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Exploring Communication Ability in Individuals with Angelman Syndrome: Findings from Qualitative Interviews with Caregivers

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Title: Exploring Communication Ability in Individuals with Angelman Syndrome: Findings from Qualitative Interviews with Caregivers
Language: English
Authors: Zigler, Christina K., Lucas, Nicole, McFatrich, Molly, Gordon, Kelly L., Jones, Harrison N., Berent, Allyson, Panagoulias, Jennifer, Evans, Paula, Reeve, Bryce B.
Source: American Journal on Intellectual and Developmental Disabilities. May 2023 128(3):185-203.
Availability: American Association on Intellectual and Developmental Disabilities. P.O. Box 1897, Lawrence, KS 66044-1897. Tel: 785-843-1235; Fax: 785-843-1274; e-mail: AJMR@allenpress.com; Web site: https://meridian.allenpress.com/aaidd
Peer Reviewed: Y
Page Count: 19
Publication Date: 2023
Document Type: Journal Articles
Reports - Research
Descriptors: Genetic Disorders, Communication Skills, Caregivers, Children, Expressive Language, Receptive Language, Pragmatics, Augmentative and Alternative Communication, Qualitative Research, Interviews
DOI: 10.1352/1944-7558-128.3.185
ISSN: 1944-7515
1944-7558
Abstract: Communication deficits have a substantial impact on quality of life for individuals with Angelman syndrome (AS) and their families, but limited qualitative work exists to support the necessary content of measures aiming to assess communication for these individuals. Following best practices for concept elicitation studies, we conducted individual qualitative interviews with caregivers and clinicians to elicit meaningful aspects of communication for individuals with AS. Caregivers were able to discuss their child's specific communication behaviors within a large number of expressive, receptive, and pragmatic functions via numerous symbolic and non-symbolic modalities. These results aligned well with published literature on communication in AS and will be used to inform the design of a novel caregiver-reported measure. Future studies on communication in individuals with AS should focus on gathering quantitative data from large samples of diverse caregivers, which would allow for estimations of the frequency of specific behaviors across the population.
Abstractor: As Provided
Entry Date: 2023
Accession Number: EJ1386849
Database: ERIC
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  Value: <anid>AN0163393724;[8z1j]01may.23;2024Aug16.14:17;v2.2.500</anid> <title id="AN0163393724-1">Exploring Communication Ability in Individuals With Angelman Syndrome: Findings From Qualitative Interviews With Caregivers </title> <p>Communication deficits have a substantial impact on quality of life for individuals with Angelman syndrome (AS) and their families, but limited qualitative work exists to support the necessary content of measures aiming to assess communication for these individuals. Following best practices for concept elicitation studies, we conducted individual qualitative interviews with caregivers and clinicians to elicit meaningful aspects of communication for individuals with AS. Caregivers were able to discuss their child's specific communication behaviors within a large number of expressive, receptive, and pragmatic functions via numerous symbolic and non-symbolic modalities. These results aligned well with published literature on communication in AS and will be used to inform the design of a novel caregiver-reported measure. Future studies on communication in individuals with AS should focus on gathering quantitative data from large samples of diverse caregivers, which would allow for estimations of the frequency of specific behaviors across the population.</p> <p>Keywords: Angelman syndrome; communication; qualitative methods; caregivers</p> <p>Angelman syndrome (AS) is a rare, neurogenetic disorder caused by loss-of-function of the maternally inherited allele of the <emph>UBE3A</emph> gene ([<reflink idref="bib25" id="ref1">25</reflink>]), resulting in deficits across multiple domains including communication, cognition, sleep, and mobility ([<reflink idref="bib3" id="ref2">3</reflink>]; [<reflink idref="bib5" id="ref3">5</reflink>]; [<reflink idref="bib15" id="ref4">15</reflink>]). Individuals with AS experience high unmet clinical needs ([<reflink idref="bib27" id="ref5">27</reflink>]), although recent and ongoing preclinical studies are demonstrating exciting breakthroughs ([<reflink idref="bib9" id="ref6">9</reflink>]; [<reflink idref="bib16" id="ref7">16</reflink>]; [<reflink idref="bib29" id="ref8">29</reflink>]). Understandably, deficits in expressive and receptive communication have a substantial impact on quality of life for these families ([<reflink idref="bib27" id="ref9">27</reflink>]; [<reflink idref="bib30" id="ref10">30</reflink>]), and caregivers have identified communication ability as the most important area to target when evaluating efficacy for novel therapeutics ([<reflink idref="bib28" id="ref11">28</reflink>]). Unfortunately, there is a lack of communication measures designed specifically for these individuals, and existing measures may not include appropriate content for this population based on the "striking and unusual communication profile" ([<reflink idref="bib12" id="ref12">12</reflink>]; [<reflink idref="bib21" id="ref13">21</reflink>], p. 1272).