Eur Arch Otorhinolaryngol (2013) 270:129–134 DOI 10.1007/s00405-012-1956-0
RHINOLOGY
Endoscopic management of congenital bilateral posterior choanal atresia: value of using stents Magdy Eisa Saafan
Received: 1 December 2011 / Accepted: 31 January 2012 / Published online: 23 March 2012 Ó Springer-Verlag 2012
Abstract Objective of this study is to evaluate the efficacy and safety of using stents after endoscopic repair of bilateral posterior choanal atresia. Twenty cases of congenital bilateral posterior choanal atresia were managed by endoscopic transnasal approach which was performed using a nasal stent in 10 cases and without stenting in the other 10 cases. The stent was left for 4 weeks and patients had 1–5 years of follow-up. Patients have been evaluated subjectively and objectively: clinical and by endoscopic examination. Closure occurs in 20% of the stent group compared to 30% of the non-stent group, with no statistically significant difference between the two groups. On the other hand, choanal narrowing and stenosis occurs in 40% of the stent group compared to 20% of the non-stent group with a statistically significant difference between the two groups. Granulation tissue formation was detected in 50%, erosion of the nares in 10%, premature extrusion in 10%, and dislodgement in 10% of stent group. Granulation tissue formation was detected in 20%, hemorrhage occurred in 10% of the non-stent group patients. There was a statistically significant difference between the two groups (P-value \ 0.05) regarding granulation tissue formation. The use of stents after endoscopic repair of congenital bilateral posterior choanal atresia does not seem to decrease the incidence of re-closure and restenosis of the posterior choana. On the other hand, there are higher complication rate from using the stents like granulation tissue formation, excoriation or erosion of the nares,
M. E. Saafan (&) Otolaryngology and Head and Neck Surgery, Faculty of Medicine, Tanta University Hospitals, 61 Kafr Esam Street, El Nadi Square, Tanta, Egypt e-mail:
[email protected]
premature extrusion, dislodgement, stent blockage, and the unsightly aspect of having stents protrude from the nose. Keywords Choanal atresia Endoscopic Transnasal Stent Prospective Randomized
Introduction Congenital choanal atresia (CA) is a rare malformation that causes airway obstruction in newborns and infants, with an incidence of 1 in 7,000–8,000 births [1]. The newborn infant is an obligatory nose breather for the first few days after birth and has no ability to breathe through the mouth. Unilateral choanal atresia may go undiagnosed until childhood or even adulthood, the symptoms being mainly those of nasal obstruction, mucoid nasal discharge, anosmia, disturbed sleep and daytime fatigue. Bilateral choanal atresia, on the other hand, poses a medical emergency immediately at birth causing respiratory distress and demanding immediate intervention. Unrecognized bilateral choanal atresia will almost certainly lead to death due to neonatal asphyxia [2]. It was believed that most of the cases were bony (90%), but recent reviews of computerized tomography (CT) studies suggest that most atresias (about 70%) are mixed, and all membranous atresias have some bony component [3]. Most cases of CA are isolated malformations, but association with other congenital deformities is not exceptional, as in the CHARGE association (a malformative syndrome that includes coloboma, heart disease, CA, retarded development, genital hypoplasia, and ear anomalies, including hypoplasia of the external ear and hearing loss) [4]. There are numerous methods for correcting this condition, but the current most commonly used methods are the
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transpalatal approach [5], the transseptal approach [6], and the endoscopic transnasal approach [7]. Once the atresia is surgically corrected, the surgeon is often faced with the problem of restenosis. The rates of restenosis vary widely and range from 0 to 85% [3]. Many authors report the use of postoperative stenting for variable periods [8, 9], but stenting is not a panacea, and granulation tissue, intranasal synechiae, and long-term restenosis can develop in spite of postoperative stenting. The use of soft surgical stenting material has been thought to result in higher surgical success rates when compared with hard and inflexible material [10]. There is no uniformity of opinion as to the duration of stenting. Four to six weeks is the commonest, but periods up to 6 months have been published in the literature [1, 11]. In addition to stenting the choana, the lumen of the stent provides an airway to facilitate nasal pattern breathing in neonates. Maintenance of the stent lumen patency is thus important. There is a degree of crusting that occurs within stent lumens. The meticulous use of saline drops and suction is required to maintain stent patency [11, 12]. In this study, we try to evaluate the possible role of stents in repair of CA and whether using the stents minimizes the rate of choanal stenosis and obstruction.
