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Comparison of Hogan pharyngeal flap and sphincter pharyngoplasty in postoperative velopharyngeal function
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ScienceDirect British Journal of Oral and Maxillofacial Surgery xxx (2019) xxx–xxx
Comparison of Hogan pharyngeal flap and sphincter pharyngoplasty in postoperative velopharyngeal function X. Luo a,b,d , C. Guo a,b , H. Yin a,b , B. Shi a,b , X. Yin a,c,∗ , J. Li a,b,∗ a
State Key Laboratory of Oral Diseases & National Clinical Research Centre for Oral Diseases, West China Hospital of Stomatology, Sichuan University, 14 Ren Min Nan Road, Chengdu, 610041, China b Department of Oral and Maxillofacial Surgery, West China Hospital of Stomatology, Chengdu, 610041, China c Department of Orthodontics, West China Hospital of Stomatology, Sichuan University, Chengdu, 610041, China d Department of Oral and Maxillofacial Surgery, Hospital of Stomatology, Zunyi Medical University, Zunyi, 563003, China Accepted 25 November 2019
Abstract The purpose of this study was to compare speech and breathing after sphincter pharyngoplasty and the Hogan pharyngeal flap in the management of cleft-related velopharyngeal insufficiency (VPI). We reviewed 78 patients with VPI who had either the Hogan flap (n = 30) or sphincter pharyngoplasty (n = 48) between 2009 and 2011. Velopharyngeal function, nasal patency, and speech were compared. In the Hogan flap group, 25 patients had achieved velopharyngeal competence and nine had normal speech. In the sphincter pharyngoplasty group, 29 patients achieved velopharyngeal competence and 20 normal speech. The Hogan flap group had a higher rate of velopharyngeal competence (n = 25) than the sphincter pharyngoplasty group (p = 0.033), but there was no significant difference in intelligibility of speech. Eighteen patients in the Hogan flap group and 33 in the sphincter pharyngoplasty group reported symptoms of snoring, with no significant difference in nasal ventilation. Our results suggest that a posterior pharyngeal flap is a more effective technique for managing VPI after repair of cleft palate than sphincter pharyngoplasty, and causes no more postoperative complications in nasal breathing. © 2019 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Pharyngeal flap; Sphincter pharyngoplasty; Velopharyngeal insufficiency; Secondary cleft palate surgery; speech intelligibility
Introduction Cleft lip and palate are common craniofacial congenital deformities, with an incidence of 0.2% - 2.3% in newborns across different ethnic groups. Although primary palatoplasty could effectively close the cleft, publications still report 10% to 30% of patients with insufficient velopharyngeal function who require further surgical intervention after primary repair of the palate.1 ∗ Corresponding authors at: No. 14, Section 3, Ren Min South Road, Chengdu, Sichuan 610041, China. E-mail addresses: [email protected] (X. Yin), [email protected] (J. Li).
Velopharyngeal insufficiency (VPI) refers to the dysfunction of structures that control closure of the velum against the pharyngeal wall during speech. Surgical treatment is still the most important,1 and among the surgical techniques used, the pharyngeal flap has a long history (dating back to 1865),1 and was popularised by Hogan2 in the 1970s. Sphincter pharyngoplasty was first described in 19503 and became a commonly-used technique after some modifications were made when it was described as “dynamic and physiological”.4 Several studies have compared the outcome of the two techniques in terms of the resolution of VPI, intelligibility of speech, nasal emissions, and postoperative complications (such as nasal obstruction). Both methods have proved to be effective in correcting VPI and improving intel-
https://doi.org/10.1016/j.bjoms.2019.11.023 0266-4356/© 2019 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
Please cite this article in press as: Luo X, et al. Comparison of Hogan pharyngeal flap and sphincter pharyngoplasty in postoperative velopharyngeal function. Br J Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.bjoms.2019.11.023
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ligibility, but there is still disagreement about which method is optimal for specific patients.5 Narrowing the pharyngeal port makes it easier for the velum to close completely during speech, but at the same time it may impede the nasal airflow and result in obstructed breathing. Many cleft centres have reported airway obstruction as a major complication after secondary velopharyngoplasty.5 However, few previous reports have described both speech function and nasal ventilation when comparing the two techniques.1,5–9 We have evaluated the pros and cons of the Hogan flap and sphincter pharyngoplasty in cleft-related management of VPI by retrospectively comparing both postoperative velopharyngeal function and nasal ventilation. Material and methods All patients or guardians signed informed consent forms. The research protocol was approved by the ethics committee of the West China Hospital of Stomatology, Sichuan University (Approval No. WCHSIRB-D-2016-084R1). All methods were in accordance with the Declaration of Helsinki. Patients studied We reviewed patients with velopharyngeal insufficiency who were treated by Hogan posterior flap pharyngoplasty or sphincter pharyngoplasty between January 2009 and December 2011 in the Department of Oral and Maxillofacial Surgery, West China Hospital of Stomatology, Sichuan University. The inclusion criteria were: age between 5 and 30 years at the time of operation; native speakers of Mandarin; no syndromic symptoms; no learning or hearing impairment; those who had had primary palatoplasty and still had VPI; and had available records of speech, nasal ventilation, and velopharyngeal function preoperatively and six months postoperatively.
