[Subglottic cysts in infants: a report of 3 cases and literature review].

Subglottic cyst is a rare cause of laryngeal tinnitus in infants and young children, and only a few cases have been reported at home and abroad. In this paper, we report the clinical characteristics and treatment experience of three cases of subglottic cysts in Children's Hospital of Nanjing Medical University. All the 3 childrem were prematurechildren, with a history of tracheal intubation, and the main symptoms were coughing and wheezing.Electronic nasopharyngolaryngoscopy revealed spherical neoplasm under the glottis. Neck computed tomography （CT） showed a slightly hypodense shadow with poorly defined borders, and no significant enhancement was observed after enhancement. Under the self-retaining laryngoscope, the new organisms were clamped and nibbled, and the cyst wall was cauterized by low temperature plasma. There was no recurrence in postoperative follow-up.

Subglottic cysts in infants: a report of 3 cases and literature review / 临床耳鼻咽喉头颈外科杂志

Subglottic cyst is a rare cause of laryngeal tinnitus in infants and young children, and only a few cases have been reported at home and abroad. In this paper, we report the clinical characteristics and treatment experience of three cases of subglottic cysts in Children's Hospital of Nanjing Medical University. All the 3 childrem were prematurechildren, with a history of tracheal intubation, and the main symptoms were coughing and wheezing.Electronic nasopharyngolaryngoscopy revealed spherical neoplasm under the glottis. Neck computed tomography （CT） showed a slightly hypodense shadow with poorly defined borders, and no significant enhancement was observed after enhancement. Under the self-retaining laryngoscope, the new organisms were clamped and nibbled, and the cyst wall was cauterized by low temperature plasma. There was no recurrence in postoperative follow-up.

Inspiratory laryngeal stridor as the main feature of progressive encephalomyelitis with rigidity and myoclonus: a case report and literature review.

BACKGROUND: Progressive encephalomyelitis with rigidity and myoclonus (PERM) is an acute, potentially life-threatening, yet curable neuro-immunological disease characterized by spasms, muscular rigidity, and brainstem and autonomic dysfunction. The clinical features of glycine receptor (GlyR) antibody-positive PERM may be overlooked, particularly with some unusual symptoms. CASE PRESENTATION: A 52-year-old man was admitted to the hospital for evaluation of tension headache for 20 days and mild dysarthria. These symptoms were followed by panic, profuse sweating, severe dysarthria, dizziness, unsteady gait, and paroxysmal muscle spasms. Brain magnetic resonance imaging and cerebrospinal fluid analysis were normal. The patient's condition steadily deteriorated. He repeatedly presented with rigidity, panic attacks, severe anxiety, paroxysmal inspiratory laryngeal stridor, cyanosis of the lips, and intractable epilepsy. Electromyography showed multiple myoclonic seizures, a single generalized tonic-clonic seizure, and a single generalized tonic seizure. Screening for autoimmune encephalitis antibodies revealed anti-GlyR antibodies in his cerebrospinal fluid. Immunomodulatory pulse therapy with steroids and immunoglobulin resulted in expeditious improvement of the symptoms within 2 weeks, and a follow-up at 5 weeks showed consistent clinical improvement. CONCLUSION: Our case highlights that inspiratory laryngeal stridor is an important symptom of PERM. Our observation widens the spectrum of the clinical presentation of anti-GlyR antibody-positive PERM, where early identification is a key to improving prognosis.

Propranolol treatment of infantile subglottic hemangioma: a report of two cases and a literature review.

