Need for Velopharyngeal Management Following Palatoplasty: An Outcome Analysis of Syndromic and Nonsyndromic Patients with Robin Sequence

Witt, Peter D.; Myckatyn, Terry; Marsh, Jeffrey L.; Grames, Lynn Marty; Dowton, Bruce S.

doi:10.1097/00006534-199705010-00008

Cited by 43 publications

(35 citation statements)

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“…Furthermore, the rate of surgical correction of VPI in the syndromic PRS patients was 23% compared to 36% of the non-syndromic patients, with an average rate of 31% for all patients, which is higher than what we found but less than that found by Witt et al [6,15]. The differences between the rates of VPI after primary palate repair may be related to technique, as de Buys left their palatal flaps pedicled anteriorly, whereas we did not and Witt et al described a 2 flap technique but did not indicate whether the anterior portion of the flaps were left attached [6,15]. However, the rate of persisting hypernasality remains about 16-18% in PRS and CPO patients, which is reflected in the VP closure, suggesting that further surgery for these patients may be necessary.…”

Section: Discussioncontrasting

confidence: 87%

“…A study by de Buys et al comparing speech outcomes between syndromic and non-syndromic PRS patients showed no differences in the need for surgical intervention for VPI [15]. Furthermore, the rate of surgical correction of VPI in the syndromic PRS patients was 23% compared to 36% of the non-syndromic patients, with an average rate of 31% for all patients, which is higher than what we found but less than that found by Witt et al [6,15]. The differences between the rates of VPI after primary palate repair may be related to technique, as de Buys left their palatal flaps pedicled anteriorly, whereas we did not and Witt et al described a 2 flap technique but did not indicate whether the anterior portion of the flaps were left attached [6,15].…”

Section: Discussioncontrasting

confidence: 73%

“…Secondary surgical management of VPI is based on the anatomic type of closure that is found, and often patient's speech improves greatly [12]. Up to this point, it has been assumed that PRS patients had a 44% incidence of VPI that would require surgical intervention [6]. A correlation has been found between an increased width of the cleft palate defect, as is found in PRS children, and increased frequency of hypernasality [13].…”

Section: Discussionmentioning

confidence: 99%

“…Many studies have been performed to evaluate mandibular development and its effect on airway obstruction, but few papers have looked at speech outcomes in patients with PRS [3][4][5]. The first to strictly focus on speech outcomes in PRS patients by Witt et al found that patients with PRS had a higher incidence of velopharyngeal insufficiency (VPI) (44%) when compared to cleft palate only patients [6].…”

Section: Introductionmentioning

confidence: 99%

See 3 more Smart Citations

The occurrence of velopharyngeal insufficiency in Pierre Robin Sequence patients

Goudy¹,

Ingraham²,

Canady³

2011

International Journal of Pediatric Otorhinolaryngology

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Section: Discussioncontrasting

confidence: 87%

Section: Discussioncontrasting

confidence: 73%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 2 more Smart Citations

The occurrence of velopharyngeal insufficiency in Pierre Robin Sequence patients

Goudy¹,

Ingraham²,

Canady³

2011

International Journal of Pediatric Otorhinolaryngology

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“…Cognitive development, speech (velopharyngeal inadequacy), obstructive sleep apnea, and recurrence risk are key issues to be monitored by the pediatrician caring for a growing child with RS. 5,98,99 With appropriate interventions and treatment, the prognosis for children with RS is usually quite good.…”

Section: Longitudinal Carementioning

confidence: 99%

Robin Sequence: From Diagnosis to Development of an Effective Management Plan

et al. 2011

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The triad of micrognathia, glossoptosis, and resultant airway obstruction is known as Robin sequence (RS). Although RS is a well-recognized clinical entity, there is wide variability in the diagnosis and care of children born with RS. Systematic evaluations of treatments and clinical outcomes for children with RS are lacking despite the advances in clinical care over the past 20 years. We explore the pathogenesis, developmental and genetic models, morphology, and syndromes and malformations associated with RS. Current classification systems for RS do not account for the heterogeneity among infants with RS, and they do not allow for prediction of the optimal management course for an individual child. Although upper airway obstruction for some infants with RS can be treated adequately with positioning, other children may require a tracheostomy. Care must be customized for each patient with RS, and health care providers must understand the anatomy and mechanism of airway obstruction to develop an individualized treatment plan to improve breathing and achieve optimal growth and development. In this article we provide a comprehensive overview of evaluation strategies and therapeutic options for children born with RS. We also propose a conceptual treatment protocol to guide the provider who is caring for a child with RS. Pediatrics 2011;127:936-948

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Developmental outcome in Pierre Robin sequence: A longitudinal and prospective study of a consecutive series of severe phenotypes

Thouvenin¹,

Djadi-Prat²,

Chalouhi³

et al. 2013

American J of Med Genetics Pt A

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Pierre Robin sequence (PRS) is a congenital condition with a heterogeneous and imprecise developmental prognosis. We conducted a longitudinal prospective study analyzing the long-term developmental outcome of a consecutive series of 39 children with PRS who had an a priori good prognosis (isolated PRS or PRS associated with a Stickler syndrome) but severe neonatal disorders (respiratory and feeding difficulties). Psychomotor and cognitive levels, speech, and eating behavior were assessed at 15 months of age and 3 and 6 years of age; 24 of the oldest children were interviewed at age 11 or 12 years. Results were analyzed by diagnosis, extent of respiratory and feeding disorders, and treatment modalities. Cognitive scores were within normal ranges and increased over time, from 90.5 at 15 months of age to 109.1 at 6 years. The 24 oldest children were enrolled in the appropriate junior high school grade at the normal age. For children 15 months of age, language scores were below the average, as were scores for vocabulary at 3 years for half of the patients. At 6 years, children's speech showed persistent rhinolalia, which was mild (47%), moderate (11%), or major (11%). At 15 months of age, 74% of the children had satisfactory eating behavior, and 15% had serious difficulties. At 3 and 6 years, 18% and 6% of the children, respectively, had eating problems. Treatment modalities had no significant effect on long-term outcome. Global developmental quotient scores were lower but not significantly for children with an associated Stickler syndrome than those with isolated PRS. Children with isolated PRS showed good prognosis.

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Need for Velopharyngeal Management Following Palatoplasty: An Outcome Analysis of Syndromic and Nonsyndromic Patients with Robin Sequence

Cited by 43 publications

References 0 publications

The occurrence of velopharyngeal insufficiency in Pierre Robin Sequence patients

The occurrence of velopharyngeal insufficiency in Pierre Robin Sequence patients

Robin Sequence: From Diagnosis to Development of an Effective Management Plan

Developmental outcome in Pierre Robin sequence: A longitudinal and prospective study of a consecutive series of severe phenotypes

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