Paediatrics (RCH) - Theses
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ItemAppearance-altering facial surgery on children: An empirically informed ethical analysis( 2015)Using empirically informed ethical analysis, this thesis investigates the ethics of appearance-altering facial surgeries performed on children. These surgeries have attracted controversy in the media and ethics literature and are the subject of legislation and professional guidelines, which leave much to individual practitioners’ discretion. Despite their contentious nature, very little is known about surgeons’ practices and decision-making processes regarding these surgeries. This thesis is ‘empirically informed’ in two different ways: (1) by existing empirical data on psychosocial outcomes of children with facial differences and children who have undergone appearance-altering facial surgeries and (2) by new interview data from 22 surgeons who encounter requests for these surgeries. Using reflective equilibrium as my method of ethical analysis, these two kinds of empirical data are combined with established paediatric bioethical principles and concepts, including the zone of parental discretion and children’s assent and dissent, to arrive at a comprehensive position on the ethics of performing these surgeries. Using reflective equilibrium, I found some aspects of surgeons’ decision-making processes and practices diverged from existing ethical understandings about the relative roles of children and parents in medical decisions. One such area related to the ethical weight surgeons attach to children’s refusals. While most ethicists in the literature advocate including children in decisions, they do not necessarily recommend giving them decision-making authority. In contrast, most surgeons tended to give children a larger role in decisions about appearance-altering facial surgeries, viewing them as ultimate decision makers. In this thesis, I make normative claims about the relative levels of ethical weight surgeons should place on children’s and parents’ wishes when making decisions about these surgeries. Using the established ethical framework of the zone of parental discretion and related ethical concepts of harm and benefit, I claim that several morally relevant differences exist between these surgeries and other medical procedures requested for children. These include their uncertain risk-benefit ratio, their elective nature, uncertainty as to how the child will later perceive their facial difference and the acceptability of surgery, and the existence of alternative, less risky and invasive psychosocial interventions for alleviating appearance-related psychosocial harm. I argue these differences warrant placing greater (even absolute) ethical weight on children’s wishes, especially when children refuse these surgeries, and comparably less ethical weight on parents’ wishes, when making these decisions. Although my ethical position is similar to most of the surgeons’ judgments, I argue it would be worthwhile for surgeons to know about and use ethical principles and concepts more explicitly when making decisions and communicating with children and/or their parents. I also claim that surgeons ought to make more of a considered effort to ascertain children’s actual wishes and inform parents and/or children about non-surgical options for alleviating appearance-related psychosocial distress, and be taught strategies for saying no and how to articulate their ethical reasoning. These findings have significant implications for clinical practice, raise questions for further ethical analysis and contribute to refinement of existing understandings of children’s assent and dissent and parents’ role as proxy decision makers for their children.
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ItemCardiovascular associations of HIV infection in children( 2015)Vertically-acquired HIV infection is a devastating disease in childhood that may affect any organ, including the cardiovascular system. With increased survival of HIV infected children due to antiretroviral (ART) treatment availability, chronic cardiovascular problems become a confronting challenge, not only because HIV infection may cause cardiac problems readily manifesting in childhood but also because it potentially increases future cardiovascular disease risk in adulthood. This thesis explores various possible cardiovascular effects of HIV infection in children, particularly the differential effects of ART-naïve compared to ART-exposed HIV infection on left ventricular (LV) remodelling, pulmonary hypertension, and arterial elasticity. We conducted a cross-sectional study enrolling 56 ART-naïve, 59 ART-exposed HIV infected, and 51 healthy children in Jakarta, Indonesia and performed cardiac/vascular ultrasound, and blood tests for biomarkers. There were marked differences in the cardiovascular parameters between the two groups. We found that ART-naïve HIV infection was associated with LV dilation while the ART-exposed seemed to cause concentric hypertrophic remodelling. ART-exposed HIV infected children who showed evidence of higher pulmonary artery pressure than healthy children, whereas the ART-naïve children had reduced right ventricular function. For arterial elasticity, the ART-naïve had higher strain and lower elastic modulus, but thicker intima-media thickness, whereas the ART-exposed had similar vascular properties as healthy children. In conclusion, HIV infection in children have significant impacts on childhood cardiovascular system with particular differential effects between ART-naïve and ART exposed HIV infection. Routine cardiovascular surveillance is needed for children with HIV infection.
