Abstract
Purpose
Congenital choanal atresia (CCA) is a rare disease entity. The prevention of restenosis has been the main concerns of choanoplasty. The authors retrospectively analyzed patients with CCA to investigate clinical features and factors affecting surgical outcomes.
Material/methods
Forty sides in 27 patients with CCA from 1987 through 2009 were reviewed with medical records that included symptoms, associated anomalies, laterality of atresia, types of the atretic plate, surgical approaches, uses of stent or mitomycin C, ages at operation, and surgical outcomes.
Results
CHARGE association was the most commonly associated malformation in bilateral CCA and cleft lip and cleft palate in unilateral CCA. Age at operation was related to restenosis rate. The cases of bilateral CCA were operated on younger ages than those of unilateral CCA (4.9 months vs 11.5 years, respectively), and the restenosis appeared to be higher in bilateral cases than in unilateral ones. The use of stent did not improve preventive rate of restenosis: 42.9% of restenosis with stent and 47.4% without stent, respectively. Mitomycin C did not seem to be effective in preventing restenosis either. No significant difference in restenosis rate was observed in terms of symptoms, associated anomalies, types of the atretic plate, and surgical approaches as well.
Conclusions
Our study suggests that bilateral CCA, meaning early operation age, develops restenosis more frequently. However, the patency rate was not related to surgical approaches or postoperative use of stent and mitomycin C.
1
Introduction
Congenital choanal atresia (CCA) is a congenital obstruction of the posterior nasal apertures, which could be either unilateral or bilateral. The main symptom of the CCA is nasal obstruction. Unilateral choanal atresia is not life threatening and rarely presents itself with immediate or severe airway obstruction and is usually diagnosed in childhood rather than immediately after birth. On the other hand, neonates with bilateral CCA need immediate intervention for respiratory distress as they obligate nasal breathers . Preventing restenosis of operation site and restoring normal nasal passages are the main concerns of surgical repair of CCA .
Various surgical techniques have been proposed to repair the atresia . Although common surgical techniques of the CCA consisted of transpalatal and transnasal approaches, technical advances in endoscopic visualization and newer sinonasal powered instruments for endonasal surgical procedures have provided the opportunity to use the transnasal endoscopic route. Some reports using transnasal endoscopic techniques describe improved surgical outcomes .
Postoperative stenting is one of the most controversial issues in maintenance of choanal patency. A recent article describes stenting as a part of choanal atresia repair . However, other studies have suggested the treatment of CCA without intranasal stents . Mitomycin C has been proposed as an adjunct therapy for CCA repair for improving healing and minimizing scar formation . Recently, 1 report suggested that predictive factors of restenosis are the presence of GERD, patients’ age if younger than 10 days at the time of surgery, and insufficient postoperative endoscopic revision . However, according to previous reports, there are still limited data regarding the factors influencing the surgical outcomes.
This study was conducted to evaluate the clinical features as well as the factors affecting surgical outcome in terms of surgical approaches, use of stent or mitomycin C, and several clinical features. This study was approved by the Institutional Review Board of the Clinical Research Institute at Seoul National University Hospital.
2
Materials and methods
This study used 40 sides in 27 patients, 11 males (40.7%) and 16 females (59.3%), between 1986 and 2009 at Seoul National University Hospital. Fourteen patients were unilateral, and 13 patients were bilateral. Careful review of patient history, complete otorhinolaryngological examination, and multislice computed tomographic scans were performed in all cases to confirm the diagnosis as well as to evaluate the extent and nature of the atresia. Surgery was performed to patients at the age of 6 days to 28 years. The patients who underwent their first operation at another facility and 5 of 32 patients with less than 6 months of follow-up records were excluded. Follow-up periods varied from 7 to 180 months.
In this study group, surgical techniques such as transpalatal correction , transnasal drill out or curettage , and endoscopic transnasal approaches were used. The postoperative stenting was performed in 21 nasal cavities of 14 patients and was removed 6 to 8 weeks later. We retrospectively analyzed sex of the patients, type (bony or mixed) of the atresia, surgical approaches, use of postoperative stenting, duration of stenting, and usage of mitomycin C.
