Enfermedad de Rosai-Dorfman con compromiso laríngeo: a propósito de un caso / Rosai-Dorfman disease with laryngeal involvement: about a case

RESUMEN La enfermedad de Rosai-Dorfman (ERD) o histiocitosis sinusal con linfadenopatía masiva es una enfermedad infrecuente, de etiología desconocida caracterizada por linfadenopatías cervicales masivas bilaterales. El compromiso extraganglionar puede ocurrir en diferentes sitios, incluida la vía aérea, en la cual la localización nasosinusal es la más frecuente, pero puede comprometer también otros sitios de la vía aérea superior. Dentro de éstos, el compromiso laríngeo es muy poco frecuente. Se presenta el caso de una paciente de 82 años con antecedentes de ERD localizada en los ganglios linfáticos cervicales en remisión espontánea, que desarrolló posteriormente una obstrucción subaguda de las vías respiratorias. La tomografía computarizada y la nasofaringolaringoscopía mostraron lesiones subglóticas que obstruían severamente las vías respiratorias. Se realizó una traqueostomía de emergencia y biopsia de las lesiones, confirmando el diagnóstico de ERD extranodal.

ABSTRACT Rosai-Dorfman disease or sinus histiocytosis with massive lymphadenopathy is a rare disease of unknown etiology characterized by bilateral massive cervical lymphadenopathy. Extranodal involvement can occur in different sites including airway where nasosinusal involvement is common but this disease may occur in other sites of the upper airway. Laryngeal involvement is rare. We present the case of an 82-year-old female patient with a history of Rosai-Dorfman disease located in cervical lymph nodes in spontaneous remission that subsequently develops airway obstruction. Computed tomographic scan and nasopharyngolaryngoscopy showed subglottic lessons that severely obstructed the airway. Emergency tracheotomy and biopsy were performed, confirming the diagnosis of extranodal RDD. Patient was observed and tracheostomy was maintained with good tolerance.

A Prospective Study Of Laryngotracheal Trauma And It‘S Management

BACKGROUND: Traumatic injuries of the larynx are diverse, uncommon, and potentially life threatening. Laryngotracheal trauma can be oadly divided into External trauma , which can be blunt or penetrating trauma , and internal trauma , which can be iatrogenic , thermal , caustic and foreign body injuries .external trauma which can be blunt trauma caused by motor vehicle accidents , suicidal or homicidal strangulation and penetrating trauma caused by suicidal or homicidal cut throat injuries . Iatrogenic injuries are most common cause of internal trauma . If not adequately treated these injuries lead to significant morbidity such as dysphonia , airway stenosis , aspiration and sometimes may lead to death . Laryngotracheal trauma is often associated with concomitant cervical or intracranial trauma or with multisystem poly trauma . External laryngeal trauma is rare. It has a population incidence of 1 in 137,000 in adults and accounts for 0.5% of trauma admissions in children. Incidence of postintubation laryngotracheal stenosis requiring surgical correction is 1 in 204,000 in adults and 4.9 in 100,000 in children.Laryngeal webs , intubation granulomas , laryngeal injuries while intubation , inhalational and ingestion injuries are very rare in incidence 1 . MATERIALS AND METHODS: 20 patients who presented with external and internal laryngotracheal trauma to casualty department in Government general hospital, Kakinada, Andhra Pradesh state , during the period between June 2015 to September 2017. A detailed history was taken with emphasis on trauma.Clinical features were noted and patients were appropriately investigated . RESULTS: The age of patients in present study varied from 12-70 years . Majority of patients are present in 26-40 years age group (55%) . Among the 20 cases in our study 13 cases were males (65%) and 7 cases were females (35%). In our study of 20 patients 14 patients sustained injuries due to external trauma(70%) and 6 patients presented with internal trauma of larynx(30%) due to prolonged intubation . In our study 6 of the 20 patients presented with laryngeal stenosis due to prolonged intubation among them 3 patients presented with subglottic stenosis 2 with glottic stenosis and 1 tracheal stenosis. Amongst them 9 of 14 patients were due to penetrating neck injuries(64.3%) and 5 of 14 are due to blunt neck trauma (35.7%) . 7 of 9 patients knife infected wounds and 2 due to motor vehicle accident. 2 of 5 patients in blunt trauma are due to hanging 2 are due to strangulation and one is due to bullgore injury. Most commonly presented with pain (70%) , dyspnoea (50%) , hoarseness (45%) . stridor was present mostly in laryngeal stenosis patients. The present study 15 of 20 patients airway was initially managed with the help of tracheostomy in 3 of 20 patients with intubation and 2 patients were under observation. There was a recurrence in one case of subglottic stenosis and endoscopic laser exicision was done again .In further followup there was no recurrence. CONCLUSION: In conclusion, we believe that the management of injuries to the larynx and trachea can be individualized based on the clinical presentation and mechanism of injury. Early diagnosis and stratification of treatment based on the initial history, physical findings has improved outcomes. Our goal remains preservation of life with restoration of a normal airway and voice. Patients with blunt injuries can often be managed conservatively with close monitoring in the intensive care unit. Penetrating injuries will often have associated injuries or airway compromise that will mandate operative exploration.

