Quality of life of children with hearing loss in special and mainstream education: a longitudinal study
Tirza F.K. van der Straaten, Carolien Rieffe, Wim Soede, Anouk P. Netten, ... Johan H.M. Frijns
In Press, Journal Pre-proof, Available online 5 October 2019


https://www.sciencedirect.com/journal/international-journal-of-pediatric-otorhinolaryngology/articles-in-press

Abstract
Objectives
To compare the quality of life (QoL) of children with hearing loss (HL) and children with normal hearing (NH) and to examine how the QoL of children with HL changes over time, considering language skills, type of hearing device, degree of HL, and type of education.

Methods and materials
This longitudinal study included 62 children with HL and their parents. Developmental outcome data were collected at two time points, when the mean ages of the children were 4 and 11 years. The Pediatric Quality of Life (PedsQL™) questionnaire, which includes assessments of Physical, Emotional, Social, and School functioning, was completed by parents at both time points and by the children with HL at the second time point. Receptive and expressive language skills at 4 years were assessed by the Reynell Developmental Language Scale. Results were compared with a Dutch normative sample.

Results
The QoL of children with HL was similar to that of children with NH at both time points on two of the four QoL scales, Emotional and Physical functioning. On the other two scales, Social and School functioning, children with HL who attended special education and children who switched to mainstream education showed lower scores than children with HL who were consistently in mainstream education and lower scores than children with NH. The School QoL of children with HL decreased over time, as did the School QoL of children with NH. Social QoL of children with cochlear implants decreased over time, but this was not the case in children with hearing aids. Language skills and the degree of HL did not clinically improve the QoL over time of preschool children with HL.

Conclusions
The QoL of children with HL in mainstream education and the Physical and Emotional QoL of all children with HL were satisfactory. It is essential to develop specific guidance regarding school activities for children with HL in special education and for children with HL who switch to mainstream education in order to increase their social QoL.

select article Radiologic, Genetic, and Endocrine Findings in Isolated Congenital Nasal Pyriform Aperture Stenosis Patients
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Radiologic, Genetic, and Endocrine Findings in Isolated Congenital Nasal Pyriform Aperture Stenosis Patients
James Ruda, Jonathan Grischkan, Zahir Allarakhia
In Press, Journal Pre-proof, Available online 5 October 2019
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Abstract
Abstract
Background
Congenital nasal pyriform aperture stenosis (CNPAS) is a rare cause of upper airway obstruction in neonates. It can occur either associated with a solitary median maxillary central incisor (SMMCI) in 40-75% of cases or as an isolated morphogenic variant. Brain MRI is routinely performed in patients with CNPAS with a SCMMI due to the concomitant risks of intracranial midline defects of the hypothalamic-pituitary axis (HPA), holoprosencephaly, or associated endocrine dysfunction. However, the role of routine brain imaging and endocrinologic evaluation in patients with isolated CNPAS is not frequently studied given the assumption that isolated CNPAS is unlikely to be associated with any intracranial findings.

Objectives
To evaluate intracranial findings on dual brain MRI imaging in isolated CNPAS patients and to determine if any radiologic, endocrinologic, or genetic abnormalities existed in these patients.

Methods
We performed a single-institution retrospective review and case series of patients with isolated CNPAS from 2006-2019. Findings from dual brain MRI imaging, cytogenetic and karyotype testing, and formal endocrinologic testing were analyzed for each patient and compared to reported findings in isolated CNPAS patients within the literature.

Results
From 2006-2019, 16/29 patients were identified at our institution with isolated CNPAS. Inpatient otolaryngologic consultation, CT, and brain MRI imaging was obtained after a mean postnatal age of 3.21 days, 13.5 days, and 35.91 days, respectively. Dual brain MRI imaging was obtained in 12/16 (75%) patients. In our patients, abnormal MRI findings included hypothalamic hamartoma, pars intermedius cyst, and grades 1 and 4 germinal matrix hemorrhages. No holoprosencephaly was found in any patient. Genetic testing was performed on 10/16 (62.5%) patients. Karyotyping was normal in all patients and microarray testing was abnormal in 2/10 patients that represented a 2p16.3 deletion in one patient and a 7p36 deletion causing an unbalanced translocation mutation of the Sonic Hedgehog gene in the second patient. In the latter patient a hypothalamic hamartoma and panhypopituitarism was found. Endocrine evaluation was performed in 5 patients and was normal in 3/5 patients.

