現(xiàn)ASD嬰兒和TD嬰兒的父母均認(rèn)為ASD嬰兒的哭聲更痛苦和更不典型(English et al., 2019)。因此, 未來有必要也有可能開展哭聲在ASD兒童早期篩查中的研究。在篩查研究中, 可同時(shí)采用疼痛型和非疼痛型哭聲樣本。非疼痛型哭聲樣本可以參照以往的研究, 在嬰兒自然狀態(tài)下收集(e.g., Bornstein et al., 2016)。疼痛型哭聲可能是早期更為敏感的指標(biāo), 也更有可能在標(biāo)準(zhǔn)化的環(huán)境下獲得。例如, 可以收集嬰兒接種疫苗時(shí)誘發(fā)出的疼痛型哭聲。加強(qiáng)哭聲在ASD兒童早期篩查中的研究, 不僅有利于明確哭聲在早期篩查中的價(jià)值, 也有助于早期干預(yù)。

4.3 探索基于最具預(yù)測(cè)性聲學(xué)參數(shù)集的自動(dòng)學(xué)習(xí)分類模型

正如前文所綜述的, 發(fā)聲常用的分析指標(biāo)為類言語(yǔ)發(fā)聲復(fù)雜性、類言語(yǔ)溝通性發(fā)聲、非類言語(yǔ)哭聲基頻等。近期也有研究采用非類言語(yǔ)發(fā)聲或類言語(yǔ)發(fā)聲的聲學(xué)參數(shù)集合對(duì)ASD兒童進(jìn)行分類預(yù)測(cè)(e.g., Khozaei et al., 2020; Pokorny et al., 2017, August)。交叉驗(yàn)證也顯示, 在區(qū)分ASD兒童和TD兒童時(shí)聲學(xué)特征顯示出良好的分類準(zhǔn)確性(Pokorny et al., 2017, August; Santos et al., 2013, October)。然而, 這些研究均沒有報(bào)道最具有預(yù)測(cè)性的聲學(xué)信息。例如, Santos等人(2013)在前瞻性研究中對(duì)18個(gè)月ASD嬰兒和TD嬰兒的呀呀語(yǔ)、哭聲等發(fā)聲的聲學(xué)特征進(jìn)行分析, 并基于基頻、共振峰、音強(qiáng)等20多種聲學(xué)參數(shù)采用支持向量機(jī)(linear kernel support vector machines)和概率神經(jīng)網(wǎng)絡(luò)分類(probabilistic neural network classifiers)模型對(duì)兒童進(jìn)行分類, 后者分類準(zhǔn)確率達(dá)97%。Pokorny等人(2017)采用回溯性研究, 對(duì)年齡更小的10個(gè)月ASD嬰兒發(fā)聲的聲學(xué)特征進(jìn)行分析, 采用了包括基頻、振幅和頻譜及它們內(nèi)部更具體的88個(gè)參數(shù)的標(biāo)準(zhǔn)聲學(xué)參數(shù)集(Eyben et al., 2016), 并使用支持向量機(jī)和單層雙向長(zhǎng)短期記憶神經(jīng)網(wǎng)絡(luò)(1-layer bidirectional long short-term memory neural network)模型正確分類75%的ASD嬰兒和TD嬰兒?？梢? 研究者探究了不同類型和數(shù)量的聲學(xué)參數(shù)對(duì)ASD預(yù)測(cè)的準(zhǔn)確性, 但最優(yōu)預(yù)測(cè)聲學(xué)參數(shù)尚不明晰。

