Abstract
Keywords
Optic nerve hypoplasia (ONH), a congenital condition characterized by underdevelopment of the optic nerve, is the chief cause of permanent congenital visual impairment in the United States (Hatton et al., 2007). ONH is estimated to affect up to 17.3 per 100,000 children (Tear Fahnehjelm et al., 2014), a 2.5-fold increase in prevalence over 2 decades previously (Blohme & Tornqvist, 1997). The etiology and pathogenesis of ONH remain largely unknown, and no definite genetic causes have been identified (Garcia-Filion & Borchert, 2013b). ONH is a nonprogressive condition in which axons are missing in the optic nerve and brain, particularly in the hypothalamus (Fink et al., 2015). Neurodevelopmental disorders such as autism spectrum disorder (ASD) often co-occur with ONH (Dahl et al., 2018; Fink & Borchert, 2011; Garcia-Filion & Borchert, 2013a). These children with co-occurring visual impairment and neurodevelopmental disorders present with many complex medical and academic needs (Kancherla et al., 2013).
There is limited data available about the educational experiences of children with visual impairments; however, some research suggests that their educational needs may not be fully recognized or addressed by school personnel (Sapp & Hatlen, 2010). Children with multiple disabilities who do not receive appropriate educational services are at high risk for developing social, emotional, and behavioral problems (Lehman, 2013). Further, litigation related to challenges in developing education programs for children with ASD is both costly and on the rise (Ruble et al., 2010).
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All children receiving special education services are required to have an individualized education program (IEP), developed by a multidisciplinary team and informed by a comprehensive assessment of the students’ strengths and weaknesses. The IEP tracks the child's progress toward identified learning goals and objectives (Individuals with Disabilities Education Act [IDEA], 2004; Ruble et al., 2010). National organizations have developed comprehensive guidelines regarding educating children with visual impairments (Huebner et al., 2004; Sapp & Hatlen, 2010) and with ASD (National Research Council [NRC], 2001), which should be reflected in the student's IEP when appropriate to address an individual student's needs. An expanded core curriculum for children with visual impairments recommends adding domains beyond those typically covered by the standard academic curriculum (Huebner et al., 2004). Similarly, many students with autism will need educational support and interventions in the areas of social-emotional skills, communication skills, task engagement, work skills, as well as adaptive, cognitive, and motor skills (NRC, 2001; Ruble et al., 2010). A study of school outcomes for children with ASD found that IEP quality predicted IEP goal attainment (Ruble & McGrew, 2013). Gense and Gense (2011) proposed an expanded core curriculum for students with both autism and visual impairment, including skills of engagement, communication skills, play and social skills (including recreation and leisure), adaptive skills, organization skills, orientation and mobility and purposeful movement skills (including sensory and motor skills), career and life education skills, and self-advocacy skills.
Despite these national guidelines, children with visual impairments or ASD or both may not consistently receive adequate academic services (Anderson et al., 2018; Brown et al., 2013; Iadarola et al., 2015; Little & Saunders, 2015). Further, there is limited literature on the adequacy of academic services for students with co-occurring visual impairments and ASD. The purpose of the current study was to determine the extent to which the IEPs of children diagnosed with ONH with or without ASD meet their developmental and educational needs.
Method
Participants
Children's Hospital Los Angeles is the site of the largest prospective registry of children with ONH in the world. Since 1992, more than 400 children with ONH diagnosed prior to 2 years of age have been enrolled. They are followed annually for ophthalmic examination until age 5. Thereafter, they are followed as needed by parental choice. From this cohort, children aged 5 to 11 and seen for ophthalmic examination between August 2017 and January 2018 were recruited for this study. Out of 16 eligible subjects, 14 consented to participate in this study and 13 completed all study procedures (81% of those eligible).
Measures
Developmental Measures
The Autism Diagnostic Observation Schedule, 2nd Edition (ADOS-2; Lord et al., 2012) is a semi-structured, standardized observational assessment of social communication, reciprocal social communication, and restricted and repetitive behaviors. The Autism Diagnostic Interview-Revised (ADI-R; Rutter et al., 2003) is a semi-structured diagnostic parent interview covering the same domains. Modifications to the measures were made to address visual impairment, following the protocol developed and piloted by Williams et al. (2014) based on consultation with experts on children with visual impairments and approval from the publisher.
