Thesis (PhD)--University of Stellenbosch, 2002. / Page ii of digitised copy missing due to the condition of the original hard copy. / ENGLISH ABSTRACT: no abstract available / AFRIKAANSE OPSOMMING: Hierdie studie bestudeer die rol wat die opvoedkundige sielkundige kan speel in die ondersteuning van leerders met Downsindroom tydens die insluiting tot hoofstroomonderwys. Die studie vind plaas in 'n tydperk waar postmoderne denke en demokratisering van onderwys in Suid-Afrika te voorskyn kom. Hierdie tipe studie is relevant. aangesien huidige beleidsdokumente klem Ie op die demokratisering van onderwys en die transformasie na inklusiewe onderwys. Die Universiteit van Pretoria het in samewerking met die Universiteit van Stellenbosch 'n lootsprojek geinisieer waarin inklusiewe onderwys bestudeer is. Die projek het bestaan uit 'n gevallestudie van tien leerders met Downsindroom wat gedurende 1996 in verskillende hoofstroom kleuterskole geplaas is, waartydens hulle 'n leergereedheidsprogram gevolg het. Gedurende 1997 is die leerders in graad eenklasse in die hoofstroom geplaas. Die leerders se agtergrond was divers ten aansien van hul onderrig, maar die meeste van hulle was eers in spesiale skole. Die doel van die studie was om die leerders, ouers en onderwysers te ondersteun en verdere ondersteuning in a trans-disslplinere span te koordineer. Die waarneming en temas wat in hierdie studie na yore gekom het, beklemtoon dat diagnose, evaluasie en terapeutiese ondersteuning binne 'n ekosistemiese raamwerk belangrik is vir leerders met Downsindroom wat in die hoofstroom geplaas word. Die konsep van holistiese gesondheidsontwikkeling word beklemtoon in die transformasie van onderwys in Suid-Afrika. Opvoedkundige sielkundiges moet hulself dus ook strategies posisioneer. Gedurende die studie is die volgende rolle vir die opvoedkundige sielkundige ge"identifiseer: psigoterapeut, assessor/evalueerder, fasiliteerder, navorser/reflektiewe praktisyn, vakspesialis, konsultant, bestuurder en administratiewe rol en laastens die rol van ontwikkelingsfasiliteerder. Dit is ook belangrik dat die opvoedkundige sielkundige die rol as fasiliteerder van geestesgesondheid vertolk.

The ZNF9 gene on chromosome 3 encodes the cellular nucleic acid binding protein (CNBP), a ubiquitously expressed, 177 amino acid (≈19.5kDa) protein that is highly conserved among vertebrates. The function of the protein is largely unknown, however an expansion in the first intron of the protein results in myotonic dystrophy type 2 (DM2), a multisystemic disease featuring cardiac arrhythmia, muscle wasting, cataracts, and a range of neuropathologies. Remarkably, we recently discovered that CNBP is involved in regulating the activity of β-secretase, the enzyme that produces the first cleavage event in the generation of the amyloid-β peptide (Aβ). The progressive fibrillization and deposition of Aβ is widely believed to be the primary causal factor in the development of Alzheimer’s disease (AD), and AD-like pathology in individuals with Down syndrome (DS). DS provides a unique model for evaluating how these factors change in the aged brain as compared to young brain, and how such changes affect the proportion of DS patients with AD. In the AD brain, both BACE1 and BACE2 increased from an early stage of disease; in DS brains, BACE1 significantly decreased (p<0.04) with age, whereas BACE2 was unchanged, even though the gene for BACE2 is located within the DS obligate region of chromosome 21. BACE1 and BACE2 activity levels were highly correlated in this series (r2 = 0.95), indicating that there may be a higher degree of shared regulation than previously believed. This implicates regulators of BACE as potentially critical for the development of AD, and our data suggests that CNBP may be one such regulator. In AD, CNBP increases early in the disease process, a change that does not occur in the normal aging process or in DS. CNBP and BACE protein levels were correlated in these cases (p<0.001), while there was no relationship between CNBP and age, or CNBP and Aβ, in either the human or mouse brain, indicating that CNBP does not increase as a consequence of normal aging. Thirty day overexpression of CNBP following adeno-associated viral delivery in murine gastrocnemius muscle resulted in an increase in BACE1 protein (p<0.01) and a consequential increase in Aβ production (p<0.01). Other experiments indicated that CNBP overexpression did not affect the half-life of BACE1 mRNA or protein, but resulted in an increase in BACE1 translation. These data indicate that CNBP is an important regulator of β-secretase, and may play an important role in the onset and progression of AD.

