Audiology - Communication Research E-ISSN: 2317-6431 [email protected] Academia Brasileira de Audiologia Brasil

Fukushiro, Ana Paula; Domingues Zwicker, Carmen Vivian; Flores Genaro, Katia; Paciello Yamashita, Renata; Kiemle Trindade, Inge Elly Nasopharyngeal dimensions and respiratory symptoms after in children and adults Audiology - Communication Research, vol. 18, núm. 2, abril-junio, 2013, pp. 57-62 Academia Brasileira de Audiologia São Paulo, Brasil

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Nasopharyngeal dimensions and respiratory symptoms after pharyngeal flap surgery in children and adults

Dimensões nasofaríngeas e sintomas respiratórios após a cirurgia de retalho faríngeo em crianças e adultos

Ana Paula Fukushiro1, Carmen Vivian Domingues Zwicker2, Katia Flores Genaro1, Renata Paciello Yamashita1, Inge Elly Kiemle Trindade1

ABSTRACT RESUMO

Purpose: To investigate the influence of age on nasopharyngeal di- Objetivo: Verificar a influência da idade sobre as dimensões nasofarín- mensions and respiratory symptoms in patients undergoing surgery to geas e os sintomas respiratórios em pacientes submetidos à cirurgia para correct velopharyngeal insufficiency using the pharyngeal flap tech- a correção da insuficiência velofaríngea pela técnica de retalho faríngeo. nique. Methods: A sample comprising 103 individuals divided into Métodos: Amostra formada por 103 indivíduos distribuídos em três three groups: children (6-12 years old), young adults (18-30 years old) grupos: crianças (6 a 12 anos), adultos jovens (18 a 30 anos) e adultos de and middle-aged adults (≥ 40 years old). This was a retrospective study meia-idade (≥40 anos). Trata-se de estudo retrospectivo que analisou os that analyzed the values ​​for the nasopharyngeal area (pressure-flow valores da área nasofaríngea (técnica fluxo-pressão, valor de referência technique, reference value: 0.570 cm2) and the symptoms of oral respi- 0,570 cm2) e os sintomas de respiração oral, ronco e dificuldade respira- ration, and respiratory difficulties during sleep. The values were tória durante sono, obtidos, em média, 12 meses após a cirurgia. A área obtained, on average, 12 months after surgery. The nasopharyngeal area nasofaríngea foi comparada entre os grupos por análise de variância e os was compared between the groups using analysis of variance, and res- sintomas respiratórios pelo teste Qui-quadrado (p<0,05). Resultados: Os piratory symptoms were compared using the chi-squared test (p<0.05). valores médios da área nasofaríngea após a cirurgia foram 0,527±0,215 Results: The mean nasopharyngeal area values after surgery were cm2, 0,599±0,213 cm2 e 0,488±0,276 cm2, respectivamente para crianças, 0.527±0.215 cm2, 0.599±0.213 cm2 and 0.488±0.276 cm2 for children, adultos jovens e adultos de meia-idade, não havendo diferença entre os young adults and middle-aged adults, respectively, with no differences grupos. As proporções de respiração oral, ronco e dificuldade respiratória between groups. The proportions of oral respiration, snoring and brea- durante o sono foram, respectivamente, 66%, 69% e 9% nas crianças, thing difficulty during sleep were, respectively, 66%, 69% and 9% in 51%, 49% e 7% nos adultos jovens e 75%, 75% e 19% nos adultos de children; 51%, 49% and 7% in young adults; and 75%, 75% and 19% in meia-idade, com maior frequência de ronco nos adultos de meia-idade. middle-aged adults, with a higher frequency of snoring in middle-aged Conclusão: A idade não interferiu nas dimensões nasofaríngeas na adults. Conclusion: Age did not affect nasopharyngeal dimensions in presença de retalho faríngeo, mas foi um fator agravante de sintomas the presence of a pharyngeal flap, but it was an aggravating factor for respiratórios, principalmente nos indivíduos mais velhos. respiratory symptoms, especially in older individuals. Descritores: Fissura palatina; Insuficiência velofaríngea; Cirurgia bucal; Keywords: Cleft ; Velopharyngeal insufficiency; Surgery, oral; Sinais e sintomas respiratórios; Rinomanometria Signs and symptoms, Respiratory; Rhinomanometry

