Treatment Outcome of Supraglottoplasty Vs. Wait-And-See Policy in Patients with Laryngomalacia

Eur Arch Otorhinolaryngol DOI 10.1007/s00405-016-3943-3

LARYNGOLOGY

Treatment outcome of supraglottoplasty vs. wait-and-see policy in patients with laryngomalacia

1,2 1 1 Martijn van der Heijden • Frederik G. Dikkers • Gyorgy B. Halmos

Received: 20 October 2015 / Accepted: 18 February 2016 Ó The Author(s) 2016. This article is published with open access at Springerlink.com

Abstract In most cases, laryngomalacia presents as a laryngomalacia (38.5 weeks vs. 14.5, p = 0.043). Supra- mild disease, and the symptoms resolve after wait-and-see glottoplasty is safe and effective in treatment of severe policy. Up to 20 % of patients present with severe laryn- laryngomalacia. SALs and comorbidities are frequently gomalacia and may require surgery (i.e. supraglottoplasty); found in patients with laryngomalacia and are responsible however, the indication for surgery is not firmly established for longer onset of complaints. yet. The goal of this study is to determine whether supraglottoplasty results in a better outcome than wait-and-see Keywords Laryngomalacia Á Supraglottoplasty Á and to investigate how different comorbidities influence Synchronous airway lesions Á Wait-and-see outcome. A retrospective study of pediatric cases of in a tertiary referral center was performed. Photo and video documentation was available and revised in all cases. Introduction Electronic and paper charts were reviewed for the following variables: gender, sex, gestational age, birth weight, Congenital laryngomalacia is the most common etiology symptoms, comorbidity, date of endoscopy, severity and causing stridor in infants with a reported incidence of type of laryngomalacia, treatment modality and technique 35–75 % of patients [1]. Supraglottic structures collapse and follow-up data and a total 89 patients were included. into the airway during inspiration, causing airway Supraglottoplasty was found to lead to significantly faster obstruction. In most cases laryngomalacia presents with complete improvement of laryngomalacia than wait-and- inspiratory, sometimes with in- and expiratory, stridor. The see policy (5 weeks vs. 29, p = 0.026). Synchronous air- stridor typically worsens with feeding, crying, supine way lesions (SALs) were present in 40.4 % of patients and positioning, agitation and exercise. Typically, symptoms were associated with prolonged symptoms of are not present at birth, start within the first 4 months of life and peak at an age of 4–8 months [2]. The most common associated symptoms are swallowing dysfunction, Electronic supplementary material The online version of this article (doi:10.1007/s00405-016-3943-3) contains supplementary regurgitation, cough, sleep-disordered breathing [1, 3]. An material, which is available to authorized users. estimated 40 % of infants presenting with laryngomalacia have mild and 40 % have moderate symptoms and can be & Gyorgy B. Halmos treated conservatively, spontaneously resolving at the age [email protected] of 12–24 months [1]. Up to 20 % of laryngomalacia 1 Department of Otorhinolaryngology/Head and Neck Surgery, patients present with severe laryngomalacia. Severe University of Groningen, University Medical Center laryngomalacia is not likely to cure spontaneously, and is Groningen, P.O. Box 30.001, 9700 RB Groningen, more likely to require supraglottoplasty [4]. The Netherlands Laryngomalacia is a self-limiting disease. Nowadays, 2 Present Address: Department of Head and Neck Oncology treatment consists of either wait-and-see or endoscopic and Surgery, The Netherlands Cancer Institute-Antoni van Leeuwenhoek, Plesmanlaan 121, 1066 CX Amsterdam, surgery (supraglottoplasty). Previous studies have shown The Netherlands that endoscopic surgical treatment is of additional value in 123 Eur Arch Otorhinolaryngol the treatment of laryngomalacia [5, 6]. The experiences treatment, postoperative outcome, time from treatment with complications after supraglottoplasty are controversial until free of symptoms, comorbidity and genetic disorders. in the current literature. Studies related failure and com- When laryngomalacia was suspected, diagnosis was plications directly to comorbidities [7, 8], while other confirmed by flexible fiberoptic laryngoscopy under gen- studies have shown otherwise [5, 9, 10]. Various supra- eral anesthesia. Patients were breathing spontaneously glottic structures can be affected in cases with laryngo- throughout the procedure and evaluation of the airway was malacia, as described in the Groningen Laryngomalcia