Safety of office-based flexible endoscopic procedures of the pharynx and larynx under topical anesthesia.

INTRODUCTION: Flexible endoscopic procedures (FEP) using a working channel allowed otolaryngologists to perform more procedures on the pharynx and the larynx under local anesthesia. The purpose of this work is to demonstrate the feasibility and safety of this technique by studying the adverse effects of this practice in an office-based setting. METHODS: This is a monocentric retrospective cohort study. We searched the database using the French procedural code for FEP performed in an outpatