Publications by authors named "Amber D Shaffer"

42 Publications

Predictors of sinonasal disease onset, progression, and severity in pediatric cystic fibrosis patients.

Am J Otolaryngol 2021 Mar 31;42(5):103016. Epub 2021 Mar 31.

Department of Otolaryngology, Division of Pediatric Otolaryngology, University of Pittsburgh Medical Center Children's Hospital of Pittsburgh, Pittsburgh, PA, United States of America.

Purpose: Pediatric cystic fibrosis (CF) patients have a variable onset, severity, and progression of sinonasal disease. The objective of this study was to identify genotypic and phenotypic factors associated with CF that are predictive of sinonasal disease, recurrent nasal polyposis, and failure to respond to standard treatment.

Methods: A retrospective case series was conducted of 30 pediatric patients with CF chronic rhinosinusitis with and without polyps. Patient specific mutations were divided by class and categorized into high risk (Class I-III) and low risk (Class IV-V). Severity of pulmonary and pancreatic manifestations of CF, number of sinus surgeries, nasal polyposis and recurrence, age at presentation to Otolaryngology, and Pediatric Sinonasal Symptom Survey (SN-5)/Sinonasal Outcome Test (SNOT-22) scores were examined.

Results: 27/30 patients (90%) had high risk mutations (Class I-III). 21/30 (70.0%) patients had nasal polyposis and 10/30 (33.3%) had recurrent nasal polyposis. Dependence on pancreatic enzymes (23/27, 85.2% vs 0/3, 0.0%, p = 0.009) and worse forced expiratory volumes (FEV1%) (mean 79, SD 15 vs mean 105, SD 12, p = 0.009) were more common in patients with high risk mutations. Insulin-dependence was more common in those with recurrent polyposis (5/10, 50% vs 2/20, 10%, p = 0.026). There was no statistical difference in ages at presentation, first polyps, or sinus surgery, or in polyposis presence, recurrence, or extent of sinus surgery based on high risk vs. low risk classification.

Conclusion: CF-related diabetes was associated with nasal polyposis recurrence. Patients with more severe extra-pulmonary manifestations of CF may also be at increased risk of sinonasal disease.
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http://dx.doi.org/10.1016/j.amjoto.2021.103016DOI Listing
March 2021

Advanced Practice Provider Clinics: Expediting Care For Children Undergoing Tympanostomy Tube Placement.

Laryngoscope 2021 Feb 26. Epub 2021 Feb 26.

Division of Pediatric Otolaryngology, UPMC Children's Hospital of Pittsburgh, Pittsburgh, Pennsylvania, U.S.A.

Objective/hypothesis: Advanced practice provider (APP) employment is becoming common in pediatric otolaryngology practices, though few studies have evaluated the consequences that APP-led clinics have on access to care. The objectives of this study were: 1) to investigate whether access to bilateral myringotomy with tympanostomy tube placement (BMT) for recurrent acute otitis media (RAOM) differed between patients seen in otolaryngologist and APP-led clinics 2) to compare clinical characteristics of patients seen by provider type.

Methods: Retrospective cohort study at an academic, tertiary care pediatric otolaryngology practice. All children were <18 years old and underwent evaluation for RAOM followed by BMT. We compared time in days from scheduling pre-operative appointment to appointment date and time from appointment to BMT between patients seen by APPs and otolaryngologists using Mann-Whitney U tests and multivariate linear regression models. We compared clinical characteristics by provider type using Mann-Whitney U tests and Fisher exact tests.

Results: A total of 957 children were included. Children seen by APPs had significantly shorter wait times for appointments (median 19 vs. 39 days, P < .001) and shorter times from preoperative appointment to BMT (median 25 vs. 37 days, P < .001). Patients seen by otolaryngologists had increased prevalence of craniofacial abnormalities, Down Syndrome, hearing loss, history of otologic surgery, and higher ASA physical status classification.

Conclusions: Children seen by APPs received care more quickly than those seen by otolaryngologists. Patients seen by otolaryngologists tended to be more medically complex. Implementation of independent APP clinics may expedite and improve access to BMT for children with RAOM.

Level Of Evidence: 3 Laryngoscope, 2021.
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http://dx.doi.org/10.1002/lary.29477DOI Listing
February 2021

It's a rash: Antibiotic allergies in the modern era of antibiotic stewardship.

Int J Pediatr Otorhinolaryngol 2021 Apr 30;143:110638. Epub 2021 Jan 30.

Department of Otolaryngology, University of Pittsburgh School of Medicine, Pittsburgh, PA, United States.

Objectives: To determine whether current guidelines emphasizing antibiotic stewardship in pediatrics have been associated with reduced prevalence of antibiotic allergies in children severely affected by otitis media undergoing bilateral myringotomy with tympanostomy tube insertion (BMT) or by recurrent sinusitis or adenotonsillitis undergoing adenoidectomy with or without tonsillectomy.

Methods: Case series of consecutive patients undergoing BMT or adenoidectomy with/without tonsillectomy for recurrent acute otitis media, recurrent sinusitis, or recurrent tonsillitis during November 2008 or November 2017 at a tertiary care children's hospital. Children with primarily obstructive indications for surgery, with prior tube placement or adenoidectomy, or with surgery by an outside provider were excluded. Demographics, type of surgery, and allergies or allergic symptoms were collected from the electronic medical record. Factors associated with antibiotic allergies were compared using logistic regression, Wilcoxon rank-sum, or Chi-squared test.

Results: Seventy-five children who underwent surgery during 2008 and 75 children who underwent surgery in 2017 were included. Overall, median age at surgery was 3.24 years (range 0.56-17.49 years). Seventy-nine (52.7%) patients were female and 95 (63.3%) had private insurance. BMT was the most common surgery (82 children, 54.7%) followed by tonsillectomy with adenoidectomy (46 children, 30.7%), and adenoidectomy without tonsillectomy (39 children, 26.0%). Symptoms of allergic rhinitis were reported by 53 (35.3%) patients, and 11 (7.3%) and 5 (3.3%) had positive environmental and food allergy testing, respectively. Surprisingly, there was not a significant difference between the prevalence of antibiotic allergies in patients undergoing surgery during 2017 (17 patients, 22.7%) compared with 2008 (14 patients, 18.7%) (OR: 1.28, 95% CI: 0.578-2.82, p = 0.546). However, antibiotic allergies were less common in females (OR: 0.413, 95% CI: 0.182-0.937, p = 0.034) and more common in patients with a family history of antibiotic allergies (OR: 36.9, 95% CI: 5.12-∞, p < 0.001).

Conclusion: Pediatric otolaryngology surgical patients continue to exhibit a similar and high prevalence of antibiotic allergies in 2017 compared with 2008. Future studies are needed to determine whether this is because of overdiagnosis of antibiotic allergies or a failure of antibiotic guideline adherence to reduce antibiotic allergy prevalence.
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http://dx.doi.org/10.1016/j.ijporl.2021.110638DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7994189PMC
April 2021

Operable, Low-Cost, High-Resolution, Patient-Specific 3D Printed Temporal Bones for Surgical Simulation and Evaluation.

Ann Otol Rhinol Laryngol 2021 Feb 8:3489421993733. Epub 2021 Feb 8.

Department of Otolaryngology, University of Pittsburgh Medical Center, PA, USA.

Objectives: Three-dimensional printed models created on a consumer level printer can be used to practice mastoidectomy and to discern mastoidectomy experience level. Current models in the literature for mastoidectomy are limited by expense or operability. The aims of this study were (1) to investigate the utility of an inexpensive model for mastoidectomy and (2) to assess whether the model can be used as an evaluation tool to discern the experience level of the surgeon performing mastoidectomy.

