Stenting versus stentless repair for bilateral choanal atresia: A systematic review of the literature.

BACKGROUND: Bilateral choanal atresia requires prompt surgical intervention. Surgeons have historically used stents in the repair process, however their efficacy has come into question in recent years. We performed a systematic review to investigate, primarily, whether stents enjoy more favourable outcomes compared to stentless repair. We also explored the use of operative adjuncts, such as steroids, antibiotics, mitomycin C and KTP laser. METHODS: We performed a search of the Medline and Embase databases using a search strategy developed with the assistance of an academic librarian. Only full peer reviewed articles were included. Abstracts, posters, case reports and proceedings of academic conferences were excluded. RESULTS: We identified 48 unique articles for inclusion, composed of a meta-analysis, two randomised control trials and 45 case series. Pooled analysis of the two randomised control trials yielded no statistically significant difference in choanal patency between stented and stentless repair, but a statistically significant reduction in complications, specifically granulation tissue formation, was found in stentless repair. Data from case series were, overall, of mixed quality, making factors contributing to successful outcomes difficult to elucidate. CONCLUSION: Overall, there is a lack of high quality evidence to support the use of either a stented or stentless approach to bilateral choanal atresia repair, however stentless repair may experience fewer complications. Operative techniques, such as the use of mucosal flaps, are worthy of future study. Authors call for future high quality randomised control trials to investigate this uncommon but important condition.

A Systematic Review of Interventions for Balance Dysfunction in Patients With Vestibular Schwannoma.

OBJECTIVE: Dizziness is a major contributing factor to poor quality of life for patients with vestibular schwannoma (acoustic neuroma). We wished to review the literature on interventions for balance dysfunction in these patients. DATA SOURCES: A systematic literature review was performed identifying studies that measured balance function before and after treatment for vestibular schwannoma. Data sources include Medline (1950-present), EMBASE (1974-present), Cochrane Library (issue 3, 2008), NHS Centre of reviews and dissemination, Clinical Evidence, Cochrane central register of controlled trial, and CINAHL. STUDY SELECTION: A minimum follow-up of 6 months was required, to estimate long-term balance function. Eight articles were identified, including five studies with surgical intervention, two studies with stereotactic radiotherapy, and one comparing the two. Study design was generally poor with a high risk of bias. These studies all utilized the Dizziness Handicap Inventory (DHI) as a measure of pre- and postintervention balance function. DATA EXTRACTION: Results showed that overall DHI scores are not statistically affected by intervention irrespective of modality (surgery or stereotactic radiotherapy). Patients selected with severe dizziness, who undergo surgery, improved postoperatively. No other studies for severe dizziness were noted for comparison. CONCLUSIONS: Age, sex, and tumor size have no statistically significant effect on DHI outcomes, and no evidence to suggest which treatment modality has better dizziness related outcomes.No specific treatment modality was superior in terms of long-term balance function. Patients with severe dizziness may benefit from surgery, although no comparator studies were identified.

Subglottic acute lymphoblastic leukaemia.

Acute lymphoblastic leukaemia (ALL) is one of the the most common malignancies of childhood and can occasionally present as acute airway obstruction. We present the unusual case of a 1-year-old boy who was referred to our Paediatric Otolaryngology (ENT) clinic with a recurrent history of croup. This is the first reported case of localised ALL presenting as a subglottic mass in a paediatric patient. It highlights the need to have a broader differential diagnosis in children presenting with 'recurrent croup' including extramedullary presentation of leukaemia and to have a low threshold for performing endoscopy in such cases.

Spontaneous pneumomediastinum.

Spontaneous pneumomediastinum is the presence of free air within the mediastinum that is not associated with trauma. It is a rare, self-limiting condition that can cause widespread subcutaneous surgical emphysema. We present the case of a 12-year-old boy who presented with widespread spontaneous surgical emphysema and pneumomediastinum, with no history of trauma or respiratory tract disease. We discuss our assessment of him and management with our multidisciplinary team (MDT), and whether radiological investigations including CT are helpful in such cases. In conclusion, we hypothesised that the child's coughing was the most likely cause of the tracheal rupture leading to the spontaneous surgical emphysema. Spontaneous pneumomediastinum in the paediatric patient is extremely rare. This case highlights how spontaneous pneumomediastinum can be successfully managed conservatively, and how MDT input can be helpful in guiding management in such unusual cases.

Establishment of a normative cepstral pediatric acoustic database.