The Wechsler Intelligence Scale for Children, 5th Edition (WISC-V; Wechsler, 2014) is an individual measure of intellectual functioning for children ages 6 through 16 years; the Verbal Comprehension Index (VCI) was utilized. The VCI subtests are administered verbally and do not include any visual component. The standard score for the VCI provided a measure of verbal intellectual functioning and identified those participants functioning below the first percentile. Participants who were unable to complete the sample items for a subtest were considered to have functioned below the first percentile.
Vision Level
The best-corrected visual acuity of the better eye was used for assessing level of vision. Visual function was determined using Snellen letter recognition, or linear Allen figure recognition for participants unable to identify letters. In participants with a visual acuity of less than 20/800, a tumbling “E” was used.
IEP Coding Measure
The authors developed a measure to code the IEPs in order to quantify information about the child's education plan, including the extent to which the IEPs coincided with two sets of recommendations: (a) the Expanded Core Curriculum for Blind and Visually Impaired Children and Youths (Huebner et al., 2004; Sapp & Hatlen, 2010), which recommends goals or accommodations in the following domains: assistive technology, career education, compensatory academic skills, independent living skills, orientation and mobility, recreation and leisure, self-determination, sensory efficiency, and social interaction skills; (b) recommendations developed by the NRC's Committee on Educational Interventions for Children with Autism (NRC, 2001), including goals covering language and communication, social skills, appropriate behaviors, academic skills, and fine and gross motor skills. Finally, the authors consulted with a teacher who specializes in children with visual impairments, a special education teacher who specializes in children with ASD, and a university professor in special education, to obtain their input on the draft coding scheme. Consultation included interviews to gather information about the IEP process for this population, and then inviting written and oral feedback on the draft coding scheme.
The final coding scheme included the following areas: (a) participation in the IEP meeting (i.e., which family members and school professionals were present); (b) eligibility category or categories for special education (primary and secondary); (c) classroom placement and amount of time spent in special versus general education; (d) special education services provided (type, provider, setting, and minutes per week for speech-language therapy, occupational therapy, physical therapy, vision services, and counseling); (e) domains for which present levels of performance were described; (f) domains for which goals were provided; (g) inclusion of information about the child's level of visual impairment, and whether or not the level of visual impairment matched the level documented by the ophthalmologist for the research study; (h) inclusion of goals or accommodations meeting the recommendations in the expanded core curriculum for children with visual impairments (and if no goal was included in a domain, whether or not a clear explanation was provided in the IEP for why a goal in that domain was not needed); (i) for participants with ASD, inclusion of goals recommended by the NRC for children with ASD (and if no goal was included in a domain, whether or not a clear explanation was provided in the IEP for why a goal in that domain was not needed); and (j) whether or not a behavior intervention plan was included.
Procedures
All procedures were reviewed and approved by the Children's Hospital Los Angeles Institutional Review Board, and parent consent and child assent (when appropriate based on age and developmental level) were obtained. A neuropsychologist with research certification in the ADOS-2 administered the ADOS-2 and ADI-R and supervised doctoral trainees who administered the WISC-V verbal subtests. A Board-certified ophthalmologist completed the vision exams. Three researchers coded each IEP independently, entering the data in a RedCap database. Codes were compared after each coding session, and the three researchers reviewed discrepancies and reached a consensus. In most cases, discrepancies resulted from one researcher missing information in the IEP that was identified by one of the other researchers (e.g., one researcher identified a goal focused on social interaction, and another researcher did not code that goal; the researchers then reviewed the IEP together and concluded there was a goal focused on social interaction).
Results
Participant Characteristics
Table 1 provides participant characteristics. All 13 participants had ONH, and eight (61.5%) met the criteria for a diagnosis of ASD based on the study assessments. Six children (46.2%) had a verbal IQ below 70, based on the assessment for the study; five were children who also met the criteria for ASD. At the time of the IEP reviewed for the study, children ranged in age from 4 years, 7 months to 11 years, 3 months (M = 7 years; SD = 2.3). All were from English-speaking families. The majority of children were Hispanic (n = 7; 53.85%). All but one child lived in California. The geographic location included six counties and eight school districts. All participants had participated in early intervention programs prior to age 3. Of those eight children who were identified in the study as having ASD, four had previously been diagnosed with ASD (per parent report of a community diagnosis). Three children had autism as an eligibility category on their IEP, and one additional child had autism symptoms discussed in the IEP although it was not listed as an eligibility category.
Characteristics of the Participants.
Note: ASD = autism spectrum disorder; Dx = diagnosis; ID = intellectual disability; IEP = individualized education program; SLI = speech-language impairment; VI = visual impairment.
