AM ER IC AN JOURNAL OF OT OLARYNGOLOGY–H E A D A N D NE CK M E D IC IN E A ND S U RGE RY 3 6 (2 0 1 5) 6 97–7 0 0

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Pediatric otolaryngology: Principles and practice

Endoscopic surgical management of inspiratory stridor in newborns and infants☆ Jeffrey Cheng, MD⁎, Lee P. Smith, MD Division of Pediatric Otolaryngology, Cohen Children’s Medical Center, New Hyde Park, NY Department of Otolaryngology, Head and Neck Surgery, Hofstra North Shore-LIJ School of Medicine, Hempstead, NY

ARTI CLE I NFO

A BS TRACT

Article history:

Objective: Compare the incidence of endoscopic surgical treatment of patients with

Received 4 May 2015

laryngomalacia to other aerodigestive pathology who may present with similar symptoms. Methods: Consecutive case series with chart review of endoscopic surgical intervention in infants, aged 12 months or less, presenting with inspiratory stridor, in the absence of syndromic condition or prior history of intubation. Results: A total of 30 patients were identified. The average age at the time of surgical intervention was 2.7 months. Endoscopic surgical management was directed at laryngomalacia (70%), vallecular cysts (23.3%), and anterior glottic webs (6.7%). All patients had nearly immediate resolution of the stridor and feeding difficulties. None required revision surgery, modified diets, or alternative means of enteric nutrition. Conclusions: Laryngomalacia was the most commonly encountered surgical indication for stridulous newborns and infants with severe symptoms. Like most previous descriptions, patients responded well to supraglottoplasty. Vallecular cysts accounted for about onequarter of the infants treated. Clinicians should carefully consider the presence of other airway pathology, which may mimic laryngomalacia, in non-syndromic infants without a previous history of intubation. Endoscopic surgical management may be safe and effective. © 2015 Elsevier Inc. All rights reserved.

1.

Introduction

Evaluation of the newborn or infant presenting with noisy breathing, dysphagia and failure to thrive starts with a detailed history and physical examination. Bedside laryngoscopy often adds a critical and integral component to the clinical assessment. Noisy breathing in infants is most often attributed to laryngomalacia. Unless there are severe symptoms, such as failure to thrive or dysphagia, most patients do well with expectant management, with only approximately less than 15% requiring surgical intervention [1]. Those with ☆

severe symptoms are usually treated with supraglottoplasty, which has a high success rate [2]. However, there are several other airway pathologies that can be encountered causing symptoms that may mimic laryngomalacia, such as vallecular cysts or anterior glottic webs and should be carefully considered when evaluating these newborns and infants. Several authors have reported and described their experiences with individual pathologies but these may have overlapping symptoms that mimic each other [3–7]. Our aim is to compare the incidence of endoscopic surgical treatment in patients with laryngomalacia to other

Financial disclosures/conflicts of interest: none. ⁎ Corresponding author at: Division of Pediatric Otolaryngology, Cohen Children’s Medical Center, Department of Otolaryngology, Head and Neck Surgery, Hoftstra North Shore-LIJ School of Medicine, New Hyde Park, NY 11042. Fax: + 1 516 470 7550. E-mail address: [email protected] (J. Cheng). http://dx.doi.org/10.1016/j.amjoto.2015.05.009 0196-0709/© 2015 Elsevier Inc. All rights reserved.

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aerodigestive pathology in a pediatric otolaryngology practice with who may present with similar symptoms and hope to provide a frame of reference for consideration of pathology other than laryngomalacia that can respond well to endoscopic surgery and should be considered when evaluating infants with inspiratory stridor. When the stridor is associated with severe symptoms, such as dysphagia and failure to thrive, surgical management is often indicated and usually helpful in resolving symptomatology.

encountered. The average length until last follow up was 24.0 months [range 1–60 months]. In one newborn who underwent a vallecular cyst excision, the immediate perioperative period was complicated by postoperative respiratory distress, necessitating re-intubation. The patient was able to be extubated the following day, and the remainder of his hospital course was uneventful.

4. 2.

