Progressive Decline in Voice and Voice-Related Quality of Life in X-Linked Dystonia Parkinsonism.

OBJECTIVE: To characterize the evolution of swallowing and voice in patients with X-linked dystonia parkinsonism (XDP). STUDY DESIGN: Retrospective case series. METHODS: Retrospective review of 59 patients with XDP from January 2016 to January 2018. All patients underwent complete examinations and quality of life (QOL) surveys (Swallowing Quality of Life questionnaire [SWAL-QOL], Voice-Related Quality of Life [V-RQOL], and Voice Handicap Index [VHI]), and functional endoscopic examination of swallowing. We excluded patients with incomplete records or patients lost to follow-up. Univariate analysis was used to compare 2016 to 2018 Penetration-Aspiration Scale (PAS), SWAL-QOL, V-RQOL, and VHI scores. RESULTS: Ten patients met the inclusion criteria. Nine patients had oromandibular dystonia. Voice-related measures significantly worsened with an increase in mean VHI from 81 to 109.9 (P = 0.026) and decrease in mean V-RQOL from 58 to 28 (P = 0.013). Vocal strain also significantly worsened 0.4 to 1.4 (P = 0.001). Mean PAS scores increased from 4.2 to 5.1 (P = 0.068) and mean SWAL-QOL decreased from 50.4 to 43.5 (P = 0.157). In the SWAL-QOL, the mean Eating Duration score worsened from 0.9 to 0.4 (P = 0.052) and Mental Health score declined from 10.1 to 6.1 (P = 0.077). CONCLUSIONS: Both vocal strain and voice-related QOL measures considerably worsened over the 2-year interval in our limited group of XDP patients with no significant change in PAS scores or swallowing QOL. The findings demonstrated that the pace of disease affecting voice symptoms was different from swallowing symptoms in our study group and that changes in communication ability may be a more sensitive marker for disease progression than swallowing dysfunction.

The Poor Validity of Asymmetric Laryngoscopic Findings in Predicting Laterality in Vocal Fold Paresis.

OBJECTIVE: To determine the laryngoscopic findings most predictive of laterality in vocal fold paresis in patients with known RLN and/or SLN paresis by laryngeal electromyography (LEMG). STUDY DESIGN: Blinded, prospective video perceptual analysis study. METHODS: Patients with vocal fold paresis diagnosed by LEMG at a tertiary care hospital from 2017 to 2019 were identified. Two fellowship-trained laryngologists blinded to clinical history and LEMG results reviewed laryngostroboscopic examinations and assessed for evidence of paresis using defined criteria. Inclusion criteria were adults with laryngeal asymmetry and evidence of decreased recruitment on LEMG. Exclusion criteria were children, presence of laryngeal lesions, myasthenia gravis, vocal fold paralysis, and normal laryngeal symmetry. RESULTS: We identified 95 patients who were diagnosed with vocal fold paresis with LEMG who met inclusion and exclusion criteria (mean age 43.8 ± 20.4 years (18-88), 38.9% male). When comparing the laterality of the observed laryngoscopic finding with LEMG, we found that in patients who had severe true vocal fold (TVF) range of motion disturbance, the laterality of the finding matched the LEMG distribution of paresis in 12 out of 13 (92.3%) patients (P = 0.002). No other laryngoscopic findings reliably predicted laterality including corniculate and cuneiform cartilage asymmetry, pyriform sinus dilation, abnormal TVF show, petiole deviation, abnormal ventricular show, increased supraglottic area, and FVF hyperfunction of the opposite side. CONCLUSION: With the exception of severe TVF range of motion disturbance, there seems to be poor validity of laryngoscopic findings in predicting the affected side in vocal fold paresis. We recommend neurophysiologic testing to confirm the clinical diagnosis of vocal fold paresis.

Long-term outcomes of aminolevulinic acid photodynamic therapy for treatment of recalcitrant laryngeal premalignant lesions.

