Speech Outcomes and Velopharyngeal Function After Surgical Treatment of Velopharyngeal Insufficiency in Individuals With Signs of Velocardiofacial Syndrome

Brandão, Giovana Rinalde; Freitas, José Alberto de Souza; Genaro, Kátia Flores; Yamashita, Renata Paciello; Fukushiro, Ana Paula; Lauris, José Roberto Pereira

doi:10.1097/scs.0b013e31822e624f

Cited by 23 publications

(21 citation statements)

References 39 publications

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“…However, in this case, the analysis made by listeners was based on the presence or the absence of hypernasality and not on the grading of the symptom. From the studies that classified hypernasality using a four-point scale, most found agreement that are similar to those reported in this study, ranging from fair to moderate (21) , moderate to good (27) , and moderate (28) . With regard to the use of training and this study, other researchers also reported improvement in intra-and interrater reliability after listener training (15) .…”

Section: Discussionsupporting

confidence: 70%

Influência do treinamento dos avaliadores no julgamento perceptivo da hipernasalidade

Oliveira

Scarmagnani

Fukushiro

et al. 2016

CoDAS

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RESUMO Introdução Alto índice de concordância no julgamento perceptivo da hipernasalidade entre diferentes avaliadores é difícil de ser alcançado. O treinamento prévio dos avaliadores e a padronização dos critérios de análise podem ser estratégias eficazes para minimizar o efeito da subjetividade do julgamento perceptivo e aumentar a concordância entre os avaliadores. Objetivo Investigar a influência do treinamento prévio sobre a concordância entre diferentes avaliadores no julgamento perceptivo da hipernasalidade. Métodos Três fonoaudiólogas experientes analisaram 77 amostras de fala, de indivíduos com fissura de palato reparada. Na primeira etapa, as avaliadoras classificaram a hipernasalidade utilizando seus próprios critérios, em uma escala de quatro pontos. Setenta dias depois, foram submetidas a um treinamento para a definição das amostras utilizadas como referências para o julgamento na etapa seguinte. Na segunda etapa as avaliadoras julgaram as mesmas amostras e classificaram a hipernasalidade com a mesma escala, utilizando como critério as referências definidas no treinamento. Índices de concordância intra e interavaliadores foram estabelecidos nas duas etapas utilizando-se o coeficiente Kappa e foram comparados por meio do teste Z. Resultados Os índices de concordância intra-avaliadores obtidos entre as duas etapas variou de 0,38 para 0,92, com diferença estatisticamente significativa para uma das avaliadoras (p=0,004). O índice de concordância quanto ao grau de hipernasalidade obtido entre as três avaliadoras após o treinamento (0,54) foi significativamente maior do que o obtido antes do treinamento (0,37; p=0,044). Conclusão O treinamento das avaliadoras e a definição de critérios para a classificação da hipernasalidade levam ao aumento do índice de concordância intra e interavaliadores.

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Section: Discussionsupporting

confidence: 70%

Influência do treinamento dos avaliadores no julgamento perceptivo da hipernasalidade

Oliveira

Scarmagnani

Fukushiro

et al. 2016

CoDAS

Self Cite

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“…In a handful of studies the inter- or intrarater reliability for the speech test were reported, indicating the validity of the test [21], [45], [50], [52], [53]. Others used the previously validated Cleft Audit Protocol for Speech [24] or Pittsburgh Weighted Speech Score [44], [48], [54].…”

Section: Resultsmentioning

confidence: 99%

“…Lower primary success rates for all patients with hypodynamic velopharynges, including those with 22qDS, supports the logic of segregating this group (which constitutes up to 25% of the population with VPD) from the larger cleft palate population [67]. In general, the speech outcome after surgery has been reported to be worse in patients with 22qDS than in patients without the syndrome [25], [36], [47], [49], [56], [79], [80], [81], but some patients with 22qDS fare as well as their non-syndromic counterparts [33], [39], [40], [46], [52], [61], [82].…”

Section: Discussionmentioning

confidence: 99%

In Search of the Optimal Surgical Treatment for Velopharyngeal Dysfunction in 22q11.2 Deletion Syndrome: A Systematic Review

