A, Primary repair of cleft palate. B, Secondary palatal rerepair. In the scenario of secondary Furlow palatoplasty of a previously well-repaired soft palate, the levator muscle complex may be disrupted and its muscle fibers reoriented in a slightly more sagittal direction (bottom row of B).
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Rudnicki PA, Tsang C, Vecchiotti MA, Scott AR. Palatal Motion After Primary and Secondary Furlow Palatoplasty. JAMA Otolaryngol Head Neck Surg. 2017;143(2):111–115. doi:10.1001/jamaoto.2016.2783
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Is there a difference in postoperative soft palate motion following primary and secondary Furlow palatoplasty?
In this case series of 20 patients, powered to detect a 25% difference in palatal motion scores, no significant difference was identified between primary and secondary groups. There was fair interrater reliability, consistent with prior reports using this scale.
When examined in isolation, postoperative motion of the soft palate appears similar following both primary and secondary Furlow palatoplasty procedures.
Indications for Furlow palatoplasty include primary repair of cleft palate as well as secondary repair, or secondary palatoplasty for treatment of velopharyngeal insufficiency. Speculation exists surrounding the benefit of secondary Furlow palatoplasty in cases of a previously well-reconstructed palate or a short but otherwise anatomically normal soft palate because it has been theorized that reorientation of a previously reconstructed or normal muscular levator sling should in fact worsen palatal motion.
To compare palatal motion following primary and secondary Furlow palatoplasty using footage from postoperative nasopharyngoscopy videos.
Design, Setting, and Participants
In this retrospective case series, medical records in a database of an urban academic pediatric otolaryngology practice was used to identify patients who had undergone either primary or secondary Furlow palatoplasty. Subjects with adequate postoperative nasopharyngoscopy footage were randomized, and 2 blinded reviewers assessed soft palate motion in each video using an abbreviated version of the Golding-Kushner scale.
Main Outcomes and Measures
Reviewers' blinded ratings of soft palate motion were quantified using a modified Golding-Kushner scale to generate a mean palatal motion score for each subject (range, 0.0-2.0). Scores of primary and secondary Furlow palatoplasty patients were compared.
Over a 4-year period, 20 patients with adequate postoperative nasopharyngoscopy footage were identified (12 primary Furlow palatoplasty patients and 8 secondary Furlow palatoplasty patients). Patients consisted of 8 males and 12 females and ranged in age from 12 months to 22 years at the time of postoperative nasopharyngoscopy. Modified Golding-Kushner scores were similar between groups: mean primary group, 1.61 (range, 0.5-2.0); mean secondary group, 1.53 (range, 0.75-2.0); absolute difference in mean, 0.08 (95% CI, 0.00-0.43); effect size, Hedges g, 0.18. There was fair interrater reliability (interclass coefficient, R = 0.45), consistent with prior reports using this scale. No significant difference in postoperative palatal motion scores was identified between primary and secondary palatoplasty groups in this study.
Conclusions and Relevance
When examined in isolation, postoperative motion of the soft palate appears similar following both primary and secondary Furlow palatoplasty procedures, suggesting that there are no major deleterious effects on palatal motion following secondary Furlow palatoplasty.
