Oncocytic Papillary Cystadenoma: A Case of a Misdiagnosed Laryngeal Mass.

Cystadenomas are benign neoplasms found in major and minor salivary glands. In cases where both oncocytic cells and papillary architecture, without a lymphoid component, exist, the lesion is called oncocytic papillary cystadenoma (OPC). OPCs are rarely encountered in the laryngeal region and that is why they are usually misdiagnosed as other types of laryngeal tumors. Hereby, we present a case of a misdiagnosed laryngeal OPC in an attempt to raise awareness of this rare entity, both for the surgeon performing the excision of the laryngeal mass and for the pathologists examining the specimen.

Neuropatía laríngea y movimiento paradojal de cuerdas vocales en la edad pediátrica: reporte de un caso / Laryngeal neuropathy and paradoxical vocal fold motion in the pediatric age: case report

Resumen La neuropatía laríngea es una condición de hipersensibilidad, hiperreactividad e hiper-función laríngea secundaria a un desequilibrio entre las aferencias y eferencias laríngeas. La respuesta individual y exagerada frente a diversos gatillantes específicos puede generar síntomas como tos crónica, parestesia laríngea, carraspera, disfonía, estridor, sensación de globus faríngeo, movimiento paradojal de las cuerdas vocales (también conocido como disfunción cordal) y/o laringoespasmo. Existe abundante literatura sobre neuropatía laríngea en adultos, sin embargo, en niños es limitada. El objetivo de este artículo es dar a conocer un caso de neuropatía laríngea en la edad pediátrica y la importancia de su consideración en el enfrentamiento de estos pacientes. Se presenta caso clínico de un paciente de 13 años, con antecedente de cirugía cardiaca reciente, evoluciona con disfonía severa evidenciándose aparente inmovilidad cordal bilateral con resultados discordantes entre nasofibrolaringoscopía y electromiografía laríngea. Posteriormente presenta mejorías en su voz, sin embargo, se agregan otros síntomas laringológicos como carraspera, globus faríngeo y estridor no explicados por causas anatómicas. Se expone la evaluación y abordaje otorrinolaringológico-fonoaudiológico para el caso. Se concluye que el diagnóstico de neuropatía laríngea requiere un alto índice de sospecha clínica ante signos laringológicos sugerentes, debiendo descartarse causas orgánicas y estructurales. El abordaje otorrinolaringológico-fonoaudiológico constituye el pilar terapéutico asociado al uso de neuromoduladores en casos seleccionados.

Abstract Laryngeal neuropathy is a condition of hypersensitivity, hyperresponsiveness and laryngeal hyperfunction secondary to an imbalance between laryngeal afferent and efferent information. The individual and exaggerated response to diverse specific triggers can lead to symptoms such as chronic cough, laryngeal paresthesia, throat clearing, dysphonia, stridor, globus pharyngeus, vocal cord dysfunction, and/or laryngospasm. There is plentiful literature on laryngeal neuropathy in adults, however, in children, it is limited. Here, we present a case report of laryngeal neuropathy in the pediatric age and discuss the importance of its consideration in the approach of these patients. A case of a 13-year-old patient, recently intervened with cardiac surgery that evolves with severe dysphonia is presented. Nasofibrolaryngoscopy shows apparent bilateral vocal fold immobility with discordant results in laryngeal electromyography. Later, his voice improves but other laryngological symptoms appeared, such as throat clearing, globus pharyngeus and stridor, not explained by anatomical causes. The otolaryngological-speech therapy evaluation and approach for the case is exposed. We conclude that for the diagnosis of laryngeal neuropathy, a high index of clinical suspicion is required in the presence of suggestive laryngological symptoms, and organic and structural causes must be previously ruled out. The otorhinolaryngological-logopedic approach constitutes the mainstay of treatment associated with the use of neuromodulators in selected cases.

Localized laryngeal amyloidosis: A systematic review.