<p>Despite major advances in our understanding of the biology of childhood acute myeloid leukemia (AML) and the development of new cytotoxic drugs, the prognosis of long-term survival is still only 60-65 %.</p><p>In the present research, we studied the pharmacokinetics of drugs used in the induction therapy of childhood AML and performed in vitro drug sensitivity testing of leukemic cells from children with AML.</p><p>The aims of the studies were to correlate the results of the analysis to biological and clinical parameters and to identify subgroups of AML with specific drug sensitivity profiles in order to better understand why treatment fails in some patients and how therapy may be improved.</p><p>Blood samples were analysed to study the pharmacokinetics of doxorubicin (n=41), etoposide (n=45) and 6-thioguanine (n=50). Doxorubicin plasma concentration and total body clearance were correlated to the effect of induction therapy, and doxorubicin plasma concentration was an independent factor for complete remission, both in univariate and multivariate analysis including sex, age, and white blood cell count at diagnosis. For etoposide and 6-thioguanine no correlation was found between pharmacokinetics and clinical effect. Children with Down syndrome (DS) tended to reach higher blood concentrations of etoposide and thioguanine nucleotides, indicating that dose reduction may be reasonable to reach the same drug exposure as in children without DS.</p><p>Leukemic cells from 201 children with newly diagnosed AML, 15 of whom had DS, were successfully analysed for in vitro drug sensitivity by the fluorometric microculture cytotoxicity assay (FMCA). We found that samples from children with DS were highly sensitive to most drugs used in AML treatment. In non-DS children, the t(9;11) samples were significantly more sensitive to cytarabine (p=0.03) and doxorubicin (p=0.035) than other samples. The findings might explain the very favorable outcome reported in children with DS and t(9;11)-positive AML. A specific drug resistance profile was found for several other genetic subgroups as well. A detailed study of MLL-rearranged leukemia showed that cellular drug sensitivity is correlated both to partner genes and cell lineage, findings that support the strategy of contemporary protocols to include high-dose cytarabine in the treatment of patients with MLL-rearrangement, both in AML and acute lymphoblastic leukemia (ALL).</p><p>Our results indicate that drug resistance and pharmacokinetic studies may yield important information regarding drug response in different sub-groups of childhood AML, helping us to optimize future chemotherapy in childhood AML.</p>

Chronic otitis media (OM) is an archetypal complex disease, which is particularly prevalent in childhood. Epidemiological data suggest high heritability for disease susceptibility, but previous genetic association studies have had methodological flaws, and none have specifically focused on chronic OM phenotypes. Mouse models represent one way to ascertain candidate loci for human association testing. A number of mouse models of middle ear inflammation have been reported, but many susceptibility loci remain undiscovered. I demonstrate that oto-endoscopy is a robust and scalable phenotyping platform for OM in the mouse, and discuss its value in new model discovery. Chronic OM is also a feature of trisomy HSA21 (Down Syndrome). Through an interrogation of the mouse library of segmental trisomy models of Down Syndrome, I identify a critical trisomic region for chronic otitis media. This region may underlie OM susceptibility in Down Syndrome, but could also contribute to disease susceptibility in non-syndromic disease. Mouse models can also be used to interrogate disease mechanisms. Our previous work has shown that the chronically inflamed middle ear is hypoxic, and that hypoxia signalling is a potential therapeutic target. Exploiting the Junbo mouse model, I demonstrate that surgical ventilation of the Junbo ear improves inflammation, and that this is associated with loss of hypoxia signalling. I present preliminary results from transcript analyses of human middle ear effusions showing marked upregulation of hypoxia signalling. A systematic review of existing mouse models suggests that the loci FBXO11, EVI1, SMAD2, and TGIF1 are good candidates genes for human association testing. I detail recruitment and collection of DNA from families in the UK where a child is undergoing grommet insertion. Association testing using a variant of the transmission disequilibrium test shows susceptibility associated with polymorphisms at FBXO11, and possibly also SMAD2 and TGIF1.

