Treatment of Post-Adenotonsillectomy Velopharyngeal Stenosis With

International Journal of Pediatric Otorhinolaryngology 126 (2019) 109600

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International Journal of Pediatric Otorhinolaryngology

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Treatment of post-adenotonsillectomy velopharyngeal stenosis with ☆ T bivalved uvular flaps Mosaad Abdel-Aziza,*, Abdel-Rahman El-Tahanb, Mahmoud El-Foulya, Ahmed Kamelc, Assem Abdel-Wahidd a Department of Otolaryngology, Faculty of Medicine, Cairo University, Egypt b Department of Otolaryngology, Aswan University, Egypt c Department of Otolaryngology, Faculty of Medicine, Beni-Suef University, Egypt d Department of Otolaryngology, Faculty of Medicine, Fayoum University, Egypt

ARTICLE INFO ABSTRACT

Keywords: Objective: Velopharyngeal stenosis (VS) is a rare devastating complication of adenotonsillectomy that causes Velopharyngeal stenosis obstructive sleep apnea (OSA). Its treatment is challenging and has a high recurrence rate. The aim of this study Nasopharyngeal stenosis was to assess the efficacy of a bivalved uvular flap technique with topical application of mitomycin Cfor Adenotonsillectomy treatment of this problem. Bivalved flap Study design: Case series. Obstructive sleep apnea Methods: Fourteen children with VS after adenotonsillectomy were treated with a bivalved uvular flap technique with application of mitomycin C after release of the adhesions and removal of scar tissue. Preoperative and postoperative evaluation of patients were performed. Flexible nasopharyngoscopy was used to assess the patency

of the velopharynx, and apnea/hypopnea (A/H) index and minimum O2 saturation were measured before and after surgery. Results: Adequate patent airway was obtained in all patients as seen by oropharyngeal examination and flexible

nasopharyngoscopy. Additionally, significant improvement in A/H index and minimum O2 saturation were achieved postoperatively. Conclusion: The bivalved uvular flap technique with topical application of mitomycin C after removal ofscar tissue is an effective treatment for VS that may follow adenotonsillectomy in children.

1. Introduction sleep apnea. Additionally, it may decrease speech resonance and lead to hyponasality. It may be caused by surgical trauma as after tonsil- The velopharyngeal port is the passage between the nasopharynx lectomy, adenoidectomy, and uvulopalatoplasty or radiotherapy for and oropharynx. It is a mechanical sphincter that opens on breathing treatment of nasopharyngeal carcinoma [4,5]. Because of the high in- and shuts during swallowing to prevent regurgitation of food and fluids cidence of recurrence, treatment of this condition is challenging. Pa- and during articulation of oral phonemes to control speech resonance tients with VS may require repeated surgical interventions to obtain [1,2]. It is controlled by the action of the palatal muscles together with satisfactory results. Therefore, many treatment modalities are being superior constrictors and is lined by a mucous membrane that is re- tried to cure this problem [6]. The aim of this study was to assess the spiratory epithelium in the nasopharynx and nonkeratinized squamous efficacy of a bivalved uvular flap technique with topical application of epithelium in the oropharynx [3]. The area is closely related to the mitomycin C for treatment of VS following adenotonsillectomy in pe- adenotonsillar tissues; the adenoid is located in the supero-posterior diatric patients. wall of the nasopharynx, and the tonsils are located between the pillars of fauces in the oropharynx below the soft palate [4]. Velopharyngeal stenosis (VS) may lead to snoring and obstructive

☆ This multicenter study was conducted on the Departments of Otolaryngology of Cairo University, Aswan University, Beni Suef University and Fayoum University (Egypt). * Corresponding author. 2 el-salam st., King Faisal, above el-baraka bank, Giza, Cairo, Egypt. E-mail addresses: [email protected], [email protected] (M. Abdel-Aziz). https://doi.org/10.1016/j.ijporl.2019.109600 Received 22 April 2019; Received in revised form 12 July 2019; Accepted 19 July 2019 Available online 23 July 2019 0165-5876/ © 2019 Elsevier B.V. All rights reserved. M. Abdel-Aziz, et al. International Journal of Pediatric Otorhinolaryngology 126 (2019) 109600