IEP School Placements, Goals, and Services
The most common school placement (n = 6; 46.15%) was a special education classroom on a general education campus; four children (30.77%) were placed in classrooms on a public special education campus; and three (23.08%) were placed in a regular education classroom (see Table 1). All IEPs reflected the consideration of Extended School Year, and 11 out of the 13 (84.6%) offered this service to students.
Table 2 provides information for each study participant about which domains had IEP goals. All 13 children had IEP goals related to motor skills. Most children had IEP goals focused on academic skills of reading (n = 11; 85%), including instruction in braille (n = 9; 69%) and math (n = 10; 77%), including instruction in Nemeth code (n = 4; 31%) and the use of assistive technology (n = 12; 92%) such as magnification of computer screen or use of screen readers. More than half had goals related to communication (n = 8; 62%), including all but one of the children with ASD. Less common goal domains were writing (n = 6; 46%), vocational (n = 6; 46%), social-emotional or social interaction skills (n = 5; 38%), and self-care and independent living (n = 2; 16%).
Individualized Education Program (IEP) Goals.
Table 3 summarizes the direct services included in the IEPs. The most common was vision services (n = 10; 77%). Most children had adapted physical education (n = 8; 62%) and orientation and mobility (n = 8; 62%). About half had occupational therapy (n = 7; 54%) and speech-language services (n = 7; 54%). One child had physical therapy, and no children were provided counseling to address social-emotional or behavioral needs.
Direct Services Provided on Individualized Education Programs.
Note: ASD = autism spectrum disorder; VI = visual impairment.
IEP Participation
IEP sign-in pages were examined to identify participants in the meeting. All IEPs (n = 13; 100%) included at least one parent, and seven (54%) included a second parent or extended family member. All but one IEP (n = 12; 92%) had a district representative present, and almost all included a special education teacher (n = 11; 85%). Other common participants included a general education teacher (n = 8; 62%), occupational therapist (n = 8; 62%), teacher specializing in visual impairment (n = 7; 54%), adapted physical education specialist (n = 7; 54%), speech-language pathologist (n = 6; 46%), nurse (n = 5; 38%), and orientation and mobility specialist (n = 5; 38%). The least common were physical therapists (n = 2; 15%) and school psychologists (n = 3; 23%).
IEP Eligibility and Goals for Visual Impairment
Eight children (62%) had visual impairment as their primary eligibility category for special education, and four (31%) had visual impairment as secondary eligibility. One child with ONH with only a mild reduction of visual acuity did not have visual impairment included as an eligibility category. Eleven IEPs (85%) provided information about the level of visual impairment, which appropriately matched the visual impairment levels as measured for the current study. The IEPs were examined in comparison to the standards recommended by the expanded core curriculum to determine if the IEPs included a goal or accommodation for each of the nine recommended areas. Table 3 provides information about the definition of each domain and the number of children with goals or accommodations in each. All but one IEP included goals or accommodations regarding assistive technology and compensatory skills. More than half (n = 9; 69%) included a goal for orientation and mobility. Very few IEPs contained goals related to the other recommended standards: social interaction skills (n = 4; 31%); career education, independent living skills, self-determination, and sensory efficiency (n = 2; 15% for each area); and recreation and leisure (n = 0; Table 4).
Goals or Accommodations in Individualized Education Programs for Children With Visual Impairments.
Note. ASD = autism spectrum disorder; VI = visual impairment.
Source for definitions: Expanded Core Curriculum (Huebner et al., 2004; Sapp & Hatlen, 2010).
IEP Eligibility and Goals for Children With Both ONH and ASD
Of the eight children diagnosed with ASD in the present study, four (50%) were identified on their IEP as their having ASD. Three of the eight children (37.5%) had autism as primary eligibility on their IEP, and one other had autism symptoms discussed as an area of need although not included as an eligibility category. The other four children (50%) had no mention of ASD on their IEP. IEP goals were compared to five domains recommended by the NRC (2001) for children with ASD. In three domains (communication, academic skills, and fine and gross motor), all but one of the children with ASD had IEP goals. Two domains were not addressed in most IEPs for children with ASD: social-emotional (n = 3; 38%) and behavioral (n = 2; 25%). One child with ASD had a Behavior Intervention Plan in their IEP. For five children, the IEP included information in the Present Levels of Performance about problem behaviors or concerns about social interactions, yet only two of those had IEP goals related to those concerns.