This diploma thesis deals with the issue of children with Down syndrome. Down syndrome is the most common genetic disorder with trisomy 21st chromosome with typical overal psychomotoric delay. The thesis is focused on the specific difficultes of children with Down syndrome, with emphasis on motor skills. Children with Down syndrome are examined by standard motion test Movement Assessment Battery for Children, 2nd edition (MABC-2). There are a total of 19 children ranging in age 3-14 years. The control group of children is chosen in similar age group, a total of 20 children. Simultaneously at work is compared a parameter of achievement independent walking. This milestone is very often assessed and used in children with Down syndrome. The results show that children with Down syndrome in comparison with healthy peers did not achieve the similar level of motor skills. In the percentile rating, the children reached the level below the 5th percentile of standard norms. Powered by TCPDF (www.tcpdf.org)

Tato diplomová práce se zabývá úlohou muzikoterapie, jako podpůrného prostředku pro člověka, který potřebuje pomoc na cestě životem. Popisuje průběh a vývoj jednotlivých muzikoterapeutických setkání, jejich dopad na klienta při uvědomování si vlastních hodnot a jejich transformaci do osobního a studentského života. Práce byla vypracována na základě vývoje kazuistiky v čase, rozhovorů a reflexe celé práce. Klíčová slova: Muzikoterapie, Klient, Muzikoterapeut, Hudba, Edukace, Downův syndrom, Mentální postižení Abstract This thesis describing with the role of music therapy as a means of support for someone who needs help along the way. It describes the progress and development of individual music therapy meetings, their impact on the client in recognizing one's own values and their transformation into a personal and student life. The work was developed based on the development of case reports in time, interviews and reflection of the whole work. Keywords: Music therapy, Client, Music therapist, Music, Education, Down syndrome, Mental disability

As crianças pequenas, com idade entre 4 e 7 anos, ainda são pouco reconhecidas como protagonistas dos estudos acadêmicos, notadamente em Arquitetura e Urbanismo, embora desde a década de 1970 essa inquietação esteja presente na literatura. No Brasil é relativamente recente a preocupação em considerá-las interlocutoras competentes para fornecerem informações sobre si mesmas (as primeiras teses nesse campo datam do início dos anos 2000), participação notada sobretudo em estudos que utilizaram observação participante, nos quais ocasionalmente as crianças foram contatadas. Essa situação está se modificando, mas a passos lentos, pois ouvir crianças pequenas não é tarefa fácil, em especial aquelas com deficiência intelectual. A psicologia, a educação e a sociologia, por vezes, consideram a opinião de crianças pequenas com deficiência, apesar de poucos estudos abrirem espaço para a voz daquelas com deficiência intelectual. Os pesquisadores em campos de Arquitetura e Urbanismo se aventuraram ainda menos nessa área, logo, pouco se sabe sobre a opinião dessas crianças sobre o ambiente, principalmente em relação à percepção e à qualificação espacial. Ao investigar tal questão, esta tese objetiva adaptar e aplicar à Arquitetura e Urbanismo instrumentos de coleta de dados provenientes de psicologia, educação e sociologia, direcionados a crianças pequenas com deficiência intelectual. A tese é resultado de pesquisa de caráter qualitativo, exploratório e interdisciplinar, por meio de revisão bibliográfica e estudos de caso em quatro instituições especializadas no atendimento de pessoas com deficiência intelectual. A elaboração e a aplicação dos instrumentos de pesquisa especialmente preparados para interlocução com crianças pequenas com Síndrome de Down têm ancoragem nas entrevistas com profissionais e nos grupos focais com pais de crianças atendidas pelos mesmos. Os dados coletados permitem a elaboração de diretrizes específicas para o aperfeiçoamento do material técnico desenvolvido e indica modos para facilitar o planejamento e a realização de pesquisas voltadas para esse público-alvo. Espera-se, assim, contribuir para futuras pesquisas que considerem a opinião destes usuários no que tange à qualidade espacial dos ambientes em que se inserem e, ao mesmo tempo, apoiar arquitetos e urbanistas no exercício profissional voltado a este perfil de usuários. / Young children, ages 4 to 7, are still little recognized as protagonists of academic studies, notably in Architecture and Urbanism, even though since the 1970s this concern is already present on literature. In Brazil, the preoccupation in considering young children as competent interlocutors to provide information on themselves are relatively recent (first dissertations on this field are from the early 2000s). Such participation is noted mainly in studies that utilized participant observation, in which children were occasionally contacted. This situation is changing, however gradually, because listening to young children is no easy task, especially those with intellectual disability. Psychology, Education and Sociology often consider young children with disability\'s opinion, although few studies open space for those with intellectual disability. The researchers on the fields of Architecture and Urbanism venture even less on that particular field, thus, little is known about those children\'s opinion on the environment, mainly