最近研究還發(fā)現(xiàn), 聲學(xué)參數(shù)集合在性別上可能存在差異。Khozaei等人采用音質(zhì)、頻譜等相關(guān)聲學(xué)參數(shù)對(duì)10名男性ASD兒童和10名男性TD兒童的哭聲進(jìn)行分類模型訓(xùn)練, 形成子集實(shí)例(subset instance)自動(dòng)學(xué)習(xí)分類模型; 然后采用該模型再對(duì)28～53個(gè)月語(yǔ)言能力處在12個(gè)月左右的ASD嬰幼兒和18～51個(gè)月的TD嬰幼兒進(jìn)行了分類, 結(jié)果發(fā)現(xiàn)對(duì)男性兒童分類的準(zhǔn)確率比女性兒童高7% (Khozaei et al., 2020)。此外, 聲學(xué)參數(shù)的元分析顯示聲學(xué)特征是ASD兒童非常有希望的早期識(shí)別標(biāo)志, 自動(dòng)學(xué)習(xí)模型中多個(gè)聲學(xué)參數(shù)比單個(gè)聲學(xué)參數(shù)判別的準(zhǔn)確率更高(Fusaroli et al., 2017)。因此, 未來的研究可以構(gòu)建具有最優(yōu)預(yù)測(cè)效果的聲學(xué)參數(shù)集合并建立自動(dòng)學(xué)習(xí)分類模型, 為ASD兒童的早期篩查提供智能的方法, 從而使得早期識(shí)別的可能性更高。

4.4 深入探究動(dòng)機(jī)對(duì)ASD兒童類言語(yǔ)發(fā)聲的影響

上文綜述指出, 類言語(yǔ)發(fā)聲是在內(nèi)在動(dòng)機(jī)驅(qū)動(dòng)下, 與成人社會(huì)互動(dòng)過程中逐步發(fā)展的。雖然, 近期有研究者開始嘗試探究?jī)?nèi)在動(dòng)機(jī)和社會(huì)動(dòng)機(jī)對(duì)ASD高風(fēng)險(xiǎn)嬰兒類言語(yǔ)發(fā)聲發(fā)展的影響(Long et al., 2021)。但總體上來說, 該領(lǐng)域的實(shí)證研究極其有限, 尤其是缺乏已診斷ASD兒童的動(dòng)機(jī)與類言語(yǔ)發(fā)聲關(guān)系的實(shí)證數(shù)據(jù)。首先, 未來可深入探究?jī)?nèi)在動(dòng)機(jī)對(duì)ASD兒童類言語(yǔ)發(fā)聲復(fù)雜性的作用機(jī)制。例如, ASD兒童對(duì)特殊興趣的內(nèi)在動(dòng)機(jī)是如何影響他們的類言語(yǔ)發(fā)聲復(fù)雜性的？計(jì)算機(jī)建模作為一種自動(dòng)化且高效的方法在TD嬰兒類言語(yǔ)發(fā)聲發(fā)展中得到應(yīng)用, 未來可以嘗試采用計(jì)算機(jī)建模探究?jī)?nèi)在動(dòng)機(jī)對(duì)ASD兒童類言語(yǔ)發(fā)聲復(fù)雜性的影響。其次, 探究社會(huì)動(dòng)機(jī)如何對(duì)ASD兒童類言語(yǔ)發(fā)聲復(fù)雜性產(chǎn)生影響。社會(huì)動(dòng)機(jī)使得兒童注意社會(huì)信號(hào)、得到社會(huì)回報(bào)進(jìn)而維持社會(huì)關(guān)系(Chevallier et al., 2012), 從而使兒童在社會(huì)互動(dòng)中習(xí)得更接近言語(yǔ)的發(fā)聲。ASD兒童的社會(huì)動(dòng)機(jī)缺陷以何種機(jī)制影響他們的類言語(yǔ)發(fā)聲復(fù)雜性的呢？未來有必要繼續(xù)開展相關(guān)研究。

