Longitudinal analysis of communication repair skills across three neurodevelopmental disabilities

Abstract

BackgroundThis study is a longitudinal follow‐up to prior work examining the important pragmatic skill of communication repair (i.e., the ability to respond effectively to a request for clarification of an unclear message) across three neurodevelopmental disabilities in which language skills are impaired: fragile X syndrome with and without autism spectrum disorder (FXS‐ASD; FXS‐O), idiopathic ASD (ASD‐O), Down syndrome (DS) and controls with typical development (TD). Prior work examining communication repair skills at younger ages indicated impairments in boys with FXS‐ASD and ASD‐O, with females performing comparably with each other across groups.AimsTo characterize communication repair skills in young individuals with FXS‐ASD, FXS‐O, ASD‐O, DS and TD, across groups and over development. A secondary aim included documenting sex differences in FXS (with and without ASD) and DS.Methods & ProceduresSixty young individuals with FXS‐ASD (49 males, 11 females), 38 with FXS‐O (13 males, 25 females), 38 with ASD‐O (males only), 42 with DS (21 males, 21 females) and 41 with TD (21 males, 20 females) participated in the study, with a subsample reported on here who were retested at a second time point 2.7 years later on average. Participants completed a structured, picture‐based task designed to assess the ability to repair breakdowns in communication. Participants’ responses were compared across groups and sexes at the second time point, and interpreted with respect to previously published (Time 1) findings.Outcomes & ResultsKey findings included that, with age, male groups (including those with FXS‐ASD and ASD‐O, who showed difficulty at Time 1) performed more comparably, decreasing their use of inappropriate responses, in spite of relatively little change observed in general cognitive or structural language abilities in the clinical groups. However, girls with FXS and DS became more non‐responsive with age, and differences between boys and girls with FXS‐ASD emerged over time as well.Conclusions & ImplicationsFindings suggest that impairments in a critical pragmatic skill—the ability to repair communication breakdown—show significant change with age across three neurodevelopmental disabilities, with important sex‐specific patterns. These developments were often observed in spite of a relative plateau in cognitive and language growth, suggesting that repair skills may be more malleable and therefore an excellent target for intervention. Findings not only inform the nature of pragmatic impairment across groups but also can importantly inform clinical practice, suggesting that clinicians should monitor pragmatic skills such as repairs throughout development and also consider the role of sex in clinical efforts.

A longitudinal investigation of pragmatic language across contexts in autism and related neurodevelopmental conditions

“…There are several limitations of the current study that should inform future research. First, it is critical for future studies to examine whether the profiles identified here extend to females across these different neurodevelopmental conditions, given sex differences observed across clinical groups in previous studies (15,29,56,91,92). Sex-specific patterns are well documented in FXS, owing to females' second, unaffected X chromosome with normal FMR1-related protein expression and function.…”

“…Participants were part of a longitudinal study of pragmatic language development in which assessments were completed approximately once per year for up to three time points. Some children had fewer time points due to a "rolling enrollment" structure of the study where participants who enrolled later did not have time for multiple yearly visits before the study ended, as described previously (56). The number of participants receiving each assessment at each time point is summarized in Table 1.…”

Communication in fragile X syndrome: Patterns and implications for assessment and intervention

“…As in other areas, females with FXS frequently demonstrate less severe pragmatic impairment than males, although there is considerable variability (Abbeduto et al, 2008;Thurman et al, 2017a;Martin et al, 2020;Neal et al, 2022). Girls show deficits in signaling of non-comprehension as compared to TD peers matched on cognition, and there has been some research showing decreased responsivity in girls with FXS as they reach adolescence when asked to repair a communication breakdown (Thurman et al, 2017a;Martin et al, 2020).…”

“…As in other areas, females with FXS frequently demonstrate less severe pragmatic impairment than males, although there is considerable variability (Abbeduto et al, 2008;Thurman et al, 2017a;Martin et al, 2020;Neal et al, 2022). Girls show deficits in signaling of non-comprehension as compared to TD peers matched on cognition, and there has been some research showing decreased responsivity in girls with FXS as they reach adolescence when asked to repair a communication breakdown (Thurman et al, 2017a;Martin et al, 2020). Females with FXS who also meet criteria for ASD have been shown to be less likely to signal non-comprehension, initiate conversation, or make contingent remarks in conversation than those with FXS-O or individuals with DS and TD matched on nonverbal cognition.…”

Analysis of a Repetitive Language Coding System: Comparisons between Fragile X Syndrome, Autism, and Down Syndrome

“…Previous studies that have examined pragmatic language analysis using ELS have found that it distinguishes between various syndromes [8,10,[23][24][25]; however, some elements of pragmatic language have proven to be more challenging to characterize, including verbal self-repetition-also termed perseveration-which refers to the excessive self-repetition of a spoken word, phrase, sentence, or topic. Verbal self-repetition (hereafter, simply "repetitive language"), has been noted as prevalent in both ASD and FXS [26][27][28][29].…”