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ItemLanguage development in young children who stutter( 2015)Stuttering typically emerges at the same time as children are experiencing rapid development in their language. It is not surprising therefore that there is substantial interest in the interface between stuttering and language. Explanations for stuttering that utilise a linguistic framework are prominent in the theoretical literature, and there are numerous empirical studies focused upon understanding the potential relationship between stuttering and language development. Yet, agreement about the precise nature of this relationship does not exist. This research is focused upon the emergence of stuttering and language development in young children. The study is embedded within two pre-existing prospective longitudinal studies; the Early Language in Victoria Study (ELVS) investigating language development in a large community sample of children; and the ELVS-Stuttering study, a nested study investigating the onset and progression of stuttering. Utilising data collected within the framework of these two studies, this study has three parts. Part 1 involves a comparison of the early communication and language skills of children who started to stutter and children who did not start to stutter from ages 2 to 5 years. Part 2 focuses on a sub-sample of the children who started to stutter, describing their stuttering and language behaviours during the first 12 months after stuttering onset. This involved concurrent examination of stuttering behaviours and stuttering severity, and a range of global indicators of expressive language proficiency. Part 3 examines the association between variables related to stuttering (e.g. family history of stuttering, severity, age of onset, stuttering behaviours) and two global indicators of language ability (length of utterances, and grammatical complexity), again focusing upon a sub-sample of children who started to stutter. Overall, results indicate that children who stutter, as a group, demonstrate expressive language skills that are consistent with, and sometimes exceeding, developmental expectations. Part 1 revealed findings indicating that children who have stuttered demonstrate stronger language skills than children who have not stuttered on each of the communication and language outcomes measured. Findings from Part 2 indicated that, in the first 12 months after the report of stuttering onset, the subgroup of children who stutter demonstrated language skills that are consistent with developmental expectations, and when compared to available age-referenced data they in fact exceed expectations. Part 3 revealed a weak association between stuttering characteristics and language proficiency in the first 12 months following the report of stuttering onset. Although some associations were statistically significant, the corresponding regression coefficients tended to be small and explained very little of the variability in the language outcomes measured. Overall there was no evidence of language development being impacted by the onset or progression of stuttering in the cohort of children examined in this study. This study offers an unprecedented insight into the early stuttering and language development of a large community cohort of children who stutter. The findings provide clarification concerning the language-stuttering connection, contributing to a resolution to the lack of consensus in this area. Further research involving rigorous examination of language ability and language use in representative cohorts of children who stutter, with a particular focus on children whose stuttering is moderate-to-severe and persists into the school years, is recommended.