</p> <p>Most individuals with AS do not use words as a primary mode of communication ([<reflink idref="bib1" id="ref14">1</reflink>]; [<reflink idref="bib2" id="ref15">2</reflink>]; [<reflink idref="bib6" id="ref16">6</reflink>]; [<reflink idref="bib7" id="ref17">7</reflink>]; [<reflink idref="bib18" id="ref18">18</reflink>]; [<reflink idref="bib21" id="ref19">21</reflink>]). Instead, people with AS often use alternative modalities like gestures, non-speech vocalizations, physical manipulations, sign language, and augmentative and alternative communication (AAC) devices ([<reflink idref="bib1" id="ref20">1</reflink>]; [<reflink idref="bib6" id="ref21">6</reflink>]; [<reflink idref="bib10" id="ref22">10</reflink>]; [<reflink idref="bib12" id="ref23">12</reflink>]; [<reflink idref="bib14" id="ref24">14</reflink>]; [<reflink idref="bib22" id="ref25">22</reflink>]). Individuals with AS also may have more advanced receptive and pragmatic language skills that are seemingly unlinked to expressive language deficits or cognition ([<reflink idref="bib13" id="ref26">13</reflink>]; [<reflink idref="bib18" id="ref27">18</reflink>]; [<reflink idref="bib22" id="ref28">22</reflink>]). Taken together, the communication profile in individuals with AS is distinct from that of typically developing children, supporting the need for highly targeted and specific measurement tools.</p> <p>A truly AS-specific measure would include all the communication functions identified by parents as being important to their lives, align well with models of communication development, and account for the different modalities used by individuals within this population. Thus, this study's main goal was to conduct formative concept elicitation interviews with caregivers to obtain detailed information on key concepts that are relevant for understanding parental perception of communication ability in individuals with AS. To inform patient-centered practices broadly in this work, our study team included parents and relatives of individuals with AS, who are listed authors (A.S., P.E., & J.P.).</p> <hd id="AN0163393724-2">Methods</hd> <p></p> <hd id="AN0163393724-3">Participants</hd> <p></p> <hd id="AN0163393724-4">Caregivers</hd> <p>Caregivers or parents of individuals with AS were eligible for the study if they were ≥ 18 years, had the ability to read, speak, and understand English, could provide informed consent, and had access to a telephone for interviews. Their child had to be at least 2 years of age, and caregivers had to report that their child's diagnosis of AS was molecularly confirmed (e.g., DNA methylation test, FISH, CGH, or sequencing). The caregiver also had to live with the individual with AS and reside in the United States.</p> <p>Recruitment of caregivers was done in close partnership with an established patient advocacy organization, the Foundation for Angelman Syndrome Therapeutics (FAST). A recruitment invitation was posted to an existing Facebook page and circulated to other similar forums for parents of children with AS. Using snowball-sampling methods, FAST members could also distribute the recruitment flyer via email. Interested participants directly contacted the study team and were screened for eligibility.</p> <p>Enrollment of caregivers was stratified by their child's age using four groups to ensure representation of communication across children, adolescents, and adults. Ages of the individuals with AS were grouped as follows: 2–7, 8–12, 13–17, and ≥ 18 years old. In addition to stratification by age group, we targeted recruitment to achieve a representative mix of the genetic mechanism of AS (chromosomal deletion, UBE3A mutation, paternal uniparental disomy [UPD], or imprinting center defect [ICD]). The identified genotype has important implications for both severity and symptom presentation of AS ([<reflink idref="bib14" id="ref29">14</reflink>]; [<reflink idref="bib17" id="ref30">17</reflink>]). We also monitored caregiver race and ethnicity to support sample diversity.</p> <p>The final sample included 22 caregivers of individuals with AS (<emph>n</emph> = 6 each for ages 2–7 and 8–12 years; <emph>n</emph> = 5 each for ages 13–17 and 18+ years), with one caregiver screened as eligible but lost to follow-up prior to their interview. Caregivers were on average 45.7 years old (<emph>SD</emph> = 8.2) and were predominantly mothers/stepmothers (86%; Table 1) with 73% (16/22) of caregivers identifying as non-Hispanic White, two caregivers as Black, two as Hispanic/Latino, one as Asian, and one as American Indian/Alaskan Native, meaning around one third of caregivers in our sample were non-White or Hispanic. All caregivers reporting having at least some college/university education, with 11 completing their undergraduate degree and six reporting postgraduate degrees.</p> <p>PHOTO (COLOR)</p> <hd id="AN0163393724-5">Clinicians</hd> <p>To be eligible, clinicians were required to have an advanced degree in a communication-relevant field (e.g., speech-language pathology or augmentative and alternative communication (AAC) devices) and at least 2 years of experience working directly with individuals with AS. Eligible experts were identified by FAST study team members through their AS-specific networks and received a recruitment email from a member of the study staff. If an individual was interested in the study, they then contacted the study staff using the contact information in the recruitment email to obtain more information. Study staff conducted a screening visit, where eligibility of the clinicians were confirmed and additional demographics collected. After enrollment into the study, the qualitative interviews were scheduled and conducted.