OBJECTIVE: The larynx is the most common site of localized head and neck amyloidosis. Our study aimed to review the clinical features, treatments, and outcomes associated with localized laryngeal amyloidosis (LA). We also compared these features between two different time periods to evaluate the evolution of LA management. METHODS: A literature search using PubMed, CINAHL, Embase, and Cochrane Library identified cases of LA published between 1891 and 2021. Biopsy-proven cases of localized LA were included. Non-English studies, animal studies, and reviews were excluded. RESULTS: 282 patients (1891-1999: 142 patients, 2000-2021: 140 patients) from 129 studies were included. Results are reported as 1891-2000 vs. 2000-2021: Mean age was 48.5 years (range, 8-90 years) vs. 46.0 years (range, 9-84 years). The most common presenting symptoms were dysphonia (n = 30, 95 % vs. n = 127, 96 %) and difficulty breathing (n = 37, 27 % vs. n = 35, 27 %). A total of 62 (44 %) vs. 46 (33 %) lesions were found in the true vocal folds and 35 (25 %) vs. 59 (42 %) were found in the false vocal folds. 133 (94 %) vs. 137 (98 %) patients underwent surgical interventions to investigate and/or treat LA. Recurrent LA was reported in 27 (19 %) vs. 33 (24 %) patients with a mean time to recurrence of 25.4 months (range, 0.3-132 months) vs. 34.5 months (range, 0.8-144 months). Of cases reporting survival rate, 104 (97 %) vs. 107 (99 %) were alive at source study endpoints. CONCLUSION: LA typically exhibits an indolent course; therefore, early intervention may address longstanding symptoms. Recurrent disease poses a clinical challenge in patients with LA.

Partial laryngectomy for the management of laryngeal masses in six cats.

OBJECTIVES: Partial laryngectomy is an organ-sparing surgical procedure for the removal of laryngeal masses which has not been described in cats. The aim of this study was to report on the surgical procedure and the short- and long-term clinical outcomes of cats that underwent partial laryngectomy. METHODS: Medical records were retrospectively collected over a 4-year period in two institutions. The following data were retrieved: signalment, history, clinical signs, diagnostic test results, surgical procedure, postoperative management, complications and outcome. RESULTS: Six cats underwent partial laryngectomy. The most common clinical signs in cats with laryngeal masses were stridor (n = 4) and dyspnoea (n = 4). In all cats, a full-thickness portion of one or several laryngeal cartilages was resected, including thyroid cartilage alone (n = 2), thyroid cartilage and arytenoid (n = 2), and arytenoid cartilage and epiglottis (n = 2). The resected laryngeal masses were reported to be lymphoma (n = 3), carcinoma (n = 1), laryngeal cyst (n = 1) and inflammatory laryngeal disease (n = 1). All cats survived the surgical intervention of partial laryngectomy. Four cats showed varying degrees of respiratory distress in the short-term postoperative period. A temporary tracheostomy tube was placed in two cats. No other postoperative complications were noted in the short- or long-term. Four cats were still alive at the time of writing. These cats survived at least 252 days. CONCLUSIONS AND RELEVANCE: In a small number of cases, our results show that successful long-term outcomes after partial laryngectomy are achievable, with longer survival times than previously reported. Therefore, partial laryngectomy should be considered as a viable treatment option in cats with laryngeal masses.

Diagnostic Accuracies of Laryngeal Diseases Using a Convolutional Neural Network-Based Image Classification System.

OBJECTIVES/HYPOTHESIS: There may be an interobserver variation in the diagnosis of laryngeal disease based on laryngoscopic images according to clinical experience. Therefore, this study is aimed to perform computer-assisted diagnosis for common laryngeal diseases using deep learning-based disease classification models. STUDY DESIGN: Experimental study with retrospective data METHODS: A total of 4106 images (cysts, nodules, polyps, leukoplakia, papillomas, Reinke's edema, granulomas, palsies, and normal cases) were analyzed. After equal distribution of diseases into ninefolds, stratified eightfold cross-validation was performed for training, validation process and remaining onefold was used as a test dataset. A trained model was applied to test sets, and model performance was assessed for precision (positive predictive value), recall (sensitivity), accuracy, F1 score, precision-recall (PR) curve, and PR-area under the receiver operating characteristic curve (PR-AUC). Outcomes were compared to those of visual assessments by four trainees. RESULTS: The trained deep neural networks (DNNs) outperformed trainees' visual assessments in discriminating cysts, granulomas, nodules, normal cases, palsies, papillomas, and polyps according to the PR-AUC and F1 score. The lowest F1 score and PR-AUC of DNNs were estimated for Reinke's edema (0.720, 0.800) and nodules (0.730, 0.780) but were comparable to the mean of the two trainees' F1 score with the best performances (0.765 and 0.675, respectively). In discriminating papillomas, the F1 score was much higher for DNNs (0.870) than for trainees (0.685). Overall, DNNs outperformed all trainees (micro-average PR-AUC = 0.95; macro-average PR-AUC = 0.91). CONCLUSIONS: DNN technology could be applied to laryngoscopy to supplement clinical assessment of examiners by providing additional diagnostic clues and having a role as a reference of diagnosis. LEVEL OF EVIDENCE: 3 Laryngoscope, 131:2558-2566, 2021.

