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Stentless endoscopic transnasal repair of bilateral choanal atresia starting with resection of vomer
International Journal of Pediatric Otorhinolaryngology 76 (2012) 1002–1006
Contents lists available at SciVerse ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Stentless endoscopic transnasal repair of bilateral choanal atresia starting with resection of vomer Magdy Abdalla Sayed El-Ahl, Mohammad Waheed El-Anwar * Otorhinolaryngology, Head and Neck Surgery Department, Faculty of Medicine, Zagazig University, Egypt
A R T I C L E I N F O
A B S T R A C T
Article history: Received 25 January 2012 Received in revised form 18 March 2012 Accepted 20 March 2012 Available online 28 April 2012
Objectives: To assess the results of a transnasal endoscopic repair of congenital choanal atresia beginning by resection of the posterior portion of the vomer and ending by no stent. Methods: Seven patients with bilateral congenital choanal atresia aged ranging from 3 to 15 days were operated upon between June 2009 and September 2011. This transnasal endoscopic approach allowed resection of the posterior portion of the vomer first then the atretic plates and part of the medial pterygoid plate if needed leaving no stent. Postoperative control included office fiberoptic nasal endoscopy. Results: Adequate functional nasal breathing was maintained in all patients during follow up of 11 to 23 months. Apart from one case that complicated by palatal defect, no any other complications were detected. Conclusion: The described technique was proved to be very effective, allowing fast recovery, and one step surgery with early discharge from hospital using neither stents nor nasal packing. Good patency with no reduction in functional quality was also observed. ß 2012 Elsevier Ireland Ltd. All rights reserved.
Keywords: Choanal atresia Endoscopic repair No stent Neonate Vomer
1. Introduction Congenital choanal atersia is defined as a failure in the development of communication between the nasal cavity and nasopharynx causing complete obstruction of the nasal airway [1]. Despite that it has a rare incidence of approximately 1 in 7000 births [2], congenital choanal atresia and stenosis are the most common craniofacial abnormalities of the nose [3]. Neonates with bilateral choanal atresia need immediate interference for nasal respiratory distress as neonates are obligatory nasal breathers [4]. The neonatologists usually are the first to raise the hypothesis of choanal atresia by noticing the failure in pushing the nasal aspiration tube further through the nasal cavity into the pharynx [5]. Paranasal computed tomography (CT) scan and nasal fiberoptic endoscopy are the gold standard tests since they set the diagnosis and enables the examiner to identify the type of atresia. Historically, congenital choanal atresia was described as bony in 90% of the cases and membranous in 10%. However, recent studies suggest that the mixed form is the most common (70%) with a 30% incidence of pure bony atresia [5].
* Corresponding author. Tel.: +20 1004695197. E-mail address: [email protected] (M.W. El-Anwar). 0165-5876/$ – see front matter ß 2012 Elsevier Ireland Ltd. All rights reserved. http://dx.doi.org/10.1016/j.ijporl.2012.03.019
Associated congenital anomalies can be found in approximately 50% of the cases [6] however lower risks of associated congenital anomalies are observed in recent years [3]. Since the first description of choanal atresia in 1755 [7], multiple surgical techniques have been proposed to repair the malformation [2,8], these surgical procedures can be divided into four main approaches: transantral, transpalatal, trans-septal and transnasal [9,10]. Today, the transnasal endoscopic method is the most widely accepted repair technique [8]. It seems that bilateral choanal atresia is associated with more surgical corrections before achieving a normal nasal breathing than unilateral choanal atresia [11]. The aim of this study was to assess the results, easiness and safety of transnasal endoscopic repair of bilateral chongenital choanal atresia in solving the obstruction problem beginning by resection of the posterior portion of the vomer, without the use of nasal stent or mucosal flaps. 2. Patients and methods Seven patients had congenital bilateral choanal atresia were surgically treated from June 2009 to September 2011 at the Otorhinolaryngology Department, Zagazig University Hospitals, Egypt. All patients received a full history review and physical examination including failure of passage of six-French catheter through the nasal cavity into the nasopharynx. Patients were
M.A.S. El-Ahl, M.W. El-Anwar / International Journal of Pediatric Otorhinolaryngology 76 (2012) 1002–1006
Fig. 1. Nasal endoscopy admitted into one nasal cavity and the microburr admitted from other cavity.
examined also with fiberoptic nasal endoscopy. Axial non-contrast CT scanning with thin sections (2–3 mm) was done to all patients to confirm the diagnosis, assess type of atretic plate and thickness of vomer and to exclude any concomitant craniofacial abnormalities. Before CT examination, the nasal passages were aspirated. Preoperative examination was done by neonatologist and anesthesiologist including cardiac evaluation. Chest X-ray and echocardiography were sometimes needed to exclude congenital heart disease. The routine preoperative laboratory tests were done for all neonates. An oropharyngeal airway or orotracheal tube was secured in place in all neonates till the time of surgery. Transnasal endoscopic surgical repair was performed in a single stage procedure with no stent placement, nor nasal packing. A formal consent was signed by parents of the neonates or their relatives and the Zagazig University IRB approved this study. The surgical procedure was performed under general anesthesia using orotracheal tubes in all patients. Small pack was inserted through the oral cavity to nasopharynx. This pack was used to protect the nasopharyngeal mucosa, eustachian tube orifice and skull base and also to displace away the soft palate [12]. Video-endoscopies; a Storz rigid endoscopy 08 4 or 2.7 mm, was used. Cottonoid pads soaked in oxymetazoline hydrochloride 0.05% were used to diminish the volume of the nasal turbinates. Endoscopic examination and evaluation was done first, palpating atretic plates, examining posterior septum and nasal cavity. A curved semilunar incision was made over the posterior part of the bony nasal septum about 3 to 5 mm anterior to the atretic plate. The mucoperiosteal flap was elevated from the bony septum. The inferior aspect of middle turbinate was used as guide for superior limit of dissection.