Infantile hemangioma is one of the most common benign tumors of infancy. The natural evolution includes rapid growth followed by gradual involution. Airway hemangiomas are not that common, but they can lead to dyspnoea, as well as to life-threatening complications.Two children aged 3 months were admitted to the Pediatric Department with difficulties in breathing and with biphasic stridor. They had previously been hospitalized because of the same symptoms and misdiagnosed as having an upper respiratory tract infection. The previous treatment included intravenous or inhaled corticosteroids, without any significant improvement. Laryngoscopy was performed for both of the children. There was a mass in the subglottic area with the appearance of a hemangioma causing significant airway stenosis. We started treatment with propranolol at a dose of 1 mg/kg/day twice daily. The dose was gradually increased up to 3 mg/kg/day, under close monitoring. In the first 7 to 10 days after initiation of treatment, we observed a significant improvement of the respiratory distress. The second laryngoscopy showed an almost complete involution of the mass in the subglottis. The focus of this article will be primarily on the clinical presentation and the therapeutic response of subglottic hemangioma, along with a literature review on the subject.

The etiological diagnosis value of soft bronchoscopy in children with laryngeal stridor / 中国小儿急救医学

Objective:To evaluate the etiological diagnosis value of soft bronchoscopy in children with laryngeal stridor.Methods:The clinical data of 402 children with laryngeal stridor wheezing were retrospectively analyzed, which examined by soft bronchoscopy in Anhui Provincial Children′s Hospital from January 2016 to January 2019.Results:A total of 402 cases of laryngeal stridor were diagnosed by soft bronchoscopy, 317(78.8%) cases were diagnosed as congenital airway dysplasia, including 200(49.7%)cases of congenital laryngeal chondromalacia, which including 132 cases of single laryngeal chondromalacia and 68 cases with other respiratory tract dysplasia, and 117(29.1%) cases of respiratory dysplasia other than laryngeal chondromalacia; 46(11.5%) cases of laryngitis; 28(7.0%) cases of airway acquired stenosis and 11 (2.7%)cases of foreign body.Among 402 cases of children with laryngeal stridor who were diagnosed according to clinical feature, combined with chest X-ray, chest CT, CT angiography and color Doppler echocardiography as well as other imaging data, 335(83.3%) cases were congenital laryngeal chondromalacia, 16(4.0%) cases were other respiratory tract dysplasia (including six cases of subglottic and tracheal stenosis, five cases of laryngeal space occupying lesions, four cases of tracheobronchial malformation, and one case of subglottic hemangioma), 35 (8.7%)cases of laryngitis, acquired airway stenosis in 15 cases including 13 cases of congenital heart disease, one case of pulmonary artery sling, one case of mediastinal cyst, and one case of foreign body.Congenital laryngeal chondromalacia, other causes of respiratory dysplasia and foreign body detected by flexible bronchoscopy were not consistent with clinical examination( P<0.05). Conclusion:Congenital laryngeal chondromalacia is the main cause of laryngeal stridor, but it is often associated with other airway dysplasia.Soft bronchoscopy can provides etiological diagnosis for children with laryngeal stridor wheezing, especially in the diagnosis of respiratory tract dysplasia and airway foreign body.

Supraglottoplasty in Neonates under One Month of Age.

OBJECTIVES: Examine outcomes of patients 1 month of age or less who received supraglottoplasty. Demonstrate the feasibility and safety of supraglottoplasty in neonates <1 month of age. METHODS: Charts were reviewed from 2015-2017. Patients with previously identified laryngomalacia requiring surgical intervention and age 1 month or less were identified. We collected data on age, gender, surgical technique, other airway lesions, time to extubation and discharge, and comorbidities. RESULTS: Six patients met the inclusion criteria of which four were male and two were female. Average age at time of surgery was 19.5 days. All patients had diagnosis of laryngomalacia made on flexible or direct laryngoscopy with increased work of breathing, reports of cyanotic episodes, or oxygen desaturations. All had feeding difficulties, and three had signs and history consistent with gastroesophageal reflux. Two patients underwent supraglottoplasty by CO2 laser treatment alone, one patient received cold steel treatment alone, and the remaining three patients received cold steel with supplemental microdebrider treatment. Out of six patients, five were extubated within 5 days of their procedure. The average time to extubation was 1.8 days. The average time to discharge was 24.25 days following surgery. Two patients required revisions. CONCLUSIONS: Supraglottoplasty in children younger than 1 month of age is rare. Despite the rare occurrence, it appears to be feasible early in life in treating laryngomalacia. Neonates with respiratory failure, apneas, and cyanosis, or difficulty feeding due to laryngomalacia should be evaluated and treated with supraglottoplasty.

