Palatoplasty Outcomes in Nonsyndromic Patients with Cleft Palate: a 29-Year Assessment of One Surgeon’S Experience
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ORIGINAL ARTICLE Palatoplasty Outcomes in Nonsyndromic Patients With Cleft Palate: A 29-Year Assessment of One Surgeon’s Experience Stephen R. Sullivan, MD,* Eileen M. Marrinan, MS, MPH,Þ Richard A. LaBrie, EdD,þ Gary F. Rogers, MD, JD, MBA, MPH,* and John B. Mulliken, MD* the functional outcome of palatoplasty is not evident until the infant Abstract: The primary objective of cleft palate repair is velophar- is older. Velopharyngeal insufficiency (VPI), the audible hallmark of yngeal competence without fistula. The reported incidence of fistula a nonfunctional palate, is failed velopharyngeal sphincter closure and velopharyngeal insufficiency (VPI) is variable. Our purpose was and is characterized by hypernasal resonance and decreased intraoral to assess the senior surgeon’s 29-year palatoplasty experience with pressure for pressure-dependent consonants during speech.8 The respect to incidence of fistula and VPI. Our hypotheses were that VPI reported frequency of VPI is 5% to 30%.4,9Y16 is related to (1) age at palatoplasty, (2) cleft palate type, and (3) VPI Surgeons are obligated to intermittently and critically assess and palatal fistula incidence decrease with the surgeon’s experience. their results and redirect treatment protocols accordingly. Neverthe- We reviewed the records of all children with cleft palate treated by the less, the wide variability of published outcomes underscores the lack of standardization that permeates the literature and limits meaningful senior author between 1976 and 2004. Cleft palate was categorized interpretation of results.17 At the most basic level, some studies fail to according to Veau. Palatoplasty was performed on 449 patients, using stratify outcomes by cleft severity or other important patient variables a 2-flap technique with muscular retropositioning. The mean age at (eg, hearing, syndromic association). A more pervasive problem is the palatoplasty was 11.6 T 4.9 months (range, 7.0Y46.4 months). The definitional and nosological inconsistencies between surgeons and incidence of palatal fistula was 2.9%, and velopharyngeal sufficiency centers. Although there are several comparative studies, most are was found in 85.1% of patients. We found a significant association limited by the inclusion of multiple surgeons, institutions, and op- between age at palatoplasty and VPI (P = 0.009, odds ratio, 1.06 [95% erative techniques.4,11,16,18 There are few clinical audits document- confidence interval, 1.02Y1.10]). Velopharyngeal insufficiency was ing the long-term results of cleft palate repair by 1 surgeon using 1 10,13Y15 also associated with the Veau hierarchy (P = 0.001). Incidence of VPI technique. was independent of surgeon experience (P = 0.2). In conclusion, the The purpose of this study was to determine if age at palato- plasty, type of cleft, or surgical experience is associated with palato- incidence of palatal fistula was low. Velopharyngeal insufficiency was plasty outcome (ie, fistula, VPI). To reduce confounding variables, we associated with increasing age at palatoplasty and with the Veau analyze the senior surgeon’s 29-year experience at a single institution hierarchy. using 1 palatoplasty technique. Key Words: Velopharyngeal insufficiency, fistula, velopharyngeal incompetence, palatoplasty, cleft palate, Veau classification MATERIALS AND METHODS (J Craniofac Surg 2009;20: 612Y616) Patient Population After approval by the institutional review board of the Com- lthough many methods have been described to close a palatal mittee on Clinical Investigation, we identified and reviewed the charts Acleft, all share the common goal of establishing a competent of all patients with cleft palate repaired by the senior author at the velopharyngeal sphincter. Palatoplasty success is measured by both Children’s Hospital Boston between 1976 and 2004. We included 1 the structural and functional integrity of the repaired palate. Loss of those patients who were at least 4 years of age at the time of this structural integrity after palatoplasty results in a fistula. Palatal fis- review. Four years was chosen because children with velopharyngeal tula is evident shortly after palatoplasty and, depending on the size of sufficiency at age 4 years are unlikely to subsequently develop VPI, the defect, may affect velopharyngeal competence and speech. The and children younger than 4 years are often unable to cooperate for 2Y7 reported incidence of fistula ranges from 4.7% to 60%. In contrast, appropriate speech assessment.13,19 Exclusion criteria were submu- cous cleft palate, identified syndrome, Robin sequence, and hearing From the *Department of Plastic Surgery and the Craniofacial Centre, Chil- loss (sensorineural or