Perinatal outcome of fetuses with congenital high airway obstruction syndrome: a single-center experience.

OBJECTIVE: To report our experience with management of fetuses with congenital high airway obstruction syndrome (CHAOS). METHODS: We retrospectively reviewed the cases of fetuses who were prenatally diagnosed and postnatally confirmed with CHAOS between 2010 and 2019 at Asan Medical Center, Seoul, Korea. RESULTS: Of 13 fetuses prenatally diagnosed with CHAOS, 7 were lost to follow-up and 6 were postnatally confirmed as having CHAOS. All fetuses, except one were delivered via cesarean section with an ex utero intrapartum treatment (EXIT) procedure. Two patients had coexisting congenital heart diseases requiring several cardiac surgeries following birth. Both of these patients demonstrated developmental delay; however, the remaining 4 had a normal development except for expressive language. Two infants died of respiratory complications, and the remaining 4 were alive at the end of the follow-up period. All 4 live patients underwent tracheostomy with planned reconstruction surgery. Three children are now able to phonate, and 1 can maintain a conservation. CONCLUSION: The proper management of CHAOS using the EXIT procedure results in high survival and low hypoxemia-induced complication rates. Therefore, an accurate prenatal diagnosis is necessary for an appropriate perinatal management.

Laryngeal, Tracheal, and Bronchial Disease in the Mucopolysaccharidoses: Endoscopic Study.

Mucopolysaccharidoses (MPS) are genetically determined diseases, leading to a deficiency of enzymes in the glycosaminoglycan (GAG) degradation pathway. The accumulation of GAG occurs in connective tissue in various organs and systems of the body, including the larynx, trachea, and bronchi. Respiratory symptoms are common and severe in these patients, and respiratory disease is a frequent cause of death. A cross-sectional study with flexible bronchoscopy was conducted in 30 MPS patients (6 MPS I, 8 MPS II, 2 MPS III, 3 MPS IV-A, and 11 MPS VI). Only four patients (13.33%) had a normal airway; nine (30%) had mild to moderate disease, 12 (40%) moderate to severe, and five patients (16.67%) had severe disease. Of particular interest, neuronopathic MPS II had the largest proportion of tracheostomized patients who died due to respiratory complications; in MPS IV-A, all patients had significant tracheobronchial deformity with associated tracheomalacia, despite lacking laryngeal involvement. Laryngotracheobronchial disease (LTBD) was associated to longer disease history and was significantly more severe in older patients. Longer use of enzyme replacement therapy did not prevent the progression of LTBD, although the age of therapy introduction may be a crucial factor in lower airway involvement.

Gold laser removal of a large ductal cyst on the laryngeal surface of the epiglottis.

Laryngeal cysts are rare lesions of the larynx that are often described on incidental discovery. We report an unusual presentation of a large ductal cyst located on the laryngeal surface of the epiglottis. The patient presented with a low tone voice while displaying bilateral normal appearing vocal cords with normal mobility. Computed tomography scans and flexible laryngoscopy showed a round supraglottic mass approximately 2 cm in diameter on the right lateral laryngeal surface of the epiglottis. The mass was removed surgically during microsuspension laryngoscopy with excision of the mucocele utilizing a gold laser. Postoperatively, the patient's voice returned to baseline with no complications.

A cat with suspected laryngeal metastasis with mucosal irregularity resulting from apocrine/salivary gland adenocarcinoma in the head.

A 16-year-old castrated male mongrel cat presented with swelling under the left pinna and a 3 -month history of voice change. Laryngeal endoscopy revealed circumferential oedema around the arytenoid cartilages and hypersecretion of saliva. Histopathological examination of the mass around the left ear canal was considered the primary lesion that originated from cutaneous apocrine adenocarcinoma or parotid gland adenocarcinoma, and it metastasized to the larynx, lung and medial retropharyngeal lymph nodes. This report provides new insights into feline laryngeal diseases which could result in laryngeal metastasis with slight mucosal irregularity alone and without obvious radiographic abnormalities. Therefore, histopathological examination should be performed when a cat presents clinical signs such as stridor, dysphonia or voice change without any mass-forming laryngeal lesion.

A Population-Based Cross-Sectional Study of Alcohol Consumption and Risk of Benign Laryngeal Disease in Korean Adults.