</p> <p>In the final sample of six clinicians, five were women, average age 42.8 years, four had master's degrees and two had doctoral degrees. Of the individuals with Master's degrees, four indicated their title was speech-language pathologist (SLP) with one clinician reporting an additional title as complex communication needs specialist. The two clinicians with doctoral degrees were faculty members at academic institutions. On average, clinicians had about 10.5 years of experience working with individuals with AS (median = 7 years; range = 4–30 years). All indicated they had experience with augmentative and alternative communication (AAC) devices. When asked about average caseloads, three clinicians reported seeing around five individuals with AS per year, and the other three reported upwards of 15 individuals per year.</p> <hd id="AN0163393724-6">Interview Script Creation</hd> <p>After a review of the literature, semi-structured interview scripts were created for both caregivers and clinicians (see https://populationhealth.duke.edu/supplemental-files for copies of interview scripts utilized in this study). The caregiver interview script started with asking caregivers to describe the types of communication they see in their child on a typical day. Interviewers then queried around <emph>why</emph> and <emph>how</emph> individuals with AS communicate within expressive, receptive, and pragmatic communication functions. Caregivers were also asked about what would constitute "meaningful" changes in communication for their child. The resulting structure of the interview guide aligned with published models of communication development ([<reflink idref="bib8" id="ref31">8</reflink>]; [<reflink idref="bib20" id="ref32">20</reflink>]) and existing communication measures ([<reflink idref="bib10" id="ref33">10</reflink>]; [<reflink idref="bib24" id="ref34">24</reflink>]). The wording of the questions reflected a strength-based approach ("How does your child ...") in contrast to a deficit-based approach ("Does your child ..."). This choice was deliberate and based on discussions with relatives of individual with AS, to allow caregivers to respond in an open-ended format.</p> <p>The interview script for clinicians was developed in a similar fashion. Clinicians were asked to describe how individuals with AS typically communicate and what these individuals usually communicate about. Like the caregivers, clinicians were also asked about what constitutes meaningful changes in communication.</p> <hd id="AN0163393724-7">Data Collection</hd> <p>The relevant institutional review board approved all study procedures prior to initiation, and their review conformed to recognized standards including the Declaration of Helsinki and the U.S. Federal Policy for the Protection of Human Subjects. Participants provided verbal consent prior to the interviews. Due to the rare nature of this condition and to ensure geographical representation, all interviews were conducted over the phone. Interviewers (M.M., N.L., & C.Z.) were trained in qualitative methods and on the specific interview guide, and were not formally a part of any AS-specific organization. With permission from study participants, interviews were audio recorded and transcribed. The interviews lasted 60–90 min, and interviewers took detailed field notes that were expanded immediately following completion of the interview using a semi-structured debriefing form.</p> <p>We planned to conduct approximately 20 interviews with caregivers (five caregivers in each of the four age groups), and 5 interviews with clinicians, understanding that more interviews might be needed to recruit caregivers of individuals with more rare genotypes. Based on initial review of our sample, we recruited one additional caregiver to ensure representation from UPD and ICD genotypes in each of the youngest age groups (2–7 and 8–12 years old). We chose to enroll caregivers and clinicians to provide complementary information; caregivers could talk in detail about their own child's communication in multiple settings (e.g., home, community) and over time, whereas clinicians could speak more generally across individuals they had worked with and their knowledge of AS, communication development, and communication disorders.</p> <hd id="AN0163393724-8">Analysis</hd> <p>The goals of analysis were to (<reflink idref="bib1" id="ref35">1</reflink>) confirm the major communicative functions relevant to caregivers within expressive, receptive, and pragmatic communication, and (<reflink idref="bib2" id="ref36">2</reflink>) describe a wide breadth of communication behaviors within each function. Secondary goals were to examine the use of different modalities (e.g., verbal speech, gestures) and meaningful change in communication from the perspectives of caregivers and clinicians. It is important to note, that the goal of this analysis was not to speak to the frequency of specific communication behaviors in the larger population of individuals with AS or to identify developmental trends.</p> <p>Thematic analysis was performed on all transcripts using the framework proposed by Nowell ([<reflink idref="bib19" id="ref37">19</reflink>]). Thematic analysis is a method that is used for identifying, organizing, describing, and reporting themes ([<reflink idref="bib4" id="ref38">4</reflink>]). Nowell's framework supports trustworthiness of thematic analysis specifically through well-structured procedures that support summarization of findings ([<reflink idref="bib19" id="ref39">19</reflink>]). As fully described in Table 2, three analysts participated in the five phases of research including familiarization with the data, generating initial codes, searching for themes, reviewing themes, defining and naming themes, and producing reports (Nowel et al., 2017). The final codebook included 19 codes describing specific modalities ("words," "gestures," and "AAC Devices") as well as larger areas of communication ("expressive," "receptive," "pragmatic"). The broader communication codes also had related subcodes, representing more targeted communication functions (e.g., "expressive" => "refuse"; see https://populationhealth.duke.edu/supplemental-files for full codebook and descriptions of all codes). The goals of our analysis were primarily descriptive (and not interpretive), to report examples using the caregivers' own words.