OBJECTIVE: To assess long-term outcomes of ALA-PDT in treating recalcitrant laryngeal leukoplakia. STUDY DESIGN: Retrospective Case-Control. METHODS: We reviewed all laryngeal leukoplakia patients treated with ALA-PDT compared with angiolytic laser treatment alone (585 nm PDL or 532 nm KTP laser) from 2000 to 2019. Patients with laryngeal cancer (or a history of laryngeal cancer), leukoplakia previously treated with radiation and no pathologic report were excluded. Patient demographics, procedure details and outcomes were examined including histopathologic diagnosis, procedures performed, ALA usage, recurrence of leukoplakia and the development of cancer. RESULTS: We identified 132 patients with laryngeal leukoplakia: 42 were treated with ALA-PDT and 90 were treated with an angiolytic laser alone (Laser group). The proportion of cases of high-grade dysplasia was 57.1% in the ALA-PDT group compared to 32.2% in the Laser group. In high-grade dysplasia cases, there was a statistically significant better recurrence-free survival (RFS) at 12 months and 60 months in those who underwent ALA-PDT 71.4% and 7.1% vs Laser 25% and 0% (p = .01). However, for overall groups, there was no difference in RFS (p = .25). Voice outcomes (patient subjective report) improved or were stable in 75% of subjects with no serious side effects reported. CONCLUSION: ALA-PDT for recalcitrant and high-grade dysplasia is highly effective with improved recurrence-free survival compared to laser alone. ALA-PDT may be an appropriate therapy in patients who have failed prior angiolytic laser alone.

How Patients Choose a Laryngologist: A Pilot Stated Preference Study.

OBJECTIVE: Patients consider many factors when deciding how to receive medical care. This study used best-worst scaling (BWS), a technique novel to otolaryngology, to quantitatively examine preferences among patients choosing a laryngologist. Our objective was to quantify in a pilot cohort the relative importance patients place on a variety of attributes when seeking a laryngologist. STUDY DESIGN: BWS survey. SETTING: Academic voice clinic. METHODS: New patients were recruited to take a computerized BWS survey developed using attributes derived from patient input, expert opinion, and literature review. Attributes were grouped into 4 categories: physician reputation, physician qualifications, hospital-related factors, and other nonclinical factors. Responses were analyzed using multinomial logit regression to determine importance scores and associations with other variables. RESULTS: Eighty-seven of 93 patients recruited participated (93.5% response rate). Physician qualifications were the most important attributes to patients, with specialty laryngology training receiving the highest importance score (20.8; 95% CI, 20.2 to 21.5; P < .0001). Recommendations from referring physicians (15.6; 95% CI, 14.3 to 16.9) and use of cutting-edge technology (11.9; 95% CI, 10.7 to 13.1) were the second and third most important, respectively. Least important were nonclinical factors, including wait time to get an appointment (4.3; 95% CI, 2.8 to 5.8) and convenience of office location (1.5; 95% CI, 0.9 to 2.1). Just over half of patients (51.2%) reported willingness to wait 4 weeks for an appointment with a laryngologist. Older patients were less concerned with convenience-related factors. CONCLUSION: Nonclinical factors were less important to patients than clinical factors, and laryngology-specific training was paramount. Stated preference methodologies can elucidate underlying preferences and help providers make care more patient centered.

Assessment of side effects after serial intralesional steroid injections for idiopathic subglottic stenosis.

PURPOSE: To assess the incidence and severity of 12 systemic side effects of serial intralesional steroid injections (SILSI) in patients with idiopathic subglottic stenosis (iSGS). METHODS: This retrospective study included patients with iSGS who underwent SILSI with Triamcinolone 40 mg/dL. After SILSI, the patients were asked to answer 12 questions regarding frequently encountered systemic side effects of steroids. Each answer was rated as mild, moderate, or severe. Descriptive statistics were used to analyze and present the findings. RESULTS: The study included 49 patients (42 female and 7 male) with a mean age of 59.1 years (range 21-83 years). Post-SILSI treatment, 27 (55%) reported experiencing side effects while 22 (45%) patients reported no side effects. The most frequent side effect reported in women of reproductive age (n: 8) was menstrual irregularities (3/8, 37%). Other frequently reported side effects were feeling joyful and sleeping difficulties, each reported by 30% of the patients. All side effects resolved after the completion of SILSI. CONCLUSIONS: SILSI can be administered with minimal tolerable side effects. Clinicians should make their patients aware of the most frequent side effects. Special attention should be given to women of reproductive age to inform them of the possibility of menstrual irregularities during SILSI.

The Effectiveness of Nortriptyline and Tolerability of Side Effects in Neurogenic Cough Patients.