Work developed in the Laboratory of Physiology, Hospital for Rehabilitation of Craniofacial Anomalies, Universidade de São Paulo – USP – São Paulo (SP), Brazil. (1) Graduate Program in Rehabilitation Sciences, Hospital for Rehabilitation of Craniofacial Anomalies, Universidade de São Paulo – USP – São Paulo (SP), Brazil. (2) Graduate Program (Master) in Rehabilitation Sciences, Hospital for Rehabilitation of Craniofacial Anomalies, Universidade de São Paulo – USP – São Paulo (SP), Brazil. Conflicts of interest:No Author´s contributions: APF: lead author, participation in the original study idea, data collection, data analysis and the writing of the article; CVDZ: participa- tion in statistical data analysis and the writing of the results; KFG: participation in the statistical analysis and the writing of the article; RPY: participation in data collection and the writing of the article; and IEK: research group leader, participation in the original study idea and the writing of the article. Correspondence address: Ana Paula Fukushiro. R. Sílvio Marchione, 3/20, Vila Universitária, Bauru (SP), Brazil, CEP: 17012-900. E-mail: [email protected] Received: 4/25/2012; Accepted: 6/5/2013

ACR 2013;18(2):57-62 57 Fukushiro AP, Zwicker CVD, Genaro KF, Yamashita RP, Trindade IEK

INTRODUCTION In studying sleep-disordered in middle-aged adults who had undergone surgery to correct VPI using a Cleft /palate is a congenital deformity that can manifest pharyngeal flap, one study reported oral respiration, snoring in the lip, palate or both structures. Reconstructive surgery is re- and episodes of respiratory obstruction in 53% of cases, quired to correct this abnormality. and confirming that the pharyngeal flap can contribute to the are the first surgeries performed during the long and complex worsening of respiratory disorders in this age group(19). In treatments which, in conjunction with other therapeutic appro- young adults, the effect of the pharyngeal flap on the nasal aches, are critical for patient rehabilitation(1). and nasopharyngeal dimensions was investigated and corre- A cleft palate is considered the most frequent cause of lated with respiratory complaints, showing the emergence or velopharyngeal insufficiency (VPI), a structural change in the worsening of these complaints in 55% of cases in the short velopharyngeal mechanism. The most common symptom of term and 36% in the long term. In an aerodynamic evaluation, VPI is hypernasality, which is associated with nasal air emission a more pronounced decrease in nasopharyngeal dimensions and low intraoral air pressure(2-4). was observed in patients who only reported respiratory com- It is generally agreed that palatoplasty should have the plaints after surgery, highlighting the importance of moni- functional purpose of improving conditions for the velum to toring surgical results using objective measures(14). Another help the velopharyngeal mechanism. However, in many cases, study involving children analyzed respiratory symptoms and even after primary corrective surgery, VPI symptoms persist, investigated the surgical complications of the pharyngeal requiring a secondary procedure(5,6). Surgery using the supe- flap, identifying oxygen desaturation in 8% of the sample riorly based pharyngeal flap technique is one of the methods and positive findings of in 3% six most frequently employed to correct VPI. The technique con- months after surgery(20). sists of building a bridge of myomucosal tissue between the In terms of VPI correction, surgery should be performed