1003
The posterior part of the nasal septum was gently penetrated creating posterior septal window by sickle knife and dissector and now microdrill and backbiter or through cutting forceps could be admitted in one nasal cavity while the endoscopy could be admitted in other side getting wider space for the procedure with excellent visualization (Fig. 1). Posterior part of the nasal septum anterior to atretic plate was excised from anterior to posterior. The vomer was separated from the nasal floor by sharp dissection. Adequate resection of posterior part of the vomer was essential to create a visualization window to facilitate simultaneously access of both sides’ instrumentations in addition to prevent restenosis (Figs. 1 and 2). The new opening provided a clearer surgical field by drainage of blood and secretions to both nasal cavities. It became easy and wide to complete procedure through both nasal cavities. Using an olive-tipped suction, the atretic plate was penetrated and excised beginning at the weakest infero-medial part to establish a reference point into the packed nasopharynx. In two cases with thicker plate, the microdrill was necessary to establish this connection. The remaining bone of the atretic plate was removed using microdrill, curette, backbiter and/or sphenoid forceps. The neochoana was widened if needed laterally to medial pterygoid plate, inferiorly to the palatine bone, and superiorly to the level of the sphenoid sinus (sloping of the posterior pharyngeal wall). At that time the postnasal pack could be removed through the new choana from the nose. Hemostasis was secured by temporary packing or if diathermy No stents were used, nor nasal packing. Recovery from general anesthesia was uneventful in all cases (Table 1). Postoperative care began on the first day; the nose was washed with saline solution twice a day, then frequently saline nasal drops were used by parents of the neonates. All patients received prophylactic systemic antibiotics for one week. Feeding started immediately after recovery. Neonates were discharged without symptoms at the second day postoperatively. Patients underwent serial office nasal endoscopy weekly for one month then monthly for six months to confirm patency. Surgery was considered successful if the patient showed easy nasal breathing, non interrupted oral feeding, patent nasal airway, and opened new choana. Patency was defined as less than 50% restenosis [13] while the term narrowing was used to describe restenosis of less than 50%. Age at surgery, sex, type of choanal atresia, and postoperative outcomes were recorded and assessed. 3. Results Seven patients treated for bilateral congenital choanal atresia, three (42.9%) of them were male and 4 (57.1%) were female. The mean age at the time of surgery ranged from 4 to 15 days. The mean age at the time of surgery was 8 days. Five (71.4%) neonates
Fig. 2. (A) Nasal endoscopy showing penetration of vomer first. (B) Nasal endoscopy admitted into one side visualizing the microburr admitted from other side. (C) Nasal endoscopy showing partial excision of posterior part of vomer exposing atretic plates bilaterally.
M.A.S. El-Ahl, M.W. El-Anwar / International Journal of Pediatric Otorhinolaryngology 76 (2012) 1002–1006
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Table 1 Data of the 7 neonates included in this study. No
Sex
Age
Nature of atresia
Associated anomalies
Postoperative complication
Narrowing
Restenosis
1 2 3 4 5 6 7
Male Female Female Male Female Female Male
15 days 8 days 6 days 9 days 3 days 10 days 5 days
Bony Mixed Mixed Mixed Bony Mixed Mixed
No No No No Cardiac VSD Digital anomaly No
No No No Palatal defect No No No
No Narrowing No No Narrowing No No
No No No No No No No
Total (7)
3 males 4 females
3 to 15 days
5 mixed 2 pure bony
One case VSD One case polydactyl
1 complicated (14.3%) 6 no (85.7%)
2 narrowing (28.6%) 5 no (71.4%)
7 no (100%)
had mixed bony -membranous atretic plates shown by CT and endoscopic examination and 2 (28.6%) had pure bony atresia. Two cases (28.6%) had associated diseases; one had associated cardiac abnormalities in the form of ventricular septal defect and second case had digital anomaly (polydactyly). No intraoperative complications such as skull base damage or CSF leak were encountered. All neonates had eventless recovery .They breathe nasally and sleep peacefully .Oral lactation was begun in the same day of surgery without any respiratory distress. All cases could be discharged in the second day. During follow up period of 11 to 23 months, no crustation, epistaxis, or respiratory or feeding difficulty were reported. Neither granulation nor synechia were detected during follow up (Fig. 3). Adequate functional nasal breathing was maintained in all patients during follow up. However 2 cases showed narrowing of the neochoana but still adequate and satisfactory airway. One case (14.3%) developed small hard palate perforation one week after surgery that healed conservatively. Accurate blood loss was difficult to be calculated precisely due to the amount of saline irrigation and cottonoid pledgets application but during anesthesia duration, the clinical condition of all neonates was evaluated by anesthesiologist without need for blood transfusion. Initial postoperative bleeding during the first 24 h was minimal in all cases. The nursing staff noted that postoperative management was much simpler without stent care. 4. Discussion Bilateral choanal atresia causes complete bilateral nasal obstruction leading to immediate respiratory distress and even potential death due to asphyxia as newborn are obligate nose
Fig. 3. Endoscopic view at one month postoperatively.