Clinical profile of patients with laryngotracheal stenosis in a tertiary government hospital

@#<p style="text-align: justify;"><strong>OBJECTIVE:</strong> To describe the clinical profile of patients with laryngotracheal stenosis over a 7-year period and discuss strategies for its prevention.<br /><strong>METHODS:</strong><br /><strong>    Design:</strong> Retrospective Case Series<br /><strong>    Setting:</strong> Tertiary Government Hospital<br /><strong>  Participants:</strong> Thirteen (13) patients with laryngotracheal stenosis confirmed by laryngoscopy and/or bronchoscopy.<br /><strong>RESULTS:</strong> Twenty-one patients were evaluated for laryngotracheal stenosis from January 2008 to June  2015, but only 13 with complete data were included in this study. Of the 13 patients, nine (69.2%) belonged to the pediatric age group. Ten (77%) were males and three (23%) were females. Laryngotracheal  stenosis  following endotracheal  tube  (ET)  intubation  was  seen in 11 (84.6%) while 2 had thyroid masses and no  history  of  prior  ET  intubation.  Presenting symptoms or reasons for referral were wheezing (n=4), stridor (n=4), failure to decannulate the tracheostomy tube (n=3), and dyspnea (n=2). Duration of ET intubation was four to 60 days. The highest frequency of ET re-intubation was 5 times. Among those intubated, stenosis was glottic in one, subglottic in five and tracheal in five patients. Three had Cotton-Myer grade I stenosis, two had grade II, three had grade III and three had grade IV stenosis. Those with thyroid masses had tracheal stenosis.<br /><strong>CONCLUSION:</strong> Strategies  for  prevention  of  laryngotracheal  stenosis  should include  routine airway  endoscopy  for  patients  with  longstanding  neck  masses  and  for  those  with  prolonged ET  intubation,  for  whom  the  option  of  early  prophylactic  tracheostomy  is  worth  considering. Otherwise, immediate post-extubation endoscopy may facilitate documentation and appropriate<br />intervention.</p>

Estenosis larinootraqueal revisión de casos en el centro médico imbanaco de cali / Laryngotrachealsstenosis. case studies in centro médico imbanaco in cali, Colombia / Estenose larigottraqueal revisão de casos no centro médico imbanaco de cali

El manejo de la estenosis laringotraqueal continúa siendo un problema difícil para el otorrinolaringólogo por la multiplicidad de hallazgos en los pacientes, su manejo complejo y la falta de un tratamiento único. En el Centro Médico Imbanaco de Cali, Colombia, se realizó un análisis retrospectivo de una serie de pacientes con este diagnóstico tratados entre octubre de 2006 y enero de 2011. Se examinaron los antecedentes, síntomas y signos, tratamientos, enfermedades asociadas, resultados del tratamiento y seguimiento en el tiempo. Se identificaronun total de 44 adultos, con edad promedio de 41,4 años; 24 (55%) eran mujeres y 20 hombres. El promedio de seguimiento fue 19 meses; 28 (63,6%) pacientes mejoraron con el empleo de tratamiento endoscópico y láser de CO2. Los mejores resultados se obtuvieron en el Grupo I (100%) y en el Grupo II (83.3%). La cirugía endoscópica láser, es un tratamiento válido para lesiones delgadas, no complicadas. El uso de técnicas de resección y reconstrucción laringotraqueal es útil en las lesiones complejas y gruesas donde falla el tratamiento endoscópico. No obstante, en casos seleccionados, el tratamiento endoscópico se debe considerar inicialmente. Otras ventajas del tratamiento endoscópico son; pocas complicaciones, baja morbilidad, tiempo quirúrgico y de hospitalización cortos.