Conclusions
Our case series of isolated CNPAS patients identified multiple abnormalities on radiologic, genetic, and endocrine testing; one of which that involved a defect of the HPA with panhypopituitarism resulting from a hypothalamic hamartoma. Based upon our findings, dedicated brain MRI imaging, endocrinologic, and genetic testing, should likely be considered for any newly diagnosed, isolated CNPAS patient given the uncommon but potential risk for associated intracranial abnormalities.

select article Binaural processing and phonological awareness in Australian Indigenous children from the Northern Territory: A community based study
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Binaural processing and phonological awareness in Australian Indigenous children from the Northern Territory: A community based study
Mridula Sharma, Gillian Wigglesworth, Gemma Savage, Katherine Demuth
In Press, Journal Pre-proof, Available online 5 October 2019
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Abstract
Abstract
Objective
Research has found that otitis media (OM) is highly prevalent in Australian Indigenous children, and repeated bouts of OM is often associated with minimal-to-moderate hearing loss. However, what is not yet clear is the extent to which OM with hearing loss impacts auditory signal processing specifically, but also binaural listening, listening in noise, and the potential impact on phonological awareness (PA) – an important, emergent literacy skill. The goal of this study was to determine whether auditory abilities, especially binaural processing, were associated with PA in children from populations with a high incidence of OM, living in a remote Australian Indigenous community in the Northern Territory (NT).

Methods
Forty-seven 5-12-year-olds from a bilingual school participated in the study. All were tested to determine hearing sensitivity (pure tone audiometry and tympanometry), with PA measured on a test specifically developed in the first language of the children. OM often results in a hearing loss that can affect binaural processing: the Dichotic Digit difference Test (DDdT) was used to evaluate the children’s dichotic listening and the Listening in Spatialised Noise-sentences test (LiSN-S) was used to evaluate their abilities to listen to speech-in-noise.

Results
Seventeen (36%) and 16 (34%) had compromised middle ear compliance (combined Type-B and -C) in the right and left ear respectively. Six children demonstrated a bilateral mild hearing loss, and another five children demonstrated a unilateral mild hearing loss. Thirty-one children were able to complete the DDdT listening task, whereas only 24 completed the speech in noise task (LiSN-S). Forty-four children (94%) were able to complete the letter identification subtask, comprising part of the PA task. The findings revealed that age was significantly correlated with all tasks such that the older children performed better across the board. Once hearing thresholds were controlled for, PA also correlated significantly with both binaural processing tasks of dichotic listening (r=0.59, p<0.001) and listening to speech in noise (r=-0.56, p=0.005); indicating a potential association between early, emergent literacy and listening skills.

Conclusions
The significant correlations between phonological awareness and dichotic listening as well as phonological awareness with listening to speech-in-noise skills suggests auditory processing, rather than hearing thresholds per se, are associated to phonological awareness abilities of this cohort of children. This suggests that the ability to process the auditory signal is critical.

select article Intra-capsular Complete Tonsillectomy, a Modification of Surgical Technique to Eliminate Delayed Post-Operative Bleeding
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Intra-capsular Complete Tonsillectomy, a Modification of Surgical Technique to Eliminate Delayed Post-Operative Bleeding
James T. Albright, Newton O. Duncan, Abel M. Smerica, Joseph L. Edmonds
In Press, Journal Pre-proof, Available online 4 October 2019
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Abstract
Abstract
Pediatric tonsillectomy is a frequently performed procedure using a variety of surgical techniques. Despite decades of modern surgical experience and advances in tools and techniques, a consensus on which technique is superior for minimizing complications has not been established. In a 2014 study, the authors examined differing surgical techniques and devices to determine if there was a significant difference in the bleed rates based on technique and device.1 In that study 7,024 children were evaluated, of which a total of 1.4% (n=99) of children experienced a postoperative hemorrhage that required a second surgery. There was no significant difference found between the four different techniques that were evaluated. Although the combined rates of post-operative hemorrhage requiring a second surgery was lower compared to many other published reports, we hypothesized that continued improvement in surgical techniques could eliminate post-operative hemorrhage completely. As follow up to that study, we decided on a collective change in technique, and subsequent analysis of our post-operative results with respect to only one outcome, post-operative hemorrhage requiring a second surgery. With a new standardized technique in place, we retrospectively evaluated one year of surgical outcomes. These procedures were performed using the technique of a modified and complete, intra-capsular Coblator tonsillectomy. Of the 783 tonsillectomies done in 12 months, we found there were 0 patients that had experienced a postoperative hemorrhage requiring a second surgery. Based on our previous study with a rate of 1.4% post-operative hemorrhage we would have expected to have 11 episodes of post-operative hemorrhage requiring a second surgery.

