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Ear, Nose, & Throat Journal Jul 2023Rosai-Dorfman disease (RDD) is a rare benign systemic histiocytic proliferation characterized by massive lymph node enlargement and sometimes associated with extranodal...
Rosai-Dorfman disease (RDD) is a rare benign systemic histiocytic proliferation characterized by massive lymph node enlargement and sometimes associated with extranodal involvement. Even though it is considered to be benign, death can occur depending on the extent and location. Our case highlights a primary extranodal site of the right pinna with extension through the Eustachian tube to the subglottis. A previously healthy 15-year-old female presented with 1-year right pinna swelling, slowly enlarging and becoming more bothersome. An incisional biopsy was performed on the ear along with S100 staining yielding a diagnosis. After multidisciplinary case discussion, clofarabine monotherapy and systemic therapy for Langerhans cell histiocytosis has started. Rosai-Dorfman disease can be a general disorder, often affecting the lymph nodes. Unlike a nodal disease, extranodal disease could involve any site on the patient's anatomy. Head and neck lesions are the most common extranodal lesions. Rosai-Dorfman disease is self-limited in more than 20% of the cases with spontaneous regression without intervention; 70% of the patients have noticeable symptoms and vital organ involvement requiring treatments such as surgery, steroids, radiation, and chemotherapy. In our case, the patient had wide involvement and presented without any serious breathing difficulties; we decided to start with monotherapy with chemotherapy and systematic glucocorticoid treatment.
Topics: Female; Humans; Adolescent; Histiocytosis, Sinus; Lymphadenopathy; Neck; Lymph Nodes; Ear, External
PubMed: 33973483
DOI: 10.1177/01455613211016704 -
Otolaryngology--head and Neck Surgery :... Dec 2021Subglottic stenosis (SGS) is a known complication of granulomatosis with polyangiitis (GPA). We investigated the impact of medical and surgical interventions on the...
OBJECTIVE
Subglottic stenosis (SGS) is a known complication of granulomatosis with polyangiitis (GPA). We investigated the impact of medical and surgical interventions on the surgical dilation interval and characterized patients with glottic involvement.
STUDY DESIGN
A retrospective chart review of patients with GPA-associated SGS was performed from 2010 to 2019.
SETTING
Tertiary academic medical center.
METHODS
The impact of medical and surgical interventions on dilation interval was assessed. The prevalence of glottic involvement was assessed, and clinical characteristics and outcomes were compared with patients without glottic involvement.
RESULTS
A total of 39 patients with GPA-associated SGS were analyzed. Dilation intervals in patients receiving leflunomide (n = 4; median, 484 days; 95% CI, 405-1099) were greater than in those not receiving leflunomide (median, 155 days; 95% CI, 48-305; = .033). The surgical technique used did not affect dilation interval. Patients with glottic involvement (n = 13) had a greater incidence of dysphonia (13/13 vs 15/26 [58%], = .007) and a shorter dilation interval with involvement (median, 91 days; interquartile range, 70-277) versus without involvement (median, 377 days; interquartile range, 175-1148; hazard ratio, 3.38; 95% CI, 2.26-5.05; < .001). Of 13 patients, 8 (62%) did not have glottic involvement on first presentation.
CONCLUSION
Although GPA is classically thought to affect the subglottis, it also involves the glottis in a subset of patients. These patients have greater complaints of dysphonia and require more frequent surgery. Systemic therapy may increase dilation intervals. In this preliminary study, patients taking leflunomide demonstrated an improvement, highlighting the need for further study of immunosuppression regimens in the treatment of GPA-associated SGS.
Topics: Adult; Dilatation; Female; Granulomatosis with Polyangiitis; Humans; Immunosuppression Therapy; Immunosuppressive Agents; Laryngostenosis; Male; Retrospective Studies; Time Factors
PubMed: 33845664
DOI: 10.1177/01945998211004264 -
International Journal of Surgery Case... May 2021Leiomyosarcoma is a rare mesenchymal tumor that originates from smooth muscle cells. Head and neck LMSs represent only 3% of all leiomyosarcomas with less than 50 cases...
INTRODUCTION
Leiomyosarcoma is a rare mesenchymal tumor that originates from smooth muscle cells. Head and neck LMSs represent only 3% of all leiomyosarcomas with less than 50 cases of laryngeal LMS reported in the literature till now.