IMPORTANCE: Few studies have used objective measures to evaluate the development of the normal pediatric voice. Cepstral analysis of continuous speech samples is a reliable method for gathering acoustic data; however, it has not been used to examine the changes that occur with voice development. OBJECTIVE: To establish and characterize acoustic patterns of the normal pediatric voice using cepstral analysis of voice samples from a normal pediatric voice database. DESIGN, SETTING, AND PARTICIPANTS: Cross-sectional study of 218 children aged 4 to 17 years, for whom English was the primary language spoken at home, conducted at a pediatric otolaryngology practice and pediatric practice in a tertiary hospital (April 2012-May 2014). INTERVENTIONS AND EXPOSURES: Sustained vowel utterances and continuous speech samples (4 Consensus Auditory-Perceptual Evaluation of Voice [CAPE-V] and 2 sentences from the rainbow passage) were recorded and analyzed from children with normal voices. MAIN OUTCOMES AND MEASURES: Normal values were collected for the acoustic measures studied (ie, fundamental frequency, cepstral peak fundamental frequency, cepstral peak prominence [CPP], low-to-high spectral ratio [L/H ratio], and cepstral-spectral index of dysphonia in recorded phrases) and compiled into a normative acoustic database. RESULTS: Significant changes in fundamental frequency were observed with a distinct shift in slope at ages 11 and 14 years in boys for sustained vowel (ages 4-11 years, -6.83 Hz/y [P < .001]; 11-14 years, -27.62 Hz/y [P < .001]; and 14-17 years, -5.68 Hz/y [P = .001]), all voiced (ages 4-11 years, -4.19 Hz/y [P = .002]; 11-14 years, -29.42 Hz/y [P < .001]; and 14-17 years, -4.63 Hz/y [P < .001]), glottal attack (ages 4-11 years, -4.51 Hz/y; 11-14 years, -27.23 Hz/y; and 14-17 years, -1.70 Hz/y [P < .001 for all]), and rainbow (ages <14 years, -20.68 Hz/y [P < .001]; and 14-17 years, -4.50 Hz/y [P = .001]) recordings. A decreasing linear trend in fundamental frequency among all recordings (vowel, all voiced, easy onset, glottal attack, plosives, and rainbow) was found in girls (-2.56 Hz/y [P < .001], -3.48 Hz/y [P < .001], -2.82 Hz/y [P < .001], -3.49 Hz/y [P < .001], -2.30 Hz/y [P < .001], and -2.98 Hz/y [P = .01], respectively). A linear increase in CPP was seen with age in boys, with significant changes seen in recordings for vowel (0.10 dB/y [P = .05]), all voiced (0.2 dB/y [P < .001]), easy onset (0.13 dB/y [P < .001]), glottal attack (0.12 dB/y [P < .001]), plosives (0.15 dB/y [P < .001]), and rainbow (0.17 dB/y [P = .006]). A significant linear increase in CPP for girls was only seen in all voiced (0.13 dB/y [P < .001]). L/H ratio showed a linear increase with age among all speech samples (vowel, all voiced, easy onset, glottal attack, plosives, and rainbow) in boys (1.14 dB/y [P < .001], 0.92 dB/y [P < .001], 1.19 dB/y [P < .001], 0.79 dB/y [P < .001], 0.69 dB/y [P < .001], and 0.54 dB/y [P = .002], respectively) and girls (0.96 dB/y, 0.60 dB/y, 0.75 dB/y, 0.37 dB/y, 0.44 dB/y, and 0.58 dB/y, respectively [P ≤ .001 for all]). CONCLUSIONS AND RELEVANCE: This represents the first pediatric voice database using frequency-based acoustic measures. Our goal was to characterize the changes that occur in both male and female voices as children age. These findings help illustrate how acoustic measurements change with development and may aid in our understanding of the developing voice, pathologic changes, and response to treatment.

Type 1 laryngeal cleft: a multidimensional management algorithm.