2. Methods into oral and nasal halves. The cotton pieces were removed, and the lateral parts of the pharyngeal mucosal flap were used to cover the This study was conducted on 14 patients who presented with ve- lateral pharyngeal walls and the central part was used to cover the lopharyngeal stenosis after adenotonsillectomy. The patients were re- posterior pharyngeal wall. The uvular flaps were rotated to cover the ferred to our institutes from February 2013 to July 2017; their ages open raw areas on the posterior edge of the soft palate, the oral flap for ranged between 4 and 8.5 years with a mean of 6.3 ± 1.4 years, and the right side and the nasal flap for the left side (Fig. 1). We obtained a there were 9 girls and 5 boys. Patients who underwent surgery for VS, posterior border for the soft palate that was completely covered with who presented with any obstructive airway disease, or who presented uvular mucosa without raw areas. Avoiding overstretch of the mucosal with craniofacial anomalies were excluded. Informed consent was ob- flaps and narrowing of the velopharynx is of paramount importance. All tained from the parents that were informed that repetition of the same flaps were sutured in place, using 4-0 Vicryl as a simple interrupted surgical procedure may be needed in cases of restenosis, and the prin- suture technique. Upon awakening, patients were extubated and placed ciples outlined in the Declaration of Helsinki were followed. In addi- in a lateral position and then transferred to the postanesthesia care unit. tion, the research protocol was approved by the ethics committee of our institute. Written consents were obtained from the parents of the pa- 2.3. Postoperative care and follow-up tient presented in the video files. Patients underwent extensive pre- and postoperative assessment as All patients received oral amoxicillin (50 mg/kg/day) and para- described below: cetamol for 10 days. Patients were discharged from the hospital on the third postoperative day, and they were instructed to consume semisolid 2.1. Preoperative assessment food for one week. Children were seen weekly till wound healing was achieved then monthly for at least one year. Flexible nasopharyngo- Medical history was obtained from the parents of the patients, with scopy, PSG and measurement of BMI were performed one year post- emphasis on symptoms suggestive of obstructive sleep apnea (OSA) [7]. operatively. Only patients with a positive history were included in the study. The type of operative procedure and the duration elapsed until symptoms 2.4. Statistical methods appeared after adenotonsillectomy were recorded. Otolaryngologic examination was performed for assessment of the degree of VS and to Data were coded and summarized using the Statistical Package for exclude any associated airway disorders and/or middle ear effusion. VS Social Sciences for Windows, version 20.0 (SPSS Inc., Chicago, IL). was graded as mild when the lateral aspects of the palate adhered to the Quantitative variables are presented as the mean ± standard deviation posterior pharyngeal wall, moderate when there was circumferential as well as median ± interquartile range. Comparison of preoperative scarring with a small central opening that was 1–2 cm in diameter, and and postoperative results of both the A/H index and minimum O2 sa- severe when the entire palate fused with the posterior and lateral turation was performed using Wilcoxon matched-pair signed-rank test pharyngeal wall leaving a residual opening less than 1 cm [8]. Flexible and paired samples t-test respectively. P < 0.05 was considered sta- nasopharyngoscopic examination was performed to assess the thickness tistically significant. and extent of stenosis and to determine if there was scar tissue within the nasopharynx. The nasopharyngeal airway was also assessed by 3. Results computed tomography (CT) for all patients. All patients were assessed by overnight polysomnography (PSG) for at least 6 h (lab-based study) Fourteen children with VS and OSA were enrolled in the study. The in a quiet, dark room. The Apnea/hypopnea (A/H) index and minimum symptoms appeared between 2 and 5 months with a mean of 2.4