4.5 進(jìn)一步探究ASD兒童類言語(yǔ)發(fā)聲異常的神經(jīng)機(jī)制

如前文所述, 神經(jīng)運(yùn)動(dòng)系統(tǒng)不成熟及其與言語(yǔ)聽覺感知皮層的相互影響, 可能是ASD兒童類言語(yǔ)發(fā)聲復(fù)雜性異常的重要原因。然而, ASD兒童類言語(yǔ)發(fā)聲的神經(jīng)機(jī)制尚不清晰。未來ASD兒童類言語(yǔ)發(fā)聲的神經(jīng)機(jī)制可嘗試從兩個(gè)方面進(jìn)行研究。一方面, 可嘗試從言語(yǔ)失用角度探究ASD兒童類言語(yǔ)發(fā)聲異常的神經(jīng)機(jī)制。失用癥是一種影響言語(yǔ)所需動(dòng)作協(xié)調(diào)能力的運(yùn)動(dòng)障礙(Akhtar et al., 2016)。研究表明言語(yǔ)失用(apraxia of speech)在24～55個(gè)月大ASD兒童中的患病率可能高達(dá)60% (Tierney et al., 2015)。因此, 最近有研究嘗試將提示伴有言語(yǔ)失用的平均年齡為56個(gè)月ASD兒童和年齡匹配的言語(yǔ)失用兒童神經(jīng)系統(tǒng)進(jìn)行比較。結(jié)果發(fā)現(xiàn)言語(yǔ)失用兒童的、對(duì)言語(yǔ)產(chǎn)生起重要作用的大腦結(jié)構(gòu)存在異常, 主要表現(xiàn)為頂葉(緣上回)和額葉(中央旁、三角部)皮質(zhì)體積增加; ASD兒童皮質(zhì)體積的增加主要分布在對(duì)社交溝通技能發(fā)展至關(guān)重要的額顳葉(Conti et al., 2020)。未來可納入無口語(yǔ)或口語(yǔ)較少的ASD兒童繼續(xù)開展相關(guān)研究。另一方面, 開展ASD兒童言語(yǔ)聽覺皮層與類言語(yǔ)發(fā)聲運(yùn)動(dòng)皮層之間相互作用的機(jī)制研究。對(duì)TD嬰兒的研究表明, 類言語(yǔ)發(fā)聲運(yùn)動(dòng)和聽覺感知之間的映射在嬰兒學(xué)會(huì)說話之前已存在, 3個(gè)月嬰兒的口部運(yùn)動(dòng)抑制對(duì)規(guī)范音節(jié)的聽覺辨別的神經(jīng)反應(yīng)有影響(Choi et al., 2021)。對(duì)ASD兒童該領(lǐng)域的深入探究可能能提供支持他們類言語(yǔ)發(fā)聲發(fā)展神經(jīng)機(jī)制的新證據(jù)。

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Potential early identification markers for children with autism spectrum disorder: Unusual vocalizations and theoretical explanations

LIU Min1,2, HU Yang1, LIU Qiaoyun1,2

(1Department of Rehabilitation Sciences, Faculty of Education, East China Normal University, Shanghai 200062, China)

(2200062,)

Early identification and early intervention of children with autism spectrum disorder (ASD) are critical to their developmental outcomes. Vocalizations are sounds produced by children before they learn to talk. Studies suggested that unusual nonspeech-like and speech-like vocalizations are potential early identification makers for children with ASD before the age of 2 years. The theoretical explanations for unusual vocalizations mainly include the motivation orientation theories, the neuromotor orientation theories, the perceptual orientation theory, and the social feedback orientation theory. Future research may consider in (1) exploring the possibility of unusual vocalizations as unique early identification markers for children with ASD, (2) strengthening the study of crying in early screening of children with ASD, (3) constructing an automatic learning classification model based on the strongest predictive acoustic parameters, (4) analyzing the influence of intrinsic and social motivation on speech-like vocalizations in children with ASD, and (5) investigating the neural mechanisms of unusual speech-like vocalizations. These evidence may be helpful for early identification and intervention of children with ASD.

autism spectrum disorder (ASD), potential early identification markers, unusual vocalizations, theoretical explanations

2021-07-05

*上海市浦江人才計(jì)劃項(xiàng)目(2019PJC033), 上海市長(zhǎng)寧區(qū)衛(wèi)生健康委員會(huì)項(xiàng)目(2019CNECNUPI05-1), 江蘇省教育科學(xué)“十三五”規(guī)劃課題(C-b/2018/01/09)資助。

劉巧云, E-mail: qyliu@spe.ecnu.edu.cn

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