Methods: Three-dimensional printed temporal bone models from the CT scan of a 7-year old patient were created using a consumer-level stereolithography 3D printer for a raw material cost of $10 each. Mastoidectomy with facial recess approach was performed by 4 PGY-2 residents, 4 PGY-5 residents, and 4 attending surgeons on the models who then filled out an evaluation. The drilled models were collected and then graded in a blinded fashion by 6 attending otolaryngologists.

Results: Both residents and faculty felt the model was useful for training (mean score 4.7 out of 5; range: 4-5) and case preparation (mean score: 4.3; range: 3-5). Grading of the drilled models revealed significant differences between junior resident, senior resident, and attending surgeon scores ( = .012) with moderate to excellent interrater agreement (ICC = 0.882).

Conclusion: The described operable model that is patient-specific was rated favorably for pediatric mastoidectomy case preparation and training by residents and faculty. The model may be used to differentiate between experience levels and has promise for use in formative and summative evaluations.
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http://dx.doi.org/10.1177/0003489421993733DOI Listing
February 2021

Hearing preservation with a slim modiolar cochlear implant in a pediatric cohort.

Int J Pediatr Otorhinolaryngol 2021 Jan 1;140:110479. Epub 2020 Nov 1.

Department of Otolaryngology, UPMC Children's Hospital of Pittsburgh, 4401 Penn Ave, Faculty Pavilion, 7th Floor, Pittsburgh, PA, 15224, United States. Electronic address:

Objectives: Recent cochlear implant (CI) electrodes are designed to be atraumatic to inner ear structures. Studies in adults have demonstrated improved hearing preservation rates with the CI532/632 electrode, but none have examined this in children. Our objective is to describe the hearing preservation rate with CI532/632 in pediatric patients and determine factors that influence hearing preservation.

Methods: We conducted a retrospective cohort study of children undergoing CI over a three-year period. Inclusion criteria were: CI with the 532/632 electrode, presence of pre-operative low frequency hearing defined by pure tone average (PTA) of ≤80 dB at 250 Hz or at the average of 250 and 500 Hz, and post-operative unaided audiometry. Other data collected included demographics, otologic history, imaging, and surgical details.

Results: A total of 13 patients and 15 ears were included. Hearing was preserved in 10/15 (66%) ears at an average follow-up of 6 months, similar to that reported in the adult literature. Patients with preserved hearing post-operatively were more likely to have a positive family history of hearing loss. There was a trend towards patients with anatomic inner ear abnormalities being more likely to lose hearing after CI, but this was not statistically significant. Pre-operative thresholds were not predictive of hearing preservation. Patients with preserved hearing had a significantly smaller shift in thresholds after cochlear implantation. Therefore, hearing preserved and non-preserved groups differed more by the magnitude of change in threshold, rather than their preoperative threshold. Other factors such as age, sex, surgeon, and surgery duration were not associated with hearing preservation.

Conclusion: This study describes low frequency hearing preservation after pediatric CI532/632 implantation. The hearing preservation rate in our cohort was consistent with that reported in the adult literature. Our data suggest that preoperative thresholds do not solely determine which patients will go on to experience hearing preservation. We believe this will aid surgeons with patient-specific device selection and counseling potential pediatric CI recipients with preserved hearing.
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http://dx.doi.org/10.1016/j.ijporl.2020.110479DOI Listing
January 2021

Predictive Utility of the Penetration-Aspiration Scale in Inter-Arytenoid Injection Augmentation.

Laryngoscope 2021 May 3;131(5):E1707-E1713. Epub 2020 Oct 3.

Department of Otolaryngology, Children's Hospital of Pittsburgh, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania, U.S.A.

Objectives/hypothesis: To determine the utility of preoperative penetration-aspiration scale (PAS) scores and clinical findings on modified barium swallow (MBS) in predicting advancement of diet after interarytenoid injection augmentation (IAIA).

Study Design: Retrospective review.

Methods: In this retrospective cohort study, 372 consecutive patients who underwent IAIA for pharyngeal dysphagia between 2009 and 2019 were initially identified. Patients were excluded from the study if they had insufficient preop MBS, no postop MBS within 3 months of injection, supraglottoplasty, or underlying neurological condition. Ninety-three patients were included in the study. Pre- and postoperative PAS scores were recorded, as were pre and postop diets. PAS scores were calculated by a single pediatric speech and language pathologist.

Results: Average PAS score on MBS was 5.87 (standard deviation [SD] 2.74); median (range) = 8 (1-8). Postop average was 4.29 (SD 3.02); median (range) = 2 (1-8), P < .001. Those with worse preop PAS scores had increased odds of improvement in diet (odds ratio 1.24, 95% confidence interval [CI] 1.02-1.49, P = .029). An improvement in PAS score of 3.0 or greater predicted an improvement in diet with a sensitivity of 76.7% and a specificity of 85.7%.

Conclusions: PAS score on MBS can be a useful tool when assessing pediatric patients who may be candidates for IAIA. Prospectively comparing PAS score in patients post-IAIA to patients solely undergoing diet modification can help to better objectively assess differences in outcomes and understand the full utility of PAS score.

Level Of Evidence: Level III (Individual Cohort Study) Laryngoscope, 131:E1707-E1713, 2021.
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http://dx.doi.org/10.1002/lary.29142DOI Listing
May 2021

Sudden Sensorineural Hearing Loss in Children-Management and Outcomes: A Meta-analysis.

Laryngoscope 2021 02 16;131(2):425-434. Epub 2020 Jul 16.

Division of Pediatric Otolaryngology, University of Pittsburgh Medical Center Children's Hospital of Pittsburgh, Pittsburgh, Pennsylvania, USA.

Objective: To identify which patient characteristics and treatments are associated with hearing improvement in patients with pediatric sudden sensorineural hearing loss (SSNHL).

Methods: PubMed, Web of Science, Cochrane Library, PsycINFO, and Cumulative Index to Nursing and Allied Health Literature were systematically searched for articles published before February 5, 2019, and references of relevant articles were screened. Original English-language case series and cohort studies were included if they addressed SSNHL in patients <20 years of age. Study characteristics, patient demographics, symptoms, treatments, and hearing outcomes were extracted. The primary outcome was hearing improvement.

Results: Thirteen studies were included, totaling 605 patients (670 ears). Hearing did not improve in 46.7% of ears (95% confidence interval [CI]: 34.4% to 59.0%). Imaging (computed tomography and/or magnetic resonance imaging) results were described in three studies, and 24.2% of ears (95% CI: 7.7% to 40.6%) had abnormal findings. The most common serological finding was cytomegalovirus immunoglobulin (Ig)G or IgM (34.3% of ears tested, 95% CI: -2.9% to 71.6%). Unilateral hearing loss (odds ratio [OR]:3.85, P < .001), tinnitus (OR: 2.20, P = .003), age >12 years (OR: 2.11, P = .002), and ascending audiogram (OR: 3.66, P = .005), but not systemic or intratympanic steroids, were associated with increased odds of partial or complete improvement. In contrast, profound hearing loss (OR: 0.29, P < .001) and treatment delay of >6 days (OR: 0.27, P < .001) were associated with decreased odds of improvement.

Conclusions: Despite treatment, half of patients had no improvement in hearing. Prognostic factors associated with hearing improvement were generally consistent with those established in the adult population. Further research with consistent definitions for hearing improvement is needed to improve the understanding and treatment of pediatric SSNHL.

Level Of Evidence: Laryngoscope, 131:425-434, 2021.
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http://dx.doi.org/10.1002/lary.28829DOI Listing
February 2021

Follow-up after failed newborn hearing screening: Parental and primary care provider awareness.

Am J Otolaryngol 2020 Nov - Dec;41(6):102614. Epub 2020 Jun 18.

UPMC Children's Hospital of Pittsburgh, 4401 Penn Avenue, Pittsburgh, PA 15224, USA. Electronic address:

Purpose: The current loss to follow-up rate after failed newborn hearing screening (NBHS) is 34.4%. Previous studies have found that lack of parental and primary care provider (PCP) awareness of NBHS results are significant contributors to loss to follow-up. The objective of this study was to identify factors associated with parental and PCP awareness of NBHS results.