50% = extreme insufficiency). Pharyngoplasty was considered necessary when the velopharyngeal closure ratio was below 80%. Evaluation of speech The subjective evaluation was done before and six months after operation. A standard testing list in Mandarin,13 which included 63 commonly-used phrases comprising 21 consonants and three vowels (/a/, /i/, /u/) was used. All the voices were recorded and evaluated, and all correctly and incorrectly pronounced consonants counted to calculate accuracy. Each patient’s results were reported in three scales (up to 50%, 50%-85%, and 85% and over). Accuracy equal to or above 85% was considered normal. Hypernasality was evaluated subjectively and supplemented by the Mackay-Kummer Simplified Nasometric Assessment14 using the KayPENTAX computer-assisted Nasometer (KayPENTAX), and the severity scored on a five-point scale15 (-1: hyponasality; 0: normal; 1: slight hypernasality; 2: moderate hypernasality; 3: severe hypernasality).16 Nasal emissions were scored as normal (no emission), inaudible, and audible according to published standards.17 Evaluation of nasal ventilation Nasal ventilation was evaluated by physical examination before the operation and at discharge, The extent of obstruction18 was scored based on the facial expression, nasal airflow during inhalation and exhalation, and subjective feedback from patients. A three-point scale was used (0: no obstruction; 1: mild to moderate effort required to get sufficient ventilation; and 2: severe obstruction: difficulty in getting enough air through nostrils only, or the airway completely blocked). Six months after operation, patients and their families reported the incidence of snoring on a Yes/No questionnaire.18
Assessment of velopharyngeal function Operation Velopharyngeal function was evaluated by two experienced speech pathologists. Video nasopharyngeal endoscopy (ENFV3; Olympus Optical Co) was done to visualise and record the closure ratio and closure pattern of the velopharynx.10 Once the endoscope was properly positioned under topical anaesthesia, the patient was guided through standardised tests.11 The video nasopharyngeal endoscopy was recorded using a specialised video processor (CV-170 Video Processor; Olympus Optical Co) for subsequent evaluation. Areas of the velopharyngeal port were measured at rest and while speaking, and the severity of VPI quantified by the ratio between the sizes of the velopharyngeal port when speaking and at rest using a four-point scale12 (1: 100% = full closure; 2: 80%–100% = slight insufficiency; 3: 50%–80% = pronounced insufficiency; and 4: less than
The decision for pharyngoplasty was made when hypernasality was evident and the velopharyngeal closure ratio was below 80%. All procedures were completed by the senior author (JL). The superiorly-based pharyngeal flap was done using Hogan’s technique.9 The soft palate was divided in the midline, and posteriorly-based flaps of nasal lining of the soft palate raised. A superiorly-based posterior pharyngeal flap was raised as far as the adenoid region, and sutured to the raw nasal surface of the soft palate. A 4 mm diameter catheter was placed on each side of the flap to calibrate the size of the lateral ports. The tissue that originated on the nasal surface of the soft palate was then sutured to cover the raw surface of the pharyngeal flap. The soft palate and the posterior pharyngeal
Please cite this article in press as: Luo X, et al. Comparison of Hogan pharyngeal flap and sphincter pharyngoplasty in postoperative velopharyngeal function. Br J Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.bjoms.2019.11.023
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Table 1 Postoperative velopharyngeal function. Hogan pharyngeal flap Sphincter pharyngoplasty
Velopharyngeal competence
Velopharyngeal insufficiency
chi squared
p value
25 29
5 19
4.552
0.033
Table 2 Preoperative and postoperative consonant accuracy. Preoperative
Hogan pharyngeal flap Sphincter pharyngoplasty
Postoperative
≤50%
50%-85%
≥85%
≤50%
50%-85%
≥85%
18 14
8 25
4 9
7 10
14 18
9 20
Table 3 Preoperative and postoperative resonance/hypernasality. Preoperative
Hogan pharyngeal flap Sphincter pharyngoplasty
Postoperative
−1
0
1
2
3
−1
0
1
2
3
0 0
0 0
6 3
15 27
9 18
4 4
19 25
7 15
0 4
0 0
Score scales: (-1) hyponasality; (0) normal; (1) slight hypernasality; (2) moderate hypernasality; (3) marked and/or severe hypernasality.
wall were closed in the midline (Supplemental Figures 1. A and B, online only). For the sphincter pharyngoplasty, a horizontal mucosal incision was made across the lower portion of the adenoidal pad. Two superiorly-based myomucosal flaps including the palatopharyngeus muscle were raised on the lateral pharyngeal wall. The flaps were transposed medially to a horizontal position and sutured along the transverse incision on the posterior pharyngeal wall to form a bulge (Supplemental Figures 1 A and C, online only).14 Statistical analyses Patients’ characteristics and outcome measurements were compared between the two operative techniques. The chi squared test or Fisher’s exact test was used for the evaluation of categorical variables including rates of hypernasality, accuracy of consonants, nasal emission, nasal obstruction, and incidence of snoring. Continuous data were expressed as mean (SD) and the independent Student’s t test was used to detect the significance of the differences. All analyses were made using IBM SPSS Statistics for Windows version 19.0 (IBM Corp). P < 0.05 was accepted as statistically significant.
Results Thirty patients (17 male and 13 female, mean (SD) age 16 (7) years) were enrolled in the Hogan group, and 48 (24 male and 24 female, mean (SD) age 18 (6) years) were enrolled in the pharyngoplasty group. There were no significant differences between the groups in age, sex, type of cleft, or time after repair of primary cleft, or in the rates of velopharyngeal closure (39% (7%) in the Hogan flap group and 37%
(7%) in the pharyngoplasty group; p = 0.59), velopharyngeal closure patterns (Supplemental data table 1, online only), or the velopharyngeal function score (Supplemental data table 2, online only) between the two groups. Six months after pharyngoplasty, 25 patients in the Hogan flap group and 29 in the pharyngoplasty group achieved velopharyngeal competence, which was significantly higher in the Hogan flap group than in the sphincter pharyngoplasty group (chi squared = 4.552, p = 0.033) (Table 1, Supplemental data table 2, online only). As shown in Table 2, the accuracy of pronunciation of consonants in both groups improved significantly after operation (Hogan group chi squared = 8.399, p = 0.015 compared with sphincter pharyngoplasty group chi squared = 5.979, p = 0.048). Nine patients in the Hogan group and 20 in the sphincter pharyngoplasty group achieved normal speech according to the results of the consonant accuracy test, making a two-fold increase in the accuracy compared with preoperatively. No significant differences were found in the accuracy of consonants (chi squared = 0.677, p = 0.411) between the two groups postoperatively. Six months postoperatively the number of patients with moderate to severe hypernasality had decreased significantly in both groups (Hogan group: chi squared = 47.077, p < 0.001; sphincter pharyngoplasty: chi squared = 72.065, p < 0.001). Nineteen patients in the Hogan flap group and 25 patients in the sphincter pharyngoplasty group achieved normal resonance (Table 3). Four patients from each group still complained of hyponasality, but there was no significant difference in the outcomes between the two groups (chi squared = 0.950, p = 0.33). Nasal emission was significantly alleviated in both groups (Hogan group: chi squared = 33.626, p < 0.0001; sphincter pharyngoplasty chi squared = 33.778, p < 0.001). The occurrence of audible nasal
Please cite this article in press as: Luo X, et al. Comparison of Hogan pharyngeal flap and sphincter pharyngoplasty in postoperative velopharyngeal function. Br J Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.bjoms.2019.11.023
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Table 4 Preoperative and postoperative nasal emission. Preoperative
Hogan pharyngeal flap Sphincter pharyngoplasty
Postoperative
Audible
Inaudible
Normal
Audible
Inaudible
Normal
29 42
0 1
1 5
7 14
2 3
21 31
Table 5 Postoperative nasal breathing scores. Inhalation
Hogan pharyngeal flap Sphincter pharyngoplasty
Exhalation
0
1
2
0
1
2
19 35
9 11
2 2
20 27
8 11
2 10
Score scales: (0): no obstruction and normal smooth breathing; (1): obstruction: mild to medium strength required to get sufficient ventilation, which refers to the existence of nasal obstruction; (2): severe obstruction: patients find it difficult to get enough air, or the airway is completely blocked and so they have to use strong force or breathe through the mouth. Table 6 Incidence of snoring postoperatively. Hogan pharyngeal flap Sphincter pharyngoplasty Total
Yes
No
18 33 51
12 15 27
emissions decreased significantly in both groups. Over 60% patients had no nasal emissions (Table 4) after operation, and there was no significant difference between the two groups (chi squared = 0.542, p = 0.461). Postoperatively, over half the patients in each group had normal nasal ventilation during inhalation and exhalation, and there were no significant differences between the two groups (inhalation: chi squared = 0.831, p = 0.660; exhalation: chi squared = 2.847, p = 0.241) (Table 5). Eighteen patients in the Hogan group and 33 in the pharyngoplasty group reported snoring (chi squared = 0.625, p = 0.429) (Table 6).
Discussion Both the pharyngeal flap and sphincter pharyngoplasty have been widely used for decades to treat residual VPI after primary repair of cleft palate.5 To narrow the pharyngeal port and compensate for insufficient velar function,1 in the Hogan technique the pharyngeal airway is divided into two smaller tunnels by a superiorly-based flap.2 In the sphincter pharyngoplasty group, the vertical sphincter muscles on either side were raised and rotated in a horizontal direction, constricting the pharynx in a circular fashion.4 The most remarkable difference between these two procedures is that the pharyngoplasty maintains the raising function of the velum,4 while the Hogan flap fixes the velum, and the closure is mainly achieved by the movement of the lateral pharyngeal wall.4,19
Controversial results have been reported of comparisons of the two techniques. Some retrospective analyses6,20,21 have found more efficient velopharyngeal function with the Hogan group than with pharyngoplasty. Witt et al22 however, described a higher rate of persistent VPI postoperatively in the Hogan group than the other. We found a significantly higher rate of velopharyngeal competence (83%) in the Hogan group than the sphincter pharyngoplasty group (60%) (p = 0.033). This might be attributed to the more accurate and consistent control of the lateral port during Hogan flaps.9 The size of specific individual residual pharyngeal ports in the sphincter pharyngoplasty method23 might be compromised by the lack of a standardised technique. Velopharyngeal competence is not the sole standard for the success of management of a cleft. Among those of our patients who obtained velopharyngeal competence postoperatively, 19 patients in the Hogan group and 25 in the sphincter pharyngoplasty group achieved normal resonance, while nine in the Hogan group and 20 in the sphincter pharyngoplasty group achieved normal accuracy of consonants. There was no difference in intelligibility between the two groups (p = 0.33). Our results are consistent with those of previous studies that showed that both methods can produce a significant improvement in speech.8 Nasal obstruction is one of the most common and serious complications after secondary pharyngeal surgery,19,24 and most previous studies have suggested notable problems with ventilation after the Hogan procedure.9,19 However, reports about obstructed ventilation after the pharyngoplasty procedure have varied.20,25 We found that 30%-40% of patients reported problems with nasal ventilation in both groups, but with no significant difference detected. More than 60% of our patients snored postoperatively, which suggested the need for proper sleep care postoperatively. Severity of snoring might alleviate or disappear during the long-term follow up.7 Certain limitations to the current study remain (such as studying a retrospective group rather than doing a more powerful randomised controlled trial analysis).24 More comprehensive objective evaluations, such as polysomnography and lateral videofluoroscopy, should be included to better assess the nasal ventilation and velopharyngeal function. Our results suggested that the Hogan procedure was a more reliable technique for secondary velopharyngeal correction than sphincter pharyngoplasty. We found that the Hogan flap achieved a higher rate of postoperative velopharyngeal competence without causing more postoperative complications in nasal ventilation.