as velum and the posterior pharyngeal wall, bounded by two side soon as there is good nasal patency. However, the effects of the orifices that allow nasal breathing. The purpose of the flap is pharyngeal flap technique on respiration and the technique’s to create a mechanical obstruction to the airflow to eliminate correlation with velopharyngeal orifice dimensions in different hypernasality and nasal air emission, with increased intraoral age groups is a subject that has been minimally explored to date. pressure(7) during speech production. In addition, one must consider that respiratory changes related Several studies have demonstrated the great success of pha- to nasopharyngeal obstruction during sleep increase with age(21). ryngeal flap surgery in reducing or eliminating VPI symptoms Snoring during sleep, episodes of obstructive sleep apnea and in speech, with success rates varying between 25% and 98%(8- other daytime symptoms manifest themselves most often after 10). However, the pharyngeal flap is also associated with upper the age of 40 years(21-23). airway impairment, which may cause hyponasality, chronic The purpose of this study was therefore to determine the nasal obstruction, snoring and obstructive sleep apnea, symp- influence of age on nasopharyngeal dimensions and respiratory toms that interfere with respiratory function and, consequently, symptoms in patients who underwent surgery to correct velo- quality of life(11-14). pharyngeal insufficiency using the pharyngeal flap technique. It is also important to consider that a cleft lip/palate deter- mines nasal deformities that often reduce the dimensions of METHODS the nasal cavity(15) and increase nasal resistance to respiratory airflow. It is therefore possible that in these cases, the placement This was a retrospective and cross-sectional study. It recei- of a flap in the velopharyngeal region may further aggravate ved approval from the Ethics Committee on Human Research of impaired nasal patency(16). the Hospital for Rehabilitation of Craniofacial Anomalies of the Some studies have conducted surveys of clinical symptoms Universidade de São Paulo (USP), protocol number 168/2009. of respiratory obstruction, determining the area of the​​ velopha- Initially, through convenience sampling and in accordance ryngeal orifice and using instrumental assessment to evaluate with the institution’s service order, the records of individuals the effect of pharyngeal flap surgery on breathing in individuals with cleft palate and who had undergone surgery for VPI with a cleft palate(14,17,18). correction using the pharyngeal flap technique were selected. A preliminary sample showed that the pharyngeal flap Cases in the sample that presented syndromes, neurological had a variable effect on the nasal airway(18), and both positive changes and residual fistula of the palate were excluded, as and negative changes were induced by the flap. The authors were cases that had undergone other surgical procedures after attributed this result to the presence of prior nasal obstruction, pharyngeal flap placement that could interfere with nasopha- found in five of seven patients, and confirmed that the flap ryngeal or nasal patency, such as secondary surgical revision further decreased the nasal airway, which would explain the and nasal surgery. appearance of such breathing difficulties as snoring, chronic Thus, 103 cases were selected and assigned to three nasal obstruction and obstructive sleep apnea. groups according to age: 32 children between 6 and 12 years