Materials And Methods: Retrospective cohort study. A survey asking about demographics and knowledge of NBHS testing and results was offered to parents in the waiting room of an urban pediatric primary care office. Included were biological parents ≥18 years of age of children ≤10 years of age born in Pennsylvania. Each child's chart was reviewed for PCP documentation of NBHS results. The odds of knowing NBHS results were evaluated using logistic regression.

Results: The survey was completed by 304 parents. 74.0% were aware of their child's NBHS results. Child age ≥1 year old (OR: 0.49, 95%CI[0.29, 0.82], P = 0.007) and Hispanic ethnicity (OR: 0.38, 95%CI[0.16, 0.89], P = 0.03) were associated with decreased odds of a parent knowing NBHS results. In addition, fewer fathers knew the results of their child's NBHS compared with mothers (OR: 0.33, 95%CI[0.18, 0.62], P < 0.001). However, parental awareness was not associated with birthing facility or insurance type. 222 charts were reviewed for NBHS documentation, revealing PCP awareness in 95.5% of cases and no associations with any of the factors examined.

Conclusions: Factors associated with parents not knowing NBHS results included being the parent of an older child, Hispanic, or the father.
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http://dx.doi.org/10.1016/j.amjoto.2020.102614DOI Listing
January 2021

Long-term Impact of Middle Ear Effusion in Pediatric Tympanostomy Tubes.

Laryngoscope 2021 03 4;131(3):E993-E997. Epub 2020 Jul 4.

Department of Otolaryngology, University of Pittsburgh School of Medicine, Pittsburgh, Pennsylvania, U.S.A.

Objectives/hypothesis: Bilateral myringotomy and tympanostomy tube placement (BMT) is the most common pediatric surgery in the United States. Intraoperative middle ear effusion (MEE) is a risk factor for future BMTs in children with recurrent acute otitis media (RAOM). However, the impact of the type of MEE is unknown. Here, we assess otologic outcomes based on intraoperative MEE type and indication for surgery.

Study Design: Case series chart review.

Methods: After institutional review board approval, we performed a review of children undergoing BMTs between 2008 and 2009. Included patients had their first BMT, preoperative visit, and an operative report. Patients with cleft palate or Down syndrome were excluded. Indications for surgery included RAOM and chronic otitis media with effusion (COME). Other variables evaluated were future BMT, acquired cholesteatoma, and otorrhea. Logistic regression was used for statistical analysis.

Results: Out of 1,045 patients reviewed, 680 were included and underwent their first BMT. There were 619 patients who had RAOM. Serous effusions were present in 22.2%, mucoid in 31.3%, purulent in 12.9%, undocumented or bloody in 2.3% of patients, and 31.2% of patients had dry middle ears. Moreover, 22.7% of patients underwent future BMTs. In RAOM patients, serous effusions decreased odds of perforation (odds ratio [OR]: 0.195, 95% confidence interval [CI]: 0.0438-0.867, P = .032), and purulent effusions increased the odds of in-office otorrhea suctioning (OR: 2.13, 95% CI: 1.20-3.77, P = .010) compared to dry. Mucoid effusions had no significant effect on outcomes in COME or RAOM patients.

Conclusions: Intraoperative MEEs were noted in 68.7% of cases; purulent effusions increase the odds of in-office suctioning in RAOM patients.

Level Of Evidence: 4 Laryngoscope, 131:E993-E997, 2021.
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http://dx.doi.org/10.1002/lary.28860DOI Listing
March 2021

Long-Term Otitis Media Outcomes in Infants With Early Tympanostomy Tubes.

Otolaryngol Head Neck Surg 2020 12 16;163(6):1258-1263. Epub 2020 Jun 16.

Department of Otolaryngology, UPMC Children's Hospital of Pittsburgh, Pittsburgh, Pennsylvania, USA.

Objective: To review the otologic outcomes of infants who failed the newborn hearing screen (NBHS) and received early tympanostomy tubes for otitis media with effusion (OME).

Study Design: Retrospective case series.

Setting: Tertiary care pediatric hospital.

Subjects And Methods: Consecutive patients (2007-2018) who failed an NBHS and required tympanostomy tubes before 6 months of age were included. Variables including hearing loss and otitis media risk factors, episodes of acute otitis media (AOM), number of subsequent tympanostomy tubes, and posttympanostomy tube audiogram results were recorded.

Results: The cohort included 171 patients. Median age at referral to otolaryngology was 2.7 months. Sensorineural hearing loss (SNHL) was subsequently identified in 22 (12.9%) of infants after resolution of the effusion. The peak incidence of AOM was during the second year of life (1-1.9 years), with a median of 1 episode. Ninety-five patients (55.6%) had replacement of tubes, 41 of 171 (24.0%) had 2 or more additional sets of tubes, and long-term tubes were eventually placed in 8 of 95 (8.4%) patients. Craniofacial anomalies were identified in 43.3% of patients. Tube replacement (hazard ratio, 3.00; 95% CI, 1.95-4.63; < .01, log-rank) and AOM (β, 1.04; 95% CI, 0.43-1.65; = .04, ordered logistic regression) were more common, and SNHL less common (odds ratio, 0.17; 95% CI, 0.031-0.61; < .01, logistic regression), in children with craniofacial anomalies.

Conclusion: OME is a common cause of failed NBHS. A notable proportion was subsequently found to have SNHL, reiterating the need for postoperative hearing assessments. Infants meeting indication for early tympanostomy tubes for resolution of OME have a high incidence of recurrent AOM and require subsequent tubes.
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http://dx.doi.org/10.1177/0194599820931414DOI Listing
December 2020

Outcomes of BAHA connect vs BAHA attract in pediatric patients.

Int J Pediatr Otorhinolaryngol 2020 Aug 20;135:110125. Epub 2020 May 20.

Division of Pediatric Otolaryngology, UPMC Children's Hospital of Pittsburgh, 4401 Penn Ave, Faculty Pavilion, 7th Floor, Pittsburgh, PA, 15224, USA. Electronic address:

Introduction: Two of the most commonly employed bone-anchored hearing implant (BAHI) systems are the BAHA Connect and BAHA Attract. The BAHA Connect uses a skin-penetrating titanium abutment. The BAHA Attract uses an implanted magnet, leaving the overlying skin intact. Limited data is available on the difference in complication rates between the two systems. Our hypothesis was that there would be no difference in complications and audiologic data.

Methods: Retrospective chart review was performed of patients who had BAHA Connect vs. Attract at our tertiary care pediatric hospital from 2006 to 2018. Pre- and post-operative information, including demographics, related diagnoses, outcomes and complications were compared between the systems using Mann-Whitney U tests and Firth logistic regression for one year post-implant. Audiology data was analyzed with Wilcoxon rank-sum and Wilcoxon matched pairs signed rank tests.

Results: Twenty-four Attract and 18 Connect BAHA surgeries were identified from 37 patients. Eleven Connect patients had the surgery completed in two stages. Connect patients followed up an average of 6.5 years post-implant and 15 months for Attract. A total of 58.8% of patients with Connect surgeries had complications within a year and 82.4% had a complication by their last follow-up. Aside from magnet strength related issues, there were no major complications with Attract surgery at any time point. Patients with Connect surgeries had significantly more skin overgrowths, cultured infections, times on antibiotics, nursing phone calls, and ENT visits within the first year and for all records, p < .05. The pure-tone average was significantly lower for both Connect [unaided-M(SD) = 61.7(9.8); aided-M(SD) = 26.4(5.5) and Attract (unaided-M(SD) = 66.0(22.5); aided-M(SD) = 25.6(6.1)] after implant, p < .001.