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ItemEarly stuttering, temperament and anxiety( 2012)Many stuttering adults and adolescents experience speech-related anxiety. This relationship has not been examined in the preschool population. Given this, it is unknown whether stuttering preschool children also exhibit anxiety traits. Particular temperament traits have been identified as precursors of anxiety. Temperament refers to the way an individual reacts to or interacts with their environment. While the expression of temperament traits is largely genetically determined, it is also influenced by environmental factors. Variations in the expression of temperament traits between individuals are observed as personality traits. Two hypotheses regarding the relationship between temperament and early stuttering have been discussed in the literature. The first is that temperament is associated with the development of stuttering. Certain temperament traits are theorised to influence the onset, development and maintenance of the disorder. The second hypothesis is that temperament differences arise as an effect of stuttering. Stuttering preschool children can experience negative communication interactions. It is therefore possible that those negative interactions may influence the expression of temperament traits, particularly the precursors of anxiety. Further exploration of the relationship between temperament and early stuttering is crucial in order to ascertain which of the above hypotheses is true. Knowledge about the expression of temperament traits is required in order to determine whether stuttering children express temperament traits differently from non-stuttering children as well as to determine whether they have a heightened risk of developing anxiety. This doctoral study was embedded within the Early Language in Victoria Study (ELVS). ELVS has a prospective, longitudinal design. The overall aim of ELVS was to examine the epidemiology and evolution of communication impairments, including stuttering, from infancy through to 7 years. The aim of the present study was to investigate the expression of temperament traits, including the precursors of anxiety, among stuttering children in ELVS. Data were collected with a prospective, longitudinal cohort design. In total, 1444 children were recruited into this study. By 4 years, 173 children had been confirmed as stuttering and 1271 children were assigned to the non-stuttering group. Temperament data were collected using a parent-report scale which was included in annual parent completed questionnaires from 2-4 years. During this time, parents of stuttering participants also completed a stuttering questionnaire at 12 consecutive, monthly home visits, immediately post stuttering onset. At 6 and 7 years, temperament and stuttering behaviour data were collected from stuttering participants in annual parent completed questionnaires. The results from this study revealed little evidence of differences between stuttering and non-stuttering preschool children’s expression of temperament traits, including the precursors of anxiety, up to 4 years. There was little evidence of associations between various stuttering behaviours and the expression of temperament traits amongst stuttering participants. These findings suggest that within this study’s cohort, stuttering children were not at greater risk of developing anxiety compared to non-stuttering children and the range of stuttering characteristics exhibited by stuttering children did not influence the expression of their temperament traits, including the precursors of anxiety.
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ItemChronic constipation in children and the roles of nuclear transit scintigraphy (NTS) and transcutaneous electrical stimulation (TES) therapy( 2012)Chronic constipation in children is a challenging and difficult problem to manage for clinicians. It has huge psycho-socio-economic impact on the child, parents and the health care system. If left untreated, 1/3 of children will have their symptoms persisting into adulthood, contributing to poor quality of life. There is no standard therapy for the treatment of chronic constipation despite the numerous laxatives available in the market. Various conservative strategies like behavioural therapy, dietary modifications and biofeedback therapy have reported short-term success. Surgery is reserved as the last option for patients with intractable symptoms. Since the introduction of antegrade continence enema (ACE) by Malone in the 1990s, it was considered a less invasive surgical option to treat children with intractable constipation with faecal soiling. Various investigations have been used to characterize the underlying colonic dysmotility in patients suffering from intractable chronic constipation, with the hope of identifying a treatable cause. Ultrasound has a limited role for faecal matter and volume assessment in the rectum. Colonic transit study using scintigraphy (radioisotopes with gamma camera images) has replaced radio-opaque sitz markers (with x-rays) in our hospital, providing better characterization of segmental colonic transit. Wireless capsule motility studies and colonic manometry provide useful information on the underlying colonic dysmotility but are only available at certain centres with research interests. Electrical stimulation is available in two forms: the invasive form involving surgical implantation of electrodes, as in sacral