Spruijt

ReijmanHinze²,

Hens

et al. 2012

PLoS ONE

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BackgroundPatients with the 22q11.2 deletion syndrome (22qDS) and velopharyngeal dysfunction (VPD) tend to have residual VPD following surgery. This systematic review seeks to determine whether a particular surgical procedure results in superior speech outcome or less morbidity.Methodology/ Principal FindingsA combined computerized and hand-search yielded 70 studies, of which 27 were deemed relevant for this review, reporting on a total of 525 patients with 22qDS and VPD undergoing surgery for VPD. All studies were levels 2c or 4 evidence. The methodological quality of these studies was assessed using criteria based on the Cochrane Collaboration's tool for assessing risk of bias. Heterogeneous groups of patients were reported on in the studies. The surgical procedure was often tailored to findings on preoperative imaging. Overall, 50% of patients attained normal resonance, 48% attained normal nasal emissions scores, and 83% had understandable speech postoperatively. However, 5% became hyponasal, 1% had obstructive sleep apnea (OSA), and 17% required further surgery. There were no significant differences in speech outcome between patients who underwent a fat injection, Furlow or intravelar veloplasty, pharyngeal flap pharyngoplasty, Honig pharyngoplasty, or sphincter pharyngoplasty or Hynes procedures. There was a trend that a lower percentage of patients attained normal resonance after a fat injection or palatoplasty than after the more obstructive pharyngoplasties (11–18% versus 44–62%, p = 0.08). Only patients who underwent pharyngeal flaps or sphincter pharyngoplasties incurred OSA, yet this was not statistically significantly more often than after other procedures (p = 0.25). More patients who underwent a palatoplasty needed further surgery than those who underwent a pharyngoplasty (50% versus 7–13%, p = 0.03).Conclusions/ SignificanceIn the heterogeneous group of patients with 22qDS and VPD, a grade C recommendation can be made to minimize the morbidity of further surgery by choosing to perform a pharyngoplasty directly instead of only a palatoplasty.

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“…In some patients, these difficulties likely result from craniofacial dysmorphology including cleft palate and other anomalies that often require surgical intervention. Unfortunately, current surgical treatments are only effective in approximately 25% of patients [14, 15], suggesting mechanisms beyond oropharyngeal mechanics may contribute to pathology. Indeed, many cases of pediatric dysphagia in 22q11DS patients are not accompanied by overt craniofacial dysmorphology that requires surgical intervention [13, 16].…”

Section: Introductionmentioning

confidence: 99%

Hard to swallow: Developmental biological insights into pediatric dysphagia

LaMantia

Moody

Maynard

et al. 2016

Developmental Biology

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Pediatric dysphagia—feeding and swallowing difficulties that begin at birth, last throughout childhood, and continue into maturity—is one of the most common, least understood complications in children with developmental disorders. We argue that a major cause of pediatric dysphagia is altered hindbrain patterning during pre-natal development. Such changes can compromise craniofacial structures including oropharyngeal muscles and skeletal elements as well as motor and sensory circuits necessary for normal feeding and swallowing. Animal models of developmental disorders that include pediatric dysphagia in their phenotypic spectrum can provide mechanistic insight into pathogenesis of feeding and swallowing difficulties. A fairly common human genetic developmental disorder, DiGeorge/22q11.2 Deletion Syndrome (22q11DS) includes a substantial incidence of pediatric dysphagia in its phenotypic spectrum. Infant mice carrying a parallel deletion to 22q11DS patients have feeding and swallowing difficulties. Altered hindbrain patterning, neural crest migration, craniofacial malformations, and changes in cranial nerve growth prefigure these difficulties. Thus, in addition to craniofacial and pharyngeal anomalies that arise independently of altered neural development, pediatric dysphagia may reflect disrupted hindbrain patterning and its impact on neural circuit development critical for feeding and swallowing. The mechanisms that disrupt hindbrain patterning and circuitry may provide a foundation to develop novel therapeutic approaches for improved clinical management of pediatric dysphagia.

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Speech Outcomes and Velopharyngeal Function After Surgical Treatment of Velopharyngeal Insufficiency in Individuals With Signs of Velocardiofacial Syndrome

Cited by 23 publications

References 39 publications

Influência do treinamento dos avaliadores no julgamento perceptivo da hipernasalidade

Influência do treinamento dos avaliadores no julgamento perceptivo da hipernasalidade

In Search of the Optimal Surgical Treatment for Velopharyngeal Dysfunction in 22q11.2 Deletion Syndrome: A Systematic Review

Hard to swallow: Developmental biological insights into pediatric dysphagia

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