CASE PRESENTATION
We report a case of 50-year-old male presented at our ENT department for a chronic hoarseness. Clinical examination investigations found small submucosal lesion in the right vocal cord. Treatment consisted of CO2 Laser excision of the lesion. The evolution was marked by the appearance of a tumefaction in the left submandibular region and a severe dyspnea requiring an emergency tracheotomy. Paraclinical examination investigations found a supraglottis-glottis-subglottis tumor. A total laryngectomy with bilateral functional neck dissection was performed and the histopathological examination found a laryngeal leiomyosarcoma.
CONCLUSION
LMS of the larynx a very rare malignancy. The accurate diagnosis is histological. Surgery is the mainstay of treatment. Its prognosis is correlated to local recurrence and distant metastases.
PubMed: 33838483
DOI: 10.1016/j.ijscr.2021.105830 -
The American Journal of Case Reports Mar 2021BACKGROUND Hemangiomas are relatively rare, slow-growing, benign neoplasms that can cause necrosis, ulceration, and infection leading to airway obstruction or... (Review)
Review
Potassium-Titanyl-Phosphate (KTP) Laser Photocoagulation Combined with Resection Using an Ultrasonic Scalpel for Pharyngolaryngeal Hemangioma via a Transoral Approach: Case Report and Literature Review.
BACKGROUND Hemangiomas are relatively rare, slow-growing, benign neoplasms that can cause necrosis, ulceration, and infection leading to airway obstruction or intractable hemorrhage. Controversy persists regarding the treatment options for these tumors, which include active observation, corticosteroids, sclerotherapy, laser treatment, and surgical resection. CASE REPORT A 61-year-old man presented with a 6-month history of persistent throat clearing and foreign body sensation in the throat. He was receiving medical treatment and psychotherapy for major depressive disorder and anxiety disorder. Laryngoscopy and computed tomography revealed a large, irregular, lobulated mass covered by bluish mucosa in the hypopharynx and larynx on the right without involvement of the true vocal fold or subglottis. Orotracheal intubation was performed under general anesthesia. The hemangioma abutting the epiglottis and arytenoid was dissected by CO₂ laser. The hemangioma in the pharyngoepiglottic fold and aryepiglottic fold was resected using an ultrasonic scalpel. The hemangioma in the ventricle, false vocal fold, and paraglottic space was treated by potassium-titanyl-phosphate (KTP) laser photocoagulation. Pathological examination confirmed hemangioma. There has been no recurrence during 18 months of follow-up. CONCLUSIONS The treatment of pharyngolaryngeal hemangiomas is challenging. It is important to select treatment options considering the characteristics of the treatments and the anatomical and functional relationship between the hemangioma and the surrounding structures. Single-session KTP laser photocoagulation combined with surgical resection using an ultrasonic scalpel via a transoral approach according to anatomic site could be an effective treatment for pharyngolaryngeal hemangioma.
Topics: Depressive Disorder, Major; Hemangioma; Humans; Laser Therapy; Lasers, Solid-State; Light Coagulation; Male; Middle Aged; Neoplasm Recurrence, Local; Phosphates; Potassium; Treatment Outcome; Ultrasonics
PubMed: 33753714
DOI: 10.12659/AJCR.931042 -
Surgical Endoscopy Aug 2021We investigated a novel minimally invasive surgical platform for use in the oropharynx, hypopharynx, and larynx for single-port transoral surgery used in concert with...
BACKGROUND
We investigated a novel minimally invasive surgical platform for use in the oropharynx, hypopharynx, and larynx for single-port transoral surgery used in concert with standard transoral laryngeal and pharyngeal instrumentation.
METHODS
The preclinical investigational device by Fortimedix Surgical B.V. (Netherlands) features two channels for manually controlled flexible articulating surgical instruments. A third central channel accepts both rigid and flexible endoscopes. The system is coupled to a standard laryngoscope for transoral access. In three cadaver models, we evaluated the surgical capabilities using wristed grasping instruments, microlaryngeal scissors, monopolar cautery, and a laser fiber sheath. Procedures were performed within the oropharynx, supraglottis, glottis, subglottis, and hypopharynx.
RESULTS
Within the oropharynx, we found adequate strength, range of motion, and dexterity to perform lateral oropharyngectomy and tongue base resection. Within the larynx, visualization was achieved with a variety of instruments including a flexible, 0° and 30° rigid endoscope. The glottis, supraglottis, pyriform sinuses, post-cricoid space, and esophageal inlet were readily accessible. Visualization and manipulation of grasping, laser, and monopolar cautery instruments were also possible within the subglottis. Instrument reach and accuracy facilitated completion of a delicate micro-flap on the true vocal fold. Other procedures included vocal fold resection, cricopharyngeal myotomy, and resection of subglottic mucosa.