nerve stimulation (SNS), and the noninvasive form involving the use of adhesive electrodes applied over the skin of the abdomen and paraspinal region, as in transcutaneous electrical stimulation (TES). Slow-transit constipation (STC) is a well-recognized cause of chronic constipation in women. The diagnosis of slow-transit constipation [by nuclear transit scintigraphy (NTS)] in children, by contrast, is a new diagnosis and the use of TES to treat STC in children is a novel therapy. However, little was known about 1) the various colonic dysmotility patterns contributing to chronic constipation in childhood, 2) an optimal diagnostic algorithm in children with intractable constipation, 3) targeted therapy for children with chronic constipation if underlying colonic dysmotility is revealed, 4) whether TES is effective in the treatment of childhood STC and its long-term effects, 5) whether TES at home is possible when a clinician is supervising the treatment, and finally 6) whether home TES is effective in the treatment of the commonest form of chronic constipation in children – anorectal retention/functional faecal retention. The aims of this thesis were: (1) to address these questions using clinical information from a database in a tertiary institution, and (2) perform prospective clinical studies using TES to treat children with chronic constipation, and (3) to perform a follow-up study of children treated with TES in a previous randomised controlled trial. In Chapter 1, I provide an overview of the literature on the diagnosis and various medical and surgical treatments of children suffering from chronic constipation. Chapters 2-8 are the clinical studies comprising the thesis. In Chapter 2, there is a detailed description on the use of nuclear transit scintigraphy (NTS) to diagnose colonic dysmotility in children with chronic constipation at a tertiary institution. A database of children with chronic constipation investigated by NTS has led to the development of a standardised protocol. The identification of different types of colonic dysmotlity as the potential underlying pathological cause of chronic constipation has changed the management algorithm in these children. This has enabled the treatment to target the likely cause of the chronic constipation in children. I aimed to provide the diagnostic criteria of different colonic transit patterns using the standardised gastro-intestinal transit protocol. Interestingly, in Chapter 3a, the potential use of NTS to identify a new subgroup of children with rapid proximal colonic transit has led to a new treatment strategy in a subgroup of children with chronic constipation. In addition, Chapter 3b illustrates how we can gather useful anatomical information from a physiological study like NTS as it provides information on colonic length. In Chapter 4, I show that NTS has potential monitoring values in children with chronic constipation with NTS produces no change in gastrointestinal transit if the patient symptoms did not improve after treatment. Therefore, any physiological changes identify at the repeat study will indicate the effects of treatment. It has great potential to monitor progression of disease and also to monitor response to treatments. Chapter 5a described the key elements for a successful administration of TES at home. Close regular contacts and appropriate supervision are essential elements for positive outcome of TES. Chapter 5b is a prospective study of home STC to treat children with STC. This study shows that home TES is effective to overcome symptoms in STC children and to improve their quality of life with a small but demonstrable increase in colonic transit measured objectively by NTS. Chapter 6a is a prospective study conducted to establish the long-term effects of TES in STC children previously treated in a separate randomised, controlled trial. This study showed variable responses with long-term effect lasting as long as 3 years in some children. In contrast, Chapter 6b is a retrospective study to assess how the use of TES at a single tertiary institution has affected the role of surgery as a treatment in children with slow-transit constipation. Chapter 7 is a pilot study to examine the role of home TES to treat the commonest form of chronic constipation in children – anorectal retention (AR) or functional faecal retention (FFR). Chapter 8a is a study to examine the effect of upper gastrointestinal dysmotility in children with slow-transit constipation and whether this will affect their response to TES. Excitingly, chapter 8b describes the effect of TES on gastric motility and its possible implications as a non-invasive treatment for delayed gastric emptying or gastroparesis. In chapter 9, I discuss the major conclusions from this study and the implications of this work. The studies comprising this thesis provide the first detailed description of the use of a standardised NTS protocol to diagnose colonic dysmotility in children, changing the algorithm of investigation and management of children with chronic constipation at a tertiary institute. The use of home TES to overcome chronic constipation in children offers a promising and non-invasive treatment option without surgery in treatment-resistant cases. Moreover, the exciting finding of TES affecting gastric motility will form the ground for future studies in investigating its role as a non-invasive treatment option for patients with intractable upper gastrointestinal tract symptoms.