OBJECTIVE: To determine the effectiveness of nortriptyline and tolerability of side effects in the treatment of neurogenic cough. Secondary goal is to evaluate the association between laryngeal asymmetry and clinical response to nortriptyline. STUDY DESIGN: Retrospective case series. MATERIALS AND METHODS: Consecutive patients diagnosed with neurogenic cough at a quaternary care specialty hospital from 2001 to 2020 were identified. Subjects <18 years old, not treated with nortriptyline, did not have a nasolaryngoscopic examination and were lost to follow-up were excluded. Charts were reviewed for demographic information, clinical history, nasolaryngoscopic findings, medication dosage, side effects, and follow-up time. RESULTS: Forty-two patients met inclusion and exclusion criteria, 7 males and 35 females with an average age of 56.5 (±13.1) years. There were 26/36 (72.2%) responders and 10/36 (27.8%) non-responders; 6 patients stopped nortriptyline due to side effects and were not included in the response comparison. Laryngeal asymmetry was present in 36/42 (85.7%) patients. No factors related to laryngeal asymmetry were significantly different between responders and non-responders. Medication tolerance was observed in 3/42 (7.1%) patients. Side effects were reported in 16/42 (38.1%) patients. The most common side effects were sedation 9/42 (21.4%) and xerostomia 3/42 (7.1%). CONCLUSION: Nortriptyline is effective for treating neurogenic cough with 72% of patients reporting improvement in cough. Evidence of laryngeal asymmetry was not associated with better treatment response. Although 38% experienced side effects, the majority of patients continued nortriptyline despite side effects. LEVEL OF EVIDENCE: 4.

Reliability of peak expiratory flow percentage compared to endoscopic grading in subglottic stenosis.

OBJECTIVE: To determine the reliability of pulmonary function testing compared to endoscopic grading in the assessment of subglottic stenosis. METHODS: Consecutively treated patients with subglottic stenosis at a tertiary care specialty hospital from 2009 to 2019 were identified. Two fellowship-trained laryngologists and two otolaryngologists blinded to clinical history reviewed laryngo tracheoscopic examinations and assessed the degree of stenosis using the Cotton-Myer grading system (% stenosis). Nine full flow-volume loops were performed at the time of each exam. RESULTS: The endoscopic images of 45 subjects were graded for degree of stenosis and the spirometry data were analyzed. The kappa values for Cotton-Myer grade overall was 0.37, grade I was -0.103, grade II was 0.052, and grade III was 0.045. The overall intraclass correlation of the physician grading of estimated percent obstruction (% stenosis) was 0.712 (P < .01) whereas the overall intraclass correlation for PEF% was 0.96 (P < .01). Within each Cotton-Myer grade, the intraclass correlation for % stenosis was 0.45 (P = .02) for grade I, 0.06 (P = .30) for grade II, and 0.16 (P = .03) for grade III. The intraclass correlation for PEF% for grade I was 0.97 (P < .01), grade II was 0.92 (P < .01), and grade III was 0.96 (P < .01). CONCLUSION: Cotton-Myer grading and estimating percent obstruction (% stenosis) for adult subglottic stenosis showed poor reliability as an assessment tool compared to the excellent intraclass correlation seen with pulmonary function tests within each Cotton-Myer grade subgroup. We recommend pulmonary function testing, specifically PEF% because it is a normalized value, for the assessment and management of subglottic stenosis. LEVEL OF EVIDENCE: 4.

Successful Use of High-Flow Nasal Cannula for Concurrent Vocal Cord Electromyography and Tubeless Microlaryngeal Surgery in a Spontaneously Breathing Adult Patient: A Case Report.

Ventilation during microlaryngoscopy previously included jet ventilation, microlaryngeal endotracheal tubes, and extended apnea. Historically, apneic oxygenation provided a tubeless field but limited operative time. Increased utilization of high-flow nasal cannula in intensive care units and operating rooms has created new opportunities to expand tubeless microlaryngoscopy. Although few studies have described high-flow nasal cannula for microlaryngoscopy, there remains much to be explored. In this case report, we describe the unique setting of utilizing high-flow nasal cannula in a spontaneously breathing patient to create an optimal tubeless surgical field for both microlaryngoscopy and vocal cord electromyography.

Supraglottoplasty surgery types 1-3: A practical classification system for laryngomalacia surgery.