Patients and methods This is a prospective randomized controlled study carried out over 5 years (between February 2005 and January 2010) in the Otolaryngology Department of Tanta University Hospitals, Egypt. The study protocol and consent forms were approved by the research review committee. After the application of the exclusion criteria, the study was discussed with the parents of 20 consecutive patients with a clinical diagnosis of congenital bilateral choanal atresia referred to the emergency unit of the Otolaryngology Department, from either pediatric or obstetric units and were finally consented to be included in the study. The exclusion criteria included: unilateral choanal atresia, associated pyriform aperture stenosis, CHARGE association, severe associated malformations, preterm infants with bad general condition, severe neurological deficits either congenital or acquired due to postnatal respiratory distress to eliminate pre-existing surgical variables. The diagnosis was made clinically and confirmed by CT scanning. The immediate management of these neonates presenting with intermittent cyanosis was the insertion of an oral airway and feeding via oro-gastric tube. All patients with choanal atresia underwent a careful physical examination looking for any stigmata of CHARGE association. Prior to a general anesthetic, an echocardiogram was performed to identify any significant cardiac lesions prior to atresia correction.
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Operative procedure Patients were managed by endoscopic transnasal approach which was performed under general oral endotracheal anesthesia. The nasal cavities were decongested and anesthetized using cotton pledgets saturated with a mixture of lidocaine 1% and adrenaline 1:100,000 left for 5 min. After the secretions were aspirated, the nasal cavities were inspected endoscopically, in most cases with a 4 mm telescope. Infrequently, it was necessary to use only the 2.7 mm telescope. A straight urethral sound was used to perforate the thin central component of the atresia and then the opening was dilated until it would accommodate a small diamond burr. While protecting the nasal ala with an aural speculum, the choanae were enlarged by transnasally drilling the medial pterygoid plate and vomer. No attempt was made to preserve mucosal flaps. Special attention was given to the posterior part of the nasal septum, and the thick vomer was eroded with the backbiting forceps. Patients were randomly and equally distributed in two groups according to the use of nasal stent. In the stented group (10 neonates), stents were fashioned from a portex endotracheal tube (usually no. 3.5 for neonates and larger by age) (Fig. 1). To place the stent correctly in place, two small suction tubes were introduced into both nostrils and pulled out through the mouth. Each limb of the modified portex tube was tied to one suction tube, then the suction tubes were pulled, the stent was pushed out through the
Fig. 1 A diagram illustrating a nasal stent made from a modified endotracheal tube. The posterior (choanal) ends are patent, while the anterior ends will be sutured together in front of the columella
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nose, and the suction tubes were separated from the stent. The two limbs of the stent were then tied together with a silk suture anterior to the columella. Easy passage of a suction catheter through the stents confirmed satisfactory stent position and alignment. Postoperative care The parents were instructed to irrigate the child’s nose and stent with normal saline using a suction tube at home four times daily. Endoscopic follow up was performed weekly, and blood clots and crusts were removed or suctioned. The stent was removed within 4 weeks under general anesthesia, to allow for a second look of the neo-choana and a meticulous cleaning was performed. All patients were given postoperative antibiotic prophylaxis for the whole period of stenting. Most patients tolerated the endoscopic procedure under topical anesthesia. When the cleaning procedure was impossible under topical anesthesia, a general anesthesia was used. Patients have been evaluated subjectively and objectively: clinical and by endoscopic examination. The follow-up period was between 1 and 5 years, with a median of 3.2 years.
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Closure occurs in 20% of the stent group compared to 30% of the non-stent group, with no statistically significant difference between the two groups (P-value [ 0.05) (Table 1; Figs. 2, 3). On the other hand, choanal narrowing and stenosis occurs in 40% of the stent group compared to 20% of the non-stent group with a statistically significant difference between the two groups (P-value \ 0.05). Granulation tissue formation was detected in 50%, erosion of the nares in 10%, premature extrusion in 10%, and dislodgement in 10% of stent group (Fig. 4). Granulation tissue formation was detected in 20%, hemorrhage occurred in 10% on the non-stent group. There was a statistically significant difference between the two groups (P-value \ 0.05) regarding granulation tissue formation.
Discussion Bilateral choanal atresia is a clinical emergency that requires surgical treatment during the first days of life. Table 1 incidence of postoperative complications in both groups Complications
Statistical methods To maintain exactly equal treatment numbers in both groups, randomization was done using random blocks. At the time of randomization, the patients’ parents were not aware of the group assignment. The analysis was done using SPSS for windows statistics software package. Data were expressed as mean ± standard deviation (SD). P-values \ 0.05 were considered significant.