OBJECTIVE: Studies on alcohol consumption and benign laryngeal disease (BLD) in the general population are rare. The aim of this study was to investigate the relationship between alcohol consumption and BLD in the Korean general population. STUDY DESIGN: This was a cross-sectional study of a national health survey. MATERIALS AND METHODS: Subjects were 3141 noninstitutionalized civilian adults older than 19 years (1313 men and 1828 women) who completed the laryngeal examination of the Korea National Health and Nutrition Examination Survey of 2008. Frequency of drinking was classified into less than once per week, two to three times per week, and more than four times per week. Binge drinking was defined as five or more drinks (≥61 g of alcohol) per episode for men and as four or more drinks (≥41 g of alcohol) per episode for women. Poisson regression analyses were conducted to examine the relationship between alcohol drinking and BLD. RESULTS: Adjusting for covariates (age, sex, income, level of education, occupation, and cigarette smoking), it was found that those who drank more than four times a week, compared with those who drank less than once per week, were more likely to have BLD (risk ratio = 2.15, 95% confidence interval: 1.01-4.59, P < 0.05). However, the amount of alcohol consumed was not associated with BLD. CONCLUSIONS: Frequent drinking (ie, more than four times a week) was found to be a risk factor for BLD.

Sarcoidose laríngea: revisão de literatura / Laryngeal sarcoidosis: literature review

INTRODUÇÃO: A sarcoidose laríngea é uma doença granulomatosa que representa um desafio em relação ao diagnóstico e o tratamento. MÉTODO: Nesta revisão são abordados todos os aspectos da doença, incluindo etiologia, manifestações clínicas em adultos e crianças, diagnóstico e tratamento, a partir da base de dados Medline, Lilacs e Scielo, incluindo-se artigos de língua inglesa e portuguesa publicados no período de 1973 a 2008. COMENTÁRIOS FINAIS: Apesar dos avanços ocorridos na investigação e tratamento da sarcoidose, é uma doença que ainda se mantém sem etiologia e fisiopatologia definidas, constituindo um diagnóstico de exclusão pela ausência de exames específicos e por sua provável origem multifatorial. O acometimento laríngeo, apesar de raro, é suspeitado principalmente diante de história prévia de sarcoidose sistêmica, e a pesquisa dos diagnósticos diferenciais deve ser extensa para não passarem desapercebidas doenças graves como neoplasias malignas.

INTRODUCTION: Laryngeal sarcoidosis is a granulomatous disease representing a challenge as regards diagnosis and treatment. METHOD: In this review, all aspects of the disease are involved, including etiology, clinical manifestations in adults and children, diagnosis, and treatment, by searching through Medline, Lilacs and Scielo databases, as well as English and Portuguese articles published from 1973 to 2008. FINAL COMMENTS: Despite the breakthroughs in its diagnosis and treatment, sarcoidosis is a disease that remains without a clear etiology and physiopathology, becoming an exclusion diagnosis due to both the lack of precise exams and its likely multifactor background. Laryngeal impairment, however infrequent, is a suspicion mainly when there is a precedent history of systemic sarcoidosis, and the research for differentiated diagnosis must be wide-ranging so as to prevent severe diseases like malign neoplasia from being unnoticed.

Injeção de corticosteróide em patologias vocais inflamatórias crônicas, revisão da literatura / Steroid injection in chronic inflammatory vocal fold disorders, literature review

Os corticosteróides são potentes inibidores da inflamação e da cicatrização. Administração local de esteróides diretamente na laringe tem sido relatada em diversas patologias laríngeas. OBJETIVO: O objetivo desse estudo é realizar uma revisão da literatura sobre o uso de infiltração de corticosteróide em pregas vocais, em pacientes com patologias vocais benignas, inflamatórias e crônicas de laringe. METODOLOGIA: Realizou-se uma busca eletrônica na base de dados Medline, selecionando-se estudos clínicos que utilizavam corticosteróides em patologias benignas da laringe. RESULTADOS: Os corticosteróides são indicados nas seguintes situações: 1) doenças inflamatórias agudas, principalmente naquelas relacionadas a edema que comprometa a via aérea; 2) doenças auto-imunes com alterações laríngeas; 3) estenose laríngea; 4) em lesões benignas inflamatórias das pregas vocais, como nódulos, pólipos e edema de Reinke, para diminuir o processo inflamatório pré-cirúrgico ou na tentativa de se evitar uma intervenção cirúrgica; 5) em fonocirurgia, com o objetivo de reduzir a formação de cicatriz. Seu uso pode ser profilático, visando à prevenção da formação de cicatriz ou terapêutico em uma cicatriz já formada. CONCLUSÃO: Os corticosteróides podem ser considerados uma opção terapêutica importante no manejo de várias patologias, principalmente aquelas de origem inflamatória que causam alterações vocais.