OBJECTIVE: The objective of this study is to review the published literature on supraglottoplasty techniques for correcting laryngomalacia, and to subsequently provide a standardized classification system. METHODS: Three authors independently and systematically searched Pubmed/MEDLINE and six additional databases for all studies that included descriptions of supraglottoplasty techniques for correcting laryngomalacia. Sub-sites operated on and specific technique descriptions were reviewed, cataloged and subsequently categorized. This data was then used to develop a new classification system. RESULTS: 231 articles were identified, downloaded and reviewed in full text. 53 articles with 1669 patients from the included articles described in detail the supraglottoplasty procedure. 84 articles with 5731 patients had to be excluded secondary to not providing detail about the supraglottoplasty procedure. The resultant data identifies the need for a more standardized reporting of the supraglottoplasty procedure in order to more accurately evaluate technique specific outcomes. Currently 77% of the patients in the literature cannot be assessed for outcomes as they did not describe the details for the procedures. Eight variations of supraglottoplasty and four variations of epiglottis surgery were described. Based on the literature, we consolidated the surgery into the following types: Type 1: Debulking of arytenoids, Type 2: Division of aryepiglottic folds, Type 3: Epiglottis surgery. CONCLUSION: This descriptive review identified 53 articles with 1669 patients from the included articles detailing multiple variations of supraglottoplasty techniques. The new classification supplements a previously established system describing laryngomalacia, and simplifies the supraglottoplasty into three descriptive and logical types of categories. Our classification system would give surgeons a universal language to describe the supraglottoplasty performed, which could improve reporting of techniques, and facilitate future communication and research.

Double Barrel Nasal Trumpets to Prevent Upper Airway Obstruction after Nasal and Non-Nasal Surgery.

OBJECTIVES: During anesthesia emergence, patients are extubated and the upper airway can become vulnerable to obstruction. Nasal trumpets can help prevent obstruction. However, we have found no manuscript describing how to place nasal trumpets after nasal surgery (septoplasties or septorhinoplasties), likely because (1) the lack of space with nasal splints in place and (2) surgeons may fear that removing the trumpets could displace the splints. The objective of this manuscript is to describe how to place nasal trumpets even with nasal splints in place. MATERIALS AND METHODS: The authors describe techniques (Double Barrel Technique and Modified Double Barrel Technique) that were developed over three years ago and have been used in patients with obstructive sleep apnea (OSA) and other patients who had collapsible or narrow upper airways (i.e., morbidly obese patients). RESULTS: The technique described in the manuscript provides a method for placing one long and one short nasal trumpet in a manner that helps prevent postoperative upper airway obstruction. The modified version describes a method for placing nasal trumpets even when there are nasal splints in place. Over one-hundred patients have had nasal trumpets placed without postoperative oxygen desaturations. CONCLUSIONS: The Double Barrel Technique allows for a safe emergence from anesthesia in patients predisposed to upper airway obstruction (such as in obstructive sleep apnea and morbidly obese patients). To our knowledge, the Modified Double Barrel Technique is the first description for the use of nasal trumpets in patients who had nasal surgery and who have nasal splints in place.

Vitamin D and obstructive sleep apnea: a systematic review and meta-analysis.

OBJECTIVE/BACKGROUND: Several studies have reported an association between 25-hydroxyvitamin D (25(OH)D) levels and obstructive sleep apnea (OSA) patients. The objective of the current study was to perform a systematic review and meta-analysis of these studies and report the findings. PATIENTS/METHODS: Authors searched for studies (through January 1, 2017) reporting 25(OH)D serum levels in OSA patients. The Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) statement was followed. RESULTS: Fourteen studies with 4937 subjects met inclusion criteria. There were 1513 controls and 3424 OSA patients. The 25(OH)D serum levels for controls and mild OSA patients were 28.16 ± 9.39 ng/mL (95% CI 27.64, 28.68) and 27.41 ± 9.42 ng/mL (95% CI 26.87, 27.95), respectively. The 25(OH)D serum levels for controls and moderate OSA patients were 28.21 ± 9.38 ng/mL (95% CI 27.70, 28.72) and 25.48 ± 10.34 ng/mL (95% CI 24.68, 26.28), respectively. The 25(OH)D serum levels for controls and severe OSA patients were 28.32 ± 9.65 ng/mL (95% CI 27.80, 28.84) and 21.88 ± 10.24 ng/mL (95% CI 21.08, 22.68), respectively. Using random effects modeling, the 25(OH)D serum levels were decreased for patients with OSA when compared to control groups (mean differences were -2.7% for mild OSA, -10.1% for moderate OSA and -17.4% for severe OSA). CONCLUSIONS: There was a relative insufficiency in serum 25(OH)D levels among OSA patients compared to control patients, which was incrementally exacerbated with increasing severity of sleep apnea. It was unclear whether a low 25(OH)D was a risk factor for OSA or if OSA was a risk factor for 25(OH)D. It was also possible that the association between 25(OH)D and OSA was due to body mass index (BMI).

Oropharyngeal and tongue exercises (myofunctional therapy) for snoring: a systematic review and meta-analysis.