performed, including assessment of synchronous airway Classification System [11]. Most common findings are: lesions. Rigid endoscopy was not performed routinely, only inward collapse of arytenoids cartilages (type 1), medial on indication (e.g. therapeutic procedure). A division of displacement of aryepiglottic folds (type 2) and postero- severity was made on clinical presentation and endoscopic caudal displacement of epiglottis against the posterior findings [7]. Severe laryngomalacia was established if pharyngeal wall (type 3). Demonstrative video material of complaints included severe feeding difficulties, failure to these three types are available in the online version of the thrive, cyanosis or intercostal and/or xyphoidal retractions manuscript (Videos 1–3). Most patients show combinations or endoscopy showed significant dynamic airway of these dynamic abnormalities. The type of laryngoma- obstruction. lacia determinates the type of supraglottoplasty; type 1 Reasons for supraglottoplasty were severe clinical pre- laryngomalacia necessitate removing redundant supra-ary- sentation or insufficient spontaneous resolving of symp- tenoid tissue, type 2 incision of the shortened aryepiglottic toms during exerting a wait-and-see policy. folds, and type 3 epiglottopexy [11]. These surgical inter- Supraglottoplasty consisted of resection of supra-ary- ventions can be combined. tenoidal redundant mucosa, incision of shortened The primary purpose of the present study is to evaluate aryepiglottic folds, epiglottopexy or a combination of the best treatment policy in the treatment of laryngomala- these. Both removal of supra-arytenoidal redundant cia by comparing the short and long-term outcome of mucosa and incision of the aryepiglottic folds were per- patients with moderate and severe laryngomalacia treated formed with either cold steel instrumentation or (prefer- by either supraglottoplasty or wait-and-see policy. The ably) CO2 laser (0.8–1.5 W, superpulse, continuous mode, secondary aim was to identify predictors of treatment spot size 200 lm). Epiglottopexy consisted of de-epithe- outcome. lialization of the vallecula and suturing the lingual side of the epiglottis to the base of the tongue. Airway management consisted of either orotracheal intubation whether or Materials and methods not with interrupted apnea, or (supraglottic) high frequency jet ventilation (SHFJV). All patients diagnosed with laryngomalacia between Jan- Post-operative care depended on severity of preopera- uary 2002 and December 2012, less than 5 years of age at tive symptoms. It usually consisted of immediate extuba- the time of diagnosis, were included. All patients were tion and one night observation with cardiorespiratory treated at the Department of Otorhinolaryngology, Head monitoring. No preventive antibiotic treatment was and Neck Surgery of the University Medical Center administered. Occasionally patients were treated with Groningen, The Netherlands, a tertiary referral center. dexamethasone (0.5–1 mg/kg). If no events occurred dur- Patients were excluded if they underwent previous surgery ing observation patients were discharged the day after for other airway problems; if there was insufficient amount surgery. of data for analysis; or when reviewing the endoscopic Standard follow-up was performed 6 weeks after initial documentation revealed another diagnosis than laryngo- endoscopy. The frequency of further controls was malacia. Imaging material was reviewed by the first author depending on the complaints. Patients with no complaints (MvdH) under supervision of the two senior authors (FGD were dismissed from regular follow-up. If no/insufficient and GBH). improvement or progression of the symptoms occurred A retrospective chart view of patients with laryngoma- after wait-and-see or supraglottoplasty, a second diagnostic lacia was performed. Electronic patients’ charts were pri- endoscopy was performed (median 43 weeks; range marily reviewed, and paper charts were reviewed if 260 weeks). If laryngomalacia was still apparent after wait- additional data were needed. The following variables were and-see, this was called residual disease. If laryngomalacia collected: gender, gestational age, birth weight, age at was still apparent after supraglottoplasty, it was called diagnosis, presenting symptoms, severity of disease, type recurrent disease. During the last control, severity of of laryngomalacia (according to GLCS [11]), age at symptoms was divided into no improvement, partial endoscopy, synchronous airway lesions, performed improvement or complete improvement.