breathers until approximately 4–6 weeks at which time mouth breathing is learnt [14]. Bilateral choanal atresia is so a potential life threating condition where early diagnosis and treatment is imperative [12]. Surgical correction of choanal atresia is usually necessary early in life. Surgery of choanal atresia aims at complete removal of the atretic plate and assurance of long-term wide patency [15]. Currently, transnasal and transpalatal approaches are the most commonly used and safest methods of surgical repair. Transnasal approaches include dilations, curettage, drill outs and laser removal. Trans-septal repair is used for older patients over the age of 8 years with unilateral atresia. The transpalatal approach has the disadvantages of long operative time, risks of palatal fistula, cross bite, palatal muscle dysfunction, and dento-alveolar growth disturbance [9], so it is more invasive reserved for failed endoscopic cases [16]. The transnasal endoscopic approach is believed to be the most direct approach to the area and is performed completely under strict visualization. It helps to avoid injury to nearby important structures. It is performed as a same-day surgery and offers immediate feeding and revision surgery is possible [6]. So transnasal rout is currently the preferred procedure and can be performed in a minimal invasive fashion with endoscopic instrumentation. It is safe and rapid procedure even in neonates [16]. The principal challenge of endoscopic procedure in neonates is the narrow nasal cavity as well as further obstruction caused septal deviation, turbinate hypertrophy, and high arched palate [12]. A trial was tried in this study to minimize these difficulties by a new sequence of the steps of choanal atresia repair beginning by vomer. All neonates in this study underwent transnasal endoscopic surgical repair in a single stage procedure with no mucosal flap, no stent placement, nor nasal packing. Adequate functional nasal breathing was maintained in all patients. 57.1% of the neonates included in our study were females this similar to many previous studies [12,17], while Case and Mitchell [3] found the prevalence is similar in males and females. Mixed bony-membranous choanal atresia had been detected in 71.4% of the operated neonates while on the other hand, 28.6% had pure bony atresia. Which is in agree with most previous studies [1,12]. Many benefits of using the endoscopy were gained as direct vision, excellent illumination, and different magnification power by changing the position of the endoscopy during surgery. Moreover endoscopic approach increases safety of the surgery by avoiding palatal and dental complication, in addition to decreasing operative time. The removal of parts of the vomer is the essential step to get a good result for the creation and patency preservation of a neochoana [1]. Stankiewicz [18] attributed his failure in choanal atresia repair to inadequate vomer resection. So vomer resection was performed as a part of choanal atresia repair in many studies [1,4,8,19].
M.A.S. El-Ahl, M.W. El-Anwar / International Journal of Pediatric Otorhinolaryngology 76 (2012) 1002–1006
In an experimental study, Cupero et al. [20] concluded that resection of vomer not affects facial growth. In 1981, Freng [21] reported that the growth of the nasal septum/vomer, at least at its basal parts, is not involved in facial development during the period from infancy to adulthood. Moreover in more recent studies, safe extensive resection of vomerine septum, especially superiorly (Near total endoscopic removal of the bony septum) was done safely by Mcleod et al. [8] with no clinical facial growth disturbance. Even all vomer could be removed safely by Romeh and Albirmawy [19] over their 13 years work. So it is established that vomer resection not affects facial growth. In our study, endoscopy made it possible to remove the posterior portion of the vomer firstly. Then both atretic plates and may part of the medial pterygoids could be removed easier by admitting instrument from one nasal passage and endoscopy through the other nasal passage ending by large new choana, dispensing with the need to use stents or nasal packing or flaps. Also we could get the benefit of clearer field of blood as blood went in the two nasal cavities, allowing procedure to be more rapid and easier. It was very beneficial to admit the drill alone through one nostril avoiding injury, abrasion or crustation of anterior nostrils as our technique obviate the difficulty to operate through one nasal cavity due to anatomic deformities in bilateral choanal atresia include a narrow nasal cavity, lateral bony obstruction by the lateral ptergoid plate, and medial obstruction caused by thickening of the vomer as documented by most authors [12,22]. There is considerable debate on the benefit of using stents in surgical correction of choanal atresia. Many authors have argued that the use of stents is absolutely necessary for successful repair [9,23]. Rombaux et al. [11] considered that stents are needed in bilateral choanal atresia. The benefits of stenting are stabilizing the nasal airway; keep mucosal flaps in position, and remodeling lateral nasal wall [9]. Others indicated that the use of nasal stents can cause various complications including septal and columellar injury, intranasal synechia, infection in the nasal cavities and paranasal sinuses, proliferation of resistant bacteria because of the necessity of long-term antibiotic therapy and discomfort to the child [10]. Varying amount of granulation could be formed in relation to stent at the edge of neochoana that need to be removed later [12]. So it is preferred by many surgeons to not use nasal stent [1,8,13]. In this study by the sequences of steps of surgery offered excellent visualization of the choana and allowed maximal widening of the choana in a much easier, and faster procedure with early recovery and short hospitilization. Stentless surgery avoided stent complications and allowed to use antibiotic for only 7 days with no reported infection. No flaps were needed in agreement with many authors who considered mucosal flaps unnecessary and technically difficult to use especially transnasally in newborns [7,24,25]. In this study, patent neochoana was achieved in all neonates and no developed restenosis even in bony choanal atresia This is concomitant with Schoem [13] and Zuckerman et al. [4] but not concomitant with the studies of Durmaz et al. [25] and Jeissier et al. [26] who reported a higher relapse and restenosis especially in the first 3 months postoperatively. Postoperative failure of endoscopic approaches may be a result of prolonged mucosal trauma from stenting rather than any deficiency inherent in the surgical technique [13]. One case (14.3%) developed small hard palate perforation one week after surgery. that healed conservatively, without further surgery needed this near to study of Ibrahim et al. [1] and Nour and Rida [12]. No adjuvant therapy was used this in agreement with Schoem [13].