IMPORTANCE: Early diagnosis and assessment in children with type 1 laryngeal cleft are essential in preventing aspiration and associated comorbidity. Appropriate use of conservative and surgical interventions in an evidence-based management strategy can improve overall outcome. OBJECTIVE: To evaluate the management of care for children with type 1 laryngeal cleft in our practice and develop an updated management algorithm. DESIGN, SETTING, AND PARTICIPANTS: We performed a review of medical records at a tertiary pediatric aerodigestive center. During a period of 7 years (July 18, 2005, to July 18, 2012), 1014 children younger than 18 years were evaluated for aspiration, choking, cough, or recurrent pneumonia. Of these, 44 children (4.3%) had a type 1 laryngeal cleft. Two were lost to follow-up; thus, 42 children were included in our final sample (28 males, 14 females). INTERVENTIONS: The care of 15 patients (36%) was managed conservatively, and 27 patients (64%) underwent endoscopic surgical repair of their laryngeal cleft. MAIN OUTCOME AND MEASURE: Assessment of our current management strategy. RESULTS: Success was defined as improving when a child was able to tolerate a feeding without aspirating or resolved when the child had transitioned to tolerating thin liquids. All patients received a trial of conservative therapy. Fifteen of the 42 patients (36%) had an anatomic cleft and were able to maintain the feeding regimen; thus, conservative treatment was successful in this group. The remaining 27 patients (64%) received surgical intervention. Overall operative success rate was 21 of the 27 patients (78%). The age of the child (P < .01) and comorbid conditions (P < .001) affected the outcomes of conservative measures and surgical repair. Only 6 patients did not demonstrate resolution, 5 of whom had significant comorbidities. CONCLUSIONS AND RELEVANCE: Age, comorbidity status, severity of aspiration, and the ability to tolerate a feeding regimen should be taken into account when deciding on conservative or surgical management for children with a type 1 laryngeal cleft. A clinical pathway for conservative and surgical management is presented.

Prevalence of eosinophilic esophagitis in children with refractory aerodigestive symptoms.

IMPORTANCE: Eosinophilic esophagitis (EoE) is an increasingly important diagnosis for children; it has a remarkable impact on their quality of life and can present with aerodigestive symptoms commonly evaluated by otolaryngologists. OBJECTIVES: To evaluate the prevalence of EoE in children presenting to a pediatric aerodigestive clinic, to describe their presentation, and to review the role of subsequent food allergy evaluation and treatment. DESIGN: Review of a prospective database. SETTING: Tertiary pediatric multispecialty aerodigestive center. PATIENTS: Children with aerodigestive symptoms refractory to medical treatment who underwent direct laryngoscopy with rigid or flexible bronchoscopy and esophagoscopy with or without pH probe study. MAIN OUTCOMES AND MEASURES: Diagnosis of EoE. RESULTS: Between 2003 and 2012, 376 of 1540 children seen in the center (mean [range] age, 4.54 [0-18.6] years; male to female ratio, 1.72:1) remained symptomatic despite medical therapy and thus underwent triple endoscopic evaluation. Of the 376 children, 14 (3.7%) were eventually diagnosed as having EoE, as defined by 15 or more eosinophils per high-power field on esophageal biopsy and either a negative pH study result or nonresponse to a trial of high-dose proton pump inhibitors. The subpopulation with EoE presented with airway symptoms and diagnoses, most commonly cough (n = 6; 42.9%). Inflammatory subglottic stenosis due to EoE was identified in 1 patient. Of the 14 children with EoE, 6 presented with gastrointestinal symptomatology, most commonly choking or gagging. Subsequent treatment including food allergy challenge and elimination diet resulted in a clinical improvement in half of the cases identified. CONCLUSIONS AND RELEVANCE: This represents the largest multispecialty clinic epidemiologic study evaluating the prevalence of EoE in children presenting not strictly with gastrointestinal symptoms but rather with aerodigestive symptoms that are frequently evaluated by pediatric otolaryngologists. Although the prevalence is low, EoE should be considered for children with appropriate symptoms in whom other medical therapies fail.

The utility of histopathology in identifying structural differences among layers of the lamina propria.

OBJECTIVE: The purpose of this study is to assess the modality of laryngeal histopathology in identifying 1, 2, or 3 layers in the lamina propria. METHODS: Blinded analysis was performed, with a set of histophathologic slides where the magnification and localized regions shown were all standardized. Two senior pathologists with experience reviewing laryngologic histophathology were asked to assess whether the vocal fold lamina propria they evaluated contained 1, 2, or 3 layers. Their ability to accurately assess this was calculated. RESULTS: The first pathologist correctly identified 13 of 25 (52%) specimens. The second identified 19 of 25 (76%) specimens after receiving gold referents before the test. No significant difference was seen between the pathologists' interpretations (McNemar test; p = 0.0833). CONCLUSION: Our results show the difficulty of using histopathology to distinguish layers in the lamina propria even when the reviewers are senior pathologists. These findings imply that more objective modalities for such analyses may be beneficial.