The palate consists of a hard, static portion with contributions from the maxilla and palatine bones, as well as a soft, dynamic portion that includes the uvula. In patients with cleft palate, the levator veli palatini and other muscles of the soft palate insert into the posterior border of the palatine bone. This aberrant insertion pattern results in a sagittal orientation of the muscle fibers, which is markedly different from the normal, transverse orientation of the levator sling formed from the insertion of these fibers into one another. Any pathologic abnormalities in these muscles and/or soft tissue of the palate may have a noticeable impact on velopharyngeal function; a shorter palate may result in characteristic speech disturbances, including hypernasality and associated maladaptive compensatory speech mechanisms.1
The goals of primary cleft palate repair are 3-fold: close the defect, reorient the muscle fibers, and lengthen the soft palate. In 1986, Leonard Furlow described a double opposing Z-plasty technique that reoriented the muscle fibers of the levator sling, lengthened the soft palate, and created a nonlinear scar, limiting future shortening of the repaired palate secondary to scar contracture (Figure 1A).2 The radical, 90-degree retropositioning of the muscle fibers reestablished healthy soft palate anatomy, and ameliorated speech disturbances in a way that was more efficacious and more consistent than prior techniques. Since that time, the double opposing Z-plasty repair has gained wide acceptance, with Furlow palatoplasty now performed for both primary and secondary palatoplasty, in which the technique is used to address complications such as fistula development or persistent velopharyngeal insufficiency (VPI). However, in the case of secondary palatoplasty, the soft palate is lengthened at the cost of disrupting a previously repaired levator complex. When the previously repaired soft palate exhibits midline notching, shortening, or muscular diastasis, resulting VPI is often related to persistent sagittal orientation of the levator muscle fibers. In such cases, palatal rerepair using the Furlow technique restores healthy, transverse alignment, a soft palate configuration similar to that produced during a primary Furlow repair. However, a Furlow palatoplasty may also be used to lengthen a previously well-repaired soft palate or a short soft palate that seems anatomically healthy otherwise. Theoretically, Furlow repair of a transversely oriented levator sling, as in these cases, should result in distortion of the soft palate musculature, as well as scarring and misalignment of the velar musculature (Figure 1B).3
Given these theoretical concerns, the aim of this study was to examine palatal motion in isolation, following both primary and secondary Furlow palatoplasty. By comparing the 2 groups by way of a blinded review of postoperative nasopharyngoscopy videos, we sought to detect any differences in muscle function between primary and secondary Furlow palatoplasty patients.
Institutional review board approval was obtained from the Tufts Medical Center to conduct the study. A retrospective case series was performed with medical chart review of patients who underwent primary or secondary Furlow palatoplasty over a 4-year span at an urban tertiary pediatric hospital with an accredited multidisciplinary cleft lip and palate team. All primary and secondary Furlow palatoplasties were performed by the senior author (A.R.S), however most of the original repairs for secondary cases had been done by a different surgeon. In all of the secondary cases an intravelar veloplasty had been completed as part the original repair according to the original operative dictation. The patient pool was divided by the primary author (P.A.R.) into 2 groups: 1 consisting of patients who had undergone primary Furlow palatoplasty, and another consisting of those who had undergone secondary Furlow palatoplasty. The videos were edited for length and any identifying information, including all audio. After the videos were randomized and deidentified, 2 fellowship-trained pediatric otolaryngologists (A.R.S. and M.A.V.) independently reviewed the nasopharyngoscopy footage, scoring the motion of each hemisoft palate using a modified version of the Golding-Kushner scale (Figure 2).
The original Golding-Kushner scale grades velopharyngeal function by assessing for no movement vs movement to midline of the following structures: right and left lateral pharyngeal walls, right and left hemisoft palate, and posterior pharyngeal wall.4 The score for each of these subunits ranges from 0 to 0.5 or 0 to 1.0, in 0.25 increments, to achieve a total score ranging from 0 to 4.0. A score of 0 represents no motion, whereas a score of 0.5 or 1.0 (depending on the subunit) represents closure to the midline. The key endoscopic landmarks used for this evaluation are illustrated in Figure 2.
A modified Golding-Kushner scale was used in this study, differing from the classic scale by examining only 2 subunits: the right and left hemisoft palate. The score for each of these subunits ranged from 0 (no motion) to 1.0 (normal motion to the midline). For the sake of standardization, rating scores were assigned at 0.25 increments (Figure 2). The composite score for the left and right soft palate could range from 0 to 2.0.