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ItemEarly identification, prediction, and classification of speech sound disorders in the preschool years( 2011)Speech sound disorders (SSD) are often diagnosed at 4 to 5 years of age; however, it may be possible to identify children at risk of SSD earlier. One possible method of early identification involves using parent description of their children’s speech sound production, however, there is paucity in the literature on the validity of parent description of speech sound production, and whether early parent description can be used to predict later speech sound production and disorders. Furthermore, a wide range of prevalence rates of SSD have been reported in the literature, partly due to different definitions of SSD, and lack of agreement on a universally accepted classification system of SSD. This research was embedded within the Early Language in Victoria Study (ELVS), a prospective cohort study based in Melbourne, Australia. The research focused on data collected when the children were aged between 8 months and 4 years. Data were collected via parent questionnaires and face-to-face assessments, and different subsamples participated in the three phases of the research (n = 163 to 1689). In Phase 1 (Validation), there was a poor relationship between parent description and a face-to-face assessment of sound production at 12 months of age. At 4 years of age, there was a relationship between parent description of speech sound production and a face-to-face assessment of speech sound production. Parent description at 4 years of age did not identify an appropriate number of children with SSD (sensitivity); however, it was able to identify an appropriate number of children with typical speech (specificity). In Phase 2 of this research (Prediction), neither parent description of speech sound production at 8 or 12 months of age, nor a face-to-face assessment of sound production at 12 months were able to predict the results on a face-to-face assessment of speech sound production at 4 years. At 2 and 3 years of age, however, parent description of speech was able to predict results on a face-to-face assessment of speech sound production at 4 years, however, although specificity was appropriate, sensitivity was not. In Phase 3 (Classification), the prevalence of suspected SSD in a community sample was 27.0%. Two existing classification systems of SSD were applied to a group of children with mild-moderate to severe SSD in this study (n = 163). Not all participants could be classified according to each classification system; 84.0% were classified according to Dodd’s Subgroups of Speech Disorder (Dodd, 1995), whereas 59.62% were classified according to the Speech Disorders Classification System (Shriberg, 1993; Shriberg, 1994; Shriberg, Austin, Lewis, McSweeny, & Wilson, 1997; Shriberg et al., 2010; Shriberg & Kwiatkowski, 1982; Shriberg, Tomblin, & McSweeny, 1999). Of the participants who could be classified, they were classified according to Dodd’s Subgroups of Speech Disorder at similar proportions to what has been reported elsewhere in the literature. A framework for describing speech sound development and disorders was proposed. This framework is intended to be used by speech pathologists (clinicians and researchers) when assessing speech sound production. A number of critical parameters are suggested for consideration when assessing speech sound production: disorder/delay, inconsistency of speech sound production, parent concern, speech intelligibility, and risk and protective factors.
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ItemQuality of life assessment in children requiring oral anticoagulant therapy( 2011)The absence of robust evidence regarding the management and clinical outcomes of anticoagulant therapy in children challenges interpretation of the risks and benefits of long-term anticoagulation therapy for children. Decisions about treatment increase in complexity in light of the potential burden of treatment on children and families. Quality of life (QoL) is a recognised and validated measure of the burden of treatments for children; yet, there is a lack of data about the impact of long-term warfarin therapy on QoL for children and families. This thesis describes the first study to evaluate the impact of a home international normalised ratio (INR) self-testing (home ST) program on the QoL of children and their families. The aim of the study was to determine if participation in a home ST program improves QoL for children requiring long-term warfarin therapy and their families. Parents of children requiring long-term warfarin therapy and children aged eight to 18 years participated in the QoL study. Three validated QoL questionnaires, the Pediatric Quality of life Inventory Generic Core Scale™, the Pediatric Quality of Life Family Impact Module™ and the KIDCLOT Pediatric Anticoagulation Quality of Life © inventory were employed to measure QoL. Questionnaires were completed before commencing home ST and six to 12 months later. Participants answered four open-ended questions when completing the questionnaires for the second time. Fifty-six families entered the home ST program during the study period. The children were aged between 2 and 17 years. Fifty-five parents and 35 children completed QoL questionnaires. Results of INRs tested at home were collected. The percentage of time the children’s INRs were in their target therapeutic range was 71.3%, which is comparable to many published paediatric studies of home ST. Parents reported statistically significant improvements in QoL for themselves, their family and their child, across all questionnaires following the commencement of home ST (mean difference in score p ≤ 0.003 on all questionnaires). The children’s scores of their own QoL also improved; however, this improvement was not significant. Parents’ report of their children’s QoL was significantly lower than the children’s report of QoL on all questionnaires at both time points. As well as being the first validated baseline QoL data in an Australian population of children requiring oral anticoagulant therapy, this study reports QoL for the largest cohort of children requiring warfarin therapy worldwide. The employment of both quantitative and qualitative methods to assess QoL enhanced understanding about the impact of home ST on QoL. This study has demonstrated that home ST in a population of children requiring long-term warfarin therapy is safe, efficacious and improves the QoL of children and parents. This study confirms home ST successfully reduced the burden of therapy and maintained excellent clinical outcomes. The results of this study not only identify the impact of warfarin therapy upon QoL in children, but offer an alternative evaluation strategy to sensitively measure the impact of interventions in this population. Accurate QoL assessments ensure children’s and families’ values are integrated with the current evidence for best clinical practice.
