Carey Matsuba

Effect of secretin on children with autism: a randomized controlled trial

To determine the effect of intravenous porcine secretin on autistic behaviours in children aged 2 to 7 years, the effects of secretin on (1) performance on a standardized language measure, and (2) autistic behaviours, as rated by parents and child development professionals was examined. Employing a randomized, double‐blind, placebo‐controlled design, 95 participants were assigned to one of two groups and administered a single dose of either secretin or placebo. A follow‐up assessment was conducted 3 weeks after the injection. No significant differences in language or autistic behaviour measures were observed at the 3‐week follow‐up between the groups. Also, there was no significant difference in the proportion of individuals who improved by 6 points on the language measure at follow‐up. This study showed no significant effects of secretin on children with autism. Our results are consistent with a systematic review of randomized controlled trials evaluating the effect of secretin in children with autism.

Visual impairment due to a dyskinetic eye movement disorder in children with dyskinetic cerebral palsy

Neurological lesions that cause dyskinetic cerebral palsy (CP) commonly involve ocular movements. This report describes a group of 14 children (nine males, five females) whose CP is associated with severe dyskinetic eye movements. Ages ranged from 4 months to 13 years (mean 6.9 years). Clinical features of this eye movement disorder are discussed and defined. The visual function of these children is slow, variable, and highly inefficient. They are often misdiagnosed as blind, due to cortical visual impairment. Early recognition of dyskinetic eye moment disorder and appropriate developmental and educational management are important.

Interrater reliability and convergent validity of the American Academy for Cerebral Palsy and Developmental Medicine methodology for conducting systematic reviews

Aim  The aim of this study was to evaluate the interrater reliability and convergent validity of the American Academy for Cerebral Palsy and Developmental Medicine’s (AACPDM) methodology for conducting systematic reviews (group design studies).

The challenges of research in children with visual impairment

Population studies of infants with visual impairment are rare, as there are challenges in the assessment of vision, identification of the impairment, and the presence of multiple comorbid health and developmental conditions. In the original article by Dale et al., this population was systematically assessed using a large national cohort of 1-year-old infants. This paper highlights several meaningful clinical points. First, children with visual impairment are not all the same. They should be considered in the context of aetiological cause, and comorbid health and developmental conditions. In this study, anterior pathway conditions subdivided this group into simple and complex. This division was based on suspected risk differences of health and developmental conditions. However, it is important to recognize that this division is not absolute. There are many so-called simple conditions that are associated with a range of more complex comorbid conditions. As a result, grouping children with visual impairment is a difficult process. Second, the criteria for visual impairment are based on the International Classification of Diseases, Tenth Revision (ICD-10). It is not clear whether these criteria should be applied in infants. In many cases, infants may not meet criteria for a visual impairment in adulthood, such as ocular albinism or cataracts. Alternatively, some children initially appear to have appropriate vision, only to later develop a visual loss. Given that visual acuity in childhood can change over time, the categorization of visual impairment and its severity in infancy should be considered dynamic. In addition, the progress of vision may not always follow a similar course amongst differing conditions. So, there is no standard definition of visual impairment and severity in infancy. Third, visual acuity is a developmental/behavioural assessment which has been standardized through matching symbols at a distance. In this series, estimations of visual acuity were based on the Near Detection Scale, an assessment which requires a certain level of skill. For those clinicians less familiar with the assessment, measures of acuity are influenced by many factors. One such factor is visual attention, which is more difficult in infancy and in the presence of developmental disability. Visual attention is also influenced by other factors, including environment (e.g. lighting), health (e.g. seizures), and developmental conditions (e.g. motor impairment). All these elements contribute to make the assessment of visual acuity more challenging. Fourth, many anterior pathway conditions may be associated with posterior pathway components of visual impairment, particularly optic nerve and structural eye conditions. These conditions are often associated with complicated health and developmental issues. Grouping some of these conditions together without first identifying the presence or absence of other possible causes of visual impairment may be problematic. Finally, the developmental assessment of children with visual impairment is difficult. The Reynell–Zinkin Scale, commonly used in the assessment, lacks psychometric standardization. Current medical conditions and diagnostic tests are of course quite different from the late 1970s, when the Reynell–Zinkin Scale was first developed. Since then, there have been better diagnostic tests and detection of vision conditions. This means that the patient population under study today may not entirely represent the same population assessed during the development of the tool. Despite this, this paper by Dale et al. clearly demonstrates that limitations in vision have an impact on development, with those with more complex aetiological conditions and/or greater severity having greater developmental consequences. This present paper presents a relatively homogenous group of peripheral vision conditions. It is a good attempt to create a better understanding of this population’s differences. Clinicians should be aware that the larger more heterogenous group of children with visual impairment (the population not studied) are associated with greater comorbid health and developmental conditions.