Discussion
This exploratory study examined the special education plans of 13 children with ONH to determine to what extent the IEPs addressed the children's complex educational needs. Eight of the children also had ASD diagnosed through the present study, though only four had been previously diagnosed and were identified with ASD by their school districts. All children participated in early intervention services and had their first IEP in place by the age of 3, indicating that the special education needs of children with ONH were recognized early in life. All children had their level of visual impairment described in their IEP, and that level was consistent with the findings of the study ophthalmologist. Overall, the findings suggest that the education plans appropriately addressed the academic needs of these children, with goals and services for most children targeting functional academic skills (i.e., reading and math), communication, vision services, and assistive technology and compensatory tools to address the impact of visual impairments on school functioning. All but one child (a child with mild visual impairment) received vision services. In addition, the schools addressed the need for orientation and mobility services and adapted physical education.
When comparing the IEPs to the expanded core curriculum guidelines recommended for the education of children with visual impairments, most plans were lacking goals related to career education, independent living skills, self-determination, and recreation and leisure. Given that the study age range was 4.5 to 11 years, the IEP teams may have been postponing attention to these needs until later in the child's development. However, one purpose of attending to these needs early is so that children with visual impairments, who have difficulty acquiring skills and ideas through incidental learning, will have other opportunities to learn about adult roles and activities. For example, a child without visual impairment may watch others perform a variety of career roles and recreational activities in the course of everyday life, whereas a child with visual impairment may miss learning about various roles unless they are explicitly pointed out and the child is given opportunities to ask questions, learn more about particular roles, and practice foundational skills that lead to later success in school, work, and recreation.
In addition, only four out of 13 children received any services, support, or IEP goals to address social interaction skills. Social skills are learned by sighted children incidentally, through observing interactions between people in their environment. In contrast, children with visual impairments may need specific instruction to learn skills such as approaching and greeting peers and adults; using language and nonverbal cues to make requests, decline assistance, express needs, or start and end conversations; and expressing emotions and affection appropriately.
When considering the eight children who were diagnosed with ASD in the current study, only half were recognized by the IEP team as having autism symptoms. One of the children with autism had a primary IEP eligibility of intellectual disability, although testing for the present study indicated verbal intellectual functioning in the average range. The other children had either visual impairment (three children) or multiple disabilities (one child) as their primary eligibility and no mention of autism symptoms in the IEP. All four had a severe level of visual impairment. Therefore, diagnostic overshadowing may have led the IEP team to focus on visual impairment and miss the importance of the autism symptoms. Diagnostic overshadowing occurs when a clinician or educator focuses on a prominent diagnosis (such as visual impairment) and neglects to recognize other co-occurring diagnoses (such as autism).
Comparing the IEPs for the children with ASD to education recommendations for children with ASD (NRC, 2001), all but one child had goals related to communication, academic skills, and fine- and gross-motor functioning. However, the IEPs were lacking in goals or services related to social interaction skills, social-emotional functioning, or behavioral support. Social interaction challenges are a core symptom of ASD (American Psychiatric Association, 2015), and lead many children with ASD to experience peer rejection, isolation, and lack of reciprocal friendships in school (Bauminger et al., 2010; Locke et al., 2013). School-based interventions in preschool and elementary school targeting the social skills of children with ASD have been found to be effective in increasing initiations of social interaction, appropriate responses to peer-initiated interactions, appropriate social interactions with peers, and engagement (Whalon et al., 2015). However, only two out of the eight children with ASD in the present study had IEP goals related to social skills.
Many children with ASD demonstrate problem behaviors that may interfere with learning, social interactions, and independence (Kanne & Mazurek, 2011; Presmanes Hill et al., 2014). Further, mental health diagnoses such as attentional difficulties, anxiety, and depression are common in children with ASD and may affect school functioning if not addressed (Joshi et al., 2010; Simonoff et al., 2008). Therefore, school interventions for children with ASD are recommended to include goals and interventions to address problem behaviors and social-emotional needs (NRC, 2001). In this study, the IEP present levels of performance for five out of the eight children with ASD indicated serious concerns about social-emotional or behavioral needs (e.g., self-injurious behaviors, aggression toward others, repetitive and stereotyped behaviors interfering with learning), yet only two had behavior goals and only one had a behavior intervention plan.
Although the present study did not directly evaluate the reasons for the lack of inclusion of social-emotional, behavioral, and social skills goals, an examination of team member participation in IEP meetings revealed that school psychologists were present in only three out of the 13 IEP meetings. Team member participation in the IEP meeting likely influences what goals are developed and services proposed. Although any team member may raise concerns about social-emotional, behavioral, or social skills needs, the presence of a psychologist or other mental health professional might have made it more likely that these domains would be adequately addressed.
