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Transnasal Endoscopic Treatment of Choanal Atresia Without Prolonged Stenting
Thierry Van Den Abbeele, MD, PhD;
Martine François, MD;
Philippe Narcy, MD
Arch Otolaryngol Head Neck Surg. 2002;128:936-940.
ABSTRACT
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Objective To analyze the outcome of transnasal endoscopic repair of choanal atresia
(CA) in children without prolonged nasal stenting after surgery.
Design Retrospective study.
Setting Academic tertiary care children's hospital.
Patients Forty children aged 3 days to 15 years (mean age, 41 months) who presented
with unilateral (n = 26) or bilateral (n = 14) CA and underwent surgery between
August 1996 and December 1999.
Intervention All children underwent transnasal endoscopy with telescopes, endoscopic
instruments, and a microdebrider. Nasal tubes in neonates or infants and nasal
packing in older children were always removed after 2 days. Systematic revision
endoscopy was performed with the patients under local or general anesthesia
on days 6 to 10. All patients were then clinically and endoscopically monitored
for nasal obstruction and healing during a mean follow-up period of 18 months.
Results There were 16 patients with associated malformations, including 6 cases
of 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), and 14 patients
(9 with bilateral CA) with a history of previous choanal surgery (4 transnasal,
4 laser, and 6 transpalatine). Postoperatively, 32 patients (80%) had normal
nasal patency and a satisfactory choanal diameter, and 8 (20%) had restenosis
or complete choanal closure. Six underwent a second procedure, with success.
The results in all children who had been previously treated with laser or
transpalatine surgery were successful. The last patient, who presented with
severe Treacher Collins syndrome, is still tracheotomized. There were no significant
postoperative complications. One patient died of congenital cardiopathy 6
months after surgery.
Conclusions Transnasal endoscopic repair of CA is a safe and successful technique.
The use of powered instrumentation and routine postoperative revision endoscopy
seems to avoid prolonged nasal stenting.
INTRODUCTION
CONGENITAL CHOANAL atresia (CA) is a rare malformation that causes airway
obstruction in newborns and infants, with an incidence of 1 in 7000 to 8000
births. It seems to occur more commonly in females than males and to be more
frequently unilateral and right-sided than bilateral.1
The nature of the obstructing atretic plate has often been described as 90%
bony and 10% membranous. More recent studies using modern imaging techniques
have revealed that 30% of the malformations are composed of a purely bony
obstruction, and 70% of a mixed bony-membranous anomaly.2
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).1
Many approaches have been used to repair CA, including transpalatal
and transnasal routes. Until recently, the transpalatal technique was the
method preferred by most surgeons for reasons of excellent visualization and
success rates of around 80% to 90%.3-4
Transnasal techniques, including laser vaporization,5-7
are less effective and often require prolonged stenting, with higher failure
rates requiring revision surgery. However, the recent development of rigid
endoscopes and powered instrumentation has led to a reconsideration of transnasal
techniques,8-9 although most authors
are still using postoperative nasal stenting for several weeks. Herein, we
describe our experience, some of it in neonates, with a transnasal endoscopic
technique using telescopes and powered instrumentation without prolonged postoperative
stenting.
PATIENTS AND METHODS
Forty children (24 girls and 16 boys) aged 3 days to 15 years (mean
age, 41 months) were treated between August 1996 and December 1999. Ten children
were neonates, and 8 others were younger than 12 months. Fourteen children
(35%) had bilateral CA and 26 (65%) had unilateral CA (18 right-sided and
8 left-sided).
Sixteen children (40%) had associated deformities, including isolated
cardiac malformations (n = 2), CHARGE association (n = 6), Treacher Collins
syndrome (n = 1), and other undiagnosed polymalformative associations (n =
7). The average follow-up period was 18 ± 9 (mean ± SD) months.