Conclusion: Implantation of both systems lead to improved hearing outcomes with profoundly different complication rates.
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http://dx.doi.org/10.1016/j.ijporl.2020.110125DOI Listing
August 2020

The microbiome of pediatric patients with chronic rhinosinusitis.

Int Forum Allergy Rhinol 2021 Jan 22;11(1):31-39. Epub 2020 Jun 22.

Division of Pulmonary, Allergy and Critical Care Medicine, Department of Medicine, University of Pittsburgh School of Medicine and University of Pittsburgh Medical Center, Pittsburgh, PA.

Background: This study aimed to compare the microbiota of pediatric patients with chronic rhinosinusitis (CRS) who are undergoing adenoidectomy to treat their disease with that of healthy control patients.

Methods: Patients undergoing adenoidectomy-only for obstructive sleep apnea (n = 50) and CRS (n = 37) were recruited. Preoperative 22-item Sino-Nasal Outcome Test (SNOT-22) or Sinus and Nasal Quality of Life Survey (SN-5) were collected. Each patient had samples collected from their nasopharynx (adenoid bed) and nasal cavity (sinus) at the onset of surgery. 16S ribosomal ribonucleic acid (rRNA) gene sequencing was subsequently performed to obtain per sample taxonomic abundances. Statistical analyses included permutational multivariate analysis of variance (PERMANOVA), alpha (within sample) diversity measures, and changes in taxonomic abundance.

Results: Moraxella was the most abundant organism. Nasopharyngeal swabs demonstrated higher alpha diversity compared to the nasal cavity. The diversity was not different based on CRS vs obstructive history. There was an increase in diversity with increasing age, and eczema contributed to a greater difference in diversity between the nasopharynx and nasal cavity. Diversity was not affected by adenoid size; however, use of nasal steroids, inhaled steroids, and antihistamines influenced diversity in both the nasopharynx and nasal cavity. Nasopharyngeal samples were higher in relative abundance for Fusobacterium, Prevotella, Porphyromonas, and Campylobacter compared to the nasal cavity.

Conclusion: The nasopharynx and nasal cavity differed in both microbiota composition and diversity. In contrast, no significant difference in composition or diversity were found in CRS vs control patients. Ecological changes in the nasopharyngeal and sinus site may contribute to the etiology for adenoid hypertrophy in both healthy controls and CRS patients.
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http://dx.doi.org/10.1002/alr.22597DOI Listing
January 2021

Silent sinus syndrome in children.

Int J Pediatr Otorhinolaryngol 2020 Jul 1;134:110034. Epub 2020 Apr 1.

Department of Otolaryngology - Head and Neck Surgery, University of Pittsburgh Medical Center, Pittsburgh, PA, USA; UPMC Children's Hospital of Pittsburgh, University of Pittsburgh Medical Center, Pittsburgh, PA, USA. Electronic address:

Introduction: Silent sinus syndrome (SSS) is defined as a progressive enophthalmos and hypoglobus associated with maxillary sinus atelectasis. There is extremely limited literature describing SSS in children. The goals of this study are to characterize SSS in children through an IRB approval retrospective chart review of cases identified through a large health system-wide imaging database and to compare the presentation and outcomes of patients who underwent surgery versus those who were observed.

Methods: A radiology database of over 26 million reports from 2003 to 2017 was searched to identify children aged 1-18 years diagnosed with maxillary sinus hypoplasia or SSS on CT scan. Chart review was performed on the identified children including clinical presentation, eye symptoms, surgical treatment, and outcome.

Results: Eighty-three children were identified to have maxillary sinus hypoplasia. Eighty-one patients had maxillary sinus opacification and 57 patients had hypoglobus or enophthalmos characteristic of SSS. Thirty-two patients (47%) were seen by a specialist and 19 had surgery. The majority of patients (55%) had headache as their presenting symptom. There were no statistically significant differences in the clinical presentation between those who received surgery and those who were observed clinically.

Conclusions: Silent sinus syndrome can present at any age. The majority of cases of maxillary sinus hypoplasia will have the orbital floor changes characteristic of SSS. Headaches are a common presenting symptom. Close follow up of pediatric patients is advised and early intervention may be favorable to prevent long term orbital changes and complications.
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http://dx.doi.org/10.1016/j.ijporl.2020.110034DOI Listing
July 2020

Middle ear disease in CF? It's not just about the sinuses anymore!

Int J Pediatr Otorhinolaryngol 2020 Jul 1;134:110032. Epub 2020 Apr 1.

Division of Pediatric Otolaryngology, UPMC Children's Hospital of Pittsburgh, Pittsburgh, PA, USA; Department of Otolaryngology, University of Pittsburgh School of Medicine, Pittsburgh, PA, USA. Electronic address:

Introduction: Historically cystic fibrosis (CF), in contrast to primary ciliary dyskinesia (PCD), has been considered protective of the middle ear from otitis media and rarely were such patients "severely affected" to require tympanostomy tube placement (BMT). Previously the Pittsburgh Otitis Media Research group reported a 10% prevalence of otitis media in the pediatric CF population which is significantly lower than age matched non-CF children. We studied the prevalence of OM in pediatric CF patients to assess if the otologic phenotype has changed in parallel with changes in the diagnosis itself.

Methods: The medical records of 133 CF patients seen either inpatient or outpatient from one of the largest tertiary pediatric CF centers in the world from 2010 to 2019 were reviewed for demographics, acute otitis media (AOM) episodes, risk factors for AOM, placement of myringotomy tubes, genotype, BMI, pancreatic status, respiratory culture results, and pulmonary exacerbations.

Results: Just over half (52.6%) the patients were male. A median age for CF diagnosis was 11 days old (range 0 days-16 years). The most common genotype (49.6%) was homozygous for ΔF508 mutation. Fifty-five (41.4%) patients had 1-2 episodes of AOM, and 15 (11.3%) were severely affected (i.e. ≥3 episodes/6 months or ≥4 episodes/year). COME was diagnosed in 4 (3.0%) of the patients. Twelve (9.0%) patients had tympanostomy tubes at least once, including 3 patients with multiple sets of tubes. Having at least one AOM episode was not predicted by genetic mutation groups, BMI, age at diagnosis, or comorbidities, p > .05.

Conclusions: The time-honored adage of CF protecting against otitis media is no longer true and pediatric otolaryngologists must now prioritize the management of middle ear disease as highly as sino-nasal and pulmonary disease. Future study into mechanisms explaining the change is needed.
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http://dx.doi.org/10.1016/j.ijporl.2020.110032DOI Listing
July 2020

Evidence-based telehealth clinical pathway for pediatric tympanostomy tube otorrhea.

Int J Pediatr Otorhinolaryngol 2020 Jul 27;134:110027. Epub 2020 Mar 27.

Department of Otolaryngology, University of Pittsburgh School of Medicine, Pittsburgh, PA, USA.

Introduction: As healthcare moves away from volume-based to value-based delivery models, evidence based clinical pathways detail essential steps in patient care to reduce the costs and utilization of health care resources. Ideal pathways lead towards standardized, patient-centered care through an algorithm that is evidence-based, interventions with criteria-based progression, and measurable endpoints or quality indicators. Using these standards, a clinical pathway for managing tympanostomy tube otorrhea beginning with phone triage was developed in accordance with AAO-HNSF Guidelines.

Methods: A retrospective case series of all consecutive patients calling the otolaryngology nurse's line at a tertiary pediatric hospital 3/2018-11/2018 regarding otorrhea was performed. Nurses completed a standardized and evidence-based form based on parent responses regarding purulence, tympanostomy tubes/perforation, fever>102°, ear redness, bacterial rhinosinusitis, sore throat, and immunodeficiency, which was sent to the advanced practice providers (APPs) to assess for antibiotic drops. Otorrhea form information and tympanostomy tube history, subsequent phone calls, clinic visits, and antibiotic prescriptions for otorrhea were extracted.