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5. Conclusion The endoscopic transnasal surgery technique described in this study, beginning with removal of posterior part of the nasal septum first allowed early using both nasal passages for instruments admission so both atretic plates could be excised, without the use of neither stents nor flaps, offers advantages as clear visualization, easy technique for atretic plates removal through wide, clear and illuminated field. By this way, permanent bilateral nasal breathing was obtained in all patients with no neurological complications, less trauma, less bleeding, no age limitation, rapid recovery and less hospitalization time. Brief research significance The principal challenge of endoscopic procedure in neonates with bilateral choanal atresia is the narrow nasal cavity as well as lateral bony obstruction by the lateral ptergoid plate, and medial obstruction caused by thickening of the vomer. A new sequence of steps of choanal atresia repair beginning by vomer was tried in this study to minimize these difficulties. The posterior part of the nasal septum was gently penetrated creating posterior septal visualization window to facilitate simultaneously access of both sides’ instrumentations. The new opening provided a clearer surgical field by drainage of blood and secretions to both nasal cavities. It became safer, easier and wider to complete procedure through both nasal cavities. Appendix A. Supplementary data Supplementary data associated with this article can be found, in the online version, at http://dx.doi.org/10.1016/j.ijporl.2012.03.019. References [1] A.A. Ibrahim, E.A. Magdy, M.H. Hassab, Endoscopic choanoplasty without stenting for congenital choanal atresia repair, Int. J. Pediatr. Otorhinolaryngol. 74 (2) (2010) 144–150. [2] C.S. Gujrathi, S.J. Daniel, A.L. James, V. Forte, Management of bilateral choanal atresia in the neonate: an institutional review, Int. J. Pediatr. Otorhinolaryngol. 68 (4) (2004) 399–407. [3] A.P. Case, L.E. Mitchell, Prevalence and patterns of choanal atresia and choanal stenosis among pregnancies in Texas, 1999–2004, Am. J. Med. Genet. 155 (4) (2011) 786–791. [4] J.D. Zuckerman, S. Zapata, S.E. Sobol, Single – stage choanal atresia repaire in the neonate, Arch. Otolaryngol. Head Neck Surg. 134 (10) (2008) 1090– 1093. [5] J.F.L. Neto, M.M. Smith, P.A. de Souza, L.R. Vieira, Association between congenital nasolacrimal duct cyst and bilateral choanal atresia, Braz. J. Otorhinolaryngol. 77 (2) (2011) 267. [6] J.L. Keller, A. Kacker, CHARGE association and congenital nasal stenosis, Otolaryngol. Clin. North Am. 33 (6) (2000) 1343–1351. [7] W. Pirsig, Surgery of choanal atresia in infant and children: historical notes and updated review, Int. J. Pediatr. Otorhinolaryngol. 11 (1986) 153–170. [8] I.K. Mcleod, D.B. Brooks, E.A. Mair, Revision choanal atresia repair, Int. J. Pediatr. Otorhinolaryngol. 67 (5) (2003) 517–524. [9] G.D. Josephson, C.L. Vickery, W.C. Giles, et al., Transnasal endoscopic repair of congenital choanal atresia: long-term results, Arch. Otolaryngol. Head Neck Surg. 1243 (1998) 537–549. [10] V.L. Cumberworth, B. Djazari, I.S. Mackay, Endosocpic fenestration of choanal atresia, J. Laryngol. Otol. 109 (1995) 31–35. [11] P. Rombaux, M. Hamior, V. Gilain, G. Verellen, C. Debauche, P. Clapuyt, P. Eloy, B. Bertrand, Choanal atresia a retrospective study of 39 cases, Revue de laryngologie – Otologirhinologie 122 (3) (2001) 147–154. [12] Y. Nour, Rida H.E., Endoscopic repair of bilateral choanal atresia in neonates: anesthetic and surgical implications, Alexandria J. Med. 44 (2008) 1. [13] S.R. Schoem, Transnasal endoscopic repair of choanal atresia: why stent? Otolaryngol. Head Neck Surg. 131 (4) (2004) 362–366. [14] A. Sattar, T. Sultan, Choanal atresia, review article, Bangladesh J. Otorhinolaryngol. 17 (1) (2011) 59–61, 8. [15] K. Reda, Transnasal endoscopic approach in congenital choanal atresia, Laryngoscope 104 (1994) 642–646. [16] P. Assanasen, C. Metheetrairut, Choanal atresia, J. Med. Assoc. Thai. 92 (5) (2009) 699–706.
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[17] S. Roy, R. Vivero, Bilateral choanal atresia in a newborn, Ear, Nose Throat J. 86 (2007) 608–609. [18] A. Stankiewicz, The endoscopic repaire of choanal atresia, Otolaryngol. Head Neck Surg. 103 (1990) 931–937. [19] H.E. Romeh, O.A. Albirmawy, A 13 years experience and predictors for success in transnasal endoscopic repair of congenital choanal obliteration, Int. J. Pediatr. Otorhinolaryngol. (2010) 737–742. [20] T.M. Cupero, C.E. Middlecton, A.B. Saliva, Effects of functional septoplasty on the facial growth of ferrets, Arch. Otolaryngol. Head Neck Surg. 127 (2001) 1367– 1369. [21] A. Freng, Mid-facial sagittal growth following resection of the nasal septumvomer: a roentgencephalometric study in domestic cat, Acta Otolaryngol. 92 (3– 4) (1981) 363–370.
[22] S.G. Harner, T.J. McDonald, D.F. Reese, The anatomy of congenital choanal atresia, Otolaryngol. Head Neck Surg. 89 (1981) 7–9. [23] J.D. Osguthorpe, G.T. Singleton, W.Y. Adkins, The surgical approach to bilaterally choanal atresia: analysis of 14 cases, Arch. Otolaryngol. 108 (6) (1982) 366– 369. [24] A.S. Hengerer, M. Strome, Choanal atresia: a new embryologic theory and its influence on surgical management, Laryngoscope 92 (1982) 913–921. [25] A. Durmaz, F. Tosum, N. Yldrm, et al., Transnasal endoscopic repair of choanal atresia: results of 13 cases and meta-analysis, J. Craniomaxillofac. Surg. 19 (2008) 1270–1274. [26] N. Jeissier, F. Kaguelidou, V. Couloigner, et al., Predictive factors for success after transnasal endoscopic treatment of choanal atresia, Arch. Otolaryngol. Head Neck Surg. 134 (2008) 57–61.