O2 saturation were measured, and the A/H index was categorized as months after adenotonsillectomy. The operation was performed with follows: < 1.0, normal; 1–4.99, mild; 5 to < 9.99, moderate; and monopolar diathermy in 9 patients, bipolar diathermy in 3 patients, and ≥10.0, severe [9]. As the Body mass index (BMI) could affect the PSG coblation radiofrequency in 2 patients. Oropharyngeal examination data, we measured the BMI for all patients dividing their weight in (Fig. 2) showed adhesions between the pillars and the lateral and kilograms by their height in meters squared, and that was done at the posterior pharyngeal walls in all patients, and the VS was severe in 11 day of PSG study. According to BMI-for-age percentile growth charts, it and moderate in 3 patients. No patients presented with middle ear ef- was considered that underweight (≤5th percentile), normal weight fusion. Flexible nasopharyngoscopy showed cicatricial stenosis at the (> 5th and < 85th), overweight (≥85th and < 95th), obese (≥95th level of the velopharyngeal port extending to the oropharynx, neither and < 99th) and severely obese (≥99th) [10]. recurrent adenoid nor scar tissue was found in the nasopharynx in any patient. CT showed narrowing of the airway by condensed soft tissue 2.2. Operative procedure from the soft palate downwards to the oropharynx, and the thickness of the stenotic segment varied from 1.3 to 2.4 cm with a mean of 2 cm. Under general anesthesia with oral endotracheal intubation, a Preoperative PSG data showed OSA in all patients. The A/H index was Davis-Boyle mouth gag was inserted. Incisions were marked and the severe in 12 patients and moderate in two patients, with a median A/H palate and pharynx were injected with 0.5% Xylocaine in adrenaline index of 17.00 (interquartile range = 11), and a mean minimum O2 (1:100,000). The lateral sides of the stenotic area were incised from saturation of 85.36 ± 3.25%. each side of the uvula downwards to the lower end of the stenosis, and The operative decision was made upon diagnosis. However, the another U-shaped incision in the posterior pharyngeal wall was per- duration of symptoms prior to surgery ranged from 5 to 24 months with formed from each side of the uvula downwards around the stenotic a mean of 11.21 months. Excision of the scar tissue was achieved using velopharyngeal port. Mucosal flaps were elevated, and the underlying cold instruments with limited use of bipolar diathermy for hemostasis. scar tissue was removed from the lateral and posterior pharyngeal walls No intraoperative or postoperative complications were encountered, as well as from the posterior border of the soft palate on both sides of but, temporary nasal regurgitation of fluid and hypernasality of speech the uvula (Fig. 1). Cotton pieces soaked with mitomycin C were applied were recognized by the parents in all patients. Also, all patients suffered to the raw areas after removal of the scar tissue (in a dose of 0.4 mg/ dysphagia that gradually disappeared within two weeks post- mL) [11]. At this stage, we obtained three mucosal flaps; one palatal operatively. flap on each side of the uvula and a large lower pharyngeal flapthat Postoperatively, the parents of all children reported absence of OSA was divided into three parts. The uvula was bivalved by extending the symptoms on the first visit. Oropharyngeal examination showed release two lateral incisions medially so that the uvula was divided coronally of the adhesions between the soft palate and the pharyngeal wall

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Fig. 1. Illustrations for the procedure. A) The central white area is the stenotic velopharyngeal port and the dotted line is the incision marking. B) The pharyngeal mucosal flap is divided into three parts and the uvula is bivalved coronally into oral and nasal flaps. The arrows pointed for rotation ofeach mucosal flap to cover its corresponding raw area after removal of scar tissue.

(Fig. 2). However, six patients demonstrated scar tissue that appeared index showed significant improvement, with P = 0.001. Also, com- on the 3rd postoperative month between the soft palate and the lateral parison between the preoperative and postoperative minimum O2 sa- pharyngeal walls bilaterally, those patients showed adequate airway by turation showed significant improvement, with P < 0.001. the end of the follow-up period. Flexible nasopharyngoscopy showed an Regarding the BMI, the mean preoperative was 15.41 ± 0.56 and adequately patent velopharyngeal port. PSG data proved normalization the mean postoperative was 15.64 ± 0.67. According to BMI-for-age of the A/H index in five patients, while, seven patients demonstrated percentile growth charts, all patients were of normal weight pre and mild A/H index and two patients demonstrated moderate A/H index. postoperatively (Table 1) that excluded the effect of the BMI on the PSG The median postoperative A/H index was 1.00 (interquartile data. range = 1.3) and a mean minimum O2 saturation of 92.78 ± 1.84% (Table 1). Comparison between the preoperative and postoperative A/H

Fig. 2. Oropharyngeal examination of a patient with velopharyngeal stenosis. (A) Preoperative view with severe stenosis. (B) Postoperative view with adequate airway.

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Table 1 Clinical characteristics of the patients.

patient Age (years) Sex Severity of VS BMI AHI MIN. O2 %

Pre Post Pre Post Pre Post

1 6 Female Severe 15.34 16.12 17 0.5 85 92 2 4 Male Severe 15.19 15.08 20 1 86 92 3 5.5 Female Severe 15.15 15.12 13 0.5 88 95 4 8 Female Moderate 15.87 16.64 12 1 89 93 5 8.5 Male Severe 14.96 15.26 21 1 83 96 6 4.5 Male Severe 15.99 16.64 8 0.5 89 92 7 5 Female Moderate 15.43 15.88 19 4 80 91 8 7 Female Severe 14.37 15.28 7 1 87 93 9 6.5 Male Severe 16.39 15.98 22 5 82 90 10 6 Female Severe 15.88 15.45 10 0.5 81 92 11 7.5 Female Severe 15.03 15.36 18 0.5 87 94 12 8 Female Severe 14.96 15.38 11 1 90 95 13 5 Female Severe 15.15 14.31 20 5 86 90 14 6.5 Male Moderate 16.12 16.48 17 1 82 94

VS, velopharyngeal stenosis; AHI, apnea/hypopnea index; BMI, body mass index; Min. O2, minimum oxygen saturation; Pre, preoperative; Post, one year postoperatively.