Preoperative computed tomographic scans with axial, coronal, and sagittal
reconstructions were obtained in all cases (Figure 1). Twenty-one children (52%) had a mixed bony-membranous
atretic plate, and 19 children (48%) had a pure bony atretic plate.
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Figure 1. Endoscopic and preoperative
computed tomographic (CT) scans of a child with typical left-sided choanal
atresia (CA). A, Direct endoscopic view of the atretic wall. Note that the
inferior part is membranous (Memb) and the upper part is bony. S indicates
nasal septum; MT, middle turbinate; and IT, inferior turbinate. B, Non–contrast-enhanced
bony-windowed axial CT scan suggesting pure membranous CA. C, Bony-windowed
coronal CT reconstruction. D, Bony-windowed sagittal reconstruction revealing
the mixed bony-membranous components of the atretic plate.
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Fourteen (35%) of the patients had previously undergone surgery using
transnasal (n = 4), laser (n = 4), or transpalatal (n = 6) techniques. All
but 2 of the 14 procedures were performed at another institution; a transpalatal
technique was used in both cases. The mean age of the patients with bilateral
CA at the time of primary surgery was 9 days (age range, 3-25 days; n = 5).
The other patients with bilateral CA were referred to our department after
other transnasal and/or transpalatine techniques failed, and they underwent
surgery at a mean age of 42 months (age range, 16 days to 4 years; n = 9).
The mean age of patients with unilateral CA at the time of the initial surgery
was 40 months (age range, 22 days to 9 years; n = 19). The other patients
with unilateral CA were referred for a secondary procedure at a mean age of
67 months (n = 6). The surgical procedures, which were identical in all cases,
used 4.0-mm 0° and 30° and 2.7-mm 0° nasal telescopes (Karl Storz,
Tuttlingen, Germany). Traditional sinus instrumentation was used together
with powered instrumentation with 2- and 4-mm blades (Karl Storz). The mucosa
covering the atretic bone was gently removed using the soft tissue blades.
The cutting burr of the powered instrument was then used to remove the posterior
part of the vomer, the lateral wall of the choana, and the upper part of the
septum and its attachment to the sphenoid bone. Backbiting forceps were often
used to complete the resection of the posterior part of the vomer. In some
cases, the soft tissue shaver was also used to remove hypertrophic adenoid
pads or to reduce hypertrophic inferior turbinates. Figure 2 shows the major steps of this procedure. The mean ±
SD operating time was 50 ± 8 minutes for unilateral CA and 84 ±
20 minutes for bilateral CA. Polyethylene tracheal tubes (size 3.0, 3.5, or
4.0, according to the size of the nasal fossa; Mallinckrodt Inc, St Louis,
Mo) were used to prevent immediate postoperative nasal obstruction in children
younger than 12 months and were removed after 24 to 48 hours. A soft (calcium
alginate) nasal packing (Algosteril; Laboratoires Brothier, Nanterre, France)
was used in the older children after surgery. No prolonged nasal stenting
was used. Postoperative drainage of secretions and crusts was performed by
the nurses and the parents using an abundant isotonic sodium chloride wash.
Revision endoscopy was systematically performed 1 week after surgery with
the patient under local anesthesia (topical 1% lidocaine) or, if necessary,
under general anesthesia in the case of granulomas. Gastroesophageal reflux
(GER) was diagnosed on the basis of clinical gatrointestinal symptoms or 24-hour
pH recording when possible (in unilateral CA cases or before revision procedures).
After surgery, airway patency and the choanal size were checked regularly
in all cases using a 3.5-mm flexible nasal endoscope (Figure 3).
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Figure 2. Surgical technique (right side)
in a neonate with bilateral choanal atresia. A, Direct endoscopic exposure
of the atretic plate and opening of the atretic plate using the powered instrumentation.
B, Resection of the posterior part of the vomer using backbiting forceps.
C, Drilling of the lateral wall of the choana using the powered instrumentation.