Contents lists available at SciVerse ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Stentless endoscopic transnasal repair of bilateral choanal atresia starting with resection of vomer Magdy Abdalla Sayed El-Ahl, Mohammad Waheed El-Anwar * Otorhinolaryngology, Head and Neck Surgery Department, Faculty of Medicine, Zagazig University, Egypt
A R T I C L E I N F O
A B S T R A C T
Article history: Received 25 January 2012 Received in revised form 18 March 2012 Accepted 20 March 2012 Available online 28 April 2012
Objectives: To assess the results of a transnasal endoscopic repair of congenital choanal atresia beginning by resection of the posterior portion of the vomer and ending by no stent. Methods: Seven patients with bilateral congenital choanal atresia aged ranging from 3 to 15 days were operated upon between June 2009 and September 2011. This transnasal endoscopic approach allowed resection of the posterior portion of the vomer first then the atretic plates and part of the medial pterygoid plate if needed leaving no stent. Postoperative control included office fiberoptic nasal endoscopy. Results: Adequate functional nasal breathing was maintained in all patients during follow up of 11 to 23 months. Apart from one case that complicated by palatal defect, no any other complications were detected. Conclusion: The described technique was proved to be very effective, allowing fast recovery, and one step surgery with early discharge from hospital using neither stents nor nasal packing. Good patency with no reduction in functional quality was also observed. ß 2012 Elsevier Ireland Ltd. All rights reserved.
Keywords: Choanal atresia Endoscopic repair No stent Neonate Vomer
1. Introduction Congenital choanal atersia is defined as a failure in the development of communication between the nasal cavity and nasopharynx causing complete obstruction of the nasal airway [1]. Despite that it has a rare incidence of approximately 1 in 7000 births [2], congenital choanal atresia and stenosis are the most common craniofacial abnormalities of the nose [3]. Neonates with bilateral choanal atresia need immediate interference for nasal respiratory distress as neonates are obligatory nasal breathers [4]. The neonatologists usually are the first to raise the hypothesis of choanal atresia by noticing the failure in pushing the nasal aspiration tube further through the nasal cavity into the pharynx [5]. Paranasal computed tomography (CT) scan and nasal fiberoptic endoscopy are the gold standard tests since they set the diagnosis and enables the examiner to identify the type of atresia. Historically, congenital choanal atresia was described as bony in 90% of the cases and membranous in 10%. However, recent studies suggest that the mixed form is the most common (70%) with a 30% incidence of pure bony atresia [5].
* Corresponding author. Tel.: +20 1004695197. E-mail address: [email protected] (M.W. El-Anwar). 0165-5876/$ – see front matter ß 2012 Elsevier Ireland Ltd. All rights reserved. http://dx.doi.org/10.1016/j.ijporl.2012.03.019
Associated congenital anomalies can be found in approximately 50% of the cases [6] however lower risks of associated congenital anomalies are observed in recent years [3]. Since the first description of choanal atresia in 1755 [7], multiple surgical techniques have been proposed to repair the malformation [2,8], these surgical procedures can be divided into four main approaches: transantral, transpalatal, trans-septal and transnasal [9,10]. Today, the transnasal endoscopic method is the most widely accepted repair technique [8]. It seems that bilateral choanal atresia is associated with more surgical corrections before achieving a normal nasal breathing than unilateral choanal atresia [11]. The aim of this study was to assess the results, easiness and safety of transnasal endoscopic repair of bilateral chongenital choanal atresia in solving the obstruction problem beginning by resection of the posterior portion of the vomer, without the use of nasal stent or mucosal flaps. 2. Patients and methods Seven patients had congenital bilateral choanal atresia were surgically treated from June 2009 to September 2011 at the Otorhinolaryngology Department, Zagazig University Hospitals, Egypt. All patients received a full history review and physical examination including failure of passage of six-French catheter through the nasal cavity into the nasopharynx. Patients were
M.A.S. El-Ahl, M.W. El-Anwar / International Journal of Pediatric Otorhinolaryngology 76 (2012) 1002–1006
Fig. 1. Nasal endoscopy admitted into one nasal cavity and the microburr admitted from other cavity.
examined also with fiberoptic nasal endoscopy. Axial non-contrast CT scanning with thin sections (2–3 mm) was done to all patients to confirm the diagnosis, assess type of atretic plate and thickness of vomer and to exclude any concomitant craniofacial abnormalities. Before CT examination, the nasal passages were aspirated. Preoperative examination was done by neonatologist and anesthesiologist including cardiac evaluation. Chest X-ray and echocardiography were sometimes needed to exclude congenital heart disease. The routine preoperative laboratory tests were done for all neonates. An oropharyngeal airway or orotracheal tube was secured in place in all neonates till the time of surgery. Transnasal endoscopic surgical repair was performed in a single stage procedure with no stent placement, nor nasal packing. A formal consent was signed by parents of the neonates or their relatives and the Zagazig University IRB approved this study. The surgical procedure was performed under general anesthesia using orotracheal tubes in all patients. Small pack was inserted through the oral cavity to nasopharynx. This pack was used to protect the nasopharyngeal mucosa, eustachian tube orifice and skull base and also to displace away the soft palate [12]. Video-endoscopies; a Storz rigid endoscopy 08 4 or 2.7 mm, was used. Cottonoid pads soaked in oxymetazoline hydrochloride 0.05% were used to diminish the volume of the nasal turbinates. Endoscopic examination and evaluation was done first, palpating atretic plates, examining posterior septum and nasal cavity. A curved semilunar incision was made over the posterior part of the bony nasal septum about 3 to 5 mm anterior to the atretic plate. The mucoperiosteal flap was elevated from the bony septum. The inferior aspect of middle turbinate was used as guide for superior limit of dissection.