Please cite this article in press as: Luo X, et al. Comparison of Hogan pharyngeal flap and sphincter pharyngoplasty in postoperative velopharyngeal function. Br J Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.bjoms.2019.11.023
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Conflict of interest We have no conflicts of interest. Ethics statement/confirmation of patients’ permission The research protocol has been approved by the ethic committee of West China Hospital of Stomatology, Sichuan University (Approval No. WCHSIRB-D-2016-084R1). All patients or their guardians signed informed consent. Acknowledgements This work has been supported by the National Natural Science Foundation of China granted to J.L. (No. 81500829) and X.Y. (No. 81801019). Appendix A. Supplementary data Supplementary material related to this article can be found, in the online version, at doi:https://doi.org/10.1016/j.bjoms. 2019.11.023. References 1. Naran S, Ford M, Losee JE. What’s new in cleft palate and velopharyngeal dysfunction management? Plast Reconstr Surg 2017;139:e1343–55. 2. Hogan VM. A clarification of the surgical goals in cleft palate speech and the introduction of the lateral port control (l.p.c.) pharyngeal flap. Cleft Palate J 1973;10:331–45. 3. Raol N, Hartnick CJ. Sphincter pharyngoplasty. Adv Otorhinolaryngol 2015;76:58–66. 4. Ysunza A, Pamplona MC. Velopharyngeal function after two different types of pharyngoplasty. Int J Pediatr Otorhinolaryngol 2006;70:1031–7. 5. Abyholm F, D’Antonio L, Davidson Ward SL, et al. Pharyngeal flap and sphincterplasty for velopharyngeal insufficiency have equal outcome at 1 year postoperatively: results of a randomized trial. Cleft Palate Craniofac J 2005;42:501–11. 6. Pensler JM, Reich DS. A comparison of speech results after the pharyngeal flap and the dynamic sphincteroplasty procedures. Ann Plast Surg 1991;26:441–3. 7. Sirois M, Caouette-Laberge L, Spier S, et al. Sleep apnea following a pharyngeal flap: a feared complication. Plast Reconstr Surg 1994;93:943–7.
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8. Collins J, Cheung K, Farrokhyar F, et al. Pharyngeal flap versus sphincter pharyngoplasty for the treatment of velopharyngeal insufficiency: a metaanalysis. J Plast Reconstr Aesthet Surg 2012;65:864–8. 9. Sloan GM. Posterior pharyngeal flap and sphincter pharyngoplasty: the state of the art. Cleft Palate Craniofac J 2000;37:112–22. 10. Woo AS. Velopharyngeal dysfunction. Semin Plast Surg 2012;26:170–7. 11. Golding-Kushner KJ, Argamaso RV, Cotton RT, et al. Standardization for the reporting of nasopharyngoscopy and multiview videofluoroscopy: a report from an International Working Group. Cleft Palate J 1990;27:337–48. 12. Perkins JA, Lewis CW, Gruss JS, et al. Furlow palatoplasty for management of velopharyngeal insufficiency: a prospective study of 148 consecutive patients. Plast Reconstr Surg 2005;116:72–84. 13. Yin H, Li Y, Shi B, et al. Influence of cleft type, age and gender on occurrence of compensatory articulations. J Pract Stomatol 2008;24:583–5. 14. O’Connell BP, Dobbie AM, Oyer SL, et al. The impact of adenoid size on rate of revision sphincter pharyngoplasty. Laryngoscope 2014;124:2170–5. 15. Yoshikane F, Lai LH, Hui BK, et al. Orthognathic consequences of sphincter pharyngoplasty in cleft patients: a 2-institutional study. Plast Reconstr Surg Glob Open 2016;4:e676. 16. Wójcicki P, Wójcicka G. Prospective evaluation of the outcome of velopharyngeal insufficiency therapy after simultaneous double z-plasty and sphincter pharyngoplasty. Folia Phoniatr Logop 2010;62:271–7. 17. de Blacam C, Smith S, Orr D. Surgery for velopharyngeal dysfunction: a systematic review of interventions and outcomes. Cleft Palate Craniofac J 2018;55:405–22. 18. Zhang L, Bai X, Li Z, et al. Improvement of aesthetic and nasal airway in patients with cleft lip nasal deformities: rhinoplasty with septal cartilage graft and septoplasty. Cleft Palate Craniofac J 2018;55:554–61. 19. Swanson JW, Johnston JL, Mitchell BT, et al. Perioperative complications in posterior pharyngeal flap surgery: review of the national surgical quality improvement program pediatric (NSQIP-PEDS) database. Cleft Palate Craniofac J 2016;53:562–7. 20. de Serres LM, Deleyiannis FW, Eblen LE, et al. Results with sphincter pharyngoplasty and pharyngeal flap. Int J Pediatr Otorhinolaryngol 1999;48:17–25. 21. Williams HB, Woolhouse FM. Comparison of speech improvement in cases of cleft palate after two methods of pharyngoplasty. Plast Reconstr Surg Transplant Bull 1962;30:36–42. 22. Witt PD, Myckatyn T, Marsh JL. Salvaging the failed pharyngoplasty: intervention outcome. Cleft Palate Craniofac J 1998;35:447–53. 23. Marsh JL. Management of velopharyngeal dysfunction: differential diagnosis for differential management. J Craniofac Surg 2003;14:621–9. 24. Ettinger RE, Oppenheimer AJ, Lau D, et al. Obstructive sleep apnea after dynamic sphincter pharyngoplasty. J Craniofac Surg 2012;23 Suppl 1:1974–6. 25. Kilpatrick LA, Kline RM, Hufnagle KE, et al. Postoperative management following sphincter pharyngoplasty. Otolaryngol Head Neck Surg 2010;142:582–5.
