Neonatal iatrogenic nasal obstruction

Neonatal iatrogenic nasal obstruction

International Journal of Pediatric Otorhinolaryngology (2004) 68, 613—617 CASE REPORT Neonatal iatrogenic nasal obstruction Ari DeRowe a,*, Roee Lan...

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International Journal of Pediatric Otorhinolaryngology (2004) 68, 613—617

CASE REPORT

Neonatal iatrogenic nasal obstruction Ari DeRowe a,*, Roee Landsberg b , Gadi Fishman a , Doron Halperin c , Dan Fliss b a

Pediatric Otolaryngology Unit, ‘‘Dana’’ Children’s Hospital, Tel-Aviv Medical Center, 6 Weitzman St., Tel-Aviv 64239, Israel b Department of Otolaryngology Head and Neck Surgery, Tel-Aviv Sourasky Medical Center, Tel Aviv, Israel c Department of Otolaryngology Head and Neck Surgery, Kaplan Hospital, Rehovot, Israel Received 25 September 2003 ; received in revised form 11 December 2003; accepted 22 December 2003

KEYWORDS Nasal obstruction; Neonate; Nasal synechia; Iatrogenic

Summary Objective: To define the etiology, management and prevention of iatrogenic neonatal nasal obstruction caused by intra-nasal synechia (INS). Design: Case series retrospectively reviewed, years 2001–2002. Setting: Tertiary care children’s hospital Patients: Four neonates, ages 2 weeks to 3 months, evaluated for nasal obstruction and consequent respiratory distress. Endoscopic findings were of intra-nasal synechia caused by previous intra-nasal invasive interventions. Main outcome measures: The procedures inciting the formation of synechia, possible management problems leading to synechia formation, clinical presentation, treatment and results of treatment were documented. Results: Initial incorrect diagnosis and management with an inappropriate intervention was found to be a possible cause of nasal obstruction due to synechia formation in three of these patients (two with associated craniofacial anomalies). In one patient complete nasal obstruction due to synechia was a result of continuous positive airway pressure (CPAP) intra-nasal catheters. Treatment including endoscopic synechiolysis resulted in an improved airway but not a complete cure. All patients required multiple procedures to attain a satisfactory nasal airway. Conclusion: Careful initial diagnosis and management of neonatal nasal obstruction may prevent severe complications caused by synechia in the nasal cavity. © 2004 Elsevier Ireland Ltd. All rights reserved.

1. Introduction Neonates are obligatory nasal breathers. Nasal obstruction in the neonate requires prompt intervention [1]. The differential diagnosis includes: choanal atresia, pyriform aperture stenosis, cran-

* Corresponding author. Tel.: +972-3-6974517; fax: +972-9-7492822. E-mail address: [email protected] (A. DeRowe).

iofacial anomalies, meningo-encephalocele and others [2,3]. Intra-nasal synechia (INS) formation is a complication of nasal surgery. The narrow nasal airway of the child [4] may predispose them to synechia formation following intra-nasal procedures. There is no consideration in the literature regarding synechia complicating intra-nasal interventions in neonates despite the dire consequences of nasal obstruction in this age group. This series of patients describes possible causes, prevention

0165-5876/$ — see front matter © 2004 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2003.12.012

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strategies and treatment of iatrogenic neonatal nasal obstruction.

2. Report of cases During the period of the years 2001–2002, four neonates presenting with progressive upper airway obstruction, ages ranging between 2 weeks and 3 months, were referred to the pediatric otolaryngology service in our hospital. These patients were referred following a previous intra-nasal invasive procedure. The Initial workup included an endoscopic exam revealing bilateral INS and CT scanning in all four neonates. Table 1 summarizes clinical details and etiology of synechia in these four neonates. In three of the four cases the inciting intervention was nasal dilatations. In these three patients an initially narrow nasal airway resulted in upper airway obstruction, apnea and feeding difficulties. In patient #1 a diagnosis of ‘‘nasal stenosis’’ was made and dilatations with stent placement performed. Shortly following stent removal progressive airway obstruction, apnea and failure to thrive (FTT) appeared. The child was discharged to a chronic care hospital with supplementary oxygen and oxygen saturation monitoring. She was referred to our hospital in severe respiratory distress at 3 months of age. Endoscopy revealed bilateral INS. CT scan revealed bilateral INS but also a midline mega-incisor and pyriform aperture stenosis. In case #2 at birth respiratory distress developed that was relieved by oral airway placement. It was a term and uneventful pregnancy and delivery. CT scan was interpreted as ‘‘choanal stenosis’’. The child underwent nasal dilatations and stent placement. With the stents in place the child had no breathing difficulties. After 2 weeks the stents were removed and progressive airway obstruction devel-