related to the perception and spatial qualification. Investigating such question, this thesis aims to adapt and apply to Architecture and Urbanism tools for data collection coming from Psychology, Education and Sociology, directed to Young children with intellectual disability. Assuming the qualitative, exploratory and interdisciplinary characters of the thesis, an extensive bibliographical review supports the realization of case studies in four institutions that are specialized on taking care for people with intellectual disability. At an early stage, we developed specialist panels (interviews with professional and focal groups with parents of those children), which aided on elaboration and application of research tools specially prepared for interlocution with young children with Down Syndrome. The collected data allowed elaboration of specific guidelines to the improvement of the technical material developed indicating methods to facilitate planning and realization of researches to that target audience. It is expected, then, to contribute to future researches that consider the opinion of these users on what\'s relative to the spatial quality of the environments in which they are inserted, and at the same time, support architects and urbanists on exercising their profession referent to this user profile.

[pt] O nascimento de um bebê com síndrome de Down, sem diagnóstico prénatal, configura um momento potencialmente traumático para seus pais. Este acontecimento intervém no exercício da parentalidade, sobretudo quando esta se inaugura neste contexto. Investigamos a importância do preparo das equipes de saúde em lidar com esta experiência e, assim, propiciar a emergência do apego e dos vínculos entre o bebê e os pais. Para tal empreendimento estudamos as relações pais-bebê pelo prisma de diversas teorias, iniciando com a teoria do apego de John Bowlby, passando pelo eu-pele de Didier Anzieu e finalizando com Freud, Winnicott e Bion. Abordamos, ainda, o tema da construção da parentalidade com o bebê portador da síndrome de Down e as suas especificidades: o luto pelo bebê ideal, o narcisimo ferido dos pais e as vicissitudes do trauma. Exploramos o dispositivo de estimulação precoce e introduzimos possíveis contribuições do campo da psicanálise e da psicoterapia da relação pais-bebê. Pesquisamos este universo através de um estudo de campo, realizando entrevistas semi-estruturadas com pais e profissionais da área. Das análises do discurso dos sujeitos, cinco categorias emergiram: o momento da notícia, o luto, a formação dos laços afetivos, a síndrome de Down e a estimulação precoce. Estas categorias foram discutidas em profundidade, a partir dos capítulos teóricos. Constatamos que os profissionais dos centros obstétricos que comunicam aos pais o diagnóstico do filho e os terapeutas quando realizam a estimulação precoce do bebê, em geral, não consideram os aspectos relacionais entre os membros do conjunto pais-bebê-profissionais como parte do próprio trabalho, e isto, além de gerar diversos impasses nas maternidade e nos centros de estimulação precoce, não contribui para a elaboração do luto pelo bebê ideal por parte dos pais, nem para o acionamento do potencial maturativo do bebê e do seu advento como sujeito. Inovamos, ao propor, então, um deslocamento da estimulação precoce do bebê ao acolhimento precoce da família. / [en] The birth of a child with Down syndrome without a prenatal diagnosis configures a potentially traumatic moment for parents. This event affects the exercise of parenthood, especially when the later is inaugurated in this context. We researched the importance of the preparation of health teams in dealing with this experience and, therefore, in fostering attachment and bonds between infant and parents. For such project, we studied the parents-baby relationships trough the prism of several theories, starting with John Bowlby´s attachment theory, passing trough the skin ego from Didier Anzieu and finalizing with Freud, Winnicott and Bion. We approached also the theme of the parenthood construction with a baby with Down syndrome and its specificities: the mourning for the ideal baby, the parents´ hurt narcissism and the trauma vicissitudes. We explored the early intervention device and introduced possible contributions from the psychoanalysis field and from the infant-parent psychotherapy. We researched this universe through a field study, performing semi-structured interviews with parents and professionals from this sector. From the analysis of the speeches of these subjects five categories were raised: the moment of breaking the news, the mourning, the formation of affective bonds, the Down syndrome and the early intervention. These categories were discussed in depth at the theory chapters. We noted that the professionals from obstetrician centers that communicate the child diagnosis to the parents and the therapists when performing early intervention in the baby, in general, do not consider the relationships aspects of the parent-infant-professionals group as part of their own work, and this situation, besides generating several roadblocks at the maternity centers and at the early intervention centers, do not contribute to the elaboration of the parents´ mourning of the ideal baby, as well as to driving the maturational potential of the infant and its advent as subject. We innovated, when proposing a displacement of the early intervention of the infant to the early family welcome.