Methods

After institutional review board (IRB) approval was obtained, the operative records of both authors were reviewed between July 1, 2009 and December 22, 2014 for infants, aged 12 months or less, who underwent endoscopic surgery for failure to thrive, stridor, and/or dysphagia. Patients were identified using current procedural terminology (CPT) codes: 31535, 31536, 31540, 31541, 31560, 31561, and 31588. Infants with ICD-9 diagnoses of dysphagia (787.2x), stridor (786.1), laryngomalacia (748.3), or failure to thrive (783.41 and 779.34) and were non-syndromic, full term (>35 weeks gestation) without prior history of intubation were included. Infants with prior history of intubation and/or craniofacial abnormality were excluded. Microsurgical approach was surgeon dependent.

3.

T1 F1 F2 F3

Results

A total of 30 patients were identified, 13 (43.3%) boys and 17 (56.7%) girls (Table 1). The average age at the time of surgical intervention was 2.7 months [range 1 week to 7 months]. Endoscopic surgical management was directed at laryngomalacia in 21 (70%) (Fig. 1), vallecular cysts in 7 (23.3%) (Fig. 2), and anterior glottic webs in 2 (6.7%) (Fig. 3) patients. Surgical indications in cases of laryngomalacia, anterior glottic webs included significant feeding difficulties/dysphagia limiting oral intake or failure to thrive. All vallecular cysts were able to be diagnosed preoperatively on bedside, awake fiberoptic indirect laryngoscopy. All patients had nearly immediate resolution in the stridor and feeding difficulties. None required revision surgery, modified diets, or alternative means of enteric nutrition. All patients were able to start an oral diet within the first postoperative day. Dysphagia requiring further swallowing evaluation or therapy was not

Table 1 – Summary of newborns and infants that underwent endoscopic surgical management for inspiratory stridor. No. (%) Gender Females Males Endoscopic surgical procedures Supraglottoplasty Excision of vallecular cyst Division of anterior glottic web

17 (56.7) 13 (43.3) 21 (70) 7 (23.3) 2 (6.7)

Discussion

Inspiratory stridor in newborns and infants commonly presents to the otolaryngologist, and consideration is often directed at laryngomalacia, given the rarity of other clinical entities in these patients. Factors such as severity of symptoms, coexisting medical problems, and/or failure to thrive are calculated in the decision-making process for management recommendations. As demonstrated by Chen et al., who described seven children over a seven year experience with vallecular cysts, other lesions or pathology mimicking the inspiratory stridor of laryngmalacia are uncommon. The authors emphasized that vallecular cysts, although rare, should be considered in children with stridor and dysphagia. We also agree that awake, beside flexible fiberoptic laryngoscopy should be performed on stridolous infants and would like to emphasize that particular attention should be directed at the base of tongue and vallecula in newborns and infants to evaluate for a vallecular cyst, as this diagnosis may be missed and mistakenly attributed to laryngomalacia. Our surgical approach and outcomes also echo and confirm their success [8]. Other rare aerodigestive pathology may also present with similar symptoms to laryngomalacia. We reviewed our experience with endoscopic surgical management of infants with stridor and dysphagia to examine the presence of other aerodigestive pathology, which may present with similar and overlapping symptoms to emphasize careful consideration of awake, fiberoptic laryngoscopy evaluation of these infants. Also, in our experience and other authors’ reports, these processes may be endoscopically managed successfully with minimal associated morbidity. Inspiratory stridor in newborns and infants is often attributed to laryngomalacia, and in severe cases, benefit is achieved with supraglottoplasty. Supraglottoplasty is relatively safe with minimal associated risks [9]. Swallowing dysfunction has been reported but is transient, usually resolving within one month postoperatively [10]. We have experience using both cold steel and CO2 LASER techniques with similar results, with the chosen technique, primarily surgeon preference. For microsurgical excision of vallecular cysts, our preferred surgical approach is for complete excision, as opposed to marsupialization, with either cold steel or utilizing a bipolar radiofrequency plasma ablation (RFA) device [11]. Congenital anterior glottic webs are an infrequently encountered pathology, and our preferred surgical approach is for endoscopic lysis with cold steel and balloon dilation [7]. A complete review of the various methods and reports for management of these lesions that may present with inspiratory stridor is beyond the scope of our intended report, but we briefly introduce some of the surgical techniques utilized.

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Fig. 1 – Office-based indirect laryngoscopy demonstrating laryngomalacia (left). Operative endoscopy illustrating laryngomalacia (middle). Immediate postoperative supraglottoplasty results using cold steel (right).