Esteroides en extubación: ¿A quién? ¿Cómo? ¿Cuándo? / Steroids post-extubation: Who? How? When? / Esteróides na extubação: quem? Como e quando?

Resumen: El fracaso en la extubación es una complicación de alto riesgo para los pacientes en las Unidades de Cuidados Intensivos. El edema laríngeo y el estridor laríngeo han sido estudiados como causas que propician el fracaso en la extubación. Parte de las estrategias para la prevención del fracaso en la extubación y el manejo del estridor o edema laríngeo es el uso de esteroides. La discrepancia entre el tiempo, la dosis, el paciente candidato y el tipo de esteroide obligan a crear una propuesta que estandarice su utilización. La presente revisión se enfoca en identificar a aquellos pacientes que se benefician del uso de esteroides, el tipo de esteroide que se debe utilizar, el momento y la dosis correcta.

Abstract: Post-extubation failure is a high risk complication for patients in intensive care unit. Laryngeal edema and laryngeal stridor have been studied as causes of post-extubation failure. Part of the strategies for the prevention of post-extubation failure and management of stridor or laryngeal edema is the use of steroids. The discrepancy between the time, the dos, the patient and the type of steroid forces to create a strategy that standardizes its use. This review focuses on identify patients who benefit from the use of steroids, type of steroid that should be uses, timing and correct dose.

Resumo: O fracasso na extubação é uma complicação de alto risco para pacientes em unidades de terapias intensivas. O edema e o estridor laríngeo foram estudados como causas que propician a falha na extubação. Parte das estratégias para prevenção da falha na extubação e manejo do estridor ou edema laríngeo é o uso de esteróides. A discrepância entre o tempo, a dose, o paciente candidato e o tipo de esteróide nos obriga a criar uma proposta padrão de utilização. A presente revisão se concentra em identificar os pacientes que são beneficiados com o uso de esteróides, o tipo de esteróide que deve ser usado, o momento e a dose correta.

The Effect of S-Adenosylmethionine Treatment on Neurobehavioral Phenotypes in Lesch-Nyhan Disease: A Case Report.

Lesch-Nyhan disease (LND) is an X-linked recessive disorder caused by a deficiency in hypoxanthine-guanine phosphoribosyl transferase. Patients with LND experience involuntary movements, including dystonia, choreoathetosis, opisthotonos, ballismus, and self-injury. Alleviating these involuntary movements is important to improve the quality of life in patients with LND. Many clinicians have difficulty controlling these involuntary movements in their patients, and there are no established and effective treatments. A 6-month-old boy with LND presented with generalized dystonia and self-injury behavior that was alleviated after receiving S-adenosylmethionine (SAMe). His self-injury behavior completely resolved after he received SAMe and risperidone. Although he had often experienced inspiratory stridor because of laryngeal dystonia and frequently developed aspiration pneumonitis and bronchitis, no inspiratory stridor was noted after SAMe treatment. The patient is continuing to receive SAMe and risperidone. SAMe treatment alleviates dystonic movements and improves quality of life in pediatric patients with LND. Additional research is needed to determine the long-term safety and efficacy of SAMe and its appropriate dosage.

Diámetro de la columna de aire de cuerdas vocales como predictor de estridor laríngeo postextubación / Diameter of the vocals cord air column as a laryngeal stridor predictor post-extubation / Diâmetro da coluna de ar das cordas vocais como predictor do estridor laringeo pós-extubação

