Upper airway manifestations of anti-IgLON5 disease: Otorhinolaryngological point of view.

INTRODUCTION: Anti-IgLON5 disease is a recently described neurological disorder with multisystemic features. The disease is characterized by the presence of IgLON5 antibodies in serum and cerebrospinal fluid. Our objective is to describe in detail the otorhinolaryngological manifestations of this disease, which are frequent and may include dysphagia, dysarthria, vocal cord paralysis and laryngospasm. METHODS: In this study, we present a series of 9 patients with anti-IgLON5 disease and otolaryngological manifestations. Patients were evaluated between July 2012 and March 2022 by video-polysomnography, fiber-optic laryngoscopy, and functional endoscopic evaluation of swallowing. RESULTS: The median age was 71 years, and 5 (56%) were female. Video-polysomnography showed a NREM/REM parasomnia in 6 patients (67%), obstructive sleep apnea in 8 (88%), stridor during sleep in 7 (78%) and central apneas in 1 (11%). Six out of the 9 patients (67%) presented episodes of acute respiratory failure that required mechanical ventilation, 6 had vocal fold palsy with 4 of them requiring tracheostomy (3 had to be performed on an emergency basis). Dysphagia occurred in 8 patients (89%). Prominent upper airway secretion and sialorrhea was also present in 3 cases. CONCLUSION: The anti-IgLON5 disease exhibits extensive otolaryngological symptoms, mainly affecting the upper airway. These symptoms affect the quality of life and can be life-threatening. Prompt acute management is essential for stridor, dyspnea, and dysphagia. Given the potential severity of the symptoms and rarity of the disease, it is important for otolaryngologists to be familiar with anti-IgLON5 disease. LEVEL OF EVIDENCE: Level 4.

Utility of the continuous intraoperative neuromonitoring in the prevention of the recurrent laryngeal nerve paralysis during thyroid surgery. A prospective observational study.

BACKGROUND: The continuous intraoperative neuromonitoring (C-IONM) of the recurrent laryngeal nerve (RLN) could help reducing the incidence of nerve paralysis after thyroid surgery, in comparison with the mere anatomical visualization of the RLN. The objective is to assess the efficacy and utility of C-IONM as a predictive test for recurrent laryngeal nerve paralysis after thyroidectomy. METHODS: A prospective observational study was performed in 248 patients who underwent thyroid surgery where C-IONM was applied, between September 2018 and December 2019, in a high-volume center. A previous and later laryngoscopy was performed, which allowed to evaluate the reliability of the C-IONM as a predictive test for recurrent paralysis. Sensitivity (SE), specificity (SP), positive predictive value (PPV), negative predictive value (NPV) were studied. RESULTS: A total number of 171 thyroidectomies, 62 hemithyroidectomies, 15 totalization thyroidectomies and 27 thyroidectomy with cervical dissections were performed. Postoperative laryngoscopy was altered in 40 patients (16.12%). The SE, SP, PPV and NPV values ​​were 65%, 94.7%, 70.2% and 93.4% respectively. CONCLUSIONS: C-IONM is a safe technique that provides real-time information about anatomical and functional integrity of the RLN and can improve the results of thyroid surgery.

Utility of the continuous intraoperative neuromonitoring in the prevention of the recurrent laryngeal nerve paralysis during thyroid surgery. A prospective observational study / Utilidad de la neuromonitorización continua intraoperatoria en la prevención de la parálisis del nervio laríngeo recurrente durante la cirugía tiroidea. Un estudio observacional prospectivo

Background: The continuous intraoperative neuromonitoring (C-IONM) of the recurrent laryngeal nerve (RLN) could help reducing the incidence of nerve paralysis after thyroid surgery, in comparison with the mere anatomical visualization of the RLN. The objective is to assess the efficacy and utility of C-IONM as a predictive test for recurrent laryngeal nerve paralysis after thyroidectomy. Methods: A prospective observational study was performed in 248 patients who underwent thyroid surgery where C-IONM was applied, between September 2018 and December 2019, in a high-volume center. A previous and later laryngoscopy was performed, which allowed to evaluate the reliability of the C-IONM as a predictive test for recurrent paralysis. Sensitivity (SE), specificity (SP), positive predictive value (PPV), negative predictive value (NPV) were studied. Results: A total number of 171 thyroidectomies, 62 hemithyroidectomies, 15 totalization thyroidectomies and 27 thyroidectomy with cervical dissections were performed. Postoperative laryngoscopy was altered in 40 patients (16.12%). The SE, SP, PPV and NPV values ​​were 65%, 94.7%, 70.2% and 93.4% respectively. Conclusions: C-IONM is a safe technique that provides real-time information about anatomical and functional integrity of the RLN and can improve the results of thyroid surgery. (AU)

Introducción: La neuromonitorización continua intraoperatoria (C-IONM) del nervio laríngeo recurrente (RLN) puede contribuir a la reducción de la incidencia de parálisis nerviosa tras una cirugía tiroidea, en comparación con la mera visualización anatómica del RLN. El objetivo de este estudio es evaluar la fiabilidad de la NCIO de los nervios laríngeos recurrentes como prueba predictora de parálisis vocal postoperatoria. Material y métodos: Se trata de un estudio observacional prospectivo realizado en 248 pacientes intervenidos de patología tiroidea en los que se aplicó la C-IONM, entre Septiembre de 2018 y Diciembre de 2019 en un único centro de alto volumen. Se realizó una laringoscopia pre y post operatoria a todos los pacientes, lo que pudo determinar la fiabilidad de la C-IONM como test predictivo de la parálisis recurrencial tras cirugía tiroidea. Se hallaron los valores de sensibilidad (SE), especificidad (SP), valor predictivo positivo (PPV) y valor predictivo negativo (NPV) de dicha técnica. Resultados: Se realizaron un total de 171 tiroidectomías totales, 62 hemitiroidectomías, 15 tiroidectomías de totalización y 27 tiroidectomías con disección cervical. Se hallaron alteraciones en la laringoscopia postoperatoria en 40 pacientes (16,12%). Los valores de SE, SP, PPV y NPV fueron 65%, 94.7%, 70.2% y 93.4% respectivamente. Conclusiones: La C-IONM es una técnica segura que ofrece información anatómica y funcional del NLR en tiempo real y puede contribuir a mejorar los resultados de la cirugía tiroidea. (AU)

Bilateral recurrent laryngeal nerve injury in total thyroidectomy with or without intraoperative neuromonitoring. Systematic review and meta-analysis.

INTRODUCTION AND OBJECTIVE: The risk of producing bilateral laryngeal paralysis (BLP) in total thyroidectomy (TT) is low, but it is a concern for the surgeon and a serious safety incident that may compromise the airway, require reintubation or tracheostomy and cause serious sequelae or death. Neuromonitoring (NM), as an early diagnostic tool for the existence of injury to the recurrent laryngeal nerve (RLN), has not been shown to have reduced the risk, even though published series show lower incidences. Our objective was to estimate the risk of bilateral RLN paralysis with and without NM TT by systematic review and meta-analysis. METHOD: We performed a systematic review of clinical trials, cohort studies and case series with total thyroidectomy without NM published in the period 2000-2014. A database search was performed using PubMed, Scopus (EMBASE) and the Cochrane Library. Heterogeneity between studies was explored and weighted risks grouped according to random effects models were estimated. RESULTS: We selected 40 articles and estimates of risk were identified in 54 case series (without NM, 25; with NM, 29) with 30,922 patients. The prevalence of BLP in the series with NM was lower compared to that without NM (2.43‰, [1.55 to 3.5‰] versus 5.18‰ [2.53 to 8.7‰]). This difference is equivalent to an absolute risk reduction of 2.75‰ with a number needed to treat of 364.13. The NM group was more homogeneous (I2=7.52%) than those without NM (I2=79.32%). The observed differences in the subgroup analysis were very imprecise because the number of observed paralysis was very low. CONCLUSIONS: The risk of bilateral paralysis is lower in studies with neuromonitoring.

[Contribution of neuromonitoring to the safety of tracheal extubation after total thyroidectomy. Prospective study with needle electrodes]. / Contribución de la neuroestimulación a la seguridad en la extubación traqueal tras tiroidectomía total. Estudio prospectivo con electrodos de aguja.

INTRODUCTION AND OBJECTIVES: Bilateral laryngeal paralysis cause serious respiratory complications. In thyroid surgery, neuromonitoring helps in identifying the recurrent laryngeal nerve, reports on its functioning at the end of surgery, supports decision making, and may reduce the risk of bilateral paralysis. Our objective was to estimate the influence of neuromonitoring in operative strategy and extubation safety in total thyroidectomy. METHODS: A non-randomized prospective study was conducted on 210 patients undergoing total thyroidectomy (420 laryngeal nerves stimulated included). We collected qualitative neuromonitoring variables (presence or absence of final signal after stimulation of the vagus nerve), and postoperative indirect laryngoscopy (normal motility or paralysis), performed until 3rd day after the surgery. RESULTS: The accuracy of the test was 99.5% (95% CI 98.3 to 99.9). The positive predictive value was 100% (95% CI 99.1 to 100), which showed the high ability of neuromonitoring to predict paralysis in case of loss of signal, and the negative predictive value was 99.5% (95% CI 98.3 to 99.9), which indicated its predictive capacity for normal motility when there is a normal signal. CONCLUSIONS: In our group of patients, recurrent laryngeal nerve monitoring was useful in total thyroidectomy as it provided information on the prognosis of laryngeal motility, and helped in making decisions during surgery when there was signal loss. Due to the risk of serious respiratory complications due to bilateral recurrent laryngeal nerve paralysis, we opted for the performing of the 2-stage total thyroidectomy in case of signal loss in the first lobectomy. Thereby, neuromonitoring contributed to the safety of the airway in tracheal extubation, aiding in the prevention of a possible bilateral laryngeal paralysis.

Parálisis diafragmática y de cuerda vocal en un paciente cón Síndrome de Churg-Strauss: reporte de un caso / Diaphragmatic and vocal cord paralysis in Churg-Strauss syndrome: a case report

El Síndrome de Churg-Strauss es una vasculitis sistémica que afecta vasos de pequeño y mediano calibre y que suele presentarse con asma, fiebre, hipereosinofilia, insuficiencia cardiaca, daño renal y neuropatía periférica. esta última se observa en el 65 por ciento al 80 por ciento de los casos, siendo el compromiso de nervios craneales en una minoría, y aún más excepcional la parálisis de cuerdas vocales y el diafragma. Las neuropatías por vasculitis sistémicas pueden resultar en morbilidad grave e incluso la muerte, por esto la necesidad de instaurar un tratamiento temprano. Reportamos el caso de un paciente que padeció parálisis diafragmática y de cuerda vocal por síndrome de Churg-Strauss.

Churg-Strauss syndrome is a systemic vasculitis of the small and medium sized vessels that usually occurs with asthma, fever, hypereosinophilia, cardiac failure, renal damage and peripheral neuropathy. The latter affects 65 percent to 80 percent of patients, cranial nerves involvement is rare while vocal cord and diaphragmatic paralysis are exceptional. Neuropathies due to systemic vasculitis may result in significant disability and death, therefore the importance to institute an early treatment. We report here a patient who suffered diaphragmatic and vocal cord paralysis due to Churg-Strauss syndrome.