select article Keratinocyte Growth Factor (KGF) Induces Stem/Progenitor Cell Growth in Middle Ear Mucosa
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Keratinocyte Growth Factor (KGF) Induces Stem/Progenitor Cell Growth in Middle Ear Mucosa
Tomomi Yamamoto-Fukuda, Naotaro Akiyama, Hiromi Kojima
In Press, Journal Pre-proof, Available online 4 October 2019
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Abstract
Abstract
Objective
The middle ear epithelium is derived from the neural crest and endoderm, which line distinct regions of the middle ear cavity. In this study, we investigated the localization of stem/progenitor cells in the middle ear mucosa of adult mice and the effects of keratinocyte growth factor (KGF) on the cell kinetics of stem/progenitor cells in vivo.

Methods
In this study, after KGF-expression vector was transfected in the ear, two kinds of thymidine analogs, BrdU and EdU, were transferred at different time points. BrdU was detected by immunohistochemistry and EdU was detected by click chemistry. We also performed immunohistochemistry using anti-Keratin14 antibody (an undifferentiated epithelial cell marker), anti-p63 antibody (a stem/progenitor cell marker) and anti-acetylated α-tubulin antibody (a ciliated epithelial cell marker).

Results
A large number of EdU-positive cells were detected in the thickened mucosal epithelium of the pars flaccida (PF) and attic region at Day 1 after KGF transfection. Interestingly, in the mucosal epithelium overlying the promontory of the cochlea, many EdU-positive cells were detected. These cells were also positive for K14 and p63. The acetylated α-tubulin positive cells were reduced in the attic region at Day 1 after KGF transfection.

Conclusion
These findings indicate that KGF over-expression may increase stem/progenitor cell proliferation in the mucosal epithelium not only within the attic which is typical in middle ear cholesteatoma, but also overlying the promontory of the cochlea.

select article Selected cytokines in hypertrophic adenoids in children suffering from otitis media with effusion
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Selected cytokines in hypertrophic adenoids in children suffering from otitis media with effusion
Beata Zelazowska-Rutkowska, Bozena Skotnicka, Elzbieta Hassmann-Poznanska, Bogdan Cylwik
In Press, Journal Pre-proof, Available online 4 October 2019
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Abstract
Abstract
Objectives
The aim of the current study was to assess the levels of MMP-8, MMP-9 and TIMP-1 in the group of children with adenoids who suffered from exudative otitis media.

Methods
The study included 20 patients (10 females and 10 males) with adenoid hypertrophy coexisting with otitis media with effusion. The reference group included 24 patients (10 females and 14 males) with adenoid hypertrophy without otitis media. The levels of MMP-8, MMP-9 and TIMP-1 were determined in supernatants obtained from phytohemagglutinin (PHA)-stimulated cell cultures of the tonsils, using commercial enzyme-linked immunosorbent assay kits (R@D Systems, USA).

Results
The median MMP-8, MMP-9 and TIMP-1 concentrations (220.8 ng/mL, 311.1 ng/mL, 53.5 ng/mL, respectively) in the study group were significantly higher (p=0.000, p=0.000, p=0.048, respectively) than those in the reference group (93.5 ng/mL, 112.5 ng/mL, 36.95 ng/mL, respectively). ROC analysis revealed that the area under a curve (AUC) for both metalloproteinases MMP-8 and MMP-9 was 1 with a diagnostic sensitivity of 100% and diagnostic specificity of 95.8%, as compared to 0.690 for TIMP-1. Significant differences were found between the AUC for MMP-8 and TIMP-1 and MMP-9 and TIMP-1 (p<0.001 for both comparisons).

Conclusions
The changes in the concentrations of MMP-8, MMP-9 and TIMP-1 may indicate an increased remodeling of the extracellular matrix in children with adenoid hypertrophy and otitis media with effusion. The findings can have clinical as well as diagnostic utility. Determination of MMP-8 and MMP-9 may help qualify a child for adenoidectomy and differentiate pediatric patients affected by adenoid hypertrophy with and without otitis media.


Prevalence and Clinical Aspects of Hearing Loss among the South Korean Adolescent: Data from a Population-Based Study
Shin Hye Kim, Eun Shil Cha, Heung Eog Cha, Jae-Jun Song, Sung-Won Chae
In Press, Journal Pre-proof, Available online 4 October 2019
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Abstract
Abstract
Objectives
Slight/mild hearing loss is detrimental to communication and academic achievements. This study aimed to describe the prevalence of hearing loss, and to investigate the factors related to noise-induced hearing loss among South Korean adolescent.