Please cite this article in press as: Luo X, et al. Comparison of Hogan pharyngeal flap and sphincter pharyngoplasty in postoperative velopharyngeal function. Br J Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.bjoms.2019.11.023
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Comparison of Hogan pharyngeal flap and sphincter pharyngoplasty in postoperative velopharyngeal function X. Luo a,b,d , C. Guo a,b , H. Yin a,b , B. Shi a,b , X. Yin a,c,∗ , J. Li a,b,∗ a
State Key Laboratory of Oral Diseases & National Clinical Research Centre for Oral Diseases, West China Hospital of Stomatology, Sichuan University, 14 Ren Min Nan Road, Chengdu, 610041, China b Department of Oral and Maxillofacial Surgery, West China Hospital of Stomatology, Chengdu, 610041, China c Department of Orthodontics, West China Hospital of Stomatology, Sichuan University, Chengdu, 610041, China d Department of Oral and Maxillofacial Surgery, Hospital of Stomatology, Zunyi Medical University, Zunyi, 563003, China Accepted 25 November 2019
Abstract The purpose of this study was to compare speech and breathing after sphincter pharyngoplasty and the Hogan pharyngeal flap in the management of cleft-related velopharyngeal insufficiency (VPI). We reviewed 78 patients with VPI who had either the Hogan flap (n = 30) or sphincter pharyngoplasty (n = 48) between 2009 and 2011. Velopharyngeal function, nasal patency, and speech were compared. In the Hogan flap group, 25 patients had achieved velopharyngeal competence and nine had normal speech. In the sphincter pharyngoplasty group, 29 patients achieved velopharyngeal competence and 20 normal speech. The Hogan flap group had a higher rate of velopharyngeal competence (n = 25) than the sphincter pharyngoplasty group (p = 0.033), but there was no significant difference in intelligibility of speech. Eighteen patients in the Hogan flap group and 33 in the sphincter pharyngoplasty group reported symptoms of snoring, with no significant difference in nasal ventilation. Our results suggest that a posterior pharyngeal flap is a more effective technique for managing VPI after repair of cleft palate than sphincter pharyngoplasty, and causes no more postoperative complications in nasal breathing. © 2019 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Pharyngeal flap; Sphincter pharyngoplasty; Velopharyngeal insufficiency; Secondary cleft palate surgery; speech intelligibility
Introduction Cleft lip and palate are common craniofacial congenital deformities, with an incidence of 0.2% - 2.3% in newborns across different ethnic groups. Although primary palatoplasty could effectively close the cleft, publications still report 10% to 30% of patients with insufficient velopharyngeal function who require further surgical intervention after primary repair of the palate.1 ∗ Corresponding authors at: No. 14, Section 3, Ren Min South Road, Chengdu, Sichuan 610041, China. E-mail addresses: [email protected] (X. Yin), [email protected] (J. Li).
Velopharyngeal insufficiency (VPI) refers to the dysfunction of structures that control closure of the velum against the pharyngeal wall during speech. Surgical treatment is still the most important,1 and among the surgical techniques used, the pharyngeal flap has a long history (dating back to 1865),1 and was popularised by Hogan2 in the 1970s. Sphincter pharyngoplasty was first described in 19503 and became a commonly-used technique after some modifications were made when it was described as “dynamic and physiological”.4 Several studies have compared the outcome of the two techniques in terms of the resolution of VPI, intelligibility of speech, nasal emissions, and postoperative complications (such as nasal obstruction). Both methods have proved to be effective in correcting VPI and improving intel-
https://doi.org/10.1016/j.bjoms.2019.11.023 0266-4356/© 2019 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
Please cite this article in press as: Luo X, et al. Comparison of Hogan pharyngeal flap and sphincter pharyngoplasty in postoperative velopharyngeal function. Br J Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.bjoms.2019.11.023
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ligibility, but there is still disagreement about which method is optimal for specific patients.5 Narrowing the pharyngeal port makes it easier for the velum to close completely during speech, but at the same time it may impede the nasal airflow and result in obstructed breathing. Many cleft centres have reported airway obstruction as a major complication after secondary velopharyngoplasty.5 However, few previous reports have described both speech function and nasal ventilation when comparing the two techniques.1,5–9 We have evaluated the pros and cons of the Hogan flap and sphincter pharyngoplasty in cleft-related management of VPI by retrospectively comparing both postoperative velopharyngeal function and nasal ventilation. Material and methods All patients or guardians signed informed consent forms. The research protocol was approved by the ethics committee of the West China Hospital of Stomatology, Sichuan University (Approval No. WCHSIRB-D-2016-084R1). All methods were in accordance with the Declaration of Helsinki. Patients studied We reviewed patients with velopharyngeal insufficiency who were treated by Hogan posterior flap pharyngoplasty or sphincter pharyngoplasty between January 2009 and December 2011 in the Department of Oral and Maxillofacial Surgery, West China Hospital of Stomatology, Sichuan University. The inclusion criteria were: age between 5 and 30 years at the time of operation; native speakers of Mandarin; no syndromic symptoms; no learning or hearing impairment; those who had had primary palatoplasty and still had VPI; and had available records of speech, nasal ventilation, and velopharyngeal function preoperatively and six months postoperatively.