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ItemAttentional functioning in children with and without post-traumatic stress symptoms post traumatic brain injury( 2010)Attentional deficits are common following childhood traumatic brain injury (TBI). There is also evidence of attentional impairments in individuals with post-traumatic stress disorder (PTSD), however it remains unclear as to how attention might be impacted when a child has sustained a TBI and is also experiencing PTSD. To address this issue, the current study was comprised of three key components. Study 1 examined the presence and nature of attentional deficits in children with and without subsyndromal PTSD post TBI using hierarchical clinical and questionnaire measures; Study 2 involved developing experimental measures which have been shown to be useful in characterizing PTSD symptoms in adults and explored the potential application in children. The behavioural versions of the emotional counting stroop and faces paradigms were piloted in healthy children as part of this process; and Study 3 reported case study analyses of the newly developed experimental measures in children with and without PTSD post TBI. Participants involved in study 1 were between the ages of 6-14 years at the time of injury and were assessed six months post-injury. Results indicated that children with TBI do not exhibit a generalized pattern of attentional impairment. Instead, attentional deficits were limited to specific components and were related to factors such as injury severity and brain maturity at the time of injury. Although the incidence was low, findings confirmed that children with TBI can also develop subsyndromal PTSD, with comparable rates observed across TBI severity levels. Findings also suggested that the presence of subsyndromal PTSD post TBI does not compound the higher-order attentional difficulties experienced as a result of TBI. Poorer attentional functioning was best predicted by injury severity and age at injury, whereas SES, pre-injury emotional functioning and the presence of subsyndromal PTSD post TBI did not contribute significantly to attentional functioning outcomes post pediatric TBI. A total of 10 neurologically healthy children, 3 male and 7 female, participated in study 2. While the results need to be considered preliminary in light of the small sample size, healthy children responded significantly faster to the baseline condition when compared to other stimuli. Additionally, there were no significant differences in error rates in both paradigms. Two male children with moderate TBI and no PTSD and two male children with dual PTSD/mild TBI were included in the case study analyses. Findings on the emotional counting stroop case studies suggested that the processing of trauma-related information may be recruiting greater attentional resources in children with PTSD, but only in those who are displaying severe hyperarousal symptoms on clinical presentation. The stroop interference effect was not observed in the healthy controls or children with TBI only, highlighting the disorder-specific nature of the observed interference effect. The expected attentional bias to fearful expression was not observed in the faces paradigm. A number of possible explanations were considered to account for the findings, with strong suggestions that attentional bias in children with PTSD post TBI is specific to trauma-related information and does not extend to generally negative stimuli. Overall, the piloting and development of the emotional counting stroop and faces paradigms, together with the case studies, allowed for the establishment of optimal timing parameters for a pediatric population and marked the first time developmentally appropriate versions of these two paradigms were implemented in children. This was an important step towards validating these experimental paradigms for future use in larger clinical samples.