The management of laryngotracheal stenosis remains a challenging problem for the otolaryngologist. The complexity of the various preoperative situations implies that no single treatment modality can solve the problem. I performed a retrospective analysis of our experience in Centro Médico Imbanaco in Cali, Colombia, with a series of patients with this condition between October 2006 and January 2011. The study involved retrospective review of the records of patients with laryngotracheal stenosis. Symptoms, antecedents, treatment, associate pathology, outcomes and follow up were examined. A total of 44 adults were identified, the mean age was 41,4 years, 24 were female (55%) and 20 were male. The mean followup time was 19 months. Twenty eight (63.6%) of these cases have been controlled effectively with endoscopy laser techniques. Best results were seen in Group I (100%) and Group II (83.3%). Endoscopic laser surgery is a valid approach for thinner, uncomplicated lesions; patients with thicker, complex lesions in whom endoscopic treatment fails are best managed with laryngotracheal resection a reconstruction, but endoscopic treatment may be considered before tracheal resection in select cases. Endoscopic treatment is associated with few complications, low morbidity, a short operative time, and a short length of hospitalization.

O tratamento da estenose laringotraqueal continua sendo um problema difícil para o otorrinolaringologista pela multiplicidade de descobertas nos pacientes, seu tratamento complexo e a falta de um tratamento único. No Centro Médico Imbanaco de Cali, Colômbia, realizouse uma análise retrospectiva de uma série de pacientes com este diagnóstico, tratados entre outubro de 2006 e janeiro de 2011. Foram examinados os antecedentes, sintomas e signos, tratamentos, doenças associadas, resultados do tratamento e acompanhamento no tempo. Foram identificados um total de 44 adultos, com idade média de 41,4 anos; 24 (55%) eram mulheres e 20 homens. A média de acompanhamento foi 19 meses; 28 (63,6%) pacientes melhoraram com o emprego de tratamento endoscópico e laser de CO2. Os melhores resultados foram obtidos no Grupo I (100%) e no Grupo II (83.3%). A cirurgia endoscópica laser, é um tratamento válido para lesões finas, não complicadas; o uso de técnicas de resecção e reconstrução laringotraqueal são úteis nas lesões complexas e grossas onde falha o tratamento endoscópico; não obstante, em casos selecionados, o tratamento endoscópico deve ser considerado inicialmente. Outras vantagens do tratamento endoscópico são poucas complicações, baixa morbilidade, tempo cirúrgico e de hospitalização curtos.

Comprometimento da árvore respiratória na granulomatose de Wegener / Laryngeal and tracheobronchial involvement in Wegener's granulomatosis

INTRODUÇÃO: A granulomatose de Wegener (GW) é uma forma de vasculite sistêmica que envolve primariamente as vias aéreas superiores e inferiores e os rins. As manifestações mais frequentes nas vias aéreas são estenose subglótica e inflamações, estenoses da traqueia e dos brônquios. A visualização endoscópica das vias aéreas é a melhor ferramenta para avaliação, diagnóstico e manejo dessas alterações. OBJETIVOS: Descrever as alterações endoscópicas encontradas na mucosa das vias aéreas de um grupo de pacientes com GW submetido à broncoscopia no Hospital das Clínicas da Faculdade de Medicina da Universidade de São Paulo (HC-FMUSP) e relatar as intervenções terapêuticas broncoscópicas utilizadas em alguns casos. MÉTODOS: Foram estudados 15 pacientes com diagnóstico de GW provenientes do Ambulatório de Vasculites da Disciplina de Pneumologia do HC-FMUSP, encaminhados para a realização de broncoscopia no serviço de Endoscopia Respiratória do HC-FMUSP no período de 2003 a 2007. RESULTADOS: Dos 15 pacientes avaliados, 11 eram mulheres (73,33 por cento) com idade média de 34 ± 11,5 anos. Foram encontradas alterações das vias aéreas em 80 por cento dos pacientes, e o achado endoscópico mais frequente foi estenose subglótica (n = 6). Realizou-se broncoscopia terapêutica em três pacientes com estenose subglótica e em outros três com estenose brônquica, todos apresentando bons resultados. CONCLUSÃO: A broncoscopia permite diagnóstico, acompanhamento e tratamento das lesões de vias aéreas na GW, constituindo-se um recurso terapêutico pouco invasivo em casos selecionados.