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Statistical analysis was performed using SPSS version Table 1 Patient characteristics of 89 patients treated for 20.0. For comparison between groups we used the Mann– laryngomalacia Whitney U test. For correlations the Spearman test was Age in days, median (range) performed. Statistical significance was set at p \ 0.05. Presentation of symptoms 60 (0–1691) Diagnosis with endoscopy 101 (1–1755) Sex, n (%) Results Male 57 (64) Female 32 (36) A total of 100 patients met our inclusion criteria, based on Gestational age in weeks. Mean (SD) 38 ? 4(2? 5) the registered diagnosis. After analysis of data 11 patients Birth weight in grams. Mean (SD) 3168.6 (850) were excluded. Ten patients were excluded because of Presenting symptoms, n (%) misdiagnosis after reviewing photo/video documentation Stridor 64 (72) and OR-reports [bilateral vocal cord paralysis (N = 5), Dyspnea 24 (27) laryngeal web (N = 1), luxated arytenoids (N = 1), ret- Apnea 17 (19) rognatia (N = 1) and tracheamalacia (N = 2)]. One patient Cyanosis 18 (20) was excluded due to lack of data to objectify laryngoma- Feeding problems 12 (13) lacia. In total, 89 patients remained for statistical analysis. Failure to thrive 5 (6) Table 1 shows characteristics of the study population Respiratory distress 11 (12) and Table 2 describes all treatment characteristics. Retractions 15 (17) Comorbidity was found in 60 patients (67 %). The most Other 3 (3) commonly found comorbidity was synchronous airway lesions (SALs) with 36 cases (40 %), showing 54 airway Severity of Laryngomalacia, n (%) lesions: several patients had more than one synchronous Mild 36 (40.4) airway lesion. Table 3 shows the different synchronous Moderate 21 (23.6) airway lesions found in this population. Cardiac comor- Severe 32 (36.0) bidity was found in 12 patients (13.5 %), neurologic Total 89 (100) comorbidity in 8 patients (9.0 %), chromosomal/syndromal Comorbidities, n (%) a disease in 23 patients (25.8 %) and other comorbidities Synchronous airway lesions 36 (40.4) b were found in 10 patients (11.2 %). Cardial 12 (13.5) c Presence of any comorbidity was related to more severe Neurological 8 (9.0) d form of laryngomalacia (p = 0.003). However, no dis- Syndrome/genetic disorders 23 (25.8) tinction could be made between different types of comor- Othere 10 (11.2) bidities [SAL (p = 0.266), cardiac (p = 0.115), neurologic None 29 (32.6) (p = 0.247) or syndromal/genetic (p = 0.026)]. The co- a See Table 2 moborbidities showed no correlation with performed b Tetralogy of Fallot (n = 2), ventricular septum defect (4), trans- treatment. Gestational age, sex, birth weight and age at position of the great vessels (1), bicuspid aorta valves (1), atrioven- onset showed no correlations with performed treatment, tricular septum defect and AVSD with pulmonary hypertension (2) or severity of disease or length of disease. Table 4 shows the with overriding aorta (1) c influence of comorbidity on the median time to complete Epilepsy (n = 3), cerebral infarction (1), cerebral cyst (1), Hirschsprung’s disease (1), gastroparesis and autism (1) improvement. Patients with SALs needed a statistically d Down syndrome (n = 5). Pierre Robin (3), CHARGE (3), Door significant longer time for complete improvement syndrome (2), West syndrome (2), congenital myasthenia syndrome (p = 0.043). Although all factors showed difference in (1), Rubinstein Taybi syndrome (1), Freeman-Sheldon syndrome (1), median time to complete improvement, no other factors Goldenhar syndrome (1), Perlman syndrome (1), macrocephaly-cap- were found to have significant influence on the time to illar malformation syndrome (1), Q7 syndrome and an unknown mitochondrial syndrome (1) complete improvement. Gender, age at onset, gestational e Hydronephrosis (n = 2), pylorus hypertrophy (1), nasal teratoma age and birth weight was not related to laryngomalacia (1), polycystic kidney disease (1), thyroglossal cyst (1), congenital severity or time to complete improvement. Multivariate cataract (1), reflux and hernia umbilicalis combined with phimosis (1) analysis could not be performed due to low number of cases. Only one patient of patient treated with supraglottoplasty (7.1 %) had recurrent disease after supraglotto- Recurrent disease presented with contralateral type 1 plasty. During primary supraglottoplasty of this patient, laryngomalacia. During revision supraglottoplasty redun- redundant mucosa of only one arytenoid cartilage was dant mucosa over the arytenoid cartilage of the contralat- removed, based on asymmetric type 1 laryngomalacia. eral side was removed. In total 65 (73 %) patients were 123 Eur Arch Otorhinolaryngol

