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Teissier 2008

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Teissier 2008

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Tarek abo kammer
Copyright
© © All Rights Reserved
We take content rights seriously. If you suspect this is your content, claim it here.
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ORIGINAL ARTICLE

Predictive Factors for Success After Transnasal


Endoscopic Treatment of Choanal Atresia
Natacha Teissier, MD; Florentia Kaguelidou, MD; Vincent Couloigner, MD, PhD;
Martine François, MD; Thierry Van Den Abbeele, MD, PhD

Objective: To analyze the different factors affecting the drome, 12 with nonsyndromic malformations, and 2 with
outcome of transnasal endoscopic repair of choanal atre- 22q11 microdeletion. Three children had heart malfor-
sia (CA) in children. mations not related to CHARGE association. One child
had a congenital nasal piriform aperture stenosis. Twenty-
Design: Retrospective study. four children had undergone previous surgery; 10 un-
derwent a second procedure with success. Gastroesopha-
Setting: Academic tertiary care children’s hospital. geal reflux disease (GERD) was systematically treated in
cases of restenosis. Topical mitomycin C was used in 3
Patients: Eighty patients (48 girls and 32 boys) aged 3 patients with relapse. Two patients underwent laser treat-
days to 17 years (mean age, 3 years 8 months) who pre- ment to reduce stenotic scarring. Of the 10 patients who
sented with unilateral (n = 53: 37 right, 16 left) or bilat- needed revision surgery, 6 had bilateral CA, and 4 had
eral (n = 27) CA and underwent surgery between Sep- unilateral CA. Age younger than 10 days and presence
tember 1996 and December 2005. of GERD increased the chances of restenosis (P = .03).
Postoperative stenting negatively affected the outcome.
Intervention: All patients underwent transnasal endo- Associated anomalies and previous surgery had no effect
scopic surgery with telescopes and a microdebrider. Na- on outcome. The bony nature of the CA and bilaterality
sal tubes in neonates and nasal packing in older chil- were not significant (P =.08). However, surgeon learn-
dren were removed after 48 hours. Systematic endoscopic ing curve was an important element positively influenc-
revision was performed under local or general anesthe- ing the results (P=.04).
sia a week after surgery. Patients were then clinically and
endoscopically monitored for nasal obstruction and heal- Conclusions: Transnasal endoscopic repair of CA is a
ing for a mean follow-up of 43 months. safe and successful technique. Predictive factors of re-
stenosis are the presence of GERD, age younger than 10
Results: A total of 30 patients presented with associ- days at the time of surgery, and insufficient postopera-
ated malformations: 9 with CHARGE (coloboma, heart tive endoscopic revision. However, previous surgery and
disease, choanal atresia, retardation of postnatal growth associated malformations are not predictive of a poor sur-
and mental development, genital hypoplasia, and ear gical outcome.
anomalies), 1 with Treacher-Collins syndrome, 1 with
Kabuki syndrome, 1 with facial cleft, 1 with Down syn- Arch Otolaryngol Head Neck Surg. 2008;134(1):57-61

C
ONGENITAL CHOANAL ATRE- congenital anomalies; however, in 50% of
sia (CA) is a rare cause of cases, no genetic relationship may be found.2
upper airway obstruc- The embryologic mechanism seems to
tion. Its incidence varies be a combination of the persistence of either
from between 1 in 5000 to the nasobuccal membrane of Hochstetter or
1 in 8000 live births. Anatomically, it con- the buccopharyngeal membrane of the fore-
sists of an enlarged vomer and medialized gut, incomplete resorption of nasopharyn-
lateral pterygoid plate causing a complete geal mesoderm, and locally misdirected me-
Author Affiliations: nasal obstruction.1 It is more frequently uni- sodermal flow. This occurs between the
Department of Pediatric
lateral (usually the right side) and seems to fourth and 11th fetal week.3,4
Otorhinolaryngology
(Drs Teissier, Couloigner, affect girls more than boys. Although the Imaging has allowed a better compre-
François, and Van Den Abbeele) unilateral condition may be left undiag- hension of the nature of the nasal obstruc-
and Clinical Epidemiology Unit nosed, bilateral CA may be life-threaten- tion: In 30% of the cases, it is purely a bony
(Dr Kaguelidou), Robert Debré ing, newborns being solely nasal breath- obstruction; in 70%, the obstruction is a
Hospital, Paris, France. ers. Some CA may be associated with other mixed bony and membranous anomaly.5

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Table. Characteristics of Patients Who Underwent a Second Procedure a

Age at Surgery Disease Characteristic Concomitant Condition


Patient
No. ⱕ10 d ⬍1 y Bilateral Bony Stenosis Malformations GERD Stenting
1 x x x x x x
2 x x x
3 x x
4 x x
5 x x x x x x
6 x x
7 x x x x x x
8 x x x
9 x x x
10 x x x x x

Abbreviation: GERD, gastroesophageal reflux disease.


a An “x” indicates presence of the indicated condition; blank cell indicates condition’s absence.