All data was collected and analyzed using Microsoft Excel (version 14.0, Microsoft). The composite modified Golding-Kushner score (ie, the sum of the right and left soft palate scores) from each examiner was averaged and a composite palatal motion score was determined for each patient. The mean score for each of the 2 patient groups (primary and secondary) was then calculated followed by the absolute difference in mean values for the 2 groups. Effect size was determined using Hedges g coefficient, which provides a measure of effect size weighted according to the relative size of each sample. Interrater reliability coefficient (R) was assessed using the interclass coefficient calculation for ordinal data. Statistical significance was defined as P values less than .05. It was determined that a sample size of 20 was powered to detect a 25% difference (absolute difference = 0.54) in mean palatal motion scores between primary and secondary groups (type II error of 0.15).
There were 20 patients identified with postoperative nasopharyngoscopy videos available for review. Patients consisted of 8 males and 12 females and ranged in age from 12 months to 22 years at the time of postoperative nasopharyngoscopy. There were 12 patients in the primary group and 8 patients in the secondary group. Preoperative oral examination as well as preoperative nasopharyngoscopy findings failed to detect muscular dehiscence or midline notching in any of the secondary Furlow candidates in this study. Nevertheless, all of the palates were short and many of them exhibited poor movement during connected speech. The mean (SD) age at surgery in the primary group was 1.6 (1.49) years and the mean (SD) age at time of repair in the secondary group was 10.3 (6.73) years (Table).
The mean modified Golding-Kushner soft palatal motion score was 1.61 (range, 0.5-2.0) for the primary repair group and 1.53 (range, 0.75-2.0) for the secondary group. The absolute difference in palatal motion score between the 2 groups was 0.08 (95% CI, 0.00-0.43); effect size, g, 0.18. Agreement between the 2 raters was evaluated using an interclass coefficient calculation (for ordinal data), and R was calculated to be 0.45 (fair agreement).
Our results suggest that there may not be any deleterious effects on soft palatal elevation following palatal rerepair using the Furlow double opposing Z-plasty technique. Palatal movement following secondary Furlow palatoplasty was no different from that of patients repaired primarily using the same technique (Figure 3).
In 1986, Leonard Furlow first described a double-opposing Z-plasty repair as a technique for performing primary palatoplasty.2 Since then, several subsequent studies6-8 have demonstrated either unchanged or improved outcomes in velopharyngeal competence and/or speech intelligibility following use of Furlow palatoplasty as a means of secondary, or revision repair. A recent review by Crockett and Goudy8 demonstrated that, when independently assessed, Furlow palatoplasty resulted in improved VPI outcomes across most of the included studies. However, the positive outcomes following double-opposing Z-plasty reconstruction of a previously repaired palate are not intuitive, given the logical concern that repositioning of properly-oriented muscle fibers should in fact have a deleterious effect on soft palate motion.
Recently, Abdel-Aziz et al9 examined the effect of secondary Furlow palatoplasty in a single cohort, with the primary outcomes being palatal closure on nasopharyngoscopy and nasalance scores for oral and nasal sentences. There were significant improvements in speech outcomes from baseline after surgery, supporting the notion that a longer palate with varying orientation in muscle fibers may be more functional than a shorter palate with transverse orientation of the reconstructed levator complex. While the patients all had non-Furlow palatoplasties for primary repair, it was not clear as to why these secondary cases resulted in improved velopharyngeal closure. Was it maintained soft palate function on top of palatal lengthening or merely static closure of the velopharyngeal port through palatal lengthening alone?
More recently, Hsu et al10 also determined that velopharyngeal function is not negatively impaired by palate rerepair. In 13 patients who underwent secondary Furlow palatoplasty for VPI after primary palate repair, velopharyngeal closure was improved in all based on perceptual assessment by speech pathology. Five of these patients underwent nasopharyngoscopy as part of their postoperative assessment, and a comparison of preoperative and postoperative closure ratios was included as an objective measurement of velopharyngeal function in these 5 patients. While lacking somewhat in objective measures for the postoperative success in the other 8 study patients, the findings of the study by Hsu et al10 support our conclusions that palatal function is not negatively affected following secondary or revision Furlow repair.