Consistency of voice frequency and perturbation measures in children using cepstral analyses: a movement toward increased recording stability.

IMPORTANCE: Few studies have evaluated the pediatric voice objectively using acoustic measurements. Furthermore, consistency of these measurements across time, particularly for continuous speech, has not been evaluated. OBJECTIVES: (1) To evaluate normal pediatric voice frequency and perturbation using both time-based and frequency-based acoustic measurements, and (2) to determine if continuous speech samples facilitate increased recording stability. DESIGN Prospective, longitudinal study. SETTING: Pediatric otolaryngology practice within a tertiary hospital. PARTICIPANTS: Forty-three children, ages 4 to 17 years. INTERVENTION OR EXPOSURE: Sustained vowel utterances and continuous speech samples, which included 4 Consensus Auditory-Perceptual Evaluation of Voice (CAPE-V) sentences and the first sentence of the "rainbow passage" ("A rainbow is a division of white light into many beautiful colors that takes the shape of a long round arch, with its path high above and its 2 ends apparently beyond the horizon") were obtained at 2 time points. MAIN OUTCOME AND MEASURE: Intraclass correlation coefficients (ICCs) were calculated to assess reliability between speech samples. RESULTS: Fundamental frequency of sustained vowel utterances had excellent reliability (ICC ≥ 0.94). Time-based analyses of perturbation in sustained vowel utterances demonstrated poor reliability (ICC < 0.40), while frequency-based analyses of perturbation for these utterances demonstrated good to excellent reliability (ICC > 0.40). Fundamental frequency of continuous speech sample had excellent reliability (ICC > 0.94). Frequency-based analyses of continuous speech samples demonstrated excellent reliability (ICC > 0.75) for all but 1 variable, which demonstrated good reliability (cepstral-spectral index of dysphonia of the all voiced sample; ICC =0.72). CONCLUSIONS AND RELEVANCE: Sustained vowel utterance and continuous speech samples provide consistent measures of fundamental frequency. Frequency-based analysis of sustained vowel recordings improves the reliability of perturbation measures. Continuous speech recordings can be obtained in children and demonstrate good to excellent reliability across recordings. This suggests that frequency-based analysis of continuous speech may be more representative of a child's voice and therefore may be of use in the study both of the developmental changes of the pediatric voice as well as the study of vocal changes pretreatment and posttreatment in children with voice disorders.

Use of adjuvant intralesional bevacizumab for aggressive respiratory papillomatosis in children.

IMPORTANCE: Juvenile recurrent respiratory papillomatosis (RRP) can be an aggressive disease process necessitating frequent trips to the operating room with multiple anesthetics for tumor debulking and airway preservation. Adjuvant therapy, such as that which is reported in this article, may help reduce the number of operative procedures affected children need each year and therefore may also affect their overall quality of life (QOL). OBJECTIVE: To describe our experience with intralesional bevacizumab (Avastin) treatment for children with severe RRP by comparing median number of surgical procedures per year, median duration of time between procedures, Derkay staging, and voice QOL before and after bevacizumab treatment. DESIGN: Prospective, consecutive case series. SETTING: Tertiary care aerodigestive center. PARTICIPANTS: Ten children, aged 18 months to 18 years, with severe RRP necessitating more than 4 operative interventions in 1 year whose parents (or legal guardians) consented to intralesional bevacizumab treatment. INTERVENTIONS: Intralesional bevacizumab administered at concentration of 2.5 mg/mL for 3 consecutive injections (with 532-nm pulsed KTP [potassium titanyl phosphate] laser when necessary) at intervals of 2 to 3 weeks. MAIN OUTCOME MEASURES: Time between surgical procedures, number of procedures per year, Derkay staging, total Pediatric Voice-Related Quality of Life (PVRQOL) score, Emotional PVRQOL score, and Physical PVRQOL score defined by comparing the year leading up to first of 3 bevacizumab injections with the year following the third bevacizumab injection. RESULTS: The median duration of time between surgical procedures increased by 5.9 weeks after bevacizumab (P = .002). The median number of procedures per year decreased by 4 (P = .002). Derkay staging decreased by 6 (P = .03). The median total PVRQOL score increased by 25.5 (P = .02), the median Emotional PVRQOL score increased by 11.3 (P = .047), and the median Physical PVRQOL score increased by 14.3 (P = .047). CONCLUSIONS AND RELEVANCE: Intralesional bevacizumab treatment may increase duration of time between surgical procedures and decrease number of procedures per year, while improving voice QOL.