persistent conductive hearing loss despite tym- dren’s Hospital and Harvard Medical School, Boston, Massachusetts; panostomy). Data collected included date of birth, sex, cleft palate †Central New YorkCleft and Craniofacial Center, Upstate Medical University type (classified according to Veau:20 I [soft palate], II [hard/soft pal- Hospital, Syracuse, New York; and ‡Division on Addictions, Cambridge ate extending to the incisive foramen], III [unilateral complete cleft Health Alliance and Harvard Medical School, Boston, Massachusetts. lip/palate], and IV [bilateral complete cleft lip/palate]), age at palato- Received August 8, 2008. plasty, postoperative speech assessments, and need for a secondary Accepted for publication October 19, 2008. operation to correct VPI. Palatal fistulas were recorded (nasal-alveolar Address correspondence and reprint requests to John B. Mulliken, MD, and anterior palatal fistulas intentionally not repaired [Pittsburgh types Department of Plastic Surgery, Children’s Hospital, 300 Longwood Ave, 21 2 Hunnewell 1, Boston, MA 02115; E-mail: john.mulliken@childrens. VI and VII ], and bifid uvula [Pittsburgh type I] were excluded ). harvard.edu Copyright * 2009 by Mutaz B. Habal, MD Palatoplasty Technique ISSN: 1049-2275 The operative technique in all patients was a 2-flap palatoplasty. DOI: 10.1097/SCS.0b013e318192801b Nasal lining dissected from the palatal shelves and vomerine flap(s) 612 The Journal of Craniofacial Surgery & Volume 20, Supplement 1, March 2009 Copyright @ 2009 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited. The Journal of Craniofacial Surgery & Volume 20, Supplement 1, March 2009 Palatoplasty Outcomes were used as indicated in complete clefts. The anteriorly displaced We next compared our binary outcome variables, fistula and tensor and levator veli palatini muscles were incised from their ab- VPI requiring a secondary operation, to the cleft type using Fisher normal attachment to the posterior palatine edge, dissected from the exact test and to the age at palatoplasty using logistic regression. To oral and nasal mucosa, retropositioned, and apposed. Other maneu- evaluate for a possible association between surgeon experience and vers to facilitate closure were dissection of the greater palatine arteri- VPI requiring a secondary operation, we separated patients into es from the mucoperiosteal flaps, posterior ostectomy of the greater quartiles based on year of palatoplasty: group 1 (1976Y1990, n = palatine foramina, and hamular fracture to free the tensor veli palatini 112), group 2 (1990Y1995, n = 113), group 3 (1995Y2000, n = 112), tendon and facilitate posteromedial displacement of the velar mus- and group 4 (2000Y2004, n = 112). All calculated P values were cles and mucoperiosteal flaps for a 3-layer soft palatal repair. A gauze 2-tailed and considered significant if less than 0.05. Results were palatal pack, soaked in balsam of Peru, was sutured to the alveolar presented as odds ratios (ORs) and 95% confidence intervals (95% ridges to support the mucoperiosteal flaps and minimize bleeding, CIs) as a measure of association. Statistical analyses were per- pain, and dead space under the flaps. formed using Stata version 8 (Statacorp, College Station, TX). Palatoplasty Outcome RESULTS Patients were followed up annually in our interdisciplinary cleft palate clinic. The senior author examined all the patients in Veau Types conjunction with speech pathologists who specialize in cleft palate. We included 449 patients in this review and summarized The presence of a fistula, recorded by the senior author or any other patient characteristics overall and by cleft type in Table 1. Veau III observer (eg, dental, oral surgery), was recorded. The speech was the most common cleft type. There was a significant difference pathologist completed perceptual assessments on each patient using in sex distribution among cleft palate types: girls more commonly the Pittsburgh Weighted Values for Speech Symptoms Associated had a Veau I or II cleft, and boys more commonly had a Veau III or With Velopharyngeal Incompetence instrument.22 Outcome mea- IV (P G 0.001). The mean age at palatoplasty was 11.6 T 4.9 months sures were palatal fistula and VPI requiring a secondary operation. (range, 7.0Y46.4 months). There were no differences in age at Velopharyngeal function was classified as sufficient, border- palatoplasty among cleft palate types (P = 0.8). line, or insufficient. Velopharyngeal insufficiency was characterized by moderate or severe hypernasal resonance, audible nasal emission, Palatal Fistula and decreased intraoral pressure. A tailored superiorly based Palatal fistula was found in 13 patients (2.9%). Most were 23,24 pharyngeal flap was recommended for patients with VPI or slitlike and found at the junction between the hard and soft palate. those with borderline velopharyngeal