Results: Eighty-two patients were included. Median child age at phone call was 2.5 years (range 0.3-20.2 years), and 45.1% were female. All patients had prior tubes and active purulent otorrhea. No parents reported cellulitis or immunodeficiency. One patient had symptoms of bacterial rhinosinusitis and a sore throat but had already been seen by their primary care provider (PCP) for systemic antibiotics. Antibiotic drops were prescribed by an APP in 96.3% of cases [ofloxacin (n = 57), ciprofloxacin (n = 17), or ciprofloxacin with dexamethasone (n = 5)]. The remaining patients already had drops (2.5%) or were referred to their PCP (1.2%). Twenty (24.4%) received another antibiotic prescription and 17.1% had a subsequent clinic or urgent care visit for otorrhea.

Conclusions: This pathway obviated clinic visits in 82.9% of patients with a 75.6% treatment cure.
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http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7282932PMC
July 2020

Management and Outcomes of Button Batteries in the Aerodigestive Tract: A Multi-institutional Study.

Laryngoscope 2021 01 18;131(1):E298-E306. Epub 2020 Feb 18.

Division of Pediatric Otolaryngology, UPMC Children's Hospital of Pittsburgh, Pittsburgh, Pennsylvania.

Objectives/hypothesis: To describe the clinical presentation, management, and complications associated with button battery impaction in the aerodigestive tract in children.

Study Design: Retrospective case series.

Methods: This multi-institutional study, endorsed by the American Society of Pediatric Otolaryngology research consortium, is a retrospective medical record review, including all children at five tertiary-care institutions presenting with button batteries impacted in the aerodigestive tract between January 2002 and December 2014. Battery type/size, duration and location of impaction, presenting symptoms, treatment, complications, and outcomes were examined.

Results: Eighty-one patients were included (64.2% male), with ingestion witnessed in 20 (24.7%). Median age at presentation was 3 years (range, 1 week-14 years). Median time from diagnosis to removal was 2.5 hours (range, 0.4-72 hours). Locations included the esophagus (n = 48), hypopharynx (n = 1), stomach (n = 6), nasal cavity (n = 22), and ear canal (n = 4). Most common symptoms for esophageal/hypopharyngeal impactions included dysphagia (26.5%), nausea/vomiting (26.5%), drooling (24.5%), cough (18.4%), and fever (18.4%). Most common symptoms for nasal impactions included epistaxis (54.6%), rhinorrhea (40.9%), nasal pain (27.3%), and fever (22.7%). Almost all esophageal impactions were from 3-V (89.5%), 20-mm (81.8%) lithium batteries. Severe esophageal complications included stricture (28.6%), perforation (24.5%), tracheoesophageal fistula formation (8.2%), pneumothorax (4.1%), and bilateral true vocal fold paresis (4.1%). Nasal complications included necrosis (59.1%), septal perforation (27.3%), and saddle nose deformity (4.5%). Duration of impaction correlated with an increased likelihood of persistent symptoms only for nasal batteries (P = .049).

Conclusions: Button batteries in the upper pediatric aerodigestive tract or ear canal should be considered a surgical emergency, requiring urgent removal and careful vigilance for complications.

Level Of Evidence: 4 Laryngoscope, 131:E298-E306, 2021.
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January 2021

Utilization of Diagnostic Testing for Renal Anomalies and Congenital Heart Disease in Patients with Microtia.

Otolaryngol Head Neck Surg 2020 Apr 21;162(4):554-558. Epub 2020 Jan 21.

Department of Otolaryngology, Children's Hospital of Pittsburgh of the University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania, USA.

Objective: Congenital ear anomalies are associated with congenital cardiac and renal defects. Renal ultrasounds, electrocardiogram, and echocardiogram can be utilized for diagnosis of these concurrent defects. No standard of care exists for the workup of patients with microtia. The goals of this study were to describe the utilization of diagnostic testing for cardiac and renal anomalies and to identify their prevalence in patients with microtia.

Study Design: Case series with chart review.

Setting: Children's Hospital of Pittsburgh of the University of Pittsburgh Medical Center.

Subjects And Methods: This study is an Institutional Review Board-approved retrospective review of consecutive patients born between 2002 and 2016 who were diagnosed with microtia and seen in the otolaryngology clinic at a tertiary care children's hospital. Demographics, sidedness and grade of microtia, comorbid diagnoses, and details of renal and cardiovascular evaluations were recorded. Factors associated with retroperitoneal ultrasound and cardiac testing were assessed with logistic regression.

Results: Microtia was present in 102 patients, and 98 patients were included as they received follow-up. Microtia was associated with craniofacial syndrome in 34.7% of patients. Renal ultrasound was performed in 64.3% of patients, and 12.9% of patients with ultrasounds had renal aplasia. Cardiac workup (electrocardiogram or echocardiogram) was completed in 60.2% of patients, and of this subset, 54.2% had a congenital heart defect.

Conclusion: Diagnostic testing revealed renal anomalies and cardiac defects in patients with isolated microtia at a higher rate than in the general population. This suggests the need for further evaluation of the role of routine screening in patients with microtia.
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April 2020

The use and cost-effectiveness of intraoperative navigation in pediatric sinus surgery.

Laryngoscope 2020 12 26;130(12):E742-E749. Epub 2019 Dec 26.

Division of Pediatric Otolaryngology, University of Pittsburgh Medical Center Children's Hospital of Pittsburgh, Pittsburgh, Pennsylvania.

Objectives/hypothesis: There are consensus statements about when to use intraoperative navigation (IN) in adult sinus surgery. However, no corresponding guidelines exist for pediatrics. Our objectives included: 1) assess the demographic and operative factors associated with IN use and 2) calculate the cost-effectiveness of IN use.

Study Design: Retrospective chart review.

Methods: One hundred nineteen pediatric patients undergoing sinus surgery between 2003 and 2016 were reviewed. Demographic and surgical factors were collected from medical records. Costs associated with use of IN were gathered from billing records.

Results: Of the 119 patients, 60 underwent sinus surgery with navigation (wIN) and 59 underwent surgery without navigation (sIN). Children in the wIN group had more complex surgeries with more sinuses opened (P = .008). Individual attending surgeon and presence of trainee were associated with increased use of IN (P < .001 for both). IN resulted in a median of 31.5 minutes longer surgical time (P < .001). IN had an incremental cost/effectiveness ratio (ICER) of $22,378 for each year without revision surgery for patients with acute disease. However, for patients with chronic disease, the probability of undergoing a second surgery was the same between wIN and sIN groups, and navigation was not cost-effective (ICER of -$3,583).

Conclusions: IN use did not decrease complications or rates of revision surgery. It was used primarily as an educational tool or to increase confidence in intraoperative identification of landmarks. However, the use of IN added surgical time and was not cost-effective. Further research must be completed to determine when IN is indicated in pediatric sinus surgery.

Level Of Evidence: 4 Laryngoscope, 2019.
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December 2020

Eustachian Tube Dysfunction in Children With Unilateral Cleft Lip and Palate: Differences Between Ipsilateral and Contralateral Ears.

Cleft Palate Craniofac J 2020 06 23;57(6):723-728. Epub 2019 Dec 23.

Department of Otolaryngology, Children's Hospital of Pittsburgh of UPMC, Pittsburgh, PA, USA.

Objective: To evaluate Eustachian tube dysfunction in the ipsilateral and contralateral ears, in children with unilateral cleft lip and palate (UCLP).

Design: Retrospective chart review.

Setting: Tertiary care children's hospital.

Patients: Seventy-four consecutive patients with UCLP born between 2005 and 2011 and treated at UPMC Children's Hospital of Pittsburgh Cleft-Craniofacial Center were included.

Main Outcome Measures: Conductive hearing loss, tympanogram type, number of middle ear effusions, tympanostomy tubes, and complications. Hypothesis was formulated prior to data collection.