Evaluación del nivel glótico mediante reconstrucción multiplanar escanográfica en pacientes con parálisis unilateral de cuerda vocal / Evaluation of the glottic level through multiplanar tomographic reconstruction in patients with unilateral vocal fold paralysis

Evaluar qué utilidad tiene la reconstrucción multiplanar escanográfica de cuello en la biometría del nivel glótico en pacientes con parálisis vocal unilateral. Materiales y métodos: Se realizó un estudio observacional, descriptivo de 29 pacientes con diagnóstico de parálisis unilateral de pliegue vocal a los cuales se les realizó Tomografía Computarizada de cuello en fonación con reconstrucción multiplanar, evaluando el nivel de asimetría vertical, horizontal, las longitudes anteroposterior, transversal, y grosor y se caracterizaron las diferentes formas del pliegue vocal paralizado, así como el ángulo de asimetría del eje del pliegue y su relación con la línea media. Se evaluó también la lateralidad del pliegue vocal afectado. Con la intención de evitar sesgos en la toma de los datos se realizó una correlación intra e interobservador, mediante el análisis de medidas de kappa para variables categóricas y coeficiente de correlación interclase para variables numéricas. Resultados: Se incluyeron 29 pacientes en el estudio. La clasificación de la asimetría del nivel vertical reportó un 82% para el tipo 3 (más de 4 mm), seguido de un 10,3% y un 6,9% para los tipos 2 y 1 respectivamente. El nivel horizontal del lado no afectado reportó un promedio de 0.4 mm (DS: 0.28) y el del lado paralizado 1.46 mm (DS: 0.28) La longitud transversa, anteroposterior y grosor dieron para el pliegue vocal no afectado promedios de 4.47 mm (DS: 0.6), 8.4 mm (DS:0.8) y 1.59 mm (DS:0.26) y para el lado paralizado 2.96 (DS: 0.52), 7.5 mm (DS: 0.78) y 2.46 mm (DS: 0.32). La forma del pliegue vocal no afectado en aducción fue denominada como en dedo de guante y la del pliegue paralizado como redondeada en el 100% de los casos. Los ángulos de asimetría de los pliegues con respecto a la línea media reportaron promedios de 82° (DS: 5.41) del pliegue vocal no afectado y 66.6° (DS: 4.19°) para el pliegue vocal paralizado...

Objective: To evaluate the usefulness of neck multiplanar tomography reconstruction in the glottic level biomethrics in patients with unilateral vocal fold paralysis. Materials and methods: We conducted a descriptive observational study of 29 patients with unilateral vocal fold paralysis who underwent multiplanar reconstruction of neck computed tomography during phonation. The transverse AP lengthand the thickness of the vocal folds were established. The vertical and horizontal level of the vocal folds were assessed and classified. The various forms of the vocal folds were characterized andasymmetries of the angle formed between the vocal fold axis and the midline were determined. Frequencyof sidedness of the paralyzed vocal fold also was obtained. In order to avoid biases during data collection, variability intra and interobserver were also assessed by kappa tests for categorical variables and intraclass correlation coefficient for numeric ones. Results: 29 patients were included in the study. The asymmetry of vertical glottic level was 92% for type 3 and 8% and 2% for types 2 and 1. The horizontal level in the non-affected side reported a mean of 0.4 mm (DS: 0.28) and the affected side had a mean of 1.46 mm (DS: 0.28). Transverse, anteroposterior an width measurements were 47 mm (DS: 0.6), 8.4 mm (DS: 0.80) y 1.59 mm (DS: 0.26) on the non affected side and 2.96 (DS: 0.52), 7.50 mm (DS: 0.78) y 2.46 mm (DS: 0.32) for the paralyzed side. The normal vocal fold was tagged as “finger glove” shape and the paralyzed fold was tagged as “rounded” shape in the whole amount of subjects. Asymmetry angles reported 82° (DS: 5.41) for the non-affected side and 66.6° (DS: 4.19) for the paralyzed side...