Center for Autism Research, Children's Hospital of Philadelphia, Roberts Center for Pediatric Research, 2716 South Street, Philadelphia, PA 19146 USA.
Perelman School of Medicine, University of Pennsylvania, 3400 Civic Center Blvd, Philadelphia, PA 19104 USA.
Mol Autism. 2017 Sep 30;8:48. doi: 10.1186/s13229-017-0164-6. eCollection 2017.
Autism spectrum disorder (ASD) is diagnosed more frequently in boys than girls, even when girls are equally symptomatic. Cutting-edge behavioral imaging has detected "camouflaging" in girls with ASD, wherein social behaviors appear superficially typical, complicating diagnosis. The present study explores a new kind of camouflage based on language differences. Pauses during conversation can be filled with words like UM or UH, but research suggests that these two words are pragmatically distinct (e.g., UM is used to signal longer pauses, and may correlate with greater social communicative sophistication than UH). Large-scale research suggests that women and younger people produce higher rates of UM during conversational pauses than do men and older people, who produce relatively more UH. Although it has been argued that children and adolescents with ASD use UM less often than typical peers, prior research has not included sufficient numbers of girls to examine whether sex explains this effect. Here, we explore UM vs. UH in school-aged boys and girls with ASD, and ask whether filled pauses relate to dimensional measures of autism symptom severity.
Sixty-five verbal school-aged participants with ASD (49 boys, 16 girls, IQ estimates in the average range) participated, along with a small comparison group of typically developing children (8 boys, 9 girls). Speech samples from the Autism Diagnostic Observation Schedule were orthographically transcribed and time-aligned, with filled pauses marked. Parents completed the Social Communication Questionnaire and the Vineland Adaptive Behavior Scales.
Girls used UH less often than boys across both diagnostic groups. UH suppression resulted in higher UM ratios for girls than boys, and overall filled pause rates were higher for typical children than for children with ASD. Higher UM ratios correlated with better socialization in boys with ASD, but this effect was driven by increased use of UH by boys with greater symptoms.
Pragmatic language markers distinguish girls and boys with ASD, mirroring sex differences in the general population. One implication of this finding is that typical-sounding disfluency patterns (i.e., reduced relative UH production leading to higher UM ratios) may normalize the way girls with ASD sound relative to other children, serving as "linguistic camouflage" for a naïve listener and distinguishing them from boys with ASD. This first-of-its-kind study highlights the importance of continued commitment to understanding how sex and gender change the way that ASD manifests, and illustrates the potential of natural language to contribute to objective "behavioral imaging" diagnostics for ASD.
自闭症谱系障碍（ASD）在男孩中的诊断率高于女孩，即使女孩的症状同样明显。前沿的行为影像学检测到 ASD 女孩存在“伪装”现象，即社交行为表面上看似典型，这使得诊断变得复杂。本研究探索了一种基于语言差异的新型伪装。对话中的停顿可以用 UM 或 UH 等词来填补，但研究表明这两个词在语用学上是有区别的（例如，UM 用于表示更长的停顿，并且可能与比 UH 更高的社交沟通复杂性相关）。大规模研究表明，女性和年轻人在对话停顿时比男性和老年人产生更高的 UM 率，而男性和老年人则相对更多地产生 UH。尽管有人认为 ASD 儿童和青少年使用 UM 的频率低于典型同龄人，但之前的研究并未纳入足够数量的女孩来检验性别是否解释了这一效应。在这里，我们探讨了 ASD 学龄期男孩和女孩的 UM 与 UH，并询问填充停顿是否与自闭症症状严重程度的维度测量有关。
65 名有 ASD 的学龄期言语参与者（49 名男孩，16 名女孩，智商估计处于平均范围）以及一小部分对照组的正常发育儿童（8 名男孩，9 名女孩）参加了研究。使用孤独症诊断观察量表的语音样本进行正字法转录和时间对齐，并标记填充停顿。父母完成社会沟通问卷和文兰适应性行为量表。
在两个诊断组中，女孩的 UH 使用频率均低于男孩。抑制 UH 会导致女孩的 UM 比值高于男孩，而典型儿童的总填充停顿率高于 ASD 儿童。较高的 UM 比值与 ASD 男孩的社交能力提高相关，但这种效应是由症状较重的男孩增加使用 UH 引起的。
语用语言标记区分了 ASD 女孩和男孩，反映了普通人群中的性别差异。这一发现的一个含义是，正常的不流畅模式（即相对 UH 产生减少导致 UM 比值升高）可能使 ASD 女孩的声音相对于其他儿童正常化，成为天真听众的“语言伪装”，并将其与 ASD 男孩区分开来。这项首例研究强调了继续致力于理解性别如何改变 ASD 表现方式的重要性，并说明了自然语言为 ASD 的客观“行为影像学”诊断做出贡献的潜力。