PURPOSE: Oropharyngeal and tongue exercises (myofunctional therapy) have been shown to improve obstructive sleep apnea. However, to our knowledge, a systematic review has not been performed for snoring. The study objective is to perform a systematic review, with a meta-analysis, dedicated to snoring outcomes after myofunctional therapy. METHODS: PubMed/MEDLINE and three other databases were searched through November 25, 2017. Two authors independently searched the literature. Eligibility (1) patients: children or adults with snoring, (2) intervention: oropharyngeal and/or tongue exercises, (3) comparison: pre and post-treatment data for snoring, (4) outcomes: snoring frequency and snoring intensity, (5) study design: publications of all study designs. RESULTS: A total of 483 articles were screened, 56 were downloaded in their full text form, and nine studies reported outcomes related to snoring. There were a total of 211 patients (all adults) in these studies. The snoring intensity was reduced by 51% in 80 patients from pre-therapy to post-therapy visual analog scale values of 8.2 ± 2.1 (95% CI 7.7, 8.7) to 4.0 ± 3.7 (95% CI 3.2, 4.8). Berlin questionnaire snoring intensity reduced by 36% in 34 patients from 2.5 ± 1.0 (95% CI 2.2, 2.8) to 1.6 ± 0.8 (95% CI 1.3, 1.9). Finally, time spent snoring during sleep was reduced by 31% in 60 patients from 26.3 ± 18.7% (95% CI 21.6, 31.0) to 18.1 ± 20.5% (95% CI 12.9, 23.3) of total sleep time. CONCLUSIONS: This systematic review demonstrated that myofunctional therapy has reduced snoring in adults based on both subjective questionnaires and objective sleep studies.

Dizziness, malpractice, and the otolaryngologist.

PURPOSE: To assess malpractice claims related to the management of dizziness in otolaryngology in order to improve care and minimize the risk of litigation. MATERIALS AND METHODS: This is a retrospective review of the LexisNexis "Jury Verdicts and Settlements" database. All lawsuits and out of court adjudications related to the management of dizziness by otolaryngologists were collected. Data including patient demographics, plaintiff allegation, procedure performed, and indemnities were analyzed. RESULTS: Of 21 cases meeting inclusion criteria, 17 were decided by a trial jury and four were resolved out of court. Jury verdicts favored the plaintiff 53% of the time and a payout was made in 57% of cases overall. Average payments were higher for jury verdicts in favor of the plaintiff ($1.8 million) as compared to out of court settlements ($545,000). Two-thirds of cases involved surgery, most commonly stapes surgery. Legal allegations, including physical injury, negligence, and lack of informed consent failed to predict the legal outcome. CONCLUSIONS: Appropriate examination, testing, and referrals within a timely manner are crucial in the management of dizzy patients to avoid misdiagnoses. It is imperative that patients undergoing ear surgery are appropriately counseled that dizziness is a potential complication. The analysis of malpractice literature is complementary to clinical studies, with the potential to educate practitioners, improve patient care, and mitigate risk.

Laser-Assisted Uvulopalatoplasty for Obstructive Sleep Apnea: A Systematic Review and Meta-Analysis.

Study Objectives: Laser-assisted uvulopalatoplasty (LAUP) has been used as treatment for obstructive sleep apnea (OSA). The objective of this study was to perform a systematic review and meta-analysis for LAUP alone as treatment for OSA in adults. Methods: Three authors searched five databases (including PubMed/MEDLINE) from inception through October 30, 2016 for peer-reviewed studies, with any design/language. A study quality assessment tool was used. The PRISMA statement was followed. A meta-analysis was performed. Results: Twenty-three adult studies (717 patients) reported outcomes (age: 50 ± 9 years, body mass index: 29 ± 4 kg/m2). The pre- and post-LAUP means (M) ± standard deviations (SDs) for apnea-hypopnea index (AHI) were 28 ± 13 and 19 ± 12 events/h (32% reduction). Random effects modeling for 519 patients demonstrated an AHI mean difference (MD) of -6.56 [95% CI -10.14, -2.97] events/h. Individual patient data analyses demonstrate a 23% success rate (≥50% reduction in AHI and <20 events/h) and an 8% cure rate. Additionally, 44% of patients had worsening of their AHI after LAUP. Lowest oxygen saturation (LSAT) improved from a M ± SD of 80 ± 8% to 82 ± 7%. A limitation is that most studies were case series studies and only two were randomized controlled trials. Conclusions: In this meta-analysis, LAUP reduced AHI by 32% among all patients; while the LSAT only changed minimally. Individual data demonstrated a success rate of 23%, cure rate of 8%, and worsening of the AHI among 44% of patients. We recommend that LAUP be performed with caution or not performed at all given the unfavorable results of currently published studies.