Systematic preoperative cranial computed tomography lution was performed at least twice a day. Gastroesophageal re-
is useful to assess possible associated anomalies, such as flux disease (GERD) treatment was prescribed postoperatively.
in the CHARGE syndrome (coloboma, heart disease, CA, Regular endoscopic controls and washing of the nasal cavity
retardation of postnatal growth and mental develop- to remove crusts and secretions were performed with the pa-
tient under local anesthesia (topical 1% lidocaine) or, if nec-
ment, genital hypoplasia, and ear anomalies). Inner ear
essary, under general anesthesia in cases of granulomas or syn-
malformations and colobomas are red flags signaling the echiae. Data concerning age at the time of surgery, associated
possible presence of other elements, including heart congenital anomalies, postoperative stenting and treatment, and
anomalies, mental retardation, and genital hypoplasia. complications were collected.
Several surgical approaches may be used to treat con- Congenital CA relapse is defined by some authors as a choa-
genital CA. Historically, the transpalatal technique was nal diameter measuring less than 50% of a normal choana. We
the first one described.6 Then, laser and endoscopic stent- also took into consideration clinical tolerance (dyspnea and na-
ing were used to treat congenital CA.3,7 Recently, trans- sal obstruction). To evaluate the choanal patency, we passed a
nasal endoscopic surgery has developed as a treatment flexible, 4-mm, fiberoptic endoscope nontraumatically through
approach for this indication. Powered instruments such the passage. If no rubbing occurred on the edges of the choanae
during this passage, the child was considered asymptomatic.
as shavers have been introduced and rendered bony and
Comparisons of characteristics were based on the Fisher ex-
membranous resection easier. Image-guided surgery al- act test for categorical variables and the Wilcoxon test for con-
lows a better appreciation of the resection needed and tinuous variables. Tests were 2 sided, and the criterion for sta-
increases safety and precision in difficult anatomic cases.8 tistical significance was Pⱕ.05.
Postoperative stenting is another operative technique, al-
though the type and the duration of the stenting vary
greatly depending on the surgeon. The main complica- RESULTS
tion of the transnasal endoscopic approach is restenosis
of the choanae. Reported rates of restenosis have ranged Ten of the 80 patients presented with a relapse of the na-
from 9% to 36%.2,7,9 Herein, we describe our experience sal obstruction requiring a second surgical procedure
with the transnasal endoscopic approach in 80 children (Table). At the last follow-up, all 10 were asymptom-
presenting with unilateral or bilateral CA. atic. The mean (SD) follow-up was 43 (17) months. The
mean (SD) duration of hospital stay was 8.4 (5.9) days.
METHODS
UNILATERALITY OR BILATERALITY
Eighty patients (48 girls and 32 boys), aged 3 days to 17 years
(mean age, 44 months), who presented with unilateral (n=53: Six of the 27 patients who presented with a bilateral CA
37 right, 16 left) or bilateral (n=27) CA and underwent surgery experienced a relapse (22%), whereas only 4 of the 53
between September 1996 and December 2005 were included in cases of unilateral CA relapsed (8%). A significant age
the study. They were all treated by the same surgeon (T.V.D.A.).
difference was found between these 2 groups: mean (SD)
All patients underwent endoscopic surgery using rigid 30°
telescopes and a microdebrider. Twenty-four to 48 hours post- age, 25.2 (51.4) months for the bilateral CA vs 53.9 (61.8)
operatively, nasopharyngeal tubes were placed in babies, while months for the bilateral CA (P =.03). However, no sig-
calcium alginate nasal packing was used for older children. Both nificant difference was found when analyzing the differ-
the tubes and the packing were usually removed 48 hours af- ence in relapse rates of patients who presented with a uni-
ter placement. Nasal washing with isotonic sodium chloride so- lateral or bilateral CA (P =.08).