Resumen: Objetivo: Correlacionar la presencia de estridor laríngeo con el diámetro de la columna de aire de cuerdas vocales a través de ultrasonido de vía aérea en pacientes intubados en UCI. Material y métodos: Descriptivo. Se midió el diámetro de la columna de aire de cuerdas vocales con ultrasonido lineal en pacientes con weaning, se tomaron cinco grupos de acuerdo con el diámetro: < 5 mm, 5.1-5.5 mm, 5.5-6.0 mm, 6.1-6.4 y > 6.5 mm. Se observó la presencia o la ausencia de estridor laríngeo. Se realizó análisis estadístico tipo frecuencias y porcentajes, con variables de tipo cuantitativo se determinaron medidas de tendencia central, media, moda y desviación estándar. Resultados: Se incluyeron 30 pacientes, la media de edad fue de 39.5 años, una desviación estándar de 18.666, 78.6% de los pacientes permaneció intubado por un periodo de 2-5 días, 83.3% tuvo extubación exitosa, cinco fracasaron a la extubación, dos de ellos por estridor laríngeo, que correspondió a 6.7% con un diámetro de 5.1-5.5 mm., Pearson 0.48. Conclusiones: La medición del diámetro de la columna de aire de cuerdas vocales es una herramienta útil para el médico intensivista como predictor de estridor laríngeo postextubación.

Abstract: Objective: To correlate the presence of laryngeal stridor with the diameter of the vocal cord air column through airway ultrasound in patients intubated in the ICU. Material and methods: Descriptive. The diameter of the vocal cord air column was measured with linear ultrasound in weaning patients, five groups were taken according to the diameter: < 5 mm, 5.1-5.5 mm, 5.5-6.0 mm, 6.1-6.4 and > 6.5 mm. The presence or absence of laryngeal stridor was observed. Statistical analysis was carried out, such as frequencies and percentages, with variables of a quantitative type, measures of central tendency, mean, mode, and standard deviation were determined. Results: Thirty patients were included, the mean age was 39.5 years, a standard deviation of 18.666, 78.6% of the patients remained intubated for a period of 2-5 days, 83.3% presented successful extubation, five presented failure to extubation, two of them due to laryngeal stridor, which corresponded to 6.7% with a diameter of 5.1-5.5 mm., Pearson 0.48. Conclusions: The measurement of the diameter of the vocal cord air column is a useful tool for the intensivist physician as a predictor of laryngeal stridor after extubation.

Resumo: Objetivo: Correlacionar a presença de estridor laríngeo com o diâmetro da coluna aérea das cordas vocais por meio de ultrassonografia das vias aéreas em pacientes intubados na UTI. Material e métodos: Descritivo. O diâmetro da coluna de ar das cordas vocais foi medido com ultrassonografia linear em pacientes com desmame ventilatório. 5 grupos foram selecionados de acordo com o diâmetro: < 5 mm, 5.1-5.5 mm, 5.5-6.0 mm, 6.1-6.4 e > 6.5 mm. Observou-se a presença ou ausência de estridor laríngeo. Realizou-se análise estatística tipo frequências e porcentagens, com variáveis de tipo quantitativo, foram determinadas medidas de tendência central, média, moda e desvio padrão. Resultados: Foram incluídos 30 pacientes, a média de idade foi de 39.5 anos, um desvio padrão de 18.666, 78.6% dos pacientes permaneceram intubados por um período de 2-5 dias, 83.3% apresentaram sucesso na extubação, 5 apresentaram falha na extubação, 2 deles por estridor laríngeo, correspondendo a 6.7% com diâmetro de 5.1 a 5.5 mm. Pearson 0.48. Conclusão: A medida do diâmetro da coluna aérea das cordas vocais é uma ferramenta útil para o médico intensivista como preditor de estridor laríngeo após a extubação.

Laryngeal stridor in multiple system atrophy: Clinicopathological features and causal hypotheses.