58 ACR 2013;18(2):57-62 Pharyngeal flap in children and adults

old (12 females and 20 males), 55 young adults between 18 RESULTS and 30 years old (31 females and 24 males) and 16 middle- -aged adults aged 40 years or over (8 females and 8 males). In relation to the nasopharyngeal area, the comparison of Information on these cases regarding the nasopharyngeal values showed no difference among the three study groups area and the presence of signs and symptoms of respiratory (p>0.05). Considering the reference value of 0.570 cm2 for changes was collected on average 12 months after placement the nasopharyngeal area, the group of children and the group of the pharyngeal flap; this information was obtained from of middle-aged adults had mean values lower than expected. the institution’s medical records. One of the children had not undergone the rhinomanometric The nasopharyngeal dimensions were estimated by me- examination (Table 1). asuring the area of ​​the velopharyngeal orifice during nasal Table 1. Mean and standard deviation of the nasopharyngeal area (cm2) breathing at rest via the pressure-flow technique, utilizing the after pharyngeal flap surgery PERCI-SARS computerized system Version 3.30 (Microtronics Nasopharyngeal area Corp®.). This technique relies on the principle that the area of Groups p-value (cm2) an orifice can be estimated by simultaneously measuring the differential pressure between the two sides of the orifice and the Children (n=31) 0.527±0.215 airflow that passes through it(24). Thus, during several breathing Young adults (n=55) 0.599±0.213 p>0.05 cycles, a catheter positioned in the oral cavity and another in Middle-aged adults (n=16) 0.488±0.276 one of the nostrils used a nasal shutter to measure the static ANOVA: Analysis of variance with one criterion (p<0.05) air pressure, which was transmitted to pressure transducers. At the same time, a plastic tube connected to a heated pneumo- Regarding the survey of respiratory symptoms reported tachograph and linked to a pressure transducer was placed in after surgery, there were reports of oral respiration, snoring and the other nostril, the one with greater flow, to measure nasal breathing difficulties during sleep in all groups. The comparison airflow. The signals from the three transducers (nasal pressure, between the groups for each symptom indicated a difference oral pressure and nasal airflow) were sent to the PERCI system with respect to snoring, which was reported more frequently for analysis with specific software. by middle-aged adults (p<0.05) (Table 2). The mean reference value used was 0.570 cm2, as proposed in a study conducted on normal adults(25). Areas smaller than DISCUSSION 0.570 cm2 were therefore deemed to be probable nasopharyn- geal obstructions. The study of the effect of pharyngeal flap surgery on respi- The respiratory signs and symptoms survey was made con- ration is of fundamental importance in analyzing the surgical ducted using questions on a survey sheet, which was applied success of this procedure. Although many studies have reported routinely and filed in the medical record. The questions were surgical outcomes solely based on VPD symptoms, research based on the questionnaire proposed by Caouette-Laberge et on the occurrence of respiratory symptoms should always be al(11) and were considered information relating to daytime oral valued. It is known that such symptoms as snoring and a mild respiration and/or oral respiration during sleep, snoring during sensation of nasal obstruction are often observed during the sleep and difficulty breathing during sleep. Only reports of wor- immediate postoperative period of pharyngeal flap surgery sening preexisting symptoms or the onset of respiratory com- because of the reduced nasopharyngeal space caused by edema. plaints that were not reported before surgery were considered. However, when these symptoms persist for a long time and are The significance of the differences between the three age compounded by other factors, such as oral respiration, episodes groups was verified using analysis of variance with one criterion of obstructed breathing during sleep and daytime symptoms, for the nasopharyngeal area and using the chi-squared test for such as excessive tiredness, headaches and drowsiness, the respiratory symptoms. The results were considered significant patient should be carefully evaluated. if p<0.05. The observation and investigation of respiratory signs and

Table 2. Relative and absolute frequency of respiratory symptoms after pharyngeal flap surgery

Respiratory symptoms Groups Oral respiration Snoring Difficulty breathing during sleep Children (n=32) 66% (21) 69% (22) 9% (3) Young adults(n=55) 51% (28) 49% (27) 7% (4) Middle-aged adults (n=16) 75% (12) 75% (12)* 19% (3) Chi-squared test p<0.05 *Chi-squared test indicating greater frequency among middle-aged adults