INTRODUCTION: Wegener's granulomatosis (WG) is a form of systemic vasculitis that involves primarily the upper and lower airways and the kidneys. The most frequent airway manifestations include subglottic stenosis and inflammation, and tracheal and bronchial stenoses. The endoscopic visualization of the airways is the best tool for assessing, diagnosing and managing those changes. OBJECTIVES: To describe the endoscopic abnormalities found in the airway mucosa of a group of patients with WG undergoing bronchoscopy at Hospital das Clínicas of the Faculdade de Medicina, Universidade de São Paulo (HC-FMUSP), and to report the therapeutic bronchoscopic interventions used in some cases. METHODS: The study assessed 15 patients diagnosed with GW from the Vasculitis Outpatient Clinic of the Department of Pulmonology, HC-FMUSP, referred for bronchoscopy at the Service of Respiratory Endoscopy, HC-FMUSP, from 2003 to 2007. RESULTS: Fifteen patients were studied [11 females (73.33 percent)]; mean age, 34 ± 11.5 years. Airway changes were found in 80 percent of the patients, and the most frequent endoscopic finding was subglottic stenosis (n = 6). Therapeutic bronchoscopy was performed in three patients with subglottic stenosis and in other three patients with bronchial stenosis, all showing good results. CONCLUSION: Bronchoscopy allows for diagnosing, monitoring, and treating the airway lesions in WG, being a minimally invasive therapeutic option in selected cases.

Post-Intubation Tracheoesophageal Fistula with Posterior Glottic Web

Tracheoesophageal fistula (TEF) after prolonged intubation could present as chronic aspiration and could be mistaken as unilateral or bilateral vocal fold palsy, especially when there was combined posterior glottic synechia. We present a case of post-intubation TEF which was successfully treated with tracheal resection and anastomosis with primary esophageal closure. The accompanying posterior glottic web was treated by endoscopic technique of web lysis, with topical application of mitomycin C solution.

Sequelas laríngeas devido inalação acidental de amônia anidra / Laryngeal sequelae due to accidental inhalation of anhydrous ammonia

Introdução: Amônia Anidra (AA) é um cáustico comumente usado na indústria que, acidentalmente, pode causar queimaduras, mesmo com um breve contato. Como ocorre com outras queimaduras por álcalis, a lavagem precoce remove a AA das áreas afetadas e é fundamental para limitar a lesão tecidual. A concentração do tóxico e a duração do contato determinam o grau de destruição da pele e das mucosas. Um caminhão carregado de AA (NH3) capotou na estrada, liberando densa nuvem de gás AA. Cinquenta e quatro pessoas inalaram o gás e, depois de noventa dias, três pessoas ainda apresentavam rouquidão, sendo por nós examinadas. Relalto do Caso: Avaliamos três pacientes com sequelas laríngeas por queimadura após inalação de AA. Encontramos um caso de hiperemia e edema, um de granuloma de 1/3 posterior de prega vocal esquerda e um de sinéquia de pregas vocais. São comentados os achados de necropsia, tanto macro quanto microscopicamente. São discutidas as sequelas apresentadas, bem como os melhores tratamentos.

Introduction: Anhydrous Ammonia (AA) is a caustic compound commonly used in the industry that can cause burns, even with brief contact. As with other alkali burns, the early washing to remove the AA from burnt areas is crucial to limit tissue damage. The concentration of toxic agent and duration of its contact determine the degree of skin and mucosa destruction. A tanker truck carrying AA (NH3) fell off a highway, and released a dense cloud of AA gas. Fifty-four people inhaled the gas and after ninety days, three people were still experiencing hoarseness and were examined. Case Report: We assessed three patients with laryngeal sequelae due to AA inhalation burn. We found a case of hyperemia and edema, one case of granuloma of the posterior third portion of the left vocal cord, and one case of vocal cord adhesion. Necropsy findings are commented both macroscopically and microscopically. The sequelae and the best treatments for them are discussed herein.

Laryngotracheal Stenosis in Burned Patients with Inhalation Injury: Analysis of Risk Factors / 대한이비인후과학회지

BACKGROUND AND OBJECTIVES: Laryngotracheal stenosis (LTS) in burned patients with inhalation injury have features distinct from other stenosis after intubation or tracheotomy. However, the risk factors for LTS in burned patients with inhalation injury have not been thoroughly assessed. The aim of this study is to identify the risk factors for and evaluate the pattern of LTS in burned patients with inhalation injury. SUBJECTS AND METHOD: From May 2005 to April 2007, 227 burned patients with inhalation injury treated at the Hangang Sacred Heart Hospital, Hallym University Medical Center were investigated retrospectively. The risk factors analyzed for LTS in burned patients with inhalation injury were gender, age, duration of intubation, size of the endotracheal tube, previous tracheotomy, number of intubations, severity of inhalation injury. RESULTS: Among 81 patients who survived and could be followed up until the study period, 10 (12%) patients developed LTS. The stenosis involved the subglottis.cervical trachea (5), subglottis.cervical tracheal and glottis (1), glottis (4). According to Myer-Cotton classification, there were 6 (60%) patients with grade III, IV stenoses on subglottis.cervical trachea. The number of intubations and previous tracheotomy were found to be risk factors for the development of LTS in burned patients with inhalation injury. CONCLUSION: LTS by inhalation injury is usually shown on web at the anterior and posterior glottic areas or circumferential narrowing of the subglottis. Repeated endotracheal intubations and previous tracheotomy in patients with inhalation injury may increase the prevalence of LTS.