Laryngeal stridor is recognized as a characteristic clinical manifestation in patients with multiple system atrophy (MSA). However, the pathogenic mechanisms underlying this symptom are controversial. Neurogenic atrophy of the posterior cricoarytenoid muscle has been identified in cases of MSA, suggesting that laryngeal abductor weakness contributes to laryngeal stridor. However, dystonia in the laryngeal adductor muscles has also been reported to cause laryngeal stridor. Depletion of serotonergic neurons in the medullary raphe nuclei, which exert tonic drive to activate the posterior cricoarytenoid muscle, has recently been identified in MSA cases. This adds weight to the possibility that laryngeal abductor weakness underlies laryngeal stridor in MSA. Continuous positive airway pressure therapy is currently used in the treatment of laryngeal stridor, but should be used with caution in patients showing contraindications. Current knowledge of the clinical and neuropathological features of laryngeal stridor is summarized in this paper, and the hypothesized causes and possible therapeutic options for this symptom are discussed.

Ataxia, ophthalmoplegia and laryngeal stridor related to glutamic acid decarboxylase antibodies / Ataxia, oftalmoplegia e estridor laríngeo relacionados a anticorpos antidescarboxilase do ácido glutâmico

Glutamic acid decarboxylase (GAD) is the enzyme responsible for the conversion of glutamate to gamma-aminobutyric acid (GABA) in the central nervous system. The presence of anti-GAD antibody in cerebrospinal fluid and high levels in blood have been described in some neurological disorders, such as stiff person syndrome and cerebellar ataxia. It is postulated that African descent with anti-GAD may exhibit more severe neurological impairment. We report a case of a young adult African descent with cerebellar syndrome associ-ated with ophthalmoplegia and laryngeal stridor. We found in the literature relationship of ophthalmoplegia plus ataxia with anti-GAD, but no reports of these symptoms with laryngeal stridor, apparently being the first reported case.

Descarboxilase do ácido glutâmico (GAD) é a enzima responsável pela conversão do glutamato em ácido gama-aminobutírico (GABA) no sistema nervoso central. A presença do anticorpo anti-GAD no líquido cefalorraquidiano e em altos níveis no sangue tem sido descrita em alguns distúrbios neurológicos, tais como a síndrome da pessoa rígida e ataxia cerebelar. Postula-se que pacientes afrodescendentes podem apresentar comprometimento neurológico mais severo. Relatamos o caso de um adulto jovem afrodescendente com síndrome cerebelar associada a oftalmoplegia e estridor laríngeo. Encontramos na literatura relação entre a oftalmoplegia com ataxia e anti-GAD, mas nenhum relato desses sintomas com estridor laríngeo, sendo aparentemente o primeiro caso reportado.

Progress of laryngeal cartilage dysplasia / 国际儿科学杂志

Laryngeal cartilage dysplasia,also known as congenital laryngeal cartilage softening or con-genital laryngeal stridor,a clinical common laryngeal disease in infant. It is often shown as airflow makes special sound through larynx or tracheal stenosis,also known as laryngeal stridor. Obstruction can be caused by airway lumen or external pressure. Airway obstruction position can be located in the nose,pharynx and larynx and tra-chea. Laryngeal stridor can be caused by many diseases,such as congenital laryngeal stridor,acute laryngeal,or acute laryngotracheobronchitis,respiratory tract foreign bodies,congenital laryngeal stenosis and laryngeal web, etc. The pathogenesis,pathology,clinical manifestation,diagnosis and treatment are reviewed.

Management of a rare case of arytenoid mucosa oedema inducing stridor and cough.

OBJECTIVE: Current guidelines recommend a multidisciplinary systematic integrated approach to patient with chronic cough in which nowadays otolaryngologists play an increasingly valuable role as new procedures are used for evaluation and treatment. Surgery has been never taken into consideration to treat refractory chronic cough. METHOD: We present a rare case of arytenoid mucosa oedema inducing stridor and cough, lasting 8 months, that critically affected the quality of life of a 15-year-old adolescent arrived to our emergency department with prominent arytenoid oedema causing paroxysmal cough and laryngeal stridor and that was successfully treated by laryngeal debridement. RESULTS: In this case report we observed that prominent swelling of arytenoid mucosa might sustain a vicious cycle of cough persistence and that laryngeal surgical debridement might interrupt it resolving the particular clinical condition. CONCLUSION: The description of this case could be of some help for clinicians to draw new insight about diagnosis and therapy of rare selected cases of chronic refractory cough.