Consistency of voice frequency and perturbation measures in children.

OBJECTIVE: Evaluate normal pediatric voice frequency and perturbation measures with Voice Evaluation Suite (VES) and Multi Dimensional Voice Program (MDVP), determine the consistency of these measures over time, and understand which measures might be most useful for evaluating children with voice disorders. STUDY DESIGN: Prospective, longitudinal study of normal voices of 50 children aged 4 to 17 years. SETTING: Pediatric otolaryngology clinic within tertiary hospital. SUBJECTS AND METHODS: Two tests of sustained utterances from each child were evaluated by 2 computerized voice analysis programs for frequency and perturbation. Intraclass correlation coefficient (ICC) was used to assess the reliability between the samples. RESULTS: Children (male/female, 1.08:1) with a mean age of 8.34 years were tested on an average of 54.2 minutes apart. Each test included 4 utterances; 1 was analyzed by MDVP, and 3 grouped utterances were averaged and evaluated by VES. Fundamental frequency had excellent reliability (ICC = 0.95) in both VES and MDVP. Jitter, shimmer, and noise to harmonic ratio were poorly reliable (ICC ≤ 0.4) in MDVP but had good to excellent reliability (ICC 0.66-0.8) in VES. CONCLUSION: Single, sustained utterances in children provide consistent measures of frequency. Perturbation is not reliably measured by such testing, but averaging multiple samples yields improved consistency. Evaluating acoustic measure stability in spontaneous speech and in sustained utterances cued by a tuning frequency can provide further insight on pediatric voice consistency.

Perioperative dexamethasone administration and risk of bleeding following tonsillectomy in children: a randomized controlled trial.

CONTEXT: Corticosteroids are commonly given to children undergoing tonsillectomy to reduce postoperative nausea and vomiting; however, they might increase the risk of perioperative and postoperative hemorrhage. OBJECTIVE: To determine the effect of dexamethasone on bleeding following tonsillectomy in children. DESIGN, SETTING, AND PATIENTS: A multicenter, prospective, randomized, double-blind, placebo-controlled study at 2 tertiary medical centers of 314 children aged 3 to 18 years undergoing tonsillectomy without a history of bleeding disorder or recent corticosteroid medication use and conducted between July 15, 2010, and December 20, 2011, with 14-day follow-up. We tested the hypothesis that dexamethasone would not result in 5% more bleeding events than placebo using a noninferiority statistical design. INTERVENTION: A single perioperative dose of dexamethasone (0.5 mg/kg; maximum dose, 20 mg), with an equivalent volume of 0.9% saline administered to the placebo group. MAIN OUTCOME MEASURES: Rate and severity of posttonsillectomy hemorrhage in the 14-day postoperative period using a bleeding severity scale (level I, self-reported or parent-reported postoperative bleeding; level II, required inpatient admission for postoperative bleeding; or level III, required reoperation to control postoperative bleeding). RESULTS: One hundred fifty-seven children (median [interquartile range] age, 6 [4-8] years) were randomized into each study group, with 17 patients (10.8%) in the dexamethasone group and 13 patients (8.2%) in the placebo group reporting bleeding events. In an intention-to-treat analysis, the rates of level I bleeding were 7.0% (n = 11) in the dexamethasone group and 4.5% (n = 7) in the placebo group (difference, 2.6%; upper limit 97.5% CI, 7.7%; P for noninferiority = .17); rates of level II bleeding were 1.9% (n = 3) and 3.2% (n = 5), respectively (difference, -1.3%; upper limit 97.5% CI, 2.2%; P for noninferiority < .001); and rates of level III bleeding were 1.9% (n = 3) and 0.6% (n = 1), respectively (difference, 1.3%; upper limit 97.5% CI, 3.8%; P for noninferiority = .002). CONCLUSIONS: Perioperative dexamethasone administered during pediatric tonsillectomy was not associated with excessive, clinically significant level II or III bleeding events based on not having crossed the noninferior threshold of 5%. Increased subjective (level I) bleeding events caused by dexamethasone could not be excluded because the noninferiority threshold was crossed. TRIAL REGISTRATION: clinicaltrials.gov Identifier: NCT01415583.