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NATURE OF NASAL OBSTRUCTION younger than 1 year, 6 of 33 patients experienced a re-
lapse (18%) (P =.30). If we consider the group of chil-
Twenty-seven patients had a purely bony obstruction, dren younger than 10 days at the time of the first pro-
whereas 53 had a mixed bony and membranous obstruc- cedure, 4 of the 9 patients had relapses (44%). We noted
tion. Six patients with a purely bony obstruction had a re- that 30% of the patients whose disease relapsed were
lapse of the nasal obstruction after surgery (22%), whereas younger than 10 days compared with only 9% of those
4 patients with a mixed obstruction relapsed (8%). This with no relapse. These differences did not reach statis-
difference was not statistically significant (P=.08). tical significance (P =.08).

PREVIOUS SURGERY POSTOPERATIVE STENTING

For 24 patients, the first study procedure was their sec- The duration of postoperative stenting was very short:
ond or third operation (30%). Five of these patients had the average time with nasopharyngeal tubes or calcium
previously undergone laser surgery; 1 also had congen- alginate packing in place was less than a day (0.9 day).
ital nasal piriform aperture stenosis with a central up- Thirty-one patients had tubes, 6 of whom also had re-
per megaincisor; 7 patients had been treated by a trans- lapses. We found that 60% of the patients who relapsed
palatal approach; and 11 had undergone previous had tubes, whereas 36% of the patients who had tubes
transnasal endoscopic surgery. Two patients had been op- did not relapse (P =.17). However, the mean (SD) age of
erated on several times before and were referred to our this group was 11 (22) months, corresponding there-
department. Seven patients who presented with reste- fore to the youngest children. The other 4 children who
nosis and needed revision surgery were first-time surgi- experienced relapses received no stenting.
cal patients (12%); 3 had undergone previous surgery. Three patients who presented with restenosis under-
This difference did not reach statistical significance. went revision surgery with application of mitomycin C.

ASSOCIATED ANOMALIES LEARNING CURVE

Thirty-one children had an associated anomaly (39%). Of the first 40 patients in the study to undergo the
One of these had a facial cleft; another, Kabuki syn- transoral endoscopic procedure (between September 1996
drome. Two children presented with a 22q11 microde- and March 2000), we had 8 relapses in this group. For
letion, and 1 had Down syndrome. Nine children had the second group (April 2000 to December 2005), we had
CHARGE syndrome (11%). One child had Treacher- only 2 relapses. This is significant (P=.04) despite no sig-
Collins syndrome. Twelve children presented with non- nificant differences in age or sex between the 2 groups
syndromic polymalformations (15%). Three children had of children.
heart malformations not related to CHARGE associa-
tion. One child had a congenital nasal piriform aperture
COMMENT
stenosis. Three patients of the 31 patients with associ-
ated anomalies experienced relapse. Differences in asso-
ciated anomalies between relapse-free patients and those SURGICAL GUIDELINES
who had relapses did not reach statistical significance (40%
vs 30%) (P=.73). One patient had undergone a trache- Successful surgery depends on a wide range of factors.
otomy prior to our surgery. The surgical technique requires a wide resection of the
vomer and/or the posterior septum to obtain a large
ANTACID TREATMENT choana. However, anatomic landmarks have to be re-
spected: the choanal opening should be done below the
Fifty-four patients received systemic antacid treatment tail of the medium turbinate to prevent basisphenoid le-
for GERD, and all 10 patients who experienced relapse sions or bleeding. One has to follow the floor of the na-
were from this group. These 10 patients all had GERD sal fossa and open medially and inferiorly. Bony resec-
diagnosed retrospectively either by 24-hour pH record- tion is then completed with the backbiting forceps. The
ing or by a finding of typical posterior laryngitis at en- edges are then smoothed using the microdebrider to pre-
doscopy. Patients who presented with a relapse were more vent bony spikes that might hinder appropriate scar-
frequently undergoing antacid treatment than were re- ring. Little resection is done on the pterygoid process.
lapse-free patients (100% vs 63%) (P = .03.) Considering this technique, McLeod et al10 described, in
2003, an endoscopic procedure involving an almost com-
AGE AT SURGERY plete resection of the vomer to create a wide unique
choana. No stenting is then required, and neither is lat-
Patient age at surgery varied from 3 days to 17 years (me- eral resection.
dian age, 16 months). The difference between median ages
at surgery between the group that relapsed and the group STATISTICAL ANALYSIS
that did not was significant (4.15 vs 18.55 months)
(P=.045). We acknowledge that interpretation of our results is sub-
We also tried to determine a threshold age, younger ject to limitations related to our statistical analysis. Mul-
than which the risk of relapse was greater. In the age group tivariate analysis was not performed because of the lim-