Results: Conductive hearing loss was nearly twice as common in the ipsilateral ear (43.2%) compared with contralateral (23.0%; = .001, McNemar test). There were no significant differences in the frequency of each type of tympanogram between the contralateral and ipsilateral ears. The proportions of ipsilateral (90.5%) and contralateral (91.9%) ears with effusion were not significantly different. The total number of tubes received was not significantly different between the 2 ears (median of 2 bilaterally). When combined, complications (retractions, perforations, and cholesteatomas) were significantly more common in the ipsilateral ear (29.7%) compared with the contralateral ear (18.9%; = .039, McNemar test).

Conclusion: In children with UCLP, there were significantly more instances of conductive hearing loss and complications on the cleft side compared to the noncleft side. This suggests that Eustachian tube dysfunction may indeed be more severe on the cleft side. Considering this information, clinicians may need to be especially observant of the ipsilateral ear.
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June 2020

The Generalizability of the Clinical Assessment Score-15 for Pediatric Sleep-Disordered Breathing.

Laryngoscope 2020 09 29;130(9):2256-2262. Epub 2019 Nov 29.

Research Division, State University of New York Downstate Medical Center, Brooklyn, New York.

Objective: The Clinical Assessment Score-15 (CAS-15) has been validated as an office-based assessment for pediatric sleep-disordered breathing in otherwise healthy children. Our objective was to determine the generalizability of the CAS-15 in a multi-institutional fashion.

Methods: Five hundred and thirty children from 13 sites with suspected sleep-disordered breathing were recruited, and the investigators completed the CAS-15. Based on decisions made in the course of clinical care, investigators recommended overnight polysomnography, observation, medical therapy, and/or surgery. Two hundred and forty-seven subjects had a follow-up CAS-15.

Results: Mean age was 5.1 (2.6) years; 54.2% were male; 39.1% were white; and 37.0% were African American. Initial mean (standard deviation [SD]) CAS-15 was 37.3 (12.7), n = 508. Spearman correlation between the initial CAS-15 and the initial apnea-hypopnea index (AHI) was 0.41 (95% confidence interval [CI], 0.29, 0.51), n = 212, P < .001. A receiver-operating characteristic curve predicting positive polysomnography (AHI > 2) had an area under the curve of 0.71 (95% CI, 0.63, 0.80). A score ≥ 32 had a sensitivity of 69.0% (95% CI, 61.7, 75.5), a specificity of 63.4% (95% CI, 47.9, 76.6), a positive predictive value of 88.7% (95% CI, 82.1, 93.1), and a negative predictive value of 32.9% (95% CI, 23.5, 44.0) in predicting positive polysomnography. Among children who underwent surgery, the mean change (SD) score was 30.5 (12.6), n = 201, t = 36.85, P < .001, effect size = 3.1.

Conclusion: This study establishes the generalizability of the CAS-15 as a useful office tool for the evaluation of pediatric sleep-disordered breathing.

Level Of Evidence: 2B Laryngoscope, 130:2256-2262, 2020.
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September 2020

The Association Between Age at Palatoplasty and Speech and Language Outcomes in Children With Cleft Palate: An Observational Chart Review Study.

Cleft Palate Craniofac J 2020 02 24;57(2):148-160. Epub 2019 Oct 24.

Division of Pediatric Otolaryngology, UPMC Children's Hospital of Pittsburgh, PA, USA.

Objective: To determine whether timing of palatoplasty (early, standard, or late) is associated with speech and language outcomes in children with cleft palate.

Design: Retrospective case series.

Setting: Tertiary care children's hospital.

Participants: Records from 733 children born between 2005 and 2015 and treated at the Cleft Craniofacial Clinic of a tertiary children's hospital were retrospectively reviewed. Exclusion criteria were cleft repair at an outside hospital, intact secondary palate, absence of postpalatoplasty speech evaluation, syndromes, staged palatoplasty, and introduction to clinic after 12 months of age. Data from 232 children with cleft palate ± cleft lip were analyzed.

Interventions: Palatoplasty.

Main Outcome Measures: Speech/language delays and disorders at 20 months and 5 years of age based on formal hospital or community-based testing or screening evaluation in the Cleft Craniofacial Clinic; additional speech surgery.

Results: Median age at palatoplasty was 12.6 months (range: 8.8-21.9 months). Age at palatoplasty was classified as early (<11 months, n = 28), standard (11-13 months, n = 158), or late (>13 months, n = 46). Late palatoplasty was associated with increased odds of speech/language delays and speech therapy at 20 months, and language delays at 5 years, compared with standard or early palatoplasty ( < .05 for all comparisons). However, speech sound production disorders, velopharyngeal incompetence, tube replacement, and hearing loss were not significantly associated with age at palatoplasty.

Conclusions: Late palatoplasty may be associated with short- and long-term delays in speech/language development. Future studies with standardized surgical technique/timing and outcome measures are required to more definitively describe the impact of age at palatoplasty on speech/language development.
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February 2020

Consideration of Cough Reflex Development When Ordering Modified Barium Swallow Studies in Infants.

Dysphagia 2020 06 6;35(3):533-541. Epub 2019 Sep 6.

UPMC Children's Hospital of Pittsburgh, 4401 Penn Avenue, Pittsburgh, PA, 15224, USA.

Infants < 51 weeks post-menstrual age (< 51 PMA) are often referred for modified barium swallow (MBS) studies for suspected silent aspiration (SA) given a possible association between SA and aspiration pneumonia. Infants this young are unlikely to have developed a mature laryngeal cough reflex, most likely rendering SA an expected finding in those who aspirate. The aims of this retrospective review were to (1) determine if SA resolves in a significant proportion of infants around the expected emergence of the laryngeal cough reflex, (2) determine which factors or characteristics are associated with and without SA resolution in these infants, and (3) determine if SA, or any aspiration, is associated with increased rates of lower respiratory infection (including aspiration pneumonia) in these infants. Results from the chart review revealed that 79/148 (53.4%) infants had SA on MBS < 51 PMA. 16/48 (33.3%) infants assessed for SA by the time of the expected emergence of the cough reflex had resolution. SA resolution was less common in infants with obstructive sleep apnea (p = 0.037). A total of 50/70 (71.4%) infants with a follow-up MBS had eventual SA resolution. Aspiration was not significantly associated with LRI, including aspiration pneumonia. The results suggested that the laryngeal cough reflex might develop later than reported in the literature and there is no association between aspiration and LRI. These findings may indicate that age should be considered before ordering an MBS solely to assess for SA in this population. The study provides preliminary evidence for future prospective research regarding SA resolution.
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June 2020

Pediatric dysphonia: It's not about the nodules.

Int J Pediatr Otorhinolaryngol 2019 Oct 4;125:147-152. Epub 2019 Jul 4.

Department of Audiology and Communication Disorders, UPMC Children's Hospital of Pittsburgh, Pittsburgh, PA, USA.

Objective: Despite the fact that vocal nodules are the most common cause of chronic dysphonia in children, uncertainty and lack of consensus complicates practically every diagnostic and management decision. Selecting an optimal staging system is fundamental to understanding a disease process, mandatory for uniform reporting, and crucial to predicting natural history and treatment outcomes. The ideal prognostic model for vocal nodules is under intense debate. The purpose of this study was to analyze the predictive power of vocal nodule grade to severity of voice metrics in children.

Methods: Seventy-nine patients diagnosed with vocal cord nodules between 2006 and 2012 were drawn from UPMC Children's Hospital of Pittsburgh Voice, Resonance and Swallowing Center Research Registry. Subject age at time of diagnosis, nodule grade, relevant co-morbidities, scores on The Consensus Auditory-Perceptual Evaluation of Voice (CAPE-V), parent-reported Pediatric Voice Handicap Index (pVHI), the phonotraumatic behaviors profile, habitual speaking pitch fundamental frequencies, pitch range, volume intensity, and s/z ratio were recorded and compiled into a de-identified database for analysis.