Table 2 Treatment characteristics of 89 patients treated for performed with CO2 laser and 3 (13.6 %) with cold steel laryngomalacia instruments. Primary treatment (1st endoscopy), n (%) Table 5 summarizes the follow up data. Follow-up data Wait-and-see 75 (84.3) were available in 62 patients (70 %). Complete improve- Endoscopic surgery 14 (16.5) ment was registered in 30 patients (34 % of total group). In Tracheostomy 0 (0.0) total seven patients died during their follow-up. None of Recurrent/residual disease (2nd endoscopy), n (%) them died due to laryngomalacia: all died of other severe After wait-and-see 13 (17.3) comorbidities. Two patients received a tracheostomy. One After surgery 1 (7.1) of them underwent tracheostomy because the laryngoma- Treatment of recurrent/residual disease, n (%) lacia did not improve and supraglottoplasty was not a Wait-and-see 3 (21.4) standard procedure in our institution yet. However, the other tracheostomy was not related to laryngomalacia, but Endoscopic surgery 9 (64.3) was performed because of severe tracheamalacia. This was Tracheostomy 2 (14.3) the reason that this patient did not undergo previous Final treatment, n (%) supraglottoplasty. Wait-and-see 65 (73.0) Table 6 demonstrates the complete improvement in Endoscopy 22 (24.7) weeks in patients with different treatment and severity of Tracheostomy 2 (2.2) disease. All patients, who underwent supraglottoplasty, Used technique, n (%) achieved complete improvement within 6 weeks, except CO laser 17 (77.3) 2 three patients who achieved complete improvement in 31, Cold steel 3 (13.6) 57 and 58 weeks. Supraglottoplasty showed significant Epiglottopexy 2 (9.1) reduction of time to achieve complete improvement in the Complications 0 whole population (p = 0.026). However, no statistically significant difference was seen among the different sever- Table 3 Distribution of synchronous airway lesions (SALs) in all ities of laryngomalacia, very likely due to insufficient patients with laryngomalacia and SAL (n = 36) numbers. Groups, wait-and-see and supraglottoplasty, Synchronous airway lesion n (%) matched for gender, age at onset, birth weight and gesta- Number of patients 36 (100) tional age. Tracheamalacia 14 (39) Pharyngomalacia 13 (36) Bronchomalacia 9 (25) Discussion Tracheal/subglottic stenosis 7 (19) Micrognathia 7 (19) Laryngomalacia is a benign and self-limiting disease. The present study confirms the additional value of supraglot- Othera 4 (11) toplasty in the treatment of laryngomalacia. We have Total SALs 54 shown for the first time that supraglottoplasty shortens the a Laryngeal web (2), bilateral vocal cord paralysis (1) and missing length of symptomatic disease, compared to wait-and-see lung lobe (1) policy. This study also shows that comorbidity, especially Table 4 Median time in weeks to achieve complete improvement synchronous airway lesions, is associated with the severity related to comorbidities in patients with laryngomalacia, regardless of of disease. Comorbidity tends to negatively influence the treatment modality used resolution of symptoms. Present (n) Not present (n) p Comorbidity was found in two-thirds of our patients (67 %). According to literature comorbidity is present in Comorbidities 31.0 (23) 15.0 (7) 0.116 around 25–47 % of patient with laryngomalacia, disre- Cardial 51.0 (5) 26.0 (25) 0.373 garding synchronous airway lesions (SALs) [8, 12, 13]. Neurologic 40.0 (5) 26.0 (25) 0.845 The higher incidence of comorbidity in our practice is very Syndromal 45.0 (6) 25.5 (24) 0.243 likely due to the fact, that we also counted SALs as SAL 38.5 (18) 14.5 (12) 0.043 comorbidities and also probably because of the higher ratio Bold value indicates statistically significant p value (p \ 0.05) of complex patients in our academic setting. Although no other studies directly compared comorbidities with time to treated with a wait-and-see policy, 22 (24.7 %) with complete improvement, comorbidity is found to be risk supraglottoplasty and 2 (2.2 %) with tracheostomy. Of the factors for supraglottoplasty failure [12]. Preciado et al. patients undergoing supraglottoplasty 17 (77.3 %) were showed in their review that comorbidity is associated with 123 Eur Arch Otorhinolaryngol