Assessment of autism in children with visual impairment

It is well recognized that the prevalence of autistic spectrum disorders (ASDs) has increased substantially. However, the clinical tools to assist in the diagnosis of ASDs may not always be applicable to unique populations. Through a case series of nine children, Williams et al. have reported on their modified approach to the diagnosis of autism in children with visual impairment. This paper adds to the discussion on what clinicians need to consider when diagnostic or assessment tools may not always be applicable to special populations. Several points should be highlighted. First, the authors recognize a need to modify the diagnostic tools required. As many clinicians know, standard assessment tools may not be appropriate in specific populations. In this article, the authors assessed children with visual impairment, including eight with optic nerve disease and one with retinal disease. Unfortunately, this is far from a representative population. Of the nine children assessed, four children, all with optic nerve hypoplasia (ONH), were ‘diagnosed’ with autism. ONH is a spectrum condition that has etiological associations – some similar to autism – including systemic conditions and teratogenic agents. In addition, with ONH there are often significant associated health conditions including hypopituitarism, seizure disorders, and structural brain anomalies. Hence, the population being considered may not be truly representative and thus the modifications to the assessment tools may not apply to children with other visual impairment conditions. Second, diagnostic tools have become more widely used in the evaluation of autism. In their case series, Williams et al. chose to modify aspects of the assessment tools. At face value, this seems to be appropriate. But we must consider the possible challenges with the modifications in the diagnostic tools in children with ONH as one of the etiological conditions of visual impairment under study. ONH has its own associated developmental disabilities including cognitive, motor, and speech impairments. For this reason, some of the proposed modifications, such as using Braille books to assist in the diagnosis, may not be meaningful. Braille relies on fine motor and cognitive skills. If other impairments were present, perhaps Braille would not be a good option. When considering whether items should be modified in the context of visual impairment, it may be important to consider comorbid associations. Acquisition of developmental skills requires opportunity and experience. Many children with visual impairment may have had limited learning experiences. Braille, like many other developmental skills, relies on opportunity and experience. If these were limited, it is uncertain how modifications would be considered meaningful. Third, the usefulness of diagnostic tools is to establish a degree of certainty about the condition that is being assessed. In the current article, the authors have chosen to modify diagnostic tools along with clinical evaluation. In doing the clinical evaluation, Williams et al. recognize that autism is more than a checkbox of language, social, and stereotypic abnormalities. Therefore, the modified tool should be able to assist us in making the ultimate diagnosis. To date, others who work with visual impairment have been able to modify assessment tools to make a diagnosis of autism in that context. So the question is whether these specific modifications were needed. There is still some uncertainty. I applaud the efforts of Williams et al. in bringing this important issue to the forefront of developmental medicine. The developmental needs of children with visual impairment must not be underserved. When clinically indicated, the formal assessment of developmental disabilities should be undertaken in children, whether or not they have visual impairment. It must also be recognized that there will be a need to adjust or modify assessment tools to assist in the clinical diagnosis. How these assessments are undertaken remains, in my opinion, unclear, as it is dependent on the challenge of each presenting child. Thus, perhaps at present, the diagnosis of ASD in children with visual impairment should be based on clinical judgment.