Etiology of laryngeal stridor in 297 cases of infants / 中国小儿急救医学

ObjectiveTo summarize the cause of laryngeal stridor in infants in order to make accurate diagnosis and treatment of the disease.MethodsWe reviewed medical records of 297 cases of patients less than 3 year of age with the presenting symptom of stridor who were initially evaluated in the outpatient setting of otorhinolaryngological department from Jan 2005 to Jan 2010.The causes of stridor were clarified by examinations of ultrafine electronic laryngoscope,throat three-dimensional CT,and bronchoscopy in all cases.Patients underwent history-taking,physical examination and flexible laryngoscopy,CT examination or bronchoscopy evaluation in the operating room.ResultsOf all 297 patients,199 cases ( 67.0％ ) were diagnosed as congenital airway abnomalities for cause of stridor,which included congenital laryngeal abnomalities in 169(84.9％,169/199) and congenital tracheal abnormalities in 30 cases( 15.1％,30/199).Another 98 cases (33.0％,98/297) were diagnosed as acquired disease for cause of stridor.The most congenital laryngeal anomaly was laryngomalacia ( 159,94.1％,159/169 ).The most congenital tracheal abnormalities was tracheomalacia ( 14,46.7％,14/30 ).Sixty-four cases ( 65.3％,64/98 ) were diagnosed as foreign body in airway and 26 cases (26.5％,26/98) were respiratory infection,which were the first and second most common causes of acquired disease for stuidor.ConclusionCongenital airway structural abnormalities as a major cause of infant laryngeal stridor,followed by acquired disorders,including airway foreign body and infection.

Laringomalacia en una cohorte de seguimiento del desarrollo infantil por antecedentes de encefalopatía perinatal: Implicaciones para su conceptualización nosológica / Laryngomalacia in a Follow-Up of a Child Development Cohort for Antecendents of Perinatal Encephalophaty: Implications for Nosologic Conceptualization

Se presenta el análisis retrospectivo de 10 casos de estridor laríngeo congénito. Se discuten algunos reportes de endoscopios laríngeas con los diagnósticos elaborados. Se define la laringomalacia como una entidad caracterizada por flacidez laríngea y estridor. Algunos autores postulan, además de la inmadurez de los tejidos, la posibilidad de incoordinación laríngea y disquinesia. Se apoyan en casos de presentación tardía, de daño neurológico y casos atípicos relacionados con estado funcional o con anestesia Se realizaron endoscopios en 10 casos provenientes de una cohorte de seguimiento longitudinal de casos con diagnóstico de daño neurológico perinatal que se atienden en el Laboratorio del Seguimiento del Neurodesarrollo, del Instituto Nacional de Pediatría/Universidad Autónoma Metropolitana. En un caso se diagnosticó parálisis unilateral de cuerda vocal, postoperatoria. Un caso tuvo anillo vascular, Los ocho casos restantes cubrieron los criterios de laringomalacia, pero por sus características se insiste en que no se trata de una alteración anatómica idiopática sino de una hipotonía funcional. Se enfatiza la necesidad de realizar estudio integral y describir la comorbilidad.

We conducted a retrospective analysis of 10 cases of congenital laryngeal stridor. Reports of laryngeal endoscopy and diagnosis define laryngomalacia as laryngeal flaccidity and stridor. Some authors postulate that in addition to immaturity of cartilage, there exist the possibility o flaryngeal uncoordination and dyskinesia. They support this idea in cases of late presentation, neurological damage, and atypical cases related with functional state or anesthesia. Laryngeal endoscopies were carried out in 10 cases included in a cohort of subjects from a longitudinal follow-up diagnosed with neurologica damage of perinatal origin. One case was diagnosed with postoperative unilateral paralysis of vocal chord and another identified vascular ring. The eight remaining cases fulfilled laryngomalacia criteria of diagnosis, but because of their characteristics origin is not an anatomic alteration but a functional hypotonia. The need to carry out an integral study to describe co-morbidity is emphasized.