A síndrome de Down (SD) é uma síndrome genética causada pela trissomia do cromossomo 21 e a causa mais comum de deficiência intelectual. Apresenta alterações em todo o desenvolvimento, em particular o linguístico, que é caracterizado por atraso de linguagem, principalmente quanto aos aspectos morfossintáticos. Tais déficits são confirmados por valores de Extensão Média do Enunciado (EME) abaixo do que seria esperado tanto para a idade cronológica (IC) quanto para a mental (IM) quando se consideram crianças com desenvolvimento típico (DT). A EME tem sido considerada na literatura internacional como o índice mais aceito e efetivo para medir o desenvolvimento gramatical e morfológico, tanto de indivíduos com DT quanto de indivíduos com alterações de linguagem, como por exemplo, o Distúrbio Específico de Linguagem (DEL) e a SD. Pesquisas que comparam as duas populações apontam similaridades entre os desempenhos desses indivíduos quando se considera a EME. Na literatura nacional, estudos que abordem os aspectos morfossintáticos em indivíduos com SD e que considerem a EME como ferramenta de avaliação são escassos. O objetivo do presente estudo foi descrever as habilidades linguísticas de crianças com SD falantes do Português Brasileiro por meio da análise da EME, medida tanto em morfemas (EME-m) quanto em palavras (EME-p). Participaram do estudo três grupos de crianças, sendo um pesquisa (GP-SD) e dois controles (GC-DEL e GC-DT, cujos dados são retrospectivos de estudo anterior), cada um com 25 participantes. Os grupos foram divididos em três subgrupos, cada um, de acordo com a faixa etária (três, quatro e cinco anos). Os participantes do GP-SD foram pareados aos demais por IM. Foi realizada análise da EME de cada sujeito com SD, obtida por meio de amostras de fala, que resultou em 2500 enunciados. Os resultados indicaram que as crianças com SD apresentaram valores de EME menores que as crianças do GC-DEL e do GC-DT, o que aponta para diferenças entre os grupos, para déficits morfossintáticos e para atraso no desenvolvimento linguístico. Verificou-se ainda o desenvolvimento de habilidades morfossintáticas em idades mais avançadas. As similaridades com o GC de crianças com DEL nem sempre foram observadas, já que o GP-SD apresentou desempenho inferior e maior dificuldade quanto ao uso de palavras funcionais. Conclui-se assim, que as crianças com SD do presente estudo apresentaram déficits gramaticais importantes que se caracterizam pelo atraso do desenvolvimento morfossintático. A EME demonstrou ser uma ferramenta confiável e eficaz para identificar o desenvolvimento gramatical e linguístico da população com SD, o que confirma a validade da utilização de tal índice para essa população. Aponta se para a necessidade de estudos com populações maiores de indivíduos com SD, apesar da grande variabilidade interindividual, o que forneceria dados mais representativos para a prática clínica fonoaudiológica baseada em evidências. / Down syndrome (DS) is a genetic syndrome caused by trisomy 21 and it is the most common reason of intellectual disability. It leads to disorders in all development process, in particularly linguistic, which is characterized by language development delay, mainly regarding morphosyntax aspects. Those deficits are confirmed by values of Mean Length Utterance (MLU) lower than expected for chronological age (CA) as well as for mental age (MA) when considered typically developing children (TDC). MLU have been considered in international literature as the most acceptable and effective index to measure grammatical and morphological development to TDC as well as to children with language disorders, such as Specific Language Impairment (SLI) and DS. Researches that compare those former groups show similarities between their performances concerning MLU. National researches regarding morphosyntax aspects in DS considering MLU as an evaluation tool are scarce. The purpose of this study was depicting linguistics skills of children with DS, speakers of Brazilian Portuguese, through MLU analysis measured in morphemes (MLU-m) as well as in words (MLU-w). Participated in the study three groups of children as follows: research group (RG-DS), two control groups (CG-SLI and CG-TDC which data are retrospective from previous study), each group had 25 children. Groups were divided into three subgroups according to the age range (three, four or five years old). The participants of RG-DS group were paired by MA. It was performed MLU analysis in each child with DS that was obtained through their speech samples that leaded to 2500 statements. Results indicated that children with DS showed MLU values lower than children from CG-SLI and CG-TDC, which indicates delay in linguistic development. Further, it was found greater morphosyntax development skills in children who were older aged. Similarities to CG-SLI were not often found, once RG-DS showed lower performance and greater difficulty at using function words. It is therefore concluded that children with DS in this study presented such grammatical deficits characterized by morphosyntax development delay. MLU demonstrated being a reliable and effective tool to identify linguistic and grammatical development in children with SD, what confirms the validity of the use of such index to that population. It points to the need of more studies with larger sample of children with DS, even considering individual variability, in order to provide data more representative to the Speech-Language Pathology clinical practice based on evidence.