1003
The posterior part of the nasal septum was gently penetrated creating posterior septal window by sickle knife and dissector and now microdrill and backbiter or through cutting forceps could be admitted in one nasal cavity while the endoscopy could be admitted in other side getting wider space for the procedure with excellent visualization (Fig. 1). Posterior part of the nasal septum anterior to atretic plate was excised from anterior to posterior. The vomer was separated from the nasal floor by sharp dissection. Adequate resection of posterior part of the vomer was essential to create a visualization window to facilitate simultaneously access of both sides’ instrumentations in addition to prevent restenosis (Figs. 1 and 2). The new opening provided a clearer surgical field by drainage of blood and secretions to both nasal cavities. It became easy and wide to complete procedure through both nasal cavities. Using an olive-tipped suction, the atretic plate was penetrated and excised beginning at the weakest infero-medial part to establish a reference point into the packed nasopharynx. In two cases with thicker plate, the microdrill was necessary to establish this connection. The remaining bone of the atretic plate was removed using microdrill, curette, backbiter and/or sphenoid forceps. The neochoana was widened if needed laterally to medial pterygoid plate, inferiorly to the palatine bone, and superiorly to the level of the sphenoid sinus (sloping of the posterior pharyngeal wall). At that time the postnasal pack could be removed through the new choana from the nose. Hemostasis was secured by temporary packing or if diathermy No stents were used, nor nasal packing. Recovery from general anesthesia was uneventful in all cases (Table 1). Postoperative care began on the first day; the nose was washed with saline solution twice a day, then frequently saline nasal drops were used by parents of the neonates. All patients received prophylactic systemic antibiotics for one week. Feeding started immediately after recovery. Neonates were discharged without symptoms at the second day postoperatively. Patients underwent serial office nasal endoscopy weekly for one month then monthly for six months to confirm patency. Surgery was considered successful if the patient showed easy nasal breathing, non interrupted oral feeding, patent nasal airway, and opened new choana. Patency was defined as less than 50% restenosis [13] while the term narrowing was used to describe restenosis of less than 50%. Age at surgery, sex, type of choanal atresia, and postoperative outcomes were recorded and assessed. 3. Results Seven patients treated for bilateral congenital choanal atresia, three (42.9%) of them were male and 4 (57.1%) were female. The mean age at the time of surgery ranged from 4 to 15 days. The mean age at the time of surgery was 8 days. Five (71.4%) neonates
Fig. 2. (A) Nasal endoscopy showing penetration of vomer first. (B) Nasal endoscopy admitted into one side visualizing the microburr admitted from other side. (C) Nasal endoscopy showing partial excision of posterior part of vomer exposing atretic plates bilaterally.
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Table 1 Data of the 7 neonates included in this study. No
Sex
Age
Nature of atresia
Associated anomalies
Postoperative complication
Narrowing
Restenosis
1 2 3 4 5 6 7
Male Female Female Male Female Female Male
15 days 8 days 6 days 9 days 3 days 10 days 5 days
Bony Mixed Mixed Mixed Bony Mixed Mixed
No No No No Cardiac VSD Digital anomaly No
No No No Palatal defect No No No
No Narrowing No No Narrowing No No
No No No No No No No
Total (7)
3 males 4 females
3 to 15 days
5 mixed 2 pure bony
One case VSD One case polydactyl
1 complicated (14.3%) 6 no (85.7%)
2 narrowing (28.6%) 5 no (71.4%)
7 no (100%)
had mixed bony -membranous atretic plates shown by CT and endoscopic examination and 2 (28.6%) had pure bony atresia. Two cases (28.6%) had associated diseases; one had associated cardiac abnormalities in the form of ventricular septal defect and second case had digital anomaly (polydactyly). No intraoperative complications such as skull base damage or CSF leak were encountered. All neonates had eventless recovery .They breathe nasally and sleep peacefully .Oral lactation was begun in the same day of surgery without any respiratory distress. All cases could be discharged in the second day. During follow up period of 11 to 23 months, no crustation, epistaxis, or respiratory or feeding difficulty were reported. Neither granulation nor synechia were detected during follow up (Fig. 3). Adequate functional nasal breathing was maintained in all patients during follow up. However 2 cases showed narrowing of the neochoana but still adequate and satisfactory airway. One case (14.3%) developed small hard palate perforation one week after surgery that healed conservatively. Accurate blood loss was difficult to be calculated precisely due to the amount of saline irrigation and cottonoid pledgets application but during anesthesia duration, the clinical condition of all neonates was evaluated by anesthesiologist without need for blood transfusion. Initial postoperative bleeding during the first 24 h was minimal in all cases. The nursing staff noted that postoperative management was much simpler without stent care. 4. Discussion Bilateral choanal atresia causes complete bilateral nasal obstruction leading to immediate respiratory distress and even potential death due to asphyxia as newborn are obligate nose
Fig. 3. Endoscopic view at one month postoperatively.