Methods
As a population-based retrospective study, 1,845 South Korean adolescents aged from 12 to 19 years were analyzed using the data from Korea National Health and Nutrition Examination Survey V (KNHANES V, 2010–2012). The prevalence of hearing loss according to the side, severity, and frequency was calculated. For assessing the noise-induced hearing loss in adolescent, the prevalence of hearing loss only in high-frequency (onlyHFHL, defined as 1) thresholds at 0.5 and 1 kHz of ≤15 dB HL, and 2) maximal thresholds at 3, 4, or 6 kHz ≥15 dB HL higher than the highest threshold for 0.5 and 1 kHz) was analyzed. Moreover, the relevance of onlyHFHL in context of sociodemographic factors and noise exposure history was evaluated.

Results
The prevalence of unilateral and bilateral hearing loss based on the average of six frequencies (0.5, 1, 2, 3, 4, and 6 kHz) in South Korean adolescents were 8.56% and 1.03%, respectively, and most cases were hearing loss with slight/mild degree. The prevalence of unilateral and bilateral onlyHFHL were 32.74% and 5.53%, respectively. Factors found to be associated with onlyHFHL were sex (female) and household income (high).

Conclusions
According to this population-based study, the prevalence of slight/mild hearing loss and onlyHFHL in the South Korean adolescents were considerably high. With knowledge of the factors related with onlyHFHL, paying more attention to slight/mild hearing loss will be helpful in preventing hearing loss in adolescents.

select article International Pediatric Otolaryngology Group (IPOG): Juvenile-onset recurrent respiratory papillomatosis consensus recommendations
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International Pediatric Otolaryngology Group (IPOG): Juvenile-onset recurrent respiratory papillomatosis consensus recommendations
Claire Lawlor, Karthik Balakrishnan, Sergio Bottero, An Boudewyns, ... Roger Nuss
In Press, Journal Pre-proof, Available online 28 September 2019
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Abstract
Abstract
Objectives
To develop consensus recommendations for the evaluation and management of juvenile-onset recurrent respiratory papillomatosis (JORRP) in pediatric patients.

Methods
Expert opinion by the members of the International Pediatric Otolaryngology Group (IPOG). The mission of the IPOG is to develop expertise-based consensus recommendations for the management of pediatric otolaryngologic disorders with the goal of improving patient care. The consensus recommendations herein represent the first publication by the group.

Results
Consensus recommendations including diagnostic considerations, surgical management, systemic adjuvant therapies, postoperative management, surveillance, and voice evaluation. These recommendations are based on the collective opinion of the IPOG members and are targeted for otolaryngologists, primary care providers, pulmonologists, infectious disease specialists, and any other health care providers that manage patients with JORRP.

Conclusions
Pediatric JORRP consensus recommendations are aimed at improving care and outcomes in this patient population.

select article Inner Ear Pressure Evaluation using Wideband Tympanometry in Children with Large Vestibular Aqueduct Syndrome (LVAS): A pilot study
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Inner Ear Pressure Evaluation using Wideband Tympanometry in Children with Large Vestibular Aqueduct Syndrome (LVAS): A pilot study
Lifang Zhang, Jie Wang, Fei Zhao, Yongxin Li
In Press, Journal Pre-proof, Available online 19 September 2019
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Abstract
Abstract
Objective
To investigate middle ear function in children with Large Vestibular Aqueduct Syndrome (LVAS) to explore the feasibility of measuring inner ear pressure using Wideband tympanometry (WBT).

Methods
13 young children with LVAS were recruited. WBT and other audiological measurements i.e., Auditory Steady State Response (ASSR), Auditory Brain Stem Response (ABR), and Distorted Product Otoacoustic Emissions (DPOAE) were performed. Absorbance under ambient and peak pressure were compared with normative data, and analyzed using a one sample t-test.

Results
Average absorbance in children with LVAS was significantly lower than normative data under ambient pressure at 1000, 1189, 1296, 2000 Hz and 4000 Hz. Absorbance under peak pressure was also significantly lower at 707, 794, 917, 1000, 1189, 1297, 1498 and 2000 Hz. However, absorbance was higher than standard values above 4000 Hz under ambient and peak pressure. It was also higher under ambient pressure at frequencies below 500 Hz.