Obstructive sleep apnoea/hypopnoea syndrome (OSAHS) is characterised by repeated cycles of upper airway obstruction during sleep, leading to diurnal symptoms. Individuals with Down syndrome (DS) are predisposed to this as the DS phenotype overlaps with OSAHS risk factors. Around 2-4% of the general adult population and 55% of children with DS have OSAHS but, to date, no large-scale study has assessed OSAHS prevalence or efficacy of treatment in DS adults. This study aimed to: 1) Systematically assess subjective and objective OSAHS prevalence; 2) Assess the effectiveness of continuous positive airway pressure (CPAP) in an adult DS population. Standard questionnaires including pictorial Epworth Sleepiness Scale (pESS) and Developmental Behaviour Checklist for Adults (DBC-A) were sent to UK adults aged ≥16yr with DS and their caregivers. All questionnaire responders were invited to undergo home polygraphy. Symptomatic adults with DS with ≥10 apnoeas/hypopnoeas per hour in bed (AH) on home polygraphy were invited to participate in a prospective randomised controlled trial (RCT) of CPAP v. lifestyle advice, with review at 1, 3, 6 and 12m. Participants in the lifestyle arm were offered CPAP at 1m. Standard measurements of sleepiness, behaviour, cognitive function and general health were undertaken. Standard statistical analyses were conducted, with significance set at p < 0.001 to control for multiple testing. Of 5270 questionnaires sent, 1105 responses were valid (21%). Responders (55% males) were overweight/obese young adults: mean BMI 29.0±6.8kg/m2; mean age 28±9 years. Women had a higher BMI (p < 0.0001), but collar size was greater in men (p < 0.0001). Mean pESS scores were broadly within the normal range (7±5/24). No significant gender differences in OSAHS symptoms were noted. Individuals with probable OSAHS had higher pESS and DBC-A scores, and significantly more symptoms of OSAHS. Subjective OSAHS prevalence was estimated at 35%. Of the 790 individuals invited, 149 underwent polygraphy, with 134 valid studies obtained: mean AH 21.8(10.9-42.7); mean oximetry desaturation index (ODI) 6.6(2.3-20.0). No significant gender differences were observed. Forty-two percent of participants met standard clinical diagnostic criteria for OSAHS. Twenty-eight eligible adults with DS (19 male) were randomised: age 28±9yr; BMI 31.5±7.9kg/m2; AH 28.6(14.8-47.9); ODI 7.3(1.8-21.9); pESS 11±6/24. Groups did not differ significantly at baseline. By 12m, 4 participants had withdrawn (all remaining participants on CPAP). The pESS (p=0.001), DBC-A Disruptive (p < 0.0001) and Kaufmann Brief Intelligence Test verbal subscale (p=0.001) scores improved significantly. This first large study of OSAHS prevalence in the adult DS population estimates a prevalence of 35-42% - around 10 times higher than in the general adult population. Sustained, significant improvements in sleepiness, cognitive function and behavioural/emotional outcomes with CPAP use over a 12m period were demonstrated during this first RCT of CPAP in adults with DS. A larger trial of CPAP in this population is warranted.