oped. He was referred to our hospital. On exam the child was in respiratory distress. Endoscopic examination revealed severe bilateral synechia and a large septal perforation. CT scan revealed bilateral INS without any additional anatomic anomaly. Patient #3 had Apert’s syndrome. At birth respiratory distress developed and CT scan was interpreted as ‘‘choanal stenosis’’. Dilatations with stent placement were performed. The stents spontaneously extruded after 1 month and progressive sleep apnea appeared. She was referred to our hospital for neurosurgical consultation regarding her craniosynostosis. Due to her nasal obstruction nasal endoscopy was performed revealing severe bilateral INS. CT scan revealed bilateral hypoplasia of the maxilla and INS. The CT and re-evaluation of the initial CT showed no choanal stenosis, however, nasal cavity narrowing due to the cranio-facial anomaly was seen. In these three cases intervening in an initially narrow nasal airway by dilatations and stent placement probably resulted in trauma to the nasal mucous membranes and formation of synechia resulting in even greater airway compromise. Case #4 was referred at 6 weeks of age due to progressive upper airway obstruction. He suffered from meconium aspiration at birth which developed into sepsis and an epidural abscess near the anterior brain stem. He required respiratory assistance in the form of nasal CPAP catheters for 3 weeks. Fig. 1 shows the endoscopic exam revealing synechia that were present in both nasal cavities. Fig. 2 shows the CT scan revealing bilateral INS.

3. Results In all four patients surgical intervention was attempted to attain a patent nasal airway. The indications for surgery were upper airway obstruction,

Table 1 Clinical characteristics of four patients with iatrogenic nasal obstruction Patient #

Age

Craniofacial anomaly

Inciting intervention

Endoscopic findings

Treatment

1

3 months

Pyriform aperture stenosis

Nasal dilatations and stenting

Sublabial degloving

2

2 weeks

None

3

3 months

Apert’s

Nasal dilatations and stenting Nasal dilatations and stenting

Narrow pyriform aperture, bilateral synechia Septal perforation, bilateral synechia Bilateral nasal synechia

4

6 weeks

None

CPAP catheters

Bilateral nasal synechia

Endoscopic synechiolysis with bilateral stenting Endoscopic synechiolysis followed by repeat dilatations Endoscopic synechiolysis with bilateral stenting

Neonatal iatrogenic nasal obstruction

Fig. 1

615

Naso-endoscopy revealing synechia on the right side in patient #4.

feeding difficulties, FTT and obstructive sleep apnea. In patient #1 a sublabial approach to pyriform aperture repair with drilling of the maxillary bone was performed along with endoscopic synechiolysis. Short-term stenting with a naso-pharyngeal tube was used. A patent nasal airway with gradual resolution of symptoms resulted. In patient #2 due to the severity of the synechia following synechiolysis bilateral nasal stenting was performed.

Stents were removed after 6 weeks. There was a dramatic improvement in nasal breathing and no further intervention was required. Patient #3 had bilateral endoscopic synechiolysis and dilatation performed. Because of the previous negative result of stenting, no stenting was performed. Following surgery synechia reformed and repeat dilatations were performed in clinic. After four dilatations a patent airway in one nostril was achieved with

Fig. 2 CT showing bilateral intra-nasal synechia in patient #4. The right side shows complete obstruction. On the left obstruction is partial.

616 improvement but not resolution of symptoms. The other nasal passage remained obstructed; however, the parents declined further treatment. Patient #4 had endoscopic synechiolysis performed. Because of severity a stent was placed in the right nasal cavity. Following removal synechia reformed on both sides. Dilatations were performed in clinic three times at weekly intervals until a patent airway was achieved on both sides and symptoms resolved.