</p> <p>PHOTO (COLOR)</p> <p>Please note, we report frequency counts for specific aspects of the interview that every participant was queried about in the same way (i.e., meaningful change and number of words). For observable behaviors within a communication function, as is traditional for thematic analysis, we selectively and intentionally use semiquantitative terms (e.g., "a few", "many", "some"), when we want to reflect patterns seen in the data. Caregiver interviews were the primary data for analysis, clinician interviews were utilized to expand on and confirm concepts identified by caregivers. Thus, when we report data that was obtained from clinician interviews, this is specifically stated.</p> <hd id="AN0163393724-9">Results</hd> <p></p> <hd id="AN0163393724-10">Overview</hd> <p>Caregivers confirmed a number of communication functions relevant to individuals with AS and were able to describe a number of observable communication behaviors associated with each function. For ease of review, the results are organized by expressive, receptive, and pragmatic communication (Figure 1), with tables including additional quotations (Tables 3–5).</p> <p>PHOTO (COLOR): Figure 1 Conceptual Model of Communication Ability Based on Themes Identified by Caregivers and Clinicians During Qualitative Interviews Note. The grey oval highlights a concept that may or may not be related to "communication ability." Please see text for more discussion.</p> <p>PHOTO (COLOR)</p> <p>PHOTO (COLOR)</p> <hd id="AN0163393724-11">Expressive Communication</hd> <p>Expressive communication was defined as specific communication interactions where the individual with AS was communicating something to their communication partner (most often, their caregiver).</p> <hd id="AN0163393724-12">Requesting</hd> <p>Caregivers reported that individuals with AS requested a variety of objects and activities, including food, attention, entertainment (i.e., music, TV shows), places (outside, pool, store, etc.), baths, and social games. Individuals with AS could be skilled at requesting a small number of things that they specifically enjoyed or that were motivating for them (e.g., their favorite food, a TV show, or a game they like to play). A few children could request specific things that were not in the same room or that they could not see.</p> <p>For nonspecific communication attempts like crying, caregivers reported using context clues to help decipher what it is the child truly wanted. For example, caregivers mentioned they used context like the time of day or physical location (e.g., kitchen) to know that their child wanted food. "So, if it's 11:00 a.m. or 12:00 p.m. and she's fussing, then I know that she wants lunch." Caregivers also looked for confirmatory behaviors to help determine if their child's request was met (i.e., smiling, nodding yes, or squealing in excitement).</p> <hd id="AN0163393724-13">Seeking Attention</hd> <p>Individuals with AS sought attention from their caregiver using sounds, their body, or their AAC device. For example, one caregiver of an individual who was ambulatory stated, "He will usually approach us and tap us, or pull on our pant legs if we're standing." Caregivers reported that their child would seek their attention before communicating a request.</p> <hd id="AN0163393724-14">Requesting More of Something</hd> <p>Caregivers reported children requested "more" of something using different behaviors including reaching, pointing, vocalizations, specific signs/gestures, or by using their AAC device. One caregiver said,</p> <p>I would usually ask him, "Do you want more?" and he'll either – he might do that little head nod thing with his body – "Yes" – or he might push his plate towards you. He might just reach for whatever it is.</p> <p>Another caregiver reported use of symbolic language to request "more." "He makes the 'more' sign language. You sort of hit your two fists together in the front, and occasionally he will use the more button on his device or if he can see the item."</p> <hd id="AN0163393724-15">Toileting</hd> <p>There was variation in the sample around observed communication behaviors about toileting. Some examples of communication around toileting included using an AAC device or tapping on their front or backside to share they needed to use the bathroom or needed a diaper change. There also was heterogeneity in the relevance of the concept to different families, which varied based on the priority caregivers placed on expressing needs around toileting (high/low), how intensely caregivers were reinforcing these skills with their child, and different levels of independence the individual had around toileting (being fully potty-trained vs. on a timed schedule vs. not being potty-trained). When clinicians were asked about this concept, they reported that when nonsymbolic communication modalities were being used by the child, it is often not clear if the child was intentionally communicating or if the caregiver was identifying the individual's feelings of discomfort.