4. Discussion sample was small. Cotton [14] used a laterally-based pharyngeal flap and they achieved successful results in six out of seven patients. Nan- Adenotonsillectomy is a relatively common procedure in clinical gole and Khainga [15] described the efficacy of the facial artery mus- otolaryngology practice and usually follows referrals from primary care culomucosal flap as a treatment option for severe stenosis, four outof physicians. It is the treatment of choice for OSA in children [12]. VS is their five patients had complete relief of airway obstruction. However, an uncommon complication of the procedure that may be caused by the procedure was performed in two stages. A palatal eversion tech- approximation of raw surface areas during the healing process forming nique was reported by Abdel-Fattah [2] as a successful method for VS. scar tissues that adhere the palate to the lateral and posterior phar- He divided the soft palate at the midline and removed the scar tissue. yngeal walls [4]. VS is a challenging problem as it causes circumfer- The two palatal halves were sutured laterally to allow epithelization, ential narrowing of the airway and OSA, and it is difficult to correct and and to be reunited in a second stage after six months. Although it is a often recurs. Therefore, the severity of the symptoms must be balanced good idea and the author achieved good results in 10 patients, it is a against the difficulty of surgical repair and the possibility of recurrence. two-stage procedure with the possibility of postoperative palatal de- In this study, we treated 14 children with VS after adeno- fects. In our study, we used local mucosal flaps to cover the raw areas in tonsillectomy using a bivlaved uvular flap after release of adhesions and a single stage procedure and we achieved adequate patent airway in all removal of scar tissue. Intraoperatively, a topical application of mito- our patients. mycin C was used as a fibroblast inhibitor, and the raw areas were Other therapeutic options for VS include steroid injection, scar lysis completely covered with mucosal flaps. Bivalved uvular flaps covered with CO2 laser, plasma hook, yttrium-aluminum-garnet (YAG) laser, or the raw areas on the posterior border of the soft palate, whereas with power instruments such as shavers, Surgitron high-frequency radio pharyngeal mucosal flaps covered the lateral and posterior pharyngeal waves, skin grafts, Z-plasty repair, insertion of prosthetic stents, naso- walls. We achieved adequately patent airway as seen by oropharyngeal pharyngeal obturators, and balloon dilation of the stenotic segment examination and flexible nasopharyngoscopy. Polysomnography was [1,5,6,16,17]. Karakoc et al. [18] used endoscopy-assisted coblation in used in pre- and postoperative assessment and proved that all patients two patients with VS, they reported that the procedure was less painful had OSA preoperatively, we achieved significant improvement of PSG than other surgical options and can be successfully used without the data postoperatively. need for postoperative stents. Topical application of mitomycin C was Pharyngeal surgeries may be rarely complicated with VS. used as an adjuvant therapy by many authors. It is a fibroblast inhibitor Adenotonsillectomy, uvulopalatopharyngoplasty, and laser-assisted and might reduce scarring and recurrence [5,6,11,17]. However, there uvulopalatoplasty have emerged as the predominant causes for VS [13]. is no general consensus for treatment of VS due to the rarity of cases Adenotonsillectomy was the leading cause of VS in our children, and and limited experience of the surgeons who face this difficult problem. the operation was performed with monopolar diathermy in 9 patients, Meticulous surgical technique is the key to minimizing this complica- bipolar diathermy in 3 patients, and coblation radiofrequency in 2 tion [4]. patients. VS after adenotonsillectomy may be attributed to excessive It is worth mentioning that our study is based on a somewhat small mucosal removal of the posterior tonsillar pillars, overzealous removal sample of patients due to the rarity of the disease, and we did not assess of inferolateral adenoid tissues, inadvertent injury of the posterior the speech of the patients objectively, as our objective was to assess the pharyngeal wall, liberal use of electrocautery for hemostasis resulting efficacy of our technique for relieving the obstructive airway effectsof in coagulation necrosis of palatopharyngeal mucosa, cicatrizing or ke- VS. loidal diasthesis, and postoperative infection of the surgical wound [2,4,13]. Many techniques have been described for repair of VS, ranging from 5. Conclusion simple dilatation of the stenotic velopharyngeal port with or without the use of an indwelling obturator to the use of local or distant flaps to Velopharyngeal stenosis is a devastating complication that may cover the raw areas after scar tissue removal [5]. Toh et al. [13] re- follow adenotonsillectomy. It causes OSA, and its treatment seems to be ported a reliable technique for treatment of this problem using bivalved difficult. A bivalved uvular flap technique with topical application of palatal transposition flaps: a superiorly-based flap for the nasophar- mitomycin C after removal of scar tissue is an effective treatment for the yngeal surface of the soft palate, and an inferiorly-based flap for the problem in children. oropharyngeal side. They achieved good results, however, the study

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