50% = extreme insufficiency). Pharyngoplasty was considered necessary when the velopharyngeal closure ratio was below 80%. Evaluation of speech The subjective evaluation was done before and six months after operation. A standard testing list in Mandarin,13 which included 63 commonly-used phrases comprising 21 consonants and three vowels (/a/, /i/, /u/) was used. All the voices were recorded and evaluated, and all correctly and incorrectly pronounced consonants counted to calculate accuracy. Each patient’s results were reported in three scales (up to 50%, 50%-85%, and 85% and over). Accuracy equal to or above 85% was considered normal. Hypernasality was evaluated subjectively and supplemented by the Mackay-Kummer Simplified Nasometric Assessment14 using the KayPENTAX computer-assisted Nasometer (KayPENTAX), and the severity scored on a five-point scale15 (-1: hyponasality; 0: normal; 1: slight hypernasality; 2: moderate hypernasality; 3: severe hypernasality).16 Nasal emissions were scored as normal (no emission), inaudible, and audible according to published standards.17 Evaluation of nasal ventilation Nasal ventilation was evaluated by physical examination before the operation and at discharge, The extent of obstruction18 was scored based on the facial expression, nasal airflow during inhalation and exhalation, and subjective feedback from patients. A three-point scale was used (0: no obstruction; 1: mild to moderate effort required to get sufficient ventilation; and 2: severe obstruction: difficulty in getting enough air through nostrils only, or the airway completely blocked). Six months after operation, patients and their families reported the incidence of snoring on a Yes/No questionnaire.18
Assessment of velopharyngeal function Operation Velopharyngeal function was evaluated by two experienced speech pathologists. Video nasopharyngeal endoscopy (ENFV3; Olympus Optical Co) was done to visualise and record the closure ratio and closure pattern of the velopharynx.10 Once the endoscope was properly positioned under topical anaesthesia, the patient was guided through standardised tests.11 The video nasopharyngeal endoscopy was recorded using a specialised video processor (CV-170 Video Processor; Olympus Optical Co) for subsequent evaluation. Areas of the velopharyngeal port were measured at rest and while speaking, and the severity of VPI quantified by the ratio between the sizes of the velopharyngeal port when speaking and at rest using a four-point scale12 (1: 100% = full closure; 2: 80%–100% = slight insufficiency; 3: 50%–80% = pronounced insufficiency; and 4: less than
The decision for pharyngoplasty was made when hypernasality was evident and the velopharyngeal closure ratio was below 80%. All procedures were completed by the senior author (JL). The superiorly-based pharyngeal flap was done using Hogan’s technique.9 The soft palate was divided in the midline, and posteriorly-based flaps of nasal lining of the soft palate raised. A superiorly-based posterior pharyngeal flap was raised as far as the adenoid region, and sutured to the raw nasal surface of the soft palate. A 4 mm diameter catheter was placed on each side of the flap to calibrate the size of the lateral ports. The tissue that originated on the nasal surface of the soft palate was then sutured to cover the raw surface of the pharyngeal flap. The soft palate and the posterior pharyngeal
Please cite this article in press as: Luo X, et al. Comparison of Hogan pharyngeal flap and sphincter pharyngoplasty in postoperative velopharyngeal function. Br J Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.bjoms.2019.11.023
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Table 1 Postoperative velopharyngeal function. Hogan pharyngeal flap Sphincter pharyngoplasty
Velopharyngeal competence
Velopharyngeal insufficiency
chi squared
p value
25 29
5 19
4.552
0.033
Table 2 Preoperative and postoperative consonant accuracy. Preoperative
Hogan pharyngeal flap Sphincter pharyngoplasty
Postoperative
≤50%
50%-85%
≥85%
≤50%
50%-85%
≥85%
18 14
8 25
4 9
7 10
14 18
9 20
Table 3 Preoperative and postoperative resonance/hypernasality. Preoperative
Hogan pharyngeal flap Sphincter pharyngoplasty
Postoperative
−1
0
1
2
3
−1
0
1
2
3
0 0
0 0
6 3
15 27
9 18
4 4
19 25
7 15
0 4
0 0
Score scales: (-1) hyponasality; (0) normal; (1) slight hypernasality; (2) moderate hypernasality; (3) marked and/or severe hypernasality.
wall were closed in the midline (Supplemental Figures 1. A and B, online only). For the sphincter pharyngoplasty, a horizontal mucosal incision was made across the lower portion of the adenoidal pad. Two superiorly-based myomucosal flaps including the palatopharyngeus muscle were raised on the lateral pharyngeal wall. The flaps were transposed medially to a horizontal position and sutured along the transverse incision on the posterior pharyngeal wall to form a bulge (Supplemental Figures 1 A and C, online only).14 Statistical analyses Patients’ characteristics and outcome measurements were compared between the two operative techniques. The chi squared test or Fisher’s exact test was used for the evaluation of categorical variables including rates of hypernasality, accuracy of consonants, nasal emission, nasal obstruction, and incidence of snoring. Continuous data were expressed as mean (SD) and the independent Student’s t test was used to detect the significance of the differences. All analyses were made using IBM SPSS Statistics for Windows version 19.0 (IBM Corp). P < 0.05 was accepted as statistically significant.