Our study does have limitations. With a sample size of 20 patients, our investigation was not powered to detect minor differences (ie, less than 25%) in palatal function between primary and secondary groups. In addition, the study does not examine clinical outcomes following these repairs. As noted above, multiple prior articles including a recent review demonstrate the clinical effectiveness of secondary Furlow palatoplasty.9-11 We therefore chose to focus specifically on the question of whether palatal movement is compromised as a necessary means of achieving palatal length following secondary Furlow palatoplasty.
The Golding-Kushner scale is, at its core, a subjective assessment of velopharyngeal function. A recent international working group found that there was a significant degree of interrater variability when using the Golding-Kushner scale, even by experienced otolaryngologists who specialize in treatment of VPI.12 A more user-friendly 10-point rating scale has been posited as a potential substitute, but it has yet to be validated. A prior multicenter study5 demonstrated that interrater agreement for the endoscopic evaluation of velopharyngeal insufficiency varied by anatomic subsite. The R coefficient for soft palate motion was 0.43 in that multicenter study, a value consistent with the R coefficient of 0.45 observed in this study.
Finally, often during secondary Furlow palatoplasty, one does not encounter a truly transverse orientation of muscle fibers, rather an orientation similar to the sagittal alignment observed during repair of a submucous cleft palate is noted. During palatal rerepair of such cases, Furlow palatoplasty reorients the muscles in a proper transverse position but does not overcorrect to a more posterior position than transverse, as implied in Figure 1B.
Notably, none of the 8 patients who underwent secondary Furlow repair in this study had intraoperative findings suggestive of an inadequately repaired levator musculature. While the 8 cases within this report may not be representative of typical secondary Furlow patients, these patients may in fact have undergone overcorrection of the levator sling as depicted in Figure 1B; this overcorrection did not appear to negatively affect palatal function.
To our knowledge, this study is the first of its kind to directly compare soft palate motion among patients who had a primary vs secondary Furlow palatoplasty. Our findings suggest that there is at most a minor difference, if any difference at all, in postoperative palatal function among patients who have undergone primary Furlow palatoplasty or secondary Furlow palatoplasty of a previously repaired cleft palate.
While the concept of secondary Furlow palatoplasty is not a novel one, this study is the first directly comparing postoperative soft palate function among patients who underwent primary and secondary Furlow palatoplasties. Future studies are needed to further analyze these findings on a larger scale to assess for more minor differences in palatal function between these 2 groups. Correlating soft palate motion findings with more established metrics of speech outcomes, such as quality of life surveys and perceptual speech analyses could also be areas of future research.
Corresponding Author: Andrew R. Scott, MD, Department of Pediatric Otolaryngology and Facial Plastic Surgery, Floating Hospital for Children at Tufts Medical Center, 800 Washington St, Boston, MA 02111 (firstname.lastname@example.org).
Accepted for Publication: July 26, 2016.
Published Online: October 6, 2016. doi:10.1001/jamaoto.2016.2783
Author Contributions: Dr Scott had full access to all of the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis.
Concept and design: Rudnicki, Vecchiotti, Scott.
Acquisition, analysis, or interpretation of data: All authors.
Drafting of the manuscript: Rudnicki, Tsang, Scott.
Critical revision of the manuscript for important intellectual content: Tsang, Vecchiotti, Scott.
Statistical analysis: Tsang, Scott.
Administrative, technical, or material support: Vecchiotti, Scott.
Study supervision: Tsang, Vecchiotti, Scott.
Conflict of Interest Disclosures: All authors have completed and submitted the ICMJE Form for Disclosure of Potential Conflicts of Interest and none were reported.
Previous Presentation: This study was presented at the annual Society for Ear, Nose and Throat Advances in Children (SENTAC) meeting; December 5th, 2015; San Antonio, Texas.