CONCLUSIONS
From this initial proof of concept experience with this novel platform, we found a wide range of procedures within the oropharynx, larynx, and hypopharynx to be feasible. Further work is needed to evaluate its applicability to the clinical setting. The ability of this platform to be used with conventional instrumentation may provide an opportunity for complex transoral surgery to be performed in a facile manner at greatly reduced cost.
Topics: Endoscopes; Humans; Hypopharynx; Laryngoscopes; Microsurgery; Minimally Invasive Surgical Procedures; Robotic Surgical Procedures
PubMed: 33712940
DOI: 10.1007/s00464-021-08420-2 -
BMJ Case Reports Feb 2021Chondrosarcoma of the larynx is rare accounting for approximately 1% of laryngeal cancers; clear cell subtype is a rare variant. Given the low risk of occult nodal...
Chondrosarcoma of the larynx is rare accounting for approximately 1% of laryngeal cancers; clear cell subtype is a rare variant. Given the low risk of occult nodal disease, they present a unique opportunity to maximise tissue preservation in order to optimise both recovery and long-term functional outcomes. We present a case of laryngeal clear cell chondrosarcoma causing critical airway obstruction. An emergency tracheostomy was performed and mapping biopsies were taken. The tumour originated from the cricoid and extended into both arytenoid superstructures precluding cricotracheal resection. A modified narrow-field laryngectomy was performed, preserving all pharyngeal mucosa and neurovascularly intact infrahyoids. Organ preservation surgery is preferred in the management of laryngeal chondrosarcoma. If laryngectomy is required, the surgeon must ensure that all uninvolved, functional tissue is preserved carefully to improve swallow and voice outcomes postoperatively. We describe a novel technique used to achieve this outcome.
Topics: Airway Obstruction; Arytenoid Cartilage; Biopsy; Chondrosarcoma; Cricoid Cartilage; Humans; Laryngeal Neoplasms; Laryngectomy; Magnetic Resonance Imaging; Male; Middle Aged; Tomography, X-Ray Computed; Tracheostomy
PubMed: 33563684
DOI: 10.1136/bcr-2020-236044 -
Regenerative Therapy Dec 2020Decellularized tissue exhibits cell matrix-like properties, along with reduced antigenicity. We explored the potential of decellularized allogeneic trachea to restore...
INTRODUCTION
Decellularized tissue exhibits cell matrix-like properties, along with reduced antigenicity. We explored the potential of decellularized allogeneic trachea to restore the upper respiratory tract, focusing on pediatric application. This study specifically aimed at long-term observation of tissue regeneration using a micro-miniature pig model.
METHODS
Artificial defects (15 × 15 mm) in the subglottis and trachea of micro-miniature pigs were repaired by transplantation of either allogeneic decellularized or fresh (control) tracheal patches. Pigs were evaluated , by bronchoscopy, every three months, and sacrificed for histological examination at six and twelve months after transplantation.
RESULTS
No airway symptom was observed in any pig during the observation period. Bronchoscopy revealed the tracheal lumen to be restored by fresh grafts, showing an irregular surface with remarkable longitudinal compression; these changes were mild after restoration with decellularized grafts. Histologically, while fresh graft patches were denatured and replaced by calcified tissue, decellularized patches remained unchanged throughout the observation period. There were regeneration foci of cartilage adjacent to the grafts, and some foci joined the decellularized graft uniformly, suggesting the induction of tracheal reconstitution.
CONCLUSION
Allogeneic decellularized tracheal tissue could serve as a promising biomaterial for tracheal restoration, especially for pediatric patients at the growing stage.
PubMed: 33426203
DOI: 10.1016/j.reth.2020.04.010 -
Auris, Nasus, Larynx Aug 2021It remains elusive which factors may influence the morbidity and mortality of lung metastasis (LM) in Laryngeal Squamous Cell Carcinoma (LSCC) patients. The aim of the...
OBJECTIVE
It remains elusive which factors may influence the morbidity and mortality of lung metastasis (LM) in Laryngeal Squamous Cell Carcinoma (LSCC) patients. The aim of the present study was to investigate factors influencing LM and the survival outcomes of LSCC patients with LM.
METHODS
We identified 10,935 patients with LSCC from 2010 to 2014 using the Surveillance, Epidemiology and End Results database. Multivariate logistic regression analysis was used to determine the factors associated with the presence of LM. Multivariate cox regression analysis was used to identify covariates associated with increased all-cause mortality in patients with LM.