Results Thirty patients (17 male and 13 female, mean (SD) age 16 (7) years) were enrolled in the Hogan group, and 48 (24 male and 24 female, mean (SD) age 18 (6) years) were enrolled in the pharyngoplasty group. There were no significant differences between the groups in age, sex, type of cleft, or time after repair of primary cleft, or in the rates of velopharyngeal closure (39% (7%) in the Hogan flap group and 37%
(7%) in the pharyngoplasty group; p = 0.59), velopharyngeal closure patterns (Supplemental data table 1, online only), or the velopharyngeal function score (Supplemental data table 2, online only) between the two groups. Six months after pharyngoplasty, 25 patients in the Hogan flap group and 29 in the pharyngoplasty group achieved velopharyngeal competence, which was significantly higher in the Hogan flap group than in the sphincter pharyngoplasty group (chi squared = 4.552, p = 0.033) (Table 1, Supplemental data table 2, online only). As shown in Table 2, the accuracy of pronunciation of consonants in both groups improved significantly after operation (Hogan group chi squared = 8.399, p = 0.015 compared with sphincter pharyngoplasty group chi squared = 5.979, p = 0.048). Nine patients in the Hogan group and 20 in the sphincter pharyngoplasty group achieved normal speech according to the results of the consonant accuracy test, making a two-fold increase in the accuracy compared with preoperatively. No significant differences were found in the accuracy of consonants (chi squared = 0.677, p = 0.411) between the two groups postoperatively. Six months postoperatively the number of patients with moderate to severe hypernasality had decreased significantly in both groups (Hogan group: chi squared = 47.077, p < 0.001; sphincter pharyngoplasty: chi squared = 72.065, p < 0.001). Nineteen patients in the Hogan flap group and 25 patients in the sphincter pharyngoplasty group achieved normal resonance (Table 3). Four patients from each group still complained of hyponasality, but there was no significant difference in the outcomes between the two groups (chi squared = 0.950, p = 0.33). Nasal emission was significantly alleviated in both groups (Hogan group: chi squared = 33.626, p < 0.0001; sphincter pharyngoplasty chi squared = 33.778, p < 0.001). The occurrence of audible nasal
Please cite this article in press as: Luo X, et al. Comparison of Hogan pharyngeal flap and sphincter pharyngoplasty in postoperative velopharyngeal function. Br J Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.bjoms.2019.11.023
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Table 4 Preoperative and postoperative nasal emission. Preoperative
Hogan pharyngeal flap Sphincter pharyngoplasty
Postoperative
Audible
Inaudible
Normal
Audible
Inaudible
Normal
29 42
0 1
1 5
7 14
2 3
21 31
Table 5 Postoperative nasal breathing scores. Inhalation
Hogan pharyngeal flap Sphincter pharyngoplasty
Exhalation
0
1
2
0
1
2
19 35
9 11
2 2
20 27
8 11
2 10
Score scales: (0): no obstruction and normal smooth breathing; (1): obstruction: mild to medium strength required to get sufficient ventilation, which refers to the existence of nasal obstruction; (2): severe obstruction: patients find it difficult to get enough air, or the airway is completely blocked and so they have to use strong force or breathe through the mouth. Table 6 Incidence of snoring postoperatively. Hogan pharyngeal flap Sphincter pharyngoplasty Total
Yes
No
18 33 51
12 15 27
emissions decreased significantly in both groups. Over 60% patients had no nasal emissions (Table 4) after operation, and there was no significant difference between the two groups (chi squared = 0.542, p = 0.461). Postoperatively, over half the patients in each group had normal nasal ventilation during inhalation and exhalation, and there were no significant differences between the two groups (inhalation: chi squared = 0.831, p = 0.660; exhalation: chi squared = 2.847, p = 0.241) (Table 5). Eighteen patients in the Hogan group and 33 in the pharyngoplasty group reported snoring (chi squared = 0.625, p = 0.429) (Table 6).
Discussion Both the pharyngeal flap and sphincter pharyngoplasty have been widely used for decades to treat residual VPI after primary repair of cleft palate.5 To narrow the pharyngeal port and compensate for insufficient velar function,1 in the Hogan technique the pharyngeal airway is divided into two smaller tunnels by a superiorly-based flap.2 In the sphincter pharyngoplasty group, the vertical sphincter muscles on either side were raised and rotated in a horizontal direction, constricting the pharynx in a circular fashion.4 The most remarkable difference between these two procedures is that the pharyngoplasty maintains the raising function of the velum,4 while the Hogan flap fixes the velum, and the closure is mainly achieved by the movement of the lateral pharyngeal wall.4,19
Controversial results have been reported of comparisons of the two techniques. Some retrospective analyses6,20,21 have found more efficient velopharyngeal function with the Hogan group than with pharyngoplasty. Witt et al22 however, described a higher rate of persistent VPI postoperatively in the Hogan group than the other. We found a significantly higher rate of velopharyngeal competence (83%) in the Hogan group than the sphincter pharyngoplasty group (60%) (p = 0.033). This might be attributed to the more accurate and consistent control of the lateral port during Hogan flaps.9 The size of specific individual residual pharyngeal ports in the sphincter pharyngoplasty method23 might be compromised by the lack of a standardised technique. Velopharyngeal competence is not the sole standard for the success of management of a cleft. Among those of our patients who obtained velopharyngeal competence postoperatively, 19 patients in the Hogan group and 25 in the sphincter pharyngoplasty group achieved normal resonance, while nine in the Hogan group and 20 in the sphincter pharyngoplasty group achieved normal accuracy of consonants. There was no difference in intelligibility between the two groups (p = 0.33). Our results are consistent with those of previous studies that showed that both methods can produce a significant improvement in speech.8 Nasal obstruction is one of the most common and serious complications after secondary pharyngeal surgery,19,24 and most previous studies have suggested notable problems with ventilation after the Hogan procedure.9,19 However, reports about obstructed ventilation after the pharyngoplasty procedure have varied.20,25 We found that 30%-40% of patients reported problems with nasal ventilation in both groups, but with no significant difference detected. More than 60% of our patients snored postoperatively, which suggested the need for proper sleep care postoperatively. Severity of snoring might alleviate or disappear during the long-term follow up.7 Certain limitations to the current study remain (such as studying a retrospective group rather than doing a more powerful randomised controlled trial analysis).24 More comprehensive objective evaluations, such as polysomnography and lateral videofluoroscopy, should be included to better assess the nasal ventilation and velopharyngeal function. Our results suggested that the Hogan procedure was a more reliable technique for secondary velopharyngeal correction than sphincter pharyngoplasty. We found that the Hogan flap achieved a higher rate of postoperative velopharyngeal competence without causing more postoperative complications in nasal ventilation.