</p> <hd id="AN0163393724-16">Directing Attention</hd> <p>Caregivers reported individuals with AS using physical means to direct their attention. Examples of this included grabbing the caregiver's hand and bringing them to something, turning the caregiver's face, and handing something of interest to the caregiver. One caregiver said,</p> <p>She may point if she's wanting to show me on her iPad, she's watching kids at a water park, and she'll use her voice to get my attention and then point to that. If I still don't look because I'm doing something else or talking to someone, she may tap me and/or turn my head by pulling my chin to what it is she wants me to look at.</p> <p>Other observed behaviors for directing attention included pointing to the object/action of interest and vocalizing/squealing ("ah ah!" or "eh eh!"). Some caregivers believed that their child could not direct attention or could not think of examples of directing attention.</p> <hd id="AN0163393724-17">Refusal</hd> <p>Individuals with AS refused objects or activities for a variety of reasons and used a variety of different methods. The most common refusal behavior reported by caregivers was pushing an object away. Similarly, individuals with AS also might refuse something by walking/moving away from the object or person, whining, or fussing. Another example caregivers provided was use of a symbolic "no." One caregiver stated, "She does a sign for, 'No I don't want it.' She'll shake her head, 'No.'" We also heard caregivers discuss use of an AAC device to indicate refusal or use of a word/word approximation like "No" or "Nah." Caregivers could identify and discuss these nonsymbolic and symbolic forms of refusal and some reported they were working specifically to build and reinforce these skills.</p> <hd id="AN0163393724-18">Commenting</hd> <p>Commenting was defined as remarks expressing opinions or reactions. Typically, comments that caregivers of individuals with AS identified were simple things like a laugh, smile, or vocalization to express happiness or excitement. Happy vocalizations came in the forms of shrieks or high-pitched babbles. Conversely, caregivers reported individuals with AS also made low-pitched vocalizations (i.e., whines, cries, or growls) or used facial expressions (scowling/frowning) to comment that they were unhappy or "displeased" with someone or something. In both cases, these reactions were used by the caregiver to identify their own understanding of the child's communication (e.g., to determine if they responded appropriately to a request). Some, not all individuals, were able to tell simple stories about something that happened in the past (using a device or gestures). For caregivers who could describe this type of communication, there often was significant interpretation that occurred on behalf of the communication partner using contextual cues. For example, on caregiver said,</p> <p>The cat coughed up a hairball in the middle of the floor and it was dark in the room, [the child] stepped in it and I heard her fuss and then she walked out, found me, held her foot up and pointed to it. And then she proceeded to show me exactly where the cat threw this hairball up at.</p> <hd id="AN0163393724-19">The Importance of Communication Modality</hd> <p>Caregivers confirmed that individuals with AS used one or more of the following communication modalities: sounds/vocalizations, words or word approximations, signs or gestures (including modified gestures), and AAC devices. Caregivers reported that individuals with AS use these modalities across communication areas (e.g., expressive, receptive, and pragmatic) in varying degrees, and this was confirmed by clinicians. Some caregivers reported the use of multiple modalities within a specific function (e.g., using vocalizations, gestures, and body movements to seek attention). Individuals who could utilize symbolic forms of communication, like words/word approximations and AAC devices, were able to communicate more sophisticated messages, like answering open-ended questions, making requests for something out of view, or telling stories.</p> <hd id="AN0163393724-20">Finger Point</hd> <p>Caregivers and clinicians reported the importance of the use of finger points in advancing communication skills across a number of functions. For example, individuals might reach toward a general area to let their caregiver know they wanted something or utilize a finger point:</p> <p>She's getting better with her pointing; we've been working on her with that because a lot of times it's just the hands opening and closing at a specific item to lets us know that she wants that item. Now, she's advanced to where she can finger point to that specific item.</p> <hd id="AN0163393724-21">Words/Word Approximations</hd> <p>About half of caregivers reported that their child used words or word approximations; however, only four caregivers said their child had more than three words that they used consistently. Of note, only one child in the 2–7 age group used any words or word approximations. The most consistently used word/word approximation across all age groups was "mama." Caregivers also talked about words/word approximations that were "lost" over time. One caregiver stated, "She used to say 'dad' when she was younger but then about three years ago, she started saying mom, and she doesn't say dad anymore."