breathers until approximately 4–6 weeks at which time mouth breathing is learnt [14]. Bilateral choanal atresia is so a potential life threating condition where early diagnosis and treatment is imperative [12]. Surgical correction of choanal atresia is usually necessary early in life. Surgery of choanal atresia aims at complete removal of the atretic plate and assurance of long-term wide patency [15]. Currently, transnasal and transpalatal approaches are the most commonly used and safest methods of surgical repair. Transnasal approaches include dilations, curettage, drill outs and laser removal. Trans-septal repair is used for older patients over the age of 8 years with unilateral atresia. The transpalatal approach has the disadvantages of long operative time, risks of palatal fistula, cross bite, palatal muscle dysfunction, and dento-alveolar growth disturbance [9], so it is more invasive reserved for failed endoscopic cases [16]. The transnasal endoscopic approach is believed to be the most direct approach to the area and is performed completely under strict visualization. It helps to avoid injury to nearby important structures. It is performed as a same-day surgery and offers immediate feeding and revision surgery is possible [6]. So transnasal rout is currently the preferred procedure and can be performed in a minimal invasive fashion with endoscopic instrumentation. It is safe and rapid procedure even in neonates [16]. The principal challenge of endoscopic procedure in neonates is the narrow nasal cavity as well as further obstruction caused septal deviation, turbinate hypertrophy, and high arched palate [12]. A trial was tried in this study to minimize these difficulties by a new sequence of the steps of choanal atresia repair beginning by vomer. All neonates in this study underwent transnasal endoscopic surgical repair in a single stage procedure with no mucosal flap, no stent placement, nor nasal packing. Adequate functional nasal breathing was maintained in all patients. 57.1% of the neonates included in our study were females this similar to many previous studies [12,17], while Case and Mitchell [3] found the prevalence is similar in males and females. Mixed bony-membranous choanal atresia had been detected in 71.4% of the operated neonates while on the other hand, 28.6% had pure bony atresia. Which is in agree with most previous studies [1,12]. Many benefits of using the endoscopy were gained as direct vision, excellent illumination, and different magnification power by changing the position of the endoscopy during surgery. Moreover endoscopic approach increases safety of the surgery by avoiding palatal and dental complication, in addition to decreasing operative time. The removal of parts of the vomer is the essential step to get a good result for the creation and patency preservation of a neochoana [1]. Stankiewicz [18] attributed his failure in choanal atresia repair to inadequate vomer resection. So vomer resection was performed as a part of choanal atresia repair in many studies [1,4,8,19].
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In an experimental study, Cupero et al. [20] concluded that resection of vomer not affects facial growth. In 1981, Freng [21] reported that the growth of the nasal septum/vomer, at least at its basal parts, is not involved in facial development during the period from infancy to adulthood. Moreover in more recent studies, safe extensive resection of vomerine septum, especially superiorly (Near total endoscopic removal of the bony septum) was done safely by Mcleod et al. [8] with no clinical facial growth disturbance. Even all vomer could be removed safely by Romeh and Albirmawy [19] over their 13 years work. So it is established that vomer resection not affects facial growth. In our study, endoscopy made it possible to remove the posterior portion of the vomer firstly. Then both atretic plates and may part of the medial pterygoids could be removed easier by admitting instrument from one nasal passage and endoscopy through the other nasal passage ending by large new choana, dispensing with the need to use stents or nasal packing or flaps. Also we could get the benefit of clearer field of blood as blood went in the two nasal cavities, allowing procedure to be more rapid and easier. It was very beneficial to admit the drill alone through one nostril avoiding injury, abrasion or crustation of anterior nostrils as our technique obviate the difficulty to operate through one nasal cavity due to anatomic deformities in bilateral choanal atresia include a narrow nasal cavity, lateral bony obstruction by the lateral ptergoid plate, and medial obstruction caused by thickening of the vomer as documented by most authors [12,22]. There is considerable debate on the benefit of using stents in surgical correction of choanal atresia. Many authors have argued that the use of stents is absolutely necessary for successful repair [9,23]. Rombaux et al. [11] considered that stents are needed in bilateral choanal atresia. The benefits of stenting are stabilizing the nasal airway; keep mucosal flaps in position, and remodeling lateral nasal wall [9]. Others indicated that the use of nasal stents can cause various complications including septal and columellar injury, intranasal synechia, infection in the nasal cavities and paranasal sinuses, proliferation of resistant bacteria because of the necessity of long-term antibiotic therapy and discomfort to the child [10]. Varying amount of granulation could be formed in relation to stent at the edge of neochoana that need to be removed later [12]. So it is preferred by many surgeons to not use nasal stent [1,8,13]. In this study by the sequences of steps of surgery offered excellent visualization of the choana and allowed maximal widening of the choana in a much easier, and faster procedure with early recovery and short hospitilization. Stentless surgery avoided stent complications and allowed to use antibiotic for only 7 days with no reported infection. No flaps were needed in agreement with many authors who considered mucosal flaps unnecessary and technically difficult to use especially transnasally in newborns [7,24,25]. In this study, patent neochoana was achieved in all neonates and no developed restenosis even in bony choanal atresia This is concomitant with Schoem [13] and Zuckerman et al. [4] but not concomitant with the studies of Durmaz et al. [25] and Jeissier et al. [26] who reported a higher relapse and restenosis especially in the first 3 months postoperatively. Postoperative failure of endoscopic approaches may be a result of prolonged mucosal trauma from stenting rather than any deficiency inherent in the surgical technique [13]. One case (14.3%) developed small hard palate perforation one week after surgery. that healed conservatively, without further surgery needed this near to study of Ibrahim et al. [1] and Nour and Rida [12]. No adjuvant therapy was used this in agreement with Schoem [13].