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symptoms should therefore be characterized as clinical prac- pharyngeal flap surgery, including isolated or combined tice in services that use the pharyngeal flap, and the effects symptoms such as oral respiration, snoring and episodes of pharyngeal flap surgery on breathing should be routinely of respiratory obstruction. In 53% of cases, there was also evaluated in a subjective and instrumental manner(14,26). Based evidence of associated daytime symptoms, such as excessive on this assumption, this study aimed to verify the effect of age sleepiness, headaches, constant fatigue, hypertension and on nasopharyngeal dimensions and respiratory signs and symp- obesity. The study authors concluded that the pharyngeal flap toms in patients with cleft lip/palate who underwent pharyngeal may contribute to the worsening of respiratory disorders in flap surgery. The study compared the results obtained across this age group(19). three different age groups because surgery may be indicated In patients over 40 years with a pharyngeal flap, snoring for different ages, from six years onwards. is not only be associated with the presence of the flap but also Ideally, surgery should take place as early as possible to mi- with the effects of age, which indicates the importance of close nimize speech impediments. However, because of such factors monitoring of this population and of measures that prevent as general health, dento-occlusal changes and the presence of symptoms or stop them from worsening, such as correct posi- compensatory articulations, surgery can take place later. tioning during sleep, weight and neck circumference control No difference was found when comparing the mean naso- and the adoption of healthy habits. pharyngeal area values among the three groups. It is noteworthy, Although a difference could not be established between however, that slightly lower values with a mean of 0.488 cm2 the three groups regarding oral respiration symptoms and were observed in the group of middle-aged adults. Similarly, breathing difficulties during sleep, there was a tendency for when comparing the values obtained with the reference value middle-aged adults to have more frequent symptoms, especially of 0.570 cm2, a noticeable reduction was observed in the same sleep breathing difficulties. The proportion of individuals re- group. The nasopharyngeal area of the young adults showed porting difficulty breathing during sleep (19%) is considered no evidence of obstruction. high from a clinical point of view because given this severity A recent study performed a similar analysis of the rela- of the symptom, it can be a strong indicator of obstructive tionship between the nasopharyngeal area and respiratory sleep apnea. The gold standard test to confirm these findings symptoms in young adults (mean: 20 years old) who underwent is polysomnography. the same surgical procedure. That study found a higher fre- According to the literature, the occurrence of respiratory quency of reported respiratory symptoms in patients with a changes after pharyngoplasty is extremely diverse, ranging nasopharyngeal area below 0.550 cm2. The authors concluded from 4% to 92%(12-14). Such diversity may be related to the that values below 0.550 cm2 may be related to a pharyngeal methodology used, the age of the population studied and the flap obstruction and would call for a more careful evalua- criteria used to define the presence of respiratory changes, tion(14). Because of the nature of the flap to act as a mechanical taking into account each patient’s individual tolerance of obstruction preventing the escape of the expiratory air stream symptoms after surgery. during speech, the reduction of the nasopharyngeal space is an Another result observed in this study was the fact that expected result at any age. However, this reduction should not children have a higher frequency of symptoms than young be excessive to the point of triggering respiratory symptoms adults do. Some factors that may justify this fact could also be that compromise quality of life. investigated, such as the presence of a higher incidence of upper In the analysis of the proportion of respiratory symptoms airway infections(28) and hypertrophic tonsils and adenoids in reported by the three age groups, differences were only found this age group(29), which can aggravate respiratory symptoms(30). for snoring, which occurred in 75% of middle-aged adults, Furthermore, the presence in the children’s group of a caregiver 69% of children and 49% of young adults. The fact that these or other person responsible for information about symptoms symptoms occurred most frequently in the older population favors a detailed account of respiratory signs and symptoms confirms the results of studies that have established consensus during sleep, which is not available in the case of many young on the intensification of respiratory symptoms, especially sno- adults. In such cases, the occurrence of such symptoms may ring and obstructive sleep apnea, as age increases(21-23). be underestimated because the sleep of young adults is rarely Snoring can be defined as a fricative noise originating in the observed by others. soft tissues of the upper airways. It results from the vibration The importance of surveying respiratory signs and symp- of the uvula and palate and occasionally the lateral pharyngeal toms using instrumental evaluation is clear in candidates for wall, epiglottis and base of during sleep. It is very com- pharyngeal flap surgery because aerodynamic evaluation has mon in middle-aged men and in women after menopause(27). proven to be an indicator of change in nasopharyngeal dimen- The symptom is present in 90% to 95% of obstructive sleep sions and because the questionnaire confirmed the occurrence apnea syndrome (OSA) cases. of respiratory symptoms. However, only polysomnography is One study found a prevalence of sleep-disordered brea- able to precisely diagnose the severity of breathing difficulties thing in middle-aged adults (≥40 years) who had undergone during sleep.

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Indications for pharyngeal flap surgery should be evaluated velopharyngeal insufficiency. J Craniofac Surg. 2011Sep;22(5):1647- as early as possible to eliminate or minimize speech symptoms 51. and the concomitant psychosocial effects. Rigorous and sys- 11. Caouette-Laberge L, Egerszegi EP, de Remont AM, Ottenseyer I. tematic lifelong monitoring of these cases should be part of Long-term follow-up after division of a pharyngeal flap for severe the management of respiratory symptoms. Furthermore, other nasal obstruction. Cleft Palate Craniofac J. 1992Jan;29(1):27-31. surgical treatments that are considered more physiological 12. Wells MD, Vu TA, Luce EA. Incidence and sequelae of nocturnal from an anatomical point of view and restorative procedures respiratory obstruction following posterior pharyngeal flap operation. to correct velopharyngeal insufficiency can be used as efficient Ann Plast Surg. 1999Sep;43(3):252-7. and preventative alternatives for this population. 13. 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