</p> <hd id="AN0163393724-22">AAC Devices</hd> <p>Use of an AAC device was common in our sample in at least some capacity, whether high tech (e.g., iPad with an app) or low tech (e.g., picture book). Although we did not explicitly examine cohort effects, there were differences in device use between older and younger individuals with AS in this sample. For example, a caregiver of a child in the 2–7 years age group said, "He went through this huge spike of being able to communicate so much more with the introduction of the device." However, caregivers of adults with AS typically reported introduction later in life and less positive experiences: "Everybody was trying really hard to get him to use it. ... It just didn't really click with him." When caregivers spoke about their child's level of mastery with the device, they mentioned various factors; how much modeling the support system was doing with the child, the child's motivation to use the device, the years using the device, the robustness of the device, and the child's fine motor abilities.</p> <hd id="AN0163393724-23">Receptive Communication</hd> <p>Receptive communication is the process of understanding a message expressed by a communication partner. By definition, it is less directly observable than expressive communication. Thus, in describing examples of receptive communication, we focused on observable behaviors that indicated understanding after a specific prompt (Table 4).</p> <p>PHOTO (COLOR)</p> <hd id="AN0163393724-24">Responding to Name</hd> <p>Almost all caregivers reported their child would respond to their name in some fashion. Examples of other observed responses included physically coming to the caregiver, making a sound, looking up, or making eye contact with the communication partner. One caregiver said, "If you call him or say his name, he'll look up and look at whoever just said his name or called him. He will respond to that. He may make eye contact with you to see – 'What?'"</p> <hd id="AN0163393724-25">Answering Questions</hd> <p>When asked to respond to a <emph>yes</emph> or <emph>no</emph> question (e.g., do you want to go to the park? Do you want a banana?), caregivers in our sample were able to report if their child could make a choice using at least one modality (e.g., a head nod, an informal gesture, or sound like clapping or squealing), but this behavior was less common than other functions. Other examples of responding to questions can be found in Table 4.</p> <hd id="AN0163393724-26">Making Choices</hd> <p>Some caregivers in our sample said their child was able to consistently make personal choices, and caregivers reported using different methods to assist their child in making these choices. For example, one caregiver said,</p> <p>We actually put two or three food items or snacks in front of her and ask her to choose one ... we just set them there and then let her just pick it up. We tell her to pick it up, whichever one she wants, and that's what she does.</p> <p>Other caregivers reported that their child would utilize their device to make a choice.</p> <p>I'll say, what do you want to play, tell me what you want to play, and he'll go to the play button in his iPad where a bunch of choices exist and he'll pick the thing that he wants to do.</p> <hd id="AN0163393724-27">Responding to Directions</hd> <p>Caregivers reported children understanding the word "no" (a very simple direction), with less frequent reports of understanding more complex directions. One caregiver shared examples of simple directions that their child follows, "If I say the word 'bath', he's off to the bathroom. He understands that."</p> <p>Motivation played a role in caregivers being able to observe receptive communication behaviors, and could influence whether an individual with AS would respond appropriately to a communication partner and indicate understanding. One caregiver said,</p> <p>Like if she wants my cell phone and it's on the floor, I can say, "Hey, my cell phone's on the floor," and ... she would go over and pick it up because she wanted it. But if I was like, "pick up your toy"... she wouldn't do it.</p> <hd id="AN0163393724-28">Understanding Isolated Words</hd> <p>Caregivers reported that individuals with AS were able to indicate their understanding of familiar words when overhearing their caregiver speak about something that mattered to them. For example, one caregiver was speaking about their child's love of their favorite food and how she could not say the word aloud because the child would demand it immediately. Caregivers also reported that their child indicated name recognition by responding with excitement when someone mentioned the name of a person they like.</p> <hd id="AN0163393724-29">Taking Turns in Conversation</hd> <p>Caregivers reported examples of their child taking turns in conversations and expressed enjoyment around these interactions with their child. For example,</p> <p>When [name] babbles, I talk back to her. I try to figure out what she's babbling what she goes, "Buh-buh-bub," or "Guh-buh-buh" and then point to something. I go, "Oh. So, you like this movie?" And she'll do the nod or not a nod and it's really cool.</p> <p>Another caregiver provided an example of their child responding back to her with a question or comment: "If I say 'Dad,' ... she might say, 'When? When will I see Dad?' Or [name] – if I say '[name],' she might say, 'When?' or she might say 'movie.' She likes to watch a movie with [name]."