RESULTS
Among 10,935 patients with LSCC, 232 (2.12%) patients had LM. The median survival time of patients with LM was 8 months, and 8.37% of patients survived after 3 years. Patients with age ≥ 60 years old, unmarried status, supraglottis, overlapping lesion of larynx, subglottis, pathological grade III, T4 stage, N1 stage, N2 stage, N3 stage and bone, brain or liver metastases were more likely to have LM. Survival analysis showed that chemotherapy and radiotherapy suggested better survival of LSCC patients with LM while pathological grade IV was associated with an increased all-cause mortality.
CONCLUSION
The incidence of LSCC patients with LM varied by age, married status, and tumor subtypes. LSCC patients with LM had poor survival, and only 8.37% of patients survived after 3 years. However, chemotherapy and radiotherapy were found as independent favorable prognostic factors for survival.
Topics: Age Distribution; Aged; Carcinoma, Squamous Cell; China; Female; Humans; Incidence; Laryngeal Neoplasms; Logistic Models; Lung Neoplasms; Male; Marital Status; Middle Aged; Neoplasm Staging; Prognosis; Proportional Hazards Models; Risk Factors; SEER Program; Survival Analysis
PubMed: 33358379
DOI: 10.1016/j.anl.2020.11.009 -
Medicine Dec 2020This retrospective study aimed to investigate bronchospasm after tracheobronchial foreign body removal. Bronchoscopy is the main clinical treatment for removing airway... (Review)
Review
This retrospective study aimed to investigate bronchospasm after tracheobronchial foreign body removal. Bronchoscopy is the main clinical treatment for removing airway foreign bodies, but postoperative airway spasm is very common. In our study, we perform a risk factor analysis of bronchospasm after tracheobronchial foreign body removal. The sample was composed of 261 children with airway foreign bodies who had undergone clinical bronchoscopy for foreign body removal under general anesthesia were enrolled from the department of otolaryngology, the First Hospital of Jilin University from 2014 to 2019, of which 78 in the left bronchus, 107 in the right bronchus, 51 in the main bronchus, and 25 in the subglottis. All patients were confirmed by radiographic examination or pulmonary auscultation. All their medical records and clinical data were retrospectively analyzed; single factor and multiple factor analyses of bronchospasm were performed. The logistic regression analysis showed that age, foreign body retention time and operation time were independent risk factors for postoperative airway spasm. A history of pneumonia was not an independent risk factor for postoperative airway spasm. We should pay more attention in the preoperative period according to the specific situation of child; the right means of anesthesia and appropriate hormonal drugs should be chosen to prevent the occurrence of postoperative airway spasm.
Topics: Bronchi; Bronchial Spasm; Bronchoscopy; Child; Child, Preschool; Female; Foreign Bodies; Humans; Infant; Male; Postoperative Complications; Retrospective Studies; Risk Assessment; Risk Factors; Trachea
PubMed: 33350722
DOI: 10.1097/MD.0000000000023170 -
Lin Chuang Er Bi Yan Hou Tou Jing Wai... Dec 2020To study the anatomical distribution and tumor extent of juvenile laryngeal papilloma(JLP), and to sum up the similarity and difference in distribution between...
To study the anatomical distribution and tumor extent of juvenile laryngeal papilloma(JLP), and to sum up the similarity and difference in distribution between initiatial and recurrent tumor. Data of 65 patients with JLP who were admitted to the Ear Institute, School of Medicine, Shanghai Jiao Tong University from January 2011 to December 2018 were retrospectively analyzed. The RRP tumor assessment method proposed by Derkay et al. was referred to, in which larynx was divided in to 11 anatomical sites. 2.9±1.3 Laryngeal anatomical sites were involved in patients with initial tumor, while that was 3.1±1.5 in patients with recurrent tumor. The most common sites were true vocal fords, anterior commissure and false vocal fords in both group; and the least ones were subglottis, posterior glottis and ary-epiglottic fold. However, subglottis(=0.038) and trachea(=0.007) were more likely to be involved in recurrent tumor, with statistical differences. There were differences in distribution of JLP between initial and recurrent tumor, which might be the result of suitable areas for HPV survival or micro-lesion of residual tumor.
Topics: China; Humans; Laryngeal Neoplasms; Larynx; Papillomavirus Infections; Respiratory Tract Infections; Retrospective Studies
PubMed: 33254341
DOI: 10.13201/j.issn.2096-7993.2020.12.008