The development and implementation of IEPs are governed by Federal law (IDEA, 2004). When the IDEA was modified in 1997, there were specific changes designed to impact the education of students with visual impairment. These changes included (a) the requirement to include instruction in braille for children with visual impairments, unless the individual student evaluation indicates it is not appropriate and (b) the addition of orientation and mobility services to the list of supportive services. It is notable that the present study found that most children with ONH were provided with orientation and mobility services, and almost all had access to assistive technology and vision services, including access to braillers and instruction in braille. Therefore, it seems that the legal changes implemented in IDEA are being included in the development of IEPs for children with visual impairments. It is recommended that future re-authorizations of IDEA consider the inclusion of guidelines from the expanded core curriculum for visual impairment and the NRC recommendations for the education of children with ASD, to ensure that IEP teams consider all domains of need.
This exploratory study is the first to examine IEP quality for children with visual impairments, and additionally considers the special education needs of children with both ONH and ASD. Although there are no published studies evaluating IEP quality in children with visual impairments, a study by Lohmeier et al. (2009) reported on a national survey of 40 parents and 50 educators of children with visual impairments regarding their knowledge and use of the expanded core curriculum. Most parents had never heard of the expanded core curriculum. Although 60% of educators had been exposed to the curriculum, they reported that very few children were receiving instruction across the nonacademic domains, similar to findings in the present study.
Two published studies have examined IEPs in children with ASD (without visual impairments). Ruble et al. (2010) evaluated the IEPs of 35 students with ASD (ages 3 to 9), also considering the NRC recommendations as part of their evaluation, and Gelbar et al. (2018) examined the IEPs of 75 children with ASD with a wider age range (4 to 17 years). The rate of inclusion in IEPs of communication goals and speech-language therapy services was similar in those studies to the current study. The rate of inclusion of social skills goals was much higher in the Ruble et al. study (80%) versus the present study (25% of the children with ASD); social skills training was provided to 12% of children in the Gelbar et al. study (they did not report on the presence of social skills goals). Rates of inclusion of goals related to problem behaviors were higher in the Gelbar et al. (64.3%) and Ruble et al. (42.9%) studies versus the present study (25% of the children with ASD). Diagnostic overshadowing may account for the lower rates of services related to social skills and problem behaviors in the present study; the children with ASD in the present study also had ONH, and the IEP teams may have focused more on goals related to visual impairment while neglecting those related to ASD.
Limitations
There are limitations to the present study that should be considered. First, the small sample size and location of the majority of the children in one state (California) may limit the generalizability of the findings. In addition, objective data about the individual educational needs of each child (separate from the needs documented on the IEP) were not available; therefore, it is possible that some domains that are recommended in national guidelines were not applicable to some of the individual children in the study.
Implications for Practitioners and Parents
Despite these limitations, the study findings suggest preliminary recommendations to improve the quality of IEPs for children with complex needs. First, it is important for educators and parents of children with visual impairments to recognize the co-occurring developmental disabilities (both ASD and intellectual disability) that are common in children with visual impairments, and to consider the educational needs related to these disabilities in addition to those that relate more directly to visual impairment. Second, efforts should be made to educate parents and school personnel about the expanded core curriculum for visual impairment and the NRC recommendations for educating children with ASD. Finally, consistent inclusion of school psychologists or other mental health professionals on IEP teams may lead to greater attention to children's social-emotional and behavioral needs.
Footnotes
Contribution
Marie Johnson is now at Providence St. John's Health Center. Melody Lavian is now at MindBloom Inc, A Psychology Corporation. Drs Johnson and Lavian contributed equally to the research and manuscript.
Acknowledgment
The authors appreciate the contributions of Mark W. Reid, PhD, for database development; Nancy Hunt, PhD, for consultation regarding IEPs and special education; Aimee Mendoza for consultation regarding special education services for children with autism spectrum disorder; Anne Bell for consultation regarding special education services for children with visual impairment; and Quy Huynh-Tran, RN, for study coordination, participant recruitment and data collection. This project was completed in partial fulfillment of the requirements of Drs Johnson's and Lavian's participation in the Clinical Child Psychology Postdoctoral Fellowship at the USC University Center for Excellence in Developmental Disabilities (UCEDD) and in the California Leadership Education in Neurodevelopmental Disabilities (CA-LEND) Interdisciplinary Training Program.
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding
The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This work was supported by the Marie and Robert E. Peterson Foundation and the Fahs-Beck Fund for Research and Experimentation.