Restenosis was defined as follow: any procedure or operation including removal of granulation or polyp and scar revision for somewhat widening of nasal airway patency after their first operation to prevent restenosis, although choana was kept in patency.
The surgical outcomes were analyzed using SPSS 12.0 for Windows (SPSS, Inc, Chicago, IL). Comparisons were based on the Fisher exact test for categorical variables. The P ≤ .05 was accepted as significant.
2
Materials and methods
This study used 40 sides in 27 patients, 11 males (40.7%) and 16 females (59.3%), between 1986 and 2009 at Seoul National University Hospital. Fourteen patients were unilateral, and 13 patients were bilateral. Careful review of patient history, complete otorhinolaryngological examination, and multislice computed tomographic scans were performed in all cases to confirm the diagnosis as well as to evaluate the extent and nature of the atresia. Surgery was performed to patients at the age of 6 days to 28 years. The patients who underwent their first operation at another facility and 5 of 32 patients with less than 6 months of follow-up records were excluded. Follow-up periods varied from 7 to 180 months.
In this study group, surgical techniques such as transpalatal correction , transnasal drill out or curettage , and endoscopic transnasal approaches were used. The postoperative stenting was performed in 21 nasal cavities of 14 patients and was removed 6 to 8 weeks later. We retrospectively analyzed sex of the patients, type (bony or mixed) of the atresia, surgical approaches, use of postoperative stenting, duration of stenting, and usage of mitomycin C.
Restenosis was defined as follow: any procedure or operation including removal of granulation or polyp and scar revision for somewhat widening of nasal airway patency after their first operation to prevent restenosis, although choana was kept in patency.
The surgical outcomes were analyzed using SPSS 12.0 for Windows (SPSS, Inc, Chicago, IL). Comparisons were based on the Fisher exact test for categorical variables. The P ≤ .05 was accepted as significant.
3
Results
3.1
General characteristics
Table 1 summarizes the demographic characteristics of the patient population. Among 14 cases of unilateral CCA, 7 cases were on the right side, and the remaining 7 were found on the left side. The number of males and females was similar in bilateral CCA (7 in male and 6 in female, respectively), whereas more females showed unilateral CCA than males. The incidence of atretic type, bony or mixed bony-membranous obstruction was similar in both unilateral CCA and bilateral CCA. Mean age at first operation was 11.5 years (6 months to 28 years) in unilateral CCA and 4.9 months (6 days to 19 months) in bilateral CCA. Six patients with bilateral CCA underwent an operation at the age of 1 month or less, and 9 (75%) of 12 sides showed higher restenosis rate.
| Bilateral CCA (n = 26 sides) | Unilateral CCA (n = 14 sides) | ||
|---|---|---|---|
| Sex | Male | 7 | 4 |
| Female | 6 | 10 | |
| Side | Right | 13 | 7 |
| Left | 13 | 7 | |
| Atretic type | Bone | 11 | 5 |
| Mixed | 15 | 9 | |
| Associated anomaly | 13/13 (100%) | 5 /14 (35.7%) | |
| Mean age of operation | 4.9 mo (0-19 mo) | 11.5 y (0.5-28 y) | |
| Chief complaint | Respiratory distress | Nasal obstruction | |
| Cyanosis | Rhinorrhea | ||
The most common associated malformation in bilateral CCA was CHARGE association, whereas that of unilateral CCA was cleft lip and cleft palate ( Table 2 ).
| Bilateral CCA (n = 13) | Unilateral CCA (n = 14) | |
|---|---|---|
| Heart disease | 1 | 1 |
| Cleft palate and lip | 2 | 3 |
| Treacher Collins syndrome | 2 | 0 |
| Craniofacial anomaly | 3 | 1 |
| CHARGE syndrome | 4 | 0 |
| Noonan syndrome | 1 | 0 |
| Total | 13 | 5 |
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