Stent group (total number 10) Number
Non-Stent group (total number 10) %
Number
P
%
Stenosis
4
40
2
20
\0.001
Closure
2
20
3
30
[0.05
Granulations
5
50
2
20
\0.001
Hemorrhage
–
–
1
10
[0.05
Stent dislodgement
1
10
–
–
[0.05
Premature extrusion
1
10
–
–
[0.05
Erosion of the nares
1
10
–
–
[0.05
Results Twenty patients with congenital bilateral choanal atresia were included in this study. Patients were randomly distributed in two equal groups according to the use of postoperative nasal stents. The study comprised 7 (35%) males and 13 (65%) females, with a mean age at time of initial surgery ± SD of 4 days [5]. The mothers’ age ranged from 21 to 40 years with a mean of 27 years. There was no history of troubles or drug intake during pregnancy. Weight at initial surgery ranged from 2 to 3.5 kg with a mean weight of 3.1 kg. None of the infants had associated congenital anomalies or other cranio-facial abnormalities. Follow up ranged from 1 to 5 years, with a mean of 3.2 years. The atretic plate proved to be bony in eight (40%), membranous in two cases (10%) and mixed osteomembranous in ten cases (50%).
Fig. 2 Postoperative endoscopic view: widely patent right posterior choana 6 months after repair in a non-stent-group infant
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Fig. 3 Postoperative endoscopic view: total closure of right posterior choana 8 months after repair in a stent- group infant
Fig. 4 Postoperative endoscopic view: extensive granulations around the right posterior choana 3 months after repair in a stent- group infant
Management of CA is still faced with the problem of restenosis. There are so many studies in the literature with tools to improve the outcome of surgical intervention, initially using different surgical approaches to the use of adjuvant tools or medications. There are numerous approaches for correcting this condition, but the current most commonly used methods are the transpalatal approach, the transseptal approach, and the endoscopic transnasal approach [5–7]. Factors that influence the type of approach selected and its subsequent success include the age of the patient, the size of the nasopharynx, the thickness of the atresia, bilateral versus unilateral atresia, the use of postoperative stenting, the surgeon’s preference of approach, and the presence of other anomalies such as found in the CHARGE association. Transseptal repair is
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still occasionally used for older patients with unilateral atresia, and the transantral route is of historical importance only [13]. The transnasal route fell out of favor because of the high rate of failure requiring revision [14]. This was attributed to the difficulty in visualizing areas of the choana that required special surgical attention, such as the vomerine septal bridge and bony narrowing of the lateral walls. As patients grow, the depth from the nasal vestibule to the posterior choana becomes longer, and thus, the transnasal approach is further complicated even with the use of an operating microscope. Septal deviation, turbinate hypertrophy, or other anatomical abnormalities can further complicate visualization during the transnasal approach [15]. The transpalatal route has, therefore, become the method preferred by many surgeons. Its use has been advocated for the repair of unilateral and bilateral atresia, in younger or older patients, and for revisions. It offers excellent exposure and high success rates [5]. The transpalatal approach, however, is not without disadvantages. Increased operative time, increased blood loss, palatal fistula, palatal muscle dysfunction, and maxillofacial growth disturbance are sequelae of this procedure [16]. Endoscopic surgery is now preferred, and the use of a postoperative stenting is one of the most controversial issues in choanal atresia management. Intranasal stenting is often used to maintain patency following repair of choanal atresia and pyriform aperture stenosis. Although there is a trend toward less frequent use of stents as endoscopic techniques have improved, more than 96% of surgeons surveyed still use them at least some of the times [17]. The stents are usually fashioned per-operatively by the surgeon. The use of various different materials has been described, such as endotracheal tubes, soft silicone, self-inflating sialastic tube, silicone suction tube and metal-reinforced rubber silicone tubes [1, 11]. There is no uniformity of opinion as to the efficacy of one type of stent or method of surgery above another. Some authors believe that stenting is necessary for all cases [18], while others believe that stenting is needed in only bilateral cases [19]. Two older case series use long-term stents for at least 3–4 weeks [15, 20], while more recent series use short-term stents for less than 1 week [21]. One report recommends using stents for only 2 days [5]; another recent report uses no stents [22]. In this study, in the stent group, we used a modified endotracheal tube, fashioned in a special manner to ensure patency at the posterior choana, fixity and stability by turning around the choana and safety with minimal pressure on the columella as both sides of the stent were sutured to each other in front of the columella avoiding septal suturing. Stents were left for 4 weeks before the removal under general anesthesia. This gives us the chance for a second look, to thoroughly clean the nose and remove possible granulations that would act as a nidus for future stenosis.