4. Discussion Iatrogenic neonatal INS is a severe complication of intra-nasal interventions in neonates. In this series initial mismanagement including misdiagnosis resulted in unnecessary procedures in three children. A similar finding was reported by Sculerati when choanal atresia was incorrectly diagnosed in cranio-facial anomalies [5]. Nasal dilatations in the narrow airway in these three children resulted in INS formation. Dilatations in a narrow nasal cavity should be discouraged seeing that more damage may result. In patient #4 INS was a complication of intra-nasal CPAP catheters. This complication in a neonate is rare and has not, to our knowledge, been previously reported. Although dilatations as a primary treatment may result in the formation of synechia we found the modality of some benefit once treatment of synechia was initiated when applied to gently prevent synechia reformation. Mitomycin may be considered as an adjuvant to prevent synechia recurrence; however, data is inconclusive [6]. We did not include mitomycin in our treatment protocol. Allthough dilatations were the cause of INS in three-fourth of these patients, once synechia formed we preferred gentle dilatations as part of the treatment protocol in order to avoid stent placement with the hope of allowing relining of the nasal mucosa before synechia reformation. Even though repeat dilatations were needed, we observed gradual improvement in the nasal airway with this modality in all four children. Previous reports have described iatrogenic nasal obstruction in children. One report described nasal vestibular stenosis following birth trauma [7] and another study described nasopharyngeal stenosis following adenotonsillectomy [8]. No previous studies have described INS formation in the neonate following intra-nasal interventions. This entity should be considered when an infant presents with nasal obstruction and a history of prior intra-nasal intervention is elicited. Endoscopy is the diagnostic procedure of choice; however, CT is important to evaluate additional anatomic anomalies.

A. DeRowe et al. The narrow nasal airway in neonates is a difficult diagnostic and therapeutic dilemma. This case series shows the importance of initial diagnosis and management. Once the damage is done and INS is caused management is difficult. From this small series it is difficult to reach conclusions regarding optimal treatment for iatrogenic INS in neonates. In this age group timely intervention to alleviate the nasal obstruction is required seeing the consequences in infants. Initial correct diagnosis and avoidance of unnecessary intra-nasal interventions is of utmost importance in the primary prevention of INS in neonates. These four patients illustrate the difficulty in the treatment of INS. Multiple procedures were necessary and not all cases resulted in complete resolution of the findings. The treatment of each neonate with INS was tailored to each case since no data was found regarding treatment of INS in this age group. In patient #1 initial misdiagnosis of pyriform aperture stenosis led to the decision to perform drill-out of the pyriform aperture using the sub-labial approach. We presumed that the widening of the framework of the nasal passage will avoid reformation of synechia. In patient #2 no abnormalities of the framework were observed on CT so drill-out was considered inappropriate. Stents were placed due to the severity of the INS. Patient #3 had Crouzon’s and skeletal intervation of the maxilla was not considered in a neonate. Because of the narrow framework stenting was avoided in this case. Patient #4 was unique in that INS was not caused by an inappropriate intervention but by CPAP nasal catheters. His CT did not show an underlying deformity. Due to the severity of INS stenting was performed as an adjuvant to endoscopic synechiolysis. Although a satisfactory airway was achieved multiple procedures were required in all cases. More experience will be required to define optimal treatment for neonatal INS. In conclusion, INS is a serious complication of intra-nasal procedures in children and may be avoided by careful pre-intervention evaluation of the neonatal nasal airway.

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Neonatal iatrogenic nasal obstruction [4] P. Contencin, L. Gumpert, J. Sleiman, L. Possel, I. De Gaudemar, C. Adamsbaum, Nasal fossae dimensions in the neonate and young infant: a computed tomographic scan study, Arch. Otolaryngol. Head Neck Surg. 125 (7) (1999) 777—781. [5] N. Sculerati, M.D. Gottlieb, M.S. Zimbler, P.D. Chibbaro, J.G. McCarthy, Airway management in children with major craniofacial anomalies, Laryngoscope 108 (12) (1998) 1806— 1812.

617 [6] C.J. Hartnick, B.E.J. Hartley, P.D. Lacy, et al., Topical mitomycin application after laryngotracheal reconstruction, Arch. Otolaryngol. Head Neck Surg. 127 (2001) 1260—1264. [7] J.H. Jablon, J.F. Hoffman, Birth trauma causing nasal vestibular stenosis, Arch. Otolaryngol. Head Neck Surg. 123 (9) (1997) 1004—1006. [8] C. Giannoni, M. Sulek, E.M. Friedman, N.O. Duncan III, Acquired nasopharyngeal stenosis: a warning and review, Arch. Otolaryngol. Head Neck Surg. 124 (2) (1998) 163—167.