Steroids are potent inhibitors of inflammation and wound repair. Local administration of steroids directly into the larynx has been reported in many laryngeal diseases. AIM: The purpose of this study is to review related literature about the use of steroid injection in patients with benign, inflammatory and chronic vocal disease. METHODOLOGY: We performed an electronic survey in Medline database and selected clinical trials regarding steroid use in benign laryngeal diseases. RESULTS: Steroids are indicated in these situations: 1) acute inflammatory diseases, mainly when edema compromises the airways; 2) auto- immune disease with laryngeal involvement; 3) laryngeal stenosis; 4) benign lesions of the vocal folds, e.g., nodules, polyps and Reinke's edema, to reduce the inflammatory reactions before phonosurgery or in an attempt to avoid surgery; 5) In phonosurgery, aiming to reduce scarring. In this case, it could be used as a preventive measure in vocal fold scarring, or for scar treatment. CONCLUSION: Steroids may be considered an important therapeutic option in the management of many diseases, specially the inflammatory ones, associated with vocal changes.

Pathogenetic Hypothesis of Benign Laryangeal Disease from Histopathologic Change in the Basement Membrane Zone and the Superficial Layer of Lamina Propria / 대한이비인후과학회지

BACKGROUND AND OBJECTIVES: The epithelium, basement membrane zone (BMZ) and superficial layer of lamina propria (SLLP) in the vocal fold possess similar mechanical properties and behave as one single tissue, which is commonly referred to as the "cover". The purpose of this study is to elucidate the ultra structural and histopathological changes and differences in the cover of benign laryngeal lesions, and also to investigate the pathogenetic response. MATERIALS AND METHOD: The cover of the vocal fold in vocal nodule, polyp and Reinke's edema was investigated on 87 patients. Immunohistochemical analysis was performed with monoclonal antibodies directed against collagen type III and VII for identification of SLLP and anchoring fiber. Control specimens were obtained from 10 uninvolved side vocal fold in the laryngeal specimen after total laryngectomy. RESULTS: One type of injury in the cover was due to a little or no change of SLLP, which was seen more often in vocal nodules. The second type of pathologic finding shows disorganized anchoring fiber and extensive interstitial injury, which was seen more often in vocal polyp. The third type shows injury in the anchoring fiber and sparse distribution of collagen type III in SLLP, which was the main finding in Reinka's edema. These findings lead us to understand the pathogenesis of the disease that developed as different entities. CONCLUSION: We conclude that collagens in BMZ and SLLP are the structures that seem to provide the pathogenetic portion of benign laryngeal diseases which develop from the same phonotrauma into clinically different disease entities.

The Changes of Voice Quality Following Laryngeal Microsurgery for Vocal Polyp and Nodule / 대한이비인후과학회지

BACKGROUND AND OBJECTIVES: Vocal polyps and nodules are representative chronic benign laryngeal diseases, and voice overuse, misuse, and abuse are felt by most authors to be the primary causative factors. Treatment options for vocal polyps and nodules in general include voice therapy by a speech-language pathologist and laryngeal microsurgery. The purpose of this study was to determine the changes in voice quality of a group of patients diagnosed with vocal polyp or vocal nodule before and after microlaryngeal surgery. MATERIALS AND METHOD: The following patients groups were studied prior to and 2 weeks after surgery: thirty-eight males, ages 27 to 62, and twenty five females, ages 23 to 47, with vocal polyp: three males, ages 29 to 38, and twenty eight females, ages 21 to 49, with vocal nodules. Voice recordings of the first sentence of a standardized paragraph "San Chaek" were performed for acoustic analyses. The recorded samples were analyzed on the Kay CSL model 4300B using Visi-Pitch II program. For the sentence, mean speaking fundamental frequency (F0), mean jitter and mean shimmer were analyzed. RESULTS: For the polyps, male patients showed decreased mean F0 (119.28 Hz), compared to the preoperative value (125.86 Hz) (p<0.05), and female patients showed increased mean F0 (197.52 Hz), compared to the preoperative value (190.07 Hz) (p=0.06). Mean jitter and shimmer improved in both males and females 2 weeks after surgery compared to their preoperative value (p<0.01). For the nodules, male patients showed decreased mean F0 (136.24 Hz) compared to the preoperative value (138.68 Hz), and female patients showed increased mean F0 (199.98 Hz) compared to the preoperative value (192.61 Hz) (p<0.05). Mean jitter and shimmer improved in both males and females 2 weeks after the surgery compared to their preoperative value. CONCLUSION: The acoustic output generally improved after laryngeal microsurgery as evidenced by the more appropriate mean F0 and reduced level of vocal perturbation. And, mean F0 showed different changes between males and female. Further research needs to be pursued on what this changes of mean F0 mean.