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Proponents of using stents claim that they are essential to stabilize the nasal airway and to hold the flaps in position during the early postoperative period. However, stents cause considerable discomfort, localized infection and ulceration, circumferential scar tissue leading to restenosis after removal, and proliferation of resistant bacteria due to the necessity of long-term antibiotic therapy [23]. To avoid these complications, several recent studies have recommended using brief stenting for several days [12, 21], or even not using stents at all [22, 24] with comparable or even better long-term results. The argument for this way of management is that one of the advantages of using modern endoscopic repairs is the greater precision and surgical safety in fashioning the intranasal flaps, thus making it possible to cover raw edges efficiently and thus avoid the use of stents and their complications. Our study further augments this view and demonstrates that the use of postoperative stenting is not a necessity for achieving favorable results in bilateral CA transnasal endoscopic repairs. In this prospective randomized comparative study, choanal re-closure occurs in 20% of the stent group compared to 30% of the non-stent group, with no statistically significant difference between the two groups. On the other hand, choanal narrowing and stenosis occurs in 40% of the stent group compared to 20% of the non-stent group with a statistically significant difference between the two groups. Granulation tissue formation was detected in 50% of the stent group compared to 20% of the non-stent group with a statistically significant difference between the two groups. Erosion of the nares was detected in 10%, premature extrusion in 10%, and dislodgement in 10% of the stent group. Gujrathi et al. [25] in 2004, in a study of 52 neonates with bilateral CA, reported a success rates of 94% after surgical repair, and use of nasal stent made using folded endotracheal tubes that were inserted into each choana through the mouth after endoscopic choanal dilatation using two catheters previously inserted into each nasal cavity as a guide. Sharma et al. in 2006, in a series of 14 patients using reinforced endotracheal tube mounted on a portex carrier tube inserted through the nostrils and fixed outside the nose. They report favorable results after leaving the stents for 4 weeks. They claim that these rigid stents may be more resistant to occlusion than PVC or sialastic tubes, but it has been suggested that soft stents may reduce the risk of granulations and scars that could favor restenosis [26]. Al-Ammar [27] in his series of 11 cases with bilateral CA, reported success rate of 60% with use of nasal stents versus 33% if stents were not used despite it was not statistically significant difference. Sadek [11] reported almost similar results for bilateral CA, and use of nasal stent. Other studies are reporting procedure revision rate
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ranging between 4.6 and 4.9 to achieve patent choana [20, 23]. On the other hand, nasal patency was reported to be achieved in bilateral CA without the use of nasal stent [22]. Zuckerman [28] suggests that the placement of stents for bilateral choanal atresia repair may result in a higher rate of restenosis as well as the need for multiple subsequent procedures during infancy. Schoem reported that the use of a stent in choanal atresia repair seems unnecessary with telescopes, high-speed drills and microdebriders. Special attention should be given to the removal of the posterior septal-vomer segment to reduce the need to protect the denuded tissue from obstructing synechiae and granulation tissue formation [24]. Cedin et al. reported that using neither stents nor nasal packing allowed for fast recovery in one stage surgery with better long-term results. Stenting should be avoided as it stimulates granulation tissue formation that frequently leads to stenosis. They claimed that the intraoperative application of Mitomycin C offers a promising adjunct in achieving a stable lumen [29]. The use of nasal stent may be associated with some complications that include patient discomfort, increased nasal and paranasal infection, and possibly septal or columellar necrosis [30]. This reflects the inconsistency of the results of use of nasal stents in bilateral CA among different studies in the literature. The use of telescopes provides a view of the posterior nasal cavity far superior to that of the microscope. Therefore, when reading the literature from the 1980s, when most surgeons used the microscope rather than the telescope, tissue trauma may have been greater than today using the transnasal, endoscopic technique. With that in mind, prior to the advent of newer, less-traumatic surgical techniques, a stent would be necessary to establish initial patency, despite the drawbacks of an indwelling stent. In the new era of miniaturized telescopes, high-speed protected drill bits, and microdebriders, lateral drilling is eliminated or minimized [24]. Focusing on careful removal of the posterior septal/ vomer segment reduces the need to protect denuded tissue from obstructing synechiae formation and exuberant granulation tissue. Therefore, the novice otolaryngologist needs to recognize that the term ‘‘transnasal’’ has a completely different connotation than even 10–15 years ago, and must carefully read the literature in comparing surgical techniques and approaches for repair of choanal atresia. This study supports the notion that stenting may not be necessary in the transnasal, endoscopic repair of bilateral choanal atresia. Our study is limited by the small number of the study population. To achieve more reliable conclusions, we recommend performing a prospective, multicenter, well-controlled study, which employs a larger number of cases.
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Conclusion The use of stents after endoscopic repair of congenital bilateral posterior choanal atresia does not seem to decrease the incidence of re-closure of the posterior choana. On the other hand, there are higher complication rate from using the stents like restenosis, granulation tissue formation, excoriation or erosion of the nares, premature extrusion, dislodgement, stent blockage, and the unsightly aspect of having stents protrude from the nose. Thus, stenting may not be necessary in the transnasal, endoscopic repair of bilateral choanal atresia. Conflict of interest The authors have no conflict of interest, financial or otherwise with any organization.
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