</p> <hd id="AN0163393724-30">Pragmatic Communication</hd> <p>Social communication was described by both caregivers and clinicians as a strength of individuals with AS (Table 5). Caregivers reported their child greeted others and played familiar games (e.g., peekaboo). For older individuals, caregivers discussed their child's requests for familiar activities they may enjoy with others, like swimming. Other social communication functions that were described by caregivers included the use of specific names for people like siblings, cousins, or aids, using specific signs, symbols on their devices, or vocalizations. Some caregivers also reported that their child would comfort others if they noticed they were upset; although, this was only described by a small number of caregivers.</p> <hd id="AN0163393724-31">Meaningful Change</hd> <p>Caregivers and clinicians all provided multiple answers for what would represent meaningful change; responses were aggregated into nine overlapping themes. The most frequently mentioned themes focused on complex communication, initiation of communication, communicating with people, verbal speech, and being able to communicate when hurt or sick. Examples of all themes can be found in Table 6.</p> <p>PHOTO (COLOR)</p> <p>Generally, caregivers and some experts mentioned that a meaningful change for an individual with AS would be gaining more complex communication skills. Some participants defined these skills in terms of more complex linguistic skills, like increased words per sentence or vocabulary. Other caregivers mentioned that this change could be independent of modality. The development of speech was also frequently mentioned by caregivers; however, most caregivers tempered these comments and suggested this goal was likely out of reach. For example, one caregiver expressed that a full vocabulary would be "amazing," but it was more important that their child used some words that "everyone could understand."</p> <p>Initiating communication was also a popular theme, which was often discussed in terms of initiating communication on an AAC device. Caregivers wished that their child would initiate use of the device in multiple settings and for multiple reasons. Relatedly, one expert discussed meaningful change as a child understanding the value in communication. In their view, "increased association of using the device and receiving desired stimulus" would reflect this understanding.</p> <p>Both caregivers and experts mentioned that the ability to communicate with other people (outside primary caregivers/family members/teachers) would be meaningful. Examples included more communication at school, with peers, with less familiar family members, or in other environments. Caregivers and experts also mentioned that social 'back-and-forth' conversation skills (i.e., "turn-taking") are really important to social well-being, even if this is entirely nonverbal.</p> <hd id="AN0163393724-32">Discussion</hd> <p>To our knowledge, this is one of the first systematic, qualitative explorations of communication ability in individuals with AS from the perspective of caregivers with an explicit focus on future measurement development. Caregivers could clearly articulate how their child communicated in the areas of expressive, receptive, and pragmatic communication, supporting multiple relevant concepts that could be included in future measures (Figure 1). Caregivers in the sample also reported a spectrum of communication behaviors within the identified functions, indicating variability within this population.</p> <p>Our results support the unique communication profile of individuals with AS identified by previous research ([<reflink idref="bib1" id="ref40">1</reflink>]; [<reflink idref="bib6" id="ref41">6</reflink>]; [<reflink idref="bib10" id="ref42">10</reflink>]; [<reflink idref="bib21" id="ref43">21</reflink>]; [<reflink idref="bib24" id="ref44">24</reflink>]). Data from both caregivers and clinicians confirmed the importance of considering alternative modalities used by individuals with AS when communicating. Words and word approximations were not common, and even when individuals used words (e.g., "mama"), they often did not rely on them for many communication functions. Any future measurement tool for this population must consider the implications of these alternative modalities and their ability to increase communication ability in the absence of verbal speech.</p> <p>In the eyes of caregivers, meaningful changes in communication consisted primarily of improvements in language complexity with a focus placed on verbal speech. Caregivers also prioritized allowing their child to be more universally understood by others and to have more freedom of expression. Clinicians also discussed these themes. Almost all caregivers in our sample reported multiple changes that could be "meaningful," including behaviors that were very specific to the family situation. Thus, any future explorations of meaningful change in communication ability within clinical trials should allow for multiple areas of improvement.</p> <p>It is important to note that recruitment occurred through collaboration with one patient advocacy organization. Strong partnerships with patient advocacy organizations are an invaluable asset to research done in rare disease populations but also have limitations ([<reflink idref="bib23" id="ref45">23</reflink>]; [<reflink idref="bib26" id="ref46">26</reflink>]). In our study, we utilized a purposeful sampling approach, stratifying our enrollment by age group, and including specific recruitment targets for genotype and race/ethnicity with the goal of diversity across these variables. Although we met our prespecified targets for enrollment, our sample does not fully represent all subgroups of caregivers and individuals with AS. Importantly, our sample consists of caregivers with overall high levels of education and only two fathers and one female legal guardian. Future work should continue to carefully gather validity evidence with diverse samples.