Conclusion
The special characteristics of middle ear function found in children with Large Vestibular Aqueduct Syndrome (LVAS) indicate that WBT offers a sensitive and non-invasive method to evaluate inner ear pressure indirectly.

select article Predicting Hearing loss in children According to the Referrer and Referral Cause
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Predicting Hearing loss in children According to the Referrer and Referral Cause
Slovik Y, Gorali R, Diziter Y, Joshua BZ, ... Kraus M
In Press, Journal Pre-proof, Available online 15 September 2019
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Abstract
Abstract
Background
Hearing assessment in children is considered necessary in all cases involving a doubt regarding hearing acuity. Due to the fact that the number of referrals may be much greater than the actual capacity of a hearing institute, it would be desirable to have a filtering tool in order to detect the cases with higher suspicion of hearing loss.

Objective
To evaluate whether anamnesis details can be used as a sorting tool for detection of highly suspicious cases of hearing loss in preschool children. The anamnesis details may be one or more of the following: the reason for performing hearing test, which caregiver indicated the need for hearing evaluation, the parent's comprehension of their child hearing, and the medical history.

Methods
the parents of preschool children, who were referred to the hearing institute at Soroka university medical center, were asked to fill a questionnaire containing 18 questions. The questions referred to details such as: the reason for performing the hearing test; who suggested the existence of hearing impairment; medical history issues and questions regarding details that may indicate a risk of hearing loss. Details such as Otoscopy results were taken from the medical record of the patient.

Results
317 preschool and school patients were recruited to the study. Hearing loss was found in 42% of the cases. The most common reason for performing the hearing test was speech disturbance (33%). In cases which the parents were those who raised the suspicion of hearing loss – hearing impairment was found in 61% of the cases, compared to 36% when the speech therapist was the one raising the doubts and to only 18% when the kindergarten teacher was the one raising the doubt. The positive predictive value of patient that their parents suspected the hearing loss combined with pathologic otoscopy result was 82.6% when the reason for the test was hearing impairment suspicion, and 91.3% when the reason for the hearing test was speech disturbance.

Conclusions
Parents are the best in assessing their child's hearing followed by the speech therapist and the least sensitive is the school teacher.

select article Injection laryngoplasty in infants with unilateral vocal cord paralysis: a survey of ASPO members
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Injection laryngoplasty in infants with unilateral vocal cord paralysis: a survey of ASPO members
Minyoung Jang, Stacie Gregory, Jad Jabbour, Thomas C. Robey, ... Robert Chun
In Press, Journal Pre-proof, Available online 6 September 2019
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Abstract
Abstract
Introduction
Unilateral vocal cord paralysis (UVCP) in infants may be managed medically or surgically, with the latter including injection laryngoplasty (IL). However, there is limited information.

regarding injection laryngoplasty in infants. We therefore proposed a survey of American Society of Pediatric Otolaryngology (ASPO) members to elucidate current practices, outcomes, and complications.

Methods
An online survey was distributed to all 548 ASPO members via email. The survey was closed for data analysis 2 months after initial distribution. Statistical analysis was deferred due to the primarily descriptive nature of the data and lack of comparative studies.

Results
We received 113 responses for a response rate of 20.6%. Only 31% of the respondents (n=36) reported performing IL in infants 12 months old or younger. The most commonly cited reasons for not injecting in this age group were preference for non-surgical management and concern for increased risk of airway obstruction. IL was most commonly performed to treat persistent aspiration despite attempts at medical management. The majority (66%) reported no complications, while the remainder noted stridor requiring intensive care unit observation. Re-intubation due to airway obstruction occurred in 6% (n=2 of 33). Carboxymethylcellulose gel (Prolaryn Gel) was the most commonly used injectable material, but a variety of other materials were used as well. Two respondents noted they perform reinnervation procedures in children, but not in infants, as they require at least a 12 month period of observation for possible spontaneous recovery before considering the option.

Discussion
There is limited data regarding management of UVCP in infants with IL, and considerable variation among those who do perform the procedure. While adverse events are rare, multi-institutional studies should be considered to help determine best practices.

select article Statistical analysis on “Early auditory skills development in Mandarin speaking children after bilateral cochlear implantation"
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Statistical analysis on “Early auditory skills development in Mandarin speaking children after bilateral cochlear implantation"
Mohammad Soleimani, Sepehr Eslami, Reza Amani-Beni, Sadra Ansaripour
In Press, Corrected Proof, Available online 10 August 2019
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