D, Final shape of the choana at the end of the procedure. MT indicates middle
turbinate; IT, inferior turbinate; S, nasal septum; and Sp, inferior part
of the sphenoid bone.
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Figure 3. Fiberscopic view of the choana
1 year after endoscopic treatment of left-sided choanal atresia in a 4-year-old
child. A, View of the right choana (normal side). B, View of the surgically
treated side.
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RESULTS
Thirty-two (80%) of the children who underwent surgery had a patent
choana after 1 transnasal endoscopic procedure. The endoscopic aspect of the
choana looked nearly normal in 27 of these cases and slightly narrowed, with
small granulomas or webs, in 5 cases. The success rate of the primary procedure
was 70% (7/10) in neonates; 71% (10/14) in children younger than 6 months,
including neonates; and 85% (22/26) in older children. None of these differences
is statistically significant (P = .37 and P = .41, Fisher exact test). The mean follow-up period was 18 months.
No postoperative complications, such as pain, infection, bleeding, or nasal
deformity, were found. Normal feeding was always possible starting the day
after surgery. Children younger than 6 months were discharged 12 ±
6 (mean ± SD) days after surgery and older children after 6 ±
2 days. Parents and nurses found that postoperative care was facilitated by
the absence of nasal stenting. One patient with a patent choana died of congenital
cardiopathy 6 months after surgery.
The success rate for bilateral CA was 71% (10/14) and for unilateral
CA 84% (22/26) and did not differ statistically (P
= .41, Fisher exact test). The clinical and radiological features and typical
outcome of bilateral CA related to CHARGE association are shown in Figure 4. Twelve (86%) of the 14 children
who had undergone surgery before endoscopic repair were successfully treated
in 1 procedure. Nine (64%) of the 14 children had bilateral CA, and 8 (89%)
of these 9 children were treated successfully. Eight (20%) of 40 patients
had restenosis, always less than 3 months after the primary repair. Six (75%)
of the 8 children had a bony atretic plate, 4 (50%) were younger than 6 months,
and 4 (50%) had bilateral CA. Gastroesophageal reflux (clinical symptoms or
positive results on 24-hour pH recording) was found in 13 (32%) of the 40
children. Postoperative granulomas or narrowing of the choanal aperture were
found in 8 of 13 children with GER and in only 5 of 27 patients without GER
(P<.01, Fisher exact test). Seven (87%) of the
8 children who had restenosis also had GER. Six of them underwent a second
procedure, which was successful in all cases. No patient underwent a third
procedure. One patient who presented with recurrent bilateral CA and a congenitally
narrow rhinopharynx was treated with topical mitomycin (1 mg/mL for 5 minutes),
which seemed to reduce postoperative scarring. Two other children did not
undergo revision surgery. In 1 case, the child's contralateral nasal patency
was normal, so the parents decided to wait. The other case involved severe
Treacher Collins syndrome with a small mandible and glossoptosis that required
a prolonged tracheostomy. The overall final result was normal choanal patency
in 38 (95%) of 40 patients.
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Figure 4. Bilateral choanal atresia in a
child with CHARGE association (a malformative syndrome that includes coloboma,
heart disease, choanal atresia, retarded development, genital hypoplasia,
and ear anomalies, including hypoplasia of the external ear and hearing loss).
A, Note the typical shape of the external ear (absence of earlobe). B, Non–contrast-enhanced
bony-windowed axial computed tomographic scan. Note the enlargement of the
posterior part of the vomer, the narrow diameter of the rhinopharynx, and
the absence of semicircular canals. C, View of the choana 6 months after surgery.
Note the good patency of the choana but the narrow size of the rhinopharynx.