</p> <hd id="AN0163393724-33">Conclusion</hd> <p>In this work, we describe caregiver identified communication behaviors within expressive, receptive, and pragmatic functions (Figure 1) and through numerous symbolic and non-symbolic modalities for individuals with AS, supporting the utility of caregiver report. These qualitative findings can be used to inform the development an AS-specific measure capturing communication from the caregiver's perspective for use in clinical trials. Future work should include also exploration of the frequency of communication behaviors in the larger population of individuals with AS, estimate developmental trajectories for communication, and confirm results in a larger sample of diverse caregivers.</p> <p> <emph>This study was presented at the International Society for Quality of Life Research (ISOQOL) 26th Annual Conference (abstract & poster). We would like to express our deep gratitude to the caregivers of individuals with Angelman syndrome and the communication professionals who participated in this work. This study was funded by the Foundation for Angelman Syndrome Therapeutics (FT2018-005)</emph>.</p> <ref id="AN0163393724-34"> <title> References </title> <blist> <bibl id="bib1" idref="ref14" type="bt">1</bibl> <bibtext> Alvares, R. L., & Downing, S. F. (1998). A survey of expressive communication skills in children with Angelman syndrome. 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  Data: Exploring Communication Ability in Individuals with Angelman Syndrome: Findings from Qualitative Interviews with Caregivers
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  Data: <searchLink fieldCode="AR" term="%22Zigler%2C+Christina+K%2E%22">Zigler, Christina K.</searchLink><br /><searchLink fieldCode="AR" term="%22Lucas%2C+Nicole%22">Lucas, Nicole</searchLink><br /><searchLink fieldCode="AR" term="%22McFatrich%2C+Molly%22">McFatrich, Molly</searchLink><br /><searchLink fieldCode="AR" term="%22Gordon%2C+Kelly+L%2E%22">Gordon, Kelly L.</searchLink><br /><searchLink fieldCode="AR" term="%22Jones%2C+Harrison+N%2E%22">Jones, Harrison N.</searchLink><br /><searchLink fieldCode="AR" term="%22Berent%2C+Allyson%22">Berent, Allyson</searchLink><br /><searchLink fieldCode="AR" term="%22Panagoulias%2C+Jennifer%22">Panagoulias, Jennifer</searchLink><br /><searchLink fieldCode="AR" term="%22Evans%2C+Paula%22">Evans, Paula</searchLink><br /><searchLink fieldCode="AR" term="%22Reeve%2C+Bryce+B%2E%22">Reeve, Bryce B.</searchLink>
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  Data: <searchLink fieldCode="SO" term="%22American+Journal+on+Intellectual+and+Developmental+Disabilities%22"><i>American Journal on Intellectual and Developmental Disabilities</i></searchLink>. May 2023 128(3):185-203.
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  Data: American Association on Intellectual and Developmental Disabilities. P.O. Box 1897, Lawrence, KS 66044-1897. Tel: 785-843-1235; Fax: 785-843-1274; e-mail: AJMR@allenpress.com; Web site: https://meridian.allenpress.com/aaidd
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  Data: 19
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  Data: Journal Articles<br />Reports - Research
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  Data: <searchLink fieldCode="DE" term="%22Genetic+Disorders%22">Genetic Disorders</searchLink><br /><searchLink fieldCode="DE" term="%22Communication+Skills%22">Communication Skills</searchLink><br /><searchLink fieldCode="DE" term="%22Caregivers%22">Caregivers</searchLink><br /><searchLink fieldCode="DE" term="%22Children%22">Children</searchLink><br /><searchLink fieldCode="DE" term="%22Expressive+Language%22">Expressive Language</searchLink><br /><searchLink fieldCode="DE" term="%22Receptive+Language%22">Receptive Language</searchLink><br /><searchLink fieldCode="DE" term="%22Pragmatics%22">Pragmatics</searchLink><br /><searchLink fieldCode="DE" term="%22Augmentative+and+Alternative+Communication%22">Augmentative and Alternative Communication</searchLink><br /><searchLink fieldCode="DE" term="%22Qualitative+Research%22">Qualitative Research</searchLink><br /><searchLink fieldCode="DE" term="%22Interviews%22">Interviews</searchLink>
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  Data: 10.1352/1944-7558-128.3.185
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  Data: Communication deficits have a substantial impact on quality of life for individuals with Angelman syndrome (AS) and their families, but limited qualitative work exists to support the necessary content of measures aiming to assess communication for these individuals. Following best practices for concept elicitation studies, we conducted individual qualitative interviews with caregivers and clinicians to elicit meaningful aspects of communication for individuals with AS. Caregivers were able to discuss their child's specific communication behaviors within a large number of expressive, receptive, and pragmatic functions via numerous symbolic and non-symbolic modalities. These results aligned well with published literature on communication in AS and will be used to inform the design of a novel caregiver-reported measure. Future studies on communication in individuals with AS should focus on gathering quantitative data from large samples of diverse caregivers, which would allow for estimations of the frequency of specific behaviors across the population.
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