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ited number of relapse cases observed in our series. This quent revision procedures: a mean of 2.7 procedures for
small number of cases also limited the interpretation of unilateral CA and 4.9 procedures in cases of bilateral CA.
some of our results: although certain results were not sig- More recently, Sharma et al13 described a new stenting tech-
nificant, we cannot exclude the possibility that some of nique that required 2 operations and 4 weeks of stenting;
the studied factors do influence surgical outcome. 4 of the 14 patients required a repeated dilatation. Schoem,5
on the contrary, showed that no stenting was necessary for
ANATOMIC CONDITIONS 13 patients. He affirms that stents cause discomfort, local-
ized infection and ulceration, circumferential scar tissue
This study showed that bilaterality and bony stenosis are formation, and injury to the surrounding normal tissue. Our
2 elements that might influence the outcome. It seems research group came to the same conclusion in a previous
that bilateral CA has a greater risk of relapse than uni- study14: revision endoscopy to remove crusts 1 week after
lateral CA, although the difference was not statistically surgery and abundant washing of the nasal cavity with iso-
significant. This greater risk results from twice as many tonic sodium chloride solution were the keys to success-
possibilities of inappropriate scarring leading to reste- ful management of congenital CA without stenting.
nosis of the CA. Risk is also increased by a bilateral me-
dialization of the pterygoid processes. Surgical bony re- AGE
section is usually limited in cases of bilateral CA because
both pterygoid processes are medialized, and the ob- In agreement with Friedman et al,11 we have shown that
tained choana is therefore narrower; whereas, by defini- for children with low weight at the time of surgery, re-
tion, 1 pterygoid process is in normal position in unilat- stenosis is a more frequent occurrence than in higher-
eral CA and is covered by a perfect mucosal lining. weight children. Therefore, age younger than 10 days is
Relapse was more frequent in cases of purely bony ob- associated with a greater risk of relapse. The risk is in-
struction than in other cases (22%; n=6), although the creased yet again with bilateral CA, and early surgical man-
difference was not significant. This higher relapse rate agement is crucial.
may be owing to the narrower choana in cases of pure Nasal patency must be obtained quickly: a McGovern
bony obstruction and the greater difficulty in obtaining nipple or a nasotracheal tube may be necessary, but use
a sufficient resection in this configuration. of these devices should be temporary because they both
Friedman et al11 have shown in a retrospective study have adverse effects. In our opinion, tracheotomy is not
that other factors may affect surgical outcome. For uni- an option.
lateral CA, it seems that neither the presence of facial Children with congenital CA are usually treated in the
anomalies nor the duration of stent placement had an first 10 days of life after a complete assessment to rule
effect on the outcome. On the contrary, for bilateral CA, out the presence of associated anomalies. The normal nar-
children with associated anomalies or low weight at the rowness of a newborn’s nose is a predisposing factor for
time of surgery usually had poorer outcomes.11 In the pre- postoperative obstruction. Moreover, neonates fre-
sent study, associated malformations did not seem to be quently present with physiologic GERD, which in-
associated with a greater risk of relapse compared with creases postoperative inflammation and leads to scar-
isolated CA: only 3 patients of the 10 who presented with ring and therefore restenosis.
restenosis had associated malformations (1 CHARGE syn- A final possible explanation for the greater restenosis
drome, 1 Treacher-Collins syndrome, and 1 unnamed as- rate in babies is that vomer resection may be proportion-
sociation of malformations). However, the assessment of ally more limited in neonates than in older children.
possible other malformations is mandatory for proper
management. USE OF MITOMYCIN C