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COMMENT
Choosing between the transnasal and transpalatal routes, as well as
the use of postoperative stenting, is still a controversial feature of the
surgical management of CA.10 Before the development
of endonasal endoscopic techniques, the transpalatal approach was considered
to be the most appropriate and successful route for bilateral or unilateral
CA in children, both young and old, and for revision cases after failure of
previous transnasal techniques.4 The transpalatal
approach can result in several adverse effects, including palatal fistulas,
maxillofacial disturbances, peroperative bleeding, and postoperative pain.11 Since 1990, some studies have reported high success
rates using endoscopic techniques and an otologic drill or conventional instruments,
but they were usually based on small numbers of children with unilateral CA.8, 12 Lazar and Younis,13
in a larger series involving 10 children and operating with transnasal endoscopic
techniques and conventional instruments, reported 2 failures in newborns with
bilateral CA. They therefore concluded that this technique was suitable for
older children with unilateral CA. Since then, a few studies have reported
cases in which neonates were successfully treated using endoscopic techniques.14-16 Interestingly, Josephson
et al16 found powered instrumentation very
useful in their last 7 cases, as previously suggested by Parsons.17
Herein, we describe the treatment of 40 children with CA, including
10 newborns, using a transnasal endoscopic technique and powered instrumentation.
This technique combines a protected soft tissue blade or drill and continuous
suction that seem particularly useful in small nasal fossae. It provides better
visualization and aspiration of blood and mucosal debris; it also seems to
cause less tissue injury and allows better healing.9
Combined with conventional instruments, such as backbiting forceps, it allows
the pterygoid process to be drilled laterally, the abnormal part of the vomer
posteriorly, and the upper part of the choana and insertion of the septum
to the sphenoid superiorly. In our opinion, this technique creates a common
posterior cavity that does not require prolonged stenting.
Stents have been used in most, if not all, reported surgical procedures.
Many authors believe that stenting prevents postoperative restenosis. The
duration and material of stenting vary from one study to another, and the
former can range from several weeks to months.2, 13, 16, 18-19
Stents are associated with local infections and pain, formation of granulation
tissue, and nasal synechia. Stent management is often complicated by migration
or excessive pressure on the nasal ala.19 Our
results compare favorably with those of other endoscopic series and clearly
suggest that postoperative stenting is not necessary, provided that the nasal
cavity is washed with abundant saline, especially in small children. Gastroesophageal
reflux has also been suspected to induce choanal restenosis20
and should be looked for and treated when present. We believe that revision
endoscopy is mandatory because removal of crusts is always necessary 1 week
after surgery. This procedure sometimes requires a short-acting general anesthetic
when there are granulomas or mucosal synechiae and makes it possible to evaluate
the healing process. The efficacy of topical mitomycin in selected cases (eg,
more than 2 failed procedures or bilateral CA with narrow rhinopharynx) needs
further investigation, but it has been suggested that this antibiotic can
reduce the risk of choanal restenosis.21
CONCLUSIONS
Transnasal endoscopic repair of congenital CA is a safe and effective
procedure. The use of powered instrumentation greatly improves the results,
especially in younger children, and does not require prolonged nasal stenting.
This technique is suitable for use in young children, even in neonates, with
bilateral CA or in revision cases after previous transnasal or transpalatal
approaches have failed. None of the children referred to our department since
1996 has required any other type of approach.
AUTHOR INFORMATION
Accepted for publication October 25, 2001.
This study was presented in part at the 15th Annual Meeting of the American
Society of Pediatric Otolaryngology, Orlando, Fla, May 16-18, 2000.
Corresponding author and reprints: Thierry Van Den Abbeele, MD, PhD,
Service d'ORL pédiatrique, Hôpital Robert Debré, 48 Boulevard
Sérurier, 75019 Paris, France (e-mail: thierry.van-den-abbeele{at}rdb.ap-hop-paris.fr).
From the Department of Pediatric Otorhinolaryngology, Robert Debré
Hospital, Paris, France.
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THIS ARTICLE HAS BEEN CITED BY OTHER ARTICLES
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Arch Otolaryngol Head Neck Surg 2008;134:57-61.
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