Please cite this article in press as: Luo X, et al. Comparison of Hogan pharyngeal flap and sphincter pharyngoplasty in postoperative velopharyngeal function. Br J Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.bjoms.2019.11.023
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Conflict of interest We have no conflicts of interest. Ethics statement/confirmation of patients’ permission The research protocol has been approved by the ethic committee of West China Hospital of Stomatology, Sichuan University (Approval No. WCHSIRB-D-2016-084R1). All patients or their guardians signed informed consent. Acknowledgements This work has been supported by the National Natural Science Foundation of China granted to J.L. (No. 81500829) and X.Y. (No. 81801019). Appendix A. Supplementary data Supplementary material related to this article can be found, in the online version, at doi:https://doi.org/10.1016/j.bjoms. 2019.11.023. References 1. Naran S, Ford M, Losee JE. What’s new in cleft palate and velopharyngeal dysfunction management? Plast Reconstr Surg 2017;139:e1343–55. 2. Hogan VM. A clarification of the surgical goals in cleft palate speech and the introduction of the lateral port control (l.p.c.) pharyngeal flap. Cleft Palate J 1973;10:331–45. 3. Raol N, Hartnick CJ. Sphincter pharyngoplasty. Adv Otorhinolaryngol 2015;76:58–66. 4. Ysunza A, Pamplona MC. Velopharyngeal function after two different types of pharyngoplasty. Int J Pediatr Otorhinolaryngol 2006;70:1031–7. 5. Abyholm F, D’Antonio L, Davidson Ward SL, et al. Pharyngeal flap and sphincterplasty for velopharyngeal insufficiency have equal outcome at 1 year postoperatively: results of a randomized trial. Cleft Palate Craniofac J 2005;42:501–11. 6. Pensler JM, Reich DS. A comparison of speech results after the pharyngeal flap and the dynamic sphincteroplasty procedures. Ann Plast Surg 1991;26:441–3. 7. Sirois M, Caouette-Laberge L, Spier S, et al. Sleep apnea following a pharyngeal flap: a feared complication. Plast Reconstr Surg 1994;93:943–7.
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8. Collins J, Cheung K, Farrokhyar F, et al. Pharyngeal flap versus sphincter pharyngoplasty for the treatment of velopharyngeal insufficiency: a metaanalysis. J Plast Reconstr Aesthet Surg 2012;65:864–8. 9. Sloan GM. Posterior pharyngeal flap and sphincter pharyngoplasty: the state of the art. Cleft Palate Craniofac J 2000;37:112–22. 10. Woo AS. Velopharyngeal dysfunction. Semin Plast Surg 2012;26:170–7. 11. Golding-Kushner KJ, Argamaso RV, Cotton RT, et al. Standardization for the reporting of nasopharyngoscopy and multiview videofluoroscopy: a report from an International Working Group. Cleft Palate J 1990;27:337–48. 12. Perkins JA, Lewis CW, Gruss JS, et al. Furlow palatoplasty for management of velopharyngeal insufficiency: a prospective study of 148 consecutive patients. Plast Reconstr Surg 2005;116:72–84. 13. Yin H, Li Y, Shi B, et al. Influence of cleft type, age and gender on occurrence of compensatory articulations. J Pract Stomatol 2008;24:583–5. 14. O’Connell BP, Dobbie AM, Oyer SL, et al. The impact of adenoid size on rate of revision sphincter pharyngoplasty. Laryngoscope 2014;124:2170–5. 15. Yoshikane F, Lai LH, Hui BK, et al. Orthognathic consequences of sphincter pharyngoplasty in cleft patients: a 2-institutional study. Plast Reconstr Surg Glob Open 2016;4:e676. 16. Wójcicki P, Wójcicka G. Prospective evaluation of the outcome of velopharyngeal insufficiency therapy after simultaneous double z-plasty and sphincter pharyngoplasty. Folia Phoniatr Logop 2010;62:271–7. 17. de Blacam C, Smith S, Orr D. Surgery for velopharyngeal dysfunction: a systematic review of interventions and outcomes. Cleft Palate Craniofac J 2018;55:405–22. 18. Zhang L, Bai X, Li Z, et al. Improvement of aesthetic and nasal airway in patients with cleft lip nasal deformities: rhinoplasty with septal cartilage graft and septoplasty. Cleft Palate Craniofac J 2018;55:554–61. 19. Swanson JW, Johnston JL, Mitchell BT, et al. Perioperative complications in posterior pharyngeal flap surgery: review of the national surgical quality improvement program pediatric (NSQIP-PEDS) database. Cleft Palate Craniofac J 2016;53:562–7. 20. de Serres LM, Deleyiannis FW, Eblen LE, et al. Results with sphincter pharyngoplasty and pharyngeal flap. Int J Pediatr Otorhinolaryngol 1999;48:17–25. 21. Williams HB, Woolhouse FM. Comparison of speech improvement in cases of cleft palate after two methods of pharyngoplasty. Plast Reconstr Surg Transplant Bull 1962;30:36–42. 22. Witt PD, Myckatyn T, Marsh JL. Salvaging the failed pharyngoplasty: intervention outcome. Cleft Palate Craniofac J 1998;35:447–53. 23. Marsh JL. Management of velopharyngeal dysfunction: differential diagnosis for differential management. J Craniofac Surg 2003;14:621–9. 24. Ettinger RE, Oppenheimer AJ, Lau D, et al. Obstructive sleep apnea after dynamic sphincter pharyngoplasty. J Craniofac Surg 2012;23 Suppl 1:1974–6. 25. Kilpatrick LA, Kline RM, Hufnagle KE, et al. Postoperative management following sphincter pharyngoplasty. Otolaryngol Head Neck Surg 2010;142:582–5.
Please cite this article in press as: Luo X, et al. Comparison of Hogan pharyngeal flap and sphincter pharyngoplasty in postoperative velopharyngeal function. Br J Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.bjoms.2019.11.023