POSTOPERATIVE STENTING Three patients who presented with restenosis were treated
with topical mitomycin C during revision surgery. All 3 had
Stenting is another factor affecting successful surgery. In a satisfactory choanal patency during follow-up. The num-
our experience, postoperative stenting is not necessary. It ber of patients is too low to draw conclusions concerning
does not seem to prevent obstructive scarring; on the con- the effect of mitomycin C on choanal scarring. Kubba et
trary, better results were obtained without stenting. In the al,15 in a retrospective study, did not find any difference in
present study, nasopharyngeal tubes were preferred for neo- the outcome when 22 patients treated with mitomycin C
nates to ensure postoperative nasal permeability for these were compared with 24 control patients. The authors sug-
obligate nasal breathers. These tubes were kept in place for gested that the use of mitomycin C might just be a marker
less than 2 days. Alginates were usually used for older pa- of refractory disease, since it seems to be used in cases of
tients to prevent postoperative bleeding, and they were re- children with poorer overall outcome.
trieved 24 to 48 hours postoperatively.
Pasquini et al12 showed that a shortened period of stent- HOSPITAL STAY
ing and the use of soft stents diminish the likelihood of either
granulation tissue formation or the risk of postoperative Little is said in other publications concerning the length
infection, both of which may induce restenosis. This con- of hospital stay. In the present study, we considered the
clusion seems true in the light of the findings of Samadi et time for presurgical assessment for neonates with bilat-
al,3 who report in a retrospective study that the average du- eral CA and the first few days of postoperative nasal wash-
ration of stent placement was 41 days, and this led to fre- ing with isotonic sodium chloride solution. Efficient wash-

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ing is a key element for success. Teaching parents to do sis and interpretation of data: Teissier, Kaguelidou, and
it properly may require a few days. A first revision en- Van Den Abbeele. Drafting of the manuscript: Teissier.
doscopy was usually performed at 4 to 5 days after sur- Critical revision of the manuscript for important intellec-
gery to eliminate granulations, inappropriate scarring tis- tual content: Kaguelidou, Couloigner, François, and Van
sue, and false membranes. General anesthesia may be Den Abbeele. Statistical analysis: Kaguelidou. Study su-
required for revision endoscopy to ensure a proper toi- pervision: Van Den Abbeele.
lette of the surgical site. In cases of abundant granulat- Financial Disclosure: None reported.
ing tissue, several endoscopic examinations may be nec- Previous Presentation: This article was presented at The
essary, as frequently as every week or 2 weeks. When a American Society of Pediatric Otolaryngology 2007 An-
good choanal diameter is obtained, simple surveillance nual Meeting; April 29, 2007; San Diego, California.
may be done in the clinic.

LEARNING CURVE
REFERENCES
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half of our study, from September 1996 to March 2000 1. Gujrathi CS, Daniel SD, James AL, Forte V. Management of bilateral choanal atre-
(8 patients). Starting in April 2000, only 2 children needed sia in the neonate: an institutional review. Int J Pediatr Otorhinolaryngol. 2004;
68(4):399-407.
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necessary revision procedures is probably owing to the Surg. 1990;103(6):931-937.
surgeon’s better understanding of prognostic factors: sur- 3. Samadi DS, Shah UK, Handler SD. Choanal atresia: a twenty-year review of medi-
gical resection, absence of stenting, frequent isotonic so- cal comorbidities and surgical outcomes. Laryngoscope. 2003;113(2):
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with the procedures outlined by Kubba et al,15 we used nol Laryngol. 1992;101(11):916-919.
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important element. However, previous surgery and as- 1998;124(5):537-540.
sociated malformations are not predictive of restenosis. 10. McLeod IK, Brooks DB, Mair EA. Revision choanal atresia repair. Int J Pediatr
Otorhinolaryngol. 2003;67(5):517-524.
11. Friedman NR, Mitchell RB, Bailey CM, Albert DM, Leighton SE. Management and
Submitted for Publication: January 31, 2007; final re- outcome of choanal atresia correction. Int J Pediatr Otorhinolaryngol. 2000;
vision received September 3, 2007; accepted September 52(1):45-51.
21, 2007. 12. Pasquini E, Sciarretta V, Saggese D, Cantaroni C, Macri G, Farneti G. Endo-
Correspondence: Natacha Teissier, MD, Department of scopic treatment of congenital choanal atresia. Int J Pediatr Otorhinolaryngol.
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Pediatric Otorhinolaryngology, Robert Debré Hospital, 13. Sharma RK, Lee CA, Gunasekaran S, Knight LC, Bielby M. Stenting for bilateral
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.[Link]). 70(5):869-874.
Author Contributions: Dr Van Den Abbeele had full ac- 14. Van Den Abbeele T, François M, Narcy P. Transnasal endoscopic treatment of
cess to all the data in the study and takes responsibility choanal atresia without prolonged stenting. Arch Otolaryngol Head Neck Surg.
2002;128(8):936-940.
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