Significant other limitations of our study include its retrospective nature and lack of standardized patient selection for surgical intervention. Given our observed extremely high success rate with supraglottoplasty, we may have had much more strict surgical indications when performing this procedure. However, we also excluded from study, those patients known to have higher incidence of failure, such as syndromic or hypotonic newborns and infants. We also recognize that our experience may not completely represent all of the neonatal and infant etiologies for inspiratory stridor that may present to the otolaryngologist. We have evaluated a significantly larger number of patients with the presenting symptom of stridor but did not include or examine all patients presenting to our practice with the chief complaint of stridor as a comparison group, as we wanted to focus on the cohort that was managed with endoscopic surgical techniques. Other pathology presenting with inspiratory stridor and may mimick laryngomalacia also includes but is not limited to vocal fold mobility issues, respiratory papillomas, rare upper aerodigestive lesions like hamartomas, laryngeal saccular cysts, and other base of tongue lesions, such as lingual thyroid [12–15]. Lingual thyroid is often managed medically after the diagnosis is made [16]. Unilateral vocal fold mobility in this age group may present with airway or feeding symptoms but generally, is not surgically managed [17].

Bilateral vocal fold mobility problems may present with inspiratory stridor and have been reported to be amenable to endoscopic surgical management but may often result in tracheotomy [18,19]. The incidence of other pathologies mimicking the inspiratory stridor of laryngomalacia, which may be managed by endoscopic surgical techniques, is quite uncommon, and our aim was to provide some perspective regarding etiologies that we have encountered in patients that may mimick laryngomalacia and underwent endoscopic surgical management. In our experience, other pathology accounted for almost one-third of newborns and infants undergoing endoscopic surgery.

5.

Conclusion

Laryngomalacia is often the most commonly encountered surgical indication for newborns and infants with stridor and severe clinical symptoms. Like most previous descriptions, patients responded well to supraglottoplasty. Vallecular cysts also accounted for about one-quarter of the infants treated. Clinicians should carefully consider the presence of other airway pathology, which may mimic laryngomalacia, in nonsyndromic infants without a previous history of intubation. Endoscopic surgical management may be safe and effective.

Fig. 2 – Operative endoscopy of vallecular cyst (left) and immediate postoperative results with complete excision using cold steel (right).

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Fig. 3 – Operative endoscopy of congenital anterior glottic web (left) and immediate postoperative result after lysis with cold steel and balloon dilation (right).

REFERENCES

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[10] Chun RH, Wittkopf M, Sulman C, et al. Transient swallowing dysfunction in typically developing children following supraglottoplasty for laryngomalacia. Int J Pediatr Otorhinolaryngol Nov 2014;78:1883–5. [11] Gonik N, Smith LP. Radiofrequency ablation of pediatric vallecular cysts. Int J Pediatr Otorhinolaryngol Dec 2012;76:1819–22. [12] Chan FL, Low LC, Yeung HW, et al. Case report: lingual thyroid, a cause of neonatal stridor. Br J Radiol May 1993;66:462–4. [13] Ucar S, Zorlu P, Yildirim I, et al. Hamartoma of the larynx: an unusual cause of stridor. Balk Med J Dec 2014;31:349–51. [14] Hasniah AL, Asiah K, Mariana D, et al. Congenital bilateral vocal cord paralysis. Med J Malaysia Dec 2006;61:626–9. [15] Lesnik M, Thierry B, Blanchard M, et al. Idiopathic bilateral vocal cord paralysis in infants: case series and literature review. Laryngoscope 2014. http://dx.doi.org/10.1002/lary. 25076 [Epub ahead of print]. [16] Dutta D, Kumar M, Thukral A, et al. Medical management of thyroid ectopia: report of three cases. J Clin Res Pediatr Endocrinol 2013;5:212–5. [17] Garcia-Lopez I, Penorrocha-Teres J, Perez-Ortin M, et al. Paediatric vocal fold paralysis. Acta Otorrinolaringol Esp Jul-Aug 2013;64:283–8. [18] Daniel SJ, Cardona I. Cricothyroid onabotulinum toxin A injection to avert tracheostomy in bilateral vocal fold paralysis. JAMA Otolaryngol Head Neck Surg Sep 2014;140:867–9. [19] Jabbour J, Martin T, Beste D, et al. Pediatric vocal fold immobility: natural history and the need for long-term followup. JAMA Otolaryngol Head Neck Surg May 2014;140:428–33.

Endoscopic surgical management of inspiratory stridor in newborns and infants.

Compare the incidence of endoscopic surgical treatment of patients with laryngomalacia to other aerodigestive pathology who may present with similar s...
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