Management of Pediatric Airway Stenosis using Cold Instruments and Mitomycin-C / 대한이비인후과학회지

BACKGROUND AND OBJECTIVES: Mitomycin-C is becoming one of the most important agents in the treatment of glottic and subglottic stenosis. This study was conducted to investigate the clinical outcome of endoscopic management in glottic and subglottic stenosis patients using cold instruments and Mitomycin-C. SUBJECTS AND METHODS: A total of 47 patients with glottic and subglottic stenosis who were diagnosed and surgically treated in Seoul National University Children's Hospital from Jan. 2000 through Aug. 2003 were included. All were treated for endoscopic laryngomicrosurgery using cold instruments and bougienage. Thereafter, 0.4 mg/ml Mitomycin-C was directly applied for 5minutes on the surgical site. The surgical treatment outcome was retrospectively analyzed by reviewing medical records. RESULTS: All the studied patients showed improved airway stenosis and subjective symptoms, and twenty-one out of 47 patients have been decannulated successfully without any special complications. Mean number of operation for decannulation was 5.1, and the mean duration from the initial operation to decannulation was 17.3 months. CONCLUSION: Our data suggest that the use of cold instrumentation and topical mitomycin-C application might be a promising, initial choice of management in patients with airway stenosis.

Subglottic Laryngeal Stenosis in Association with Congenital Tracheo-esophageal Fistula: A case report / 대한마취과학회지

Tracheo-esophageal fistula (TEF) is a well known and a relatively common congenital anomaly (1 in 3,000 4,500 live births). Half of the patients with TEF may have other congenital anomalies, VATER syndrome. However, the presence of subglottic laryngeal stenosis in patients with TEF is uncommon. We report a case of TEF (type C) combined with subglottic laryngeal stenosis. We anesthetized a 2.74 kg neonate for reconstruction surgery. But, it was impossible to pass an uncuffed endotracheal tube (2.5 mm inner diameter and 3.6 mm outer diameter) beyond the vocal cord. A gastrostomy was performed only under mask ventilation, and emergent tracheostomy was done. Endotracheal endoscopy revealed TEF combined with subglottic laryngeal stenosis. In this case, she can be managed successfully by the performance of an early tracheostomy.

Preliminary Results of Mitomycin-C Application in the Treatment of Laryngeal Stenosis and Granuloma / 대한이비인후과학회지

BACKGROUND AND OBJECTIVES: Mitomycin-C is an antibiotic, antineoplastic agent that inhibits DNA and protein synthesis, and fibroblast proliferation. It has been successfully used in adjunction to glaucoma surgery, dacryocystorhinostomy, pterygium surgery, and middle meatal antrostomy. The purpose of this study is to evaluate the efficacy and safety of intraoperative Mitomycin-C application as an adjunct therapy in the endoscopic treatment of laryngeal stenosis and granulation. MATERIALS AND METHOD: A total 13 subjects, of whom 2 had anterior glottic web, 4 posterior laryngeal stenosis, and 7 laryngeal granuloma were included. All underwent stroboscopy and were treated with endoscopic laryngomicrosurgery with CO2 laser. Then, 1 cc of 0.4 mg/mL Mitomycin-C was directly applied for 4 minutes on the surgical site. The patients' symptoms were assessed, and the size of the airway was graded on a scale of I (< or =50%) to IV (total occlusion) after a mean follow-up period of 5 months. The recurrence of the laryngeal granuloma was checked. RESULTS: There was a significant improvement in postoperative symptoms in the group of posterior laryngeal stenosis. Two with tracheotomy underwent decannulation. The postoperative size of airway was markedly increased and restenosis was not noted. There was only one case of recurrence in the granuloma group. CONCLUSION: According to these preliminary results, it is suggested that application of Mitomycin-C can be used as a beneficial adjunct therapy in the endoscopic COc laser excision for laryngeal stenosis and granuloma.