Results: Based on the Kruskal-Wallis H Test, there was no statistically significant correlation between nodule grade and total pitch range (p = .21), s/z ratio (p = .50), volume intensity (p = .33), overall CAPE-V Scores (p = .15), or pVHI Scores (p = .29). Chi-squared tests also revealed no significant associations between nodule grade and abnormality in habitual speaking pitch (p = .14 for fundamental frequency while sustaining a vowel sound, p = .37 for fundamental frequency while speaking structured tasks i.e. counting, or p = .76 while speaking in conversation).

Conclusion: The current "gold-standard" for grading vocal nodule size suggests that the nodules themselves are not driving the standard dysphonic metrics that are most commonly collected and monitored in such children. This outcome is consistent with other studies reporting similar findings and was expected based on the inconsistencies in the reported literature to date. By extension, the conventional wisdom of avoiding surgical treatment of vocal nodules in children seems prudent as there is little evidence to suggest that the nodules themselves are "driving" the severity of the dysphonia. Ultimately identifying the true "drivers" of dysphonia in children will suggest alternative therapies that are more specific and directed to the pathophysiology. Most pediatric voice care professionals will welcome such discoveries as those in the front line of patient care are often rendered helpless and frustrated.
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October 2019

Pediatric nasal septal perforation.

Int J Pediatr Otorhinolaryngol 2019 Mar 4;118:15-20. Epub 2018 Dec 4.

Department of Otolaryngology, University of Pittsburgh Medical Center, 200 Lothrop Street Suite 500, Pittsburgh, PA, 15213, USA; Department of Pediatric Otolaryngology, Children's Hospital of Pittsburgh, 4401 Penn Avenue #3, Pittsburgh, PA, 15224, USA.

Objective: 1.) Describe demographic and clinical characteristics of pediatric nasal septal perforations (NSP), 2.) Analyze efficacy of treatment modalities in symptom management and resolution of pediatric NSP, 3.) Describe the surgical technique of external rhinoplasty with vascularized nasal septal flaps in the treatment of nasal septal perforations.

Methods: IRB-approved retrospective chart review of pediatric patients ages 0-18 years with nasal septal perforations treated at a tertiary care pediatric otolaryngology practice. Demographic and clinical characteristics including gender, age, race, and presenting symptoms, and location, size, and etiology of perforation were collected. Outcomes including persistence of perforation and symptoms at 1 year and most recent visit were recorded. A total of 20 patients were included. Statistical analysis used Fisher's t-test for categorical variables and Wilcoxon rank-sum for continuous variables.

Results: Median age was 167.5 months (1.5-221.0). The most common etiology was iatrogenic (40%), followed by button battery (20%). Thirty percent of patients underwent surgical repair. Fifty percent of patients who underwent surgical repair achieved closure of their perforation at most recent follow up.

Conclusion: Pediatric NSP is a challenging issue with limited literature to date. Iatrogenic causes (40%) and button batteries (20%) were the most common etiologies of nasal septal perforation in our study. We introduce an advancement in our center's surgical technique with a case illustration with repair via external rhinoplasty and bilateral vascularized nasal septal flaps. Future larger studies may further elucidate characteristics and treatment modalities associated with successful closure.
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March 2019

Airway anomalies in patients with craniosynostosis.

Laryngoscope 2019 Nov 19;129(11):2594-2602. Epub 2018 Nov 19.

Department of Otolaryngology, Children's Hospital of Pittsburgh of UPMC, University of Pittsburgh School of Medicine, Pittsburgh, Pennsylvania, U.S.A.

Objectives: 1) Characterize the spectrum of airway anomalies in patients with craniosynostosis, and 2) identify clinical characteristics of these patients that may be associated with the development of airway anomalies.

Methods: This study is a retrospective case series assessing the type and frequency of airway anomalies in all patients with craniosynostosis seen at a tertiary-care children's hospital between 2000 and 2016. Cohort analyses were then performed to identify differences in airway anomalies dependent on syndromic associations, multisutural fusion, and location of suture fusion. Clinical characteristics examined included demographics and additional neurologic and craniofacial abnormalities.

Results: Four hundred and ninety-six patients with craniosynostosis (83.5% white, 64.5% male; 33.9% sagittal, 28.8% metopic, 11.5% coronal, 1.2% lambdoid, and 24.6% multisutural) were included. Notable airway anomalies included the following: 13.3% adenotonsillar hypertrophy, 8.9% laryngomalacia, 7.3% tracheomalacia, 7.1% subglottic stenosis, 4.0% bronchomalacia, 3.8% laryngeal cleft, and 1.2% vocal fold paresis. Multisutural craniosynostosis patients (n = 122) were more likely to have obstructive sleep apnea (P = 0.005), adenotonsillar hypertrophy (P = 0.014), tracheomalacia (P = 0.011), subglottic stenosis (P < 0.001), and epiglottic/base of tongue collapse (P = 0.003) and require tracheostomy (P = 0.001) and mechanical ventilation (P = 0.017) compared with single suture craniosynostosis. Syndromic craniosynostosis patients (n = 33) were more likely to have obstructive sleep apnea (P < 0.001), laryngomalacia (P = 0.047), and subglottic stenosis (P = 0.009) compared with nonsyndromic patients.

Conclusion: Airway anomalies are prevalent in patients with craniosynostosis; patients with multisutural or syndromic types have an increased risk of developing certain abnormalities. There should be a lower threshold for referral for airway evaluation in these populations.

Level Of Evidence: 4. Laryngoscope, 129:2594-2602, 2019.
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November 2019

The Impact of Timing of Tympanostomy Tube Placement on Sequelae in Children With Cleft Palate.

Cleft Palate Craniofac J 2019 07 11;56(6):720-728. Epub 2018 Nov 11.

1 Department of Otolaryngology, Children's Hospital of Pittsburgh of UPMC, Pittsburgh, PA, USA.

Objective: To describe the impact of timing of tympanostomy tube insertion on the number of tubes received and complications in children with routine tube placement.

Design: Retrospective case series.

Setting: Tertiary care children's hospital.

Participants: Records from a consecutive sample of 401 children with cleft palate were reviewed. Sixty-five patients with isolated cleft palate and 82 patients with cleft lip and palate had follow-up until 5 years of age and were included.

Interventions: Tympanostomy tubes.

Main Outcome Measure(s): Number of tubes received and tube-related complications. The hypothesis was formulated prior to data collection.

Results: Males comprised 55.8% of included patients, and tubes were placed in 98.6% of patients at a median age of 6.5 months. Effusion was documented at first tube placement for 96.5% of patients. Most (67.4%) patients required replacement of tubes, and 10.6% required long-term tubes. Complications included otorrhea (71.0%), myringosclerosis (35.2%), granulation (22.8%), perforation (17.9%), retained tubes (5.5%), and cholesteatoma (1.4%). Cleft lip and palate ( < .001) and otorrhea ( = .023) were associated with tube placement before palatoplasty. Patients with tube placement before palatoplasty ( = .033), genetic disorders ( = .007), failed newborn hearing screen ( = .012), otorrhea ( < .001), and granulation ( < .001) received more tubes.

Conclusions: Nearly universal effusion in patients with cleft palate supports the need for routine tube placement. The potential for otorrhea and requiring more tubes should be weighed against the risks associated with prolonged effusion when considering tube placement before palatoplasty.
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July 2019

Outcomes and swallowing evaluations after injection laryngoplasty for type I laryngeal cleft: Does age matter?

Int J Pediatr Otorhinolaryngol 2018 Dec 13;115:10-18. Epub 2018 Sep 13.

Paul C. Gaffney Division of Pediatric Hospital Medicine, UPMC Children's Hospital of Pittsburgh, USA; University of Pittsburgh School of Medicine, USA. Electronic address:

Objectives: To improve the recognition of differences in presentation amongst patients with type 1 laryngeal clefts of various ages and better understand the age dependent outcomes of injection laryngoplasty. A second aim was to analyze the discrepancies between swallow assessment modalities in various age groups with type I laryngeal clefts undergoing injection laryngoplasty.