Table 5 Outcome of performed Final treatment Total treatment in complete group of 62 patients with Supraglottoplasty Wait-and-see Tracheotomy laryngomalacia, depending on ﬁnal treatment (n (%)) Deceaseda 2507 No improvement 1 (4,5) 2 (5.3) 1 4 Partial improvement 9 (40.1) 12 (31.6) 0 21 Complete improvement 10 (45.4) 19 (50.0) 1 30 Total 22 (100) 38 (100) 2 62 a Causes of death (all causes occurred once): kidney cancer, epilepsy, tracheamalacia, Freeman-Sheldon syndrome, sepsis, unidentiﬁed syndrome, unknown (but not suspicious for laryngomalacia)

Table 6 Median time in weeks (n) to achieve complete improve- improvement, but this correlation was not statistically ment, depending on severity of disease, and depending on treatment applied significant (p = 0.373). Neurologic impairment is thought to influence laryn- Laryngomalacia Wait-and-see Supraglottoplasty p gomalacia severity and outcome [2, 8, 12, 16, 17, 24, 25]. Mild 32 (13) – All these studies related neurologic comorbidity to Moderate 53 (3) 3 (1) 0.180 increased chance of revision supraglottoplasty and tra- Severe 10 (3) 6 (9) 0.778 cheostomy. However, we cannot relate neurologic comor- All severities 29 (19) 5 (10) 0.026 bidities to worse outcome in our series, as the patient with revision supraglottoplasty and the two tracheostomy cases p p Bold value indicates statistically significant value ( \ 0.05) did not have any neurologic conditions. a more than sevenfold increased risk of surgical failure. The reported incidence of syndromal and genetic disor- Furthermore, otherwise healthy patients have significantly ders ranges from 8 to 20 % [1]. Down syndrome, CHARGE lower risk of post supraglottoplasty aspiration [14, 15]. and Pierre Robin are the most frequently seen disorders in Durvasula et al. related especially neurologic and syn- literature and also in our population [8]. However, the sig- dromic comorbidities with a supraglottoplasty failure [16]. nificance of the presence of syndromal/genetic disorders has We noted relation between the severity of laryngomalacia not been well examined. Syndromes associated with ret- and occurrence of comorbidity in general. Furthermore, rognathia, like CHARGE and Pierre Robin sequence, are there also seems to be a negative relation between more likely to fail supraglottoplasty [8, 16, 26]. Our data comorbidity and time to achieve complete improvement; show that syndromal/genetic disorders are related to the however, this was not statistically significant (p = 0.116). severity of disease. Our data show a trend that patients with The clinical significance of SALs in patients with laryn- syndromal/genetic disorders need more time to achieve gomalacia is a rather controversial subject. Studies reporting complete improvement than other patients (p = 0.243). a high incidence of SALs suggest that SALs are related to We found only one patient with gastroesophageal reflux disease severity and patients with SALs are therefore more disease (GERD), where the reported incidence is between likely to require surgery [17, 18]. Our results suggest that the 65 and 100 % [1, 27, 28]. A systematic review of 27 presence of SALs in patients with laryngomalacia is asso- studies showed coexistence between GERD and laryngo- ciated with worse long-term outcome but it is not associated malacia. However, due to limited evidence no causal to the severity of disease. On the other hand some studies connection could be made [27]. Due to the retrospective report no clinical significance of SALs with no influence on nature of the underlying study it is hard to determine time to complete improvement [19, 20], and no association whether reflux was not present. However, video and photo between severity and SALs [21]. The most common repor- documentation showed no convincing signs of GERD. ted SAL in our cohort is tracheamalacia (39 %). This is in Studies reporting high incidence lack a control group, thus agreement with other studies [7, 22]. the clinical significance of the presence of GERD has not Prevalence of cardiac comorbidity ranges between 10 been well established [27]. Most probably, considering and 17.9 % in patients with laryngomalacia in the literature literature data, GER and not GERD was present in our [2, 8, 23]. Patients with cardiac comorbidities have a higher cohort but not documented. The presence of GERD and the chance of revision supraglottoplasty or tracheostomy and clinical significance of it should be further examined. are associated with increased severity [8]. This could not A noteworthy finding in our study is the low amount of be confirmed by our data. However, cardiac impairment revision supraglottoplasty, tracheostomies and complica- was of negative influence on time to achieve complete tions. Hoff et al. described a higher rate of revision

123 Eur Arch Otorhinolaryngol supraglottoplasty (15.2 vs. 47.8 %) and tracheostomy Compliance with ethical standards depending on the presence of comorbidity (0 vs. 39.1 %) Conflict of interest None. [8]. Other studies showed a range of 4.4–34.6 % of patients required either revision supraglottoplasty or tracheostomy Open Access This article is distributed under the terms of the after primary supraglottoplasty [12, 29, 30]. Although Creative Commons Attribution 4.0 International License (http://crea complications are rare [31], supraglottic stenosis is a tivecommons.org/licenses/by/4.0/), which permits unrestricted use, known complication of supraglottoplasty with a reported distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a incidence of 3.7 % [12]. link to the Creative Commons license, and indicate if changes were Most of our patients (77.3 %) were operated with CO2 made. laser, with superpulse mode, which is comparable with other authors [8, 29]. Superpulse mode allows the tissue to cool down between two impulses, therefore minimizing tissue damage. Rastatter et al. used both CO2 laser and cold References instruments techniques. Only in the CO2 laser supraglottoplasty group three patients needed tracheostomy [30], sug- 1. 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