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5. Conclusion The endoscopic transnasal surgery technique described in this study, beginning with removal of posterior part of the nasal septum first allowed early using both nasal passages for instruments admission so both atretic plates could be excised, without the use of neither stents nor flaps, offers advantages as clear visualization, easy technique for atretic plates removal through wide, clear and illuminated field. By this way, permanent bilateral nasal breathing was obtained in all patients with no neurological complications, less trauma, less bleeding, no age limitation, rapid recovery and less hospitalization time. Brief research significance The principal challenge of endoscopic procedure in neonates with bilateral choanal atresia is the narrow nasal cavity as well as lateral bony obstruction by the lateral ptergoid plate, and medial obstruction caused by thickening of the vomer. A new sequence of steps of choanal atresia repair beginning by vomer was tried in this study to minimize these difficulties. The posterior part of the nasal septum was gently penetrated creating posterior septal visualization window to facilitate simultaneously access of both sides’ instrumentations. The new opening provided a clearer surgical field by drainage of blood and secretions to both nasal cavities. It became safer, easier and wider to complete procedure through both nasal cavities. Appendix A. Supplementary data Supplementary data associated with this article can be found, in the online version, at http://dx.doi.org/10.1016/j.ijporl.2012.03.019. References [1] A.A. Ibrahim, E.A. Magdy, M.H. Hassab, Endoscopic choanoplasty without stenting for congenital choanal atresia repair, Int. J. Pediatr. Otorhinolaryngol. 74 (2) (2010) 144–150. [2] C.S. Gujrathi, S.J. Daniel, A.L. James, V. Forte, Management of bilateral choanal atresia in the neonate: an institutional review, Int. J. Pediatr. Otorhinolaryngol. 68 (4) (2004) 399–407. [3] A.P. Case, L.E. Mitchell, Prevalence and patterns of choanal atresia and choanal stenosis among pregnancies in Texas, 1999–2004, Am. J. Med. Genet. 155 (4) (2011) 786–791. [4] J.D. Zuckerman, S. Zapata, S.E. Sobol, Single – stage choanal atresia repaire in the neonate, Arch. Otolaryngol. Head Neck Surg. 134 (10) (2008) 1090– 1093. [5] J.F.L. Neto, M.M. Smith, P.A. de Souza, L.R. Vieira, Association between congenital nasolacrimal duct cyst and bilateral choanal atresia, Braz. J. Otorhinolaryngol. 77 (2) (2011) 267. [6] J.L. Keller, A. Kacker, CHARGE association and congenital nasal stenosis, Otolaryngol. Clin. North Am. 33 (6) (2000) 1343–1351. [7] W. Pirsig, Surgery of choanal atresia in infant and children: historical notes and updated review, Int. J. Pediatr. Otorhinolaryngol. 11 (1986) 153–170. [8] I.K. Mcleod, D.B. Brooks, E.A. Mair, Revision choanal atresia repair, Int. J. Pediatr. Otorhinolaryngol. 67 (5) (2003) 517–524. [9] G.D. Josephson, C.L. Vickery, W.C. Giles, et al., Transnasal endoscopic repair of congenital choanal atresia: long-term results, Arch. Otolaryngol. Head Neck Surg. 1243 (1998) 537–549. [10] V.L. Cumberworth, B. Djazari, I.S. Mackay, Endosocpic fenestration of choanal atresia, J. Laryngol. Otol. 109 (1995) 31–35. [11] P. Rombaux, M. Hamior, V. Gilain, G. Verellen, C. Debauche, P. Clapuyt, P. Eloy, B. Bertrand, Choanal atresia a retrospective study of 39 cases, Revue de laryngologie – Otologirhinologie 122 (3) (2001) 147–154. [12] Y. Nour, Rida H.E., Endoscopic repair of bilateral choanal atresia in neonates: anesthetic and surgical implications, Alexandria J. Med. 44 (2008) 1. [13] S.R. Schoem, Transnasal endoscopic repair of choanal atresia: why stent? Otolaryngol. Head Neck Surg. 131 (4) (2004) 362–366. [14] A. Sattar, T. Sultan, Choanal atresia, review article, Bangladesh J. Otorhinolaryngol. 17 (1) (2011) 59–61, 8. [15] K. Reda, Transnasal endoscopic approach in congenital choanal atresia, Laryngoscope 104 (1994) 642–646. [16] P. Assanasen, C. Metheetrairut, Choanal atresia, J. Med. Assoc. Thai. 92 (5) (2009) 699–706.
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[17] S. Roy, R. Vivero, Bilateral choanal atresia in a newborn, Ear, Nose Throat J. 86 (2007) 608–609. [18] A. Stankiewicz, The endoscopic repaire of choanal atresia, Otolaryngol. Head Neck Surg. 103 (1990) 931–937. [19] H.E. Romeh, O.A. Albirmawy, A 13 years experience and predictors for success in transnasal endoscopic repair of congenital choanal obliteration, Int. J. Pediatr. Otorhinolaryngol. (2010) 737–742. [20] T.M. Cupero, C.E. Middlecton, A.B. Saliva, Effects of functional septoplasty on the facial growth of ferrets, Arch. Otolaryngol. Head Neck Surg. 127 (2001) 1367– 1369. [21] A. Freng, Mid-facial sagittal growth following resection of the nasal septumvomer: a roentgencephalometric study in domestic cat, Acta Otolaryngol. 92 (3– 4) (1981) 363–370.
[22] S.G. Harner, T.J. McDonald, D.F. Reese, The anatomy of congenital choanal atresia, Otolaryngol. Head Neck Surg. 89 (1981) 7–9. [23] J.D. Osguthorpe, G.T. Singleton, W.Y. Adkins, The surgical approach to bilaterally choanal atresia: analysis of 14 cases, Arch. Otolaryngol. 108 (6) (1982) 366– 369. [24] A.S. Hengerer, M. Strome, Choanal atresia: a new embryologic theory and its influence on surgical management, Laryngoscope 92 (1982) 913–921. [25] A. Durmaz, F. Tosum, N. Yldrm, et al., Transnasal endoscopic repair of choanal atresia: results of 13 cases and meta-analysis, J. Craniomaxillofac. Surg. 19 (2008) 1270–1274. [26] N. Jeissier, F. Kaguelidou, V. Couloigner, et al., Predictive factors for success after transnasal endoscopic treatment of choanal atresia, Arch. Otolaryngol. Head Neck Surg. 134 (2008) 57–61.