Methods: A retrospective review of electronic medical records of patients who underwent injection laryngoplasty from 2009 through 2015 at a tertiary care children's hospital. Data extracted included: Demographics, histories and physical exam findings, diagnostic studies, and medical and surgical treatments.

Results: Most (72/102, 70.6%) patients were male with a median gestational age at birth of 37 weeks (range 24-41 weeks). Formula thickening and GERD medications were used in 94/102 (92.2%) and 97/102 (95.1%) patients, respectively. Comorbid GERD, laryngomalacia, tracheomalacia, and subglottic stenosis were present in 98/102 (96.1%), 40/102 (39.2%), 9/102 (8.8%), and 14/102 (13.7%) patients, respectively. There was no significant difference in demographics, comorbidities or medical therapy between age groups. Symptoms at presentation differed between age groups with stridor (χ(1) = 11.6, p = 0.002) and cyanosis (χ(1) = 8.13, p = 0.012) being more common in the 0-3-month group compared to the 12-36 month group. Symptom resolution and the odds of undergoing additional surgery (second injection or suture repair) over time, however, did not differ. There was a significant reduction in aspiration with thins during FEES (McNemar χ(1) = 10.7, p = 0.002) and aspiration with nectar during MBS (McNemar χ(1) = 5.26, p = 0.035) post-injection. After injection, there was significant agreement in aspiration with thins between FEES and MBS (kappa = 0.308 ± SE 0.170, p = 0.035). However, finding aspiration with thins was more common during MBS than during FEES (McNemar χ(1) = 7.00, p = 0.016). There were no differences in swallow evaluation findings between the age groups.

Conclusions: Symptoms of type I laryngeal clefts may differ by age. However, there was no impact of age on the safety and efficacy of surgical intervention.
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December 2018

Laryngomalacia in Patients With Craniosynostosis.

Ann Otol Rhinol Laryngol 2018 Aug 20;127(8):543-550. Epub 2018 Jun 20.

1 Department of Otolaryngology, University of Pittsburgh School of Medicine, Pittsburgh, Pennsylvania, USA.

Objective: To characterize differences in the clinical presentation and treatment outcomes of laryngomalacia in patients with and without craniosynostosis.

Methods: A retrospective cohort study was performed comparing all patients with concomitant laryngomalacia and craniosynostosis seen at a tertiary care children's hospital between 2000 and 2016 with a control group of patients with isolated laryngomalacia. Thirty-two patients with craniosynostosis (59% male) and 68 control patients (56% male) were included. There were no significant differences in age of diagnosis or incidence of prematurity. Symptom presentation, disease severity, swallowing function, comorbidities, treatment modalities, and outcomes were examined using logistic regression.

Results: Patients with craniosynostosis had increased odds of presenting with stertor (odds ratio [OR] = 3.41, P = .022), increased work of breathing (OR = 18.8, P = .007), obstructive sleep apnea (OR = 8.48, P = .003), dysphagia (OR = 3.40, P = .008), and aspiration (OR = 40.2, P < .001) and decreased odds of presenting with stridor (OR = 0.0804, P < .001) compared with controls. Patients with craniosynostosis had increased odds of severe laryngomalacia (OR = 5.00, P = .031) and other airway anomalies such as tracheomalacia (OR = 5.73, P = .004), bronchomalacia (OR = 15.5, P = .013), and subglottic stenosis (OR = 2.75, P = .028). Treatment of patients with craniosynostosis was more likely to include tracheostomy (OR = 24.8, P < .001) and gastrostomy tube (OR = 88.4, P < .001). There were no significant differences in rates of supraglottoplasty.

Conclusion: Clinical presentations, comorbidities, and treatments of laryngomalacia are significantly different in the context of craniosynostosis.
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August 2018

Prognostic Factors of Pediatric Revision Ossicular Chain Reconstruction.

Otol Neurotol 2018 07;39(6):724-731

Department of Otolaryngology, University of Pittsburgh School of Medicine.

Objective: Ossicular chain reconstruction (OCR) is a surgical method used to repair conductive hearing loss. In children, up to 40% of OCRs will require revisions. In this study, our aims were to validate improvement in hearing outcomes following revision OCR and identify prognostic factors for successful revision OCRs.

Design: Retrospective review at the Children's Hospital of Pittsburgh from 2003 to 2014.

Setting: Tertiary referral center.

Patients: Pediatric patients undergoing multiple OCRs who had demographic, operative, and pre- and postoperative audiogram data. Patients included in our study had ≥ 2 OCR procedures to the same ear. Forty-three of 123 patients met inclusion criteria.

Main Outcome Measures: Pre- and postoperative audiograms were used to compare the changes in pure-tone averages (PTA), speech recognition thresholds, and air-bone gaps following OCR surgeries.

Results: Revision OCRs significantly improved PTA, speech recognition thresholds, and air-bone gaps (p values= 0.003, 0.004, 0.005, respectively) and seven patients (14%) had return of normal hearing (PTA < 25 dB). Cholesteatoma at the time of first revision was associated with additional OCR revisions (hazard ratio [HR]: 10.3; p=0.013), while patients who had canal wall down or no mastoidectomy during first OCR revision were less likely to require additional revision compared with patients who had canal wall up mastoidectomy (HR: 0.170, p = 0.041 versus canal wall down; HR: 0.242, p = 0.041 versus no mastoidectomy).

Conclusions: Revision OCR(s) are an acceptable method to improve residual conductive hearing loss following primary OCR surgery in pediatric patients. Patients with cholesteatoma and canal wall up at the time of revision OCR have increased chances of requiring further revision.
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http://dx.doi.org/10.1097/MAO.0000000000001837DOI Listing
July 2018

Outcomes of adenoidectomy-alone in patients less than 3-years old.

Int J Pediatr Otorhinolaryngol 2018 Mar 2;106:46-49. Epub 2018 Jan 2.

Division of Pediatric Otolaryngology, 4401 Penn Ave, Children's Hospital of Pittsburgh of UPMC, 7th Floor Faculty Pavilion, Pittsburgh, PA 15224, USA. Electronic address:

Objectives: 1. Determine the percentage of patients under the age of 3 undergoing adenoidectomy-alone who require subsequent management of residual sleep disordered breathing (SDB).2. Characterize complications following adenoidectomy and determine if any perioperative factors are associated with intra-operative or post-operative complications and outcomes.

Methods: Case series with chart review was conducted including children seen at a tertiary care children's hospital between 2008 and 2012. Consecutive patients under the age of 3 who underwent adenoidectomy-alone were identified by billing codes. After excluding those with syndromes, partial adenoidectomies, and those without follow-up, 148 patients were included. Predictors of requiring additional surgery for SDB were evaluated using log-rank tests or Cox proportional hazards regression.

Results: Median age at time of initial adenoidectomy was 27.5 months (range 11-36 months) and the patient population was comprised of 66.2% males (n = 98/148) and 89.2% Caucasians (n = 132/148). 56.5% (n = 74/131) of patients continued to have residual symptoms of SDB and 34.5% (n = 51/148) underwent additional surgical intervention. Multivariable survival analysis revealed GERD (HR, 6.21; CI, 1.29-29.77, p = .022) and tonsil size (HR, 4.07; CI, 1.57-10.51, p = .004) were significant predictors of additional surgery in this group of patients under the age of 3. There was no observed difference in intra- and post-operative complication rates between patients with and without additional operative intervention.

Conclusions: Residual SDB symptoms following adenoidectomy in patients less than 3 years of age are common and require additional surgery at a high rate. Medical comorbidities such as GERD and large tonsil size may help predict the need for additional surgery.
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http://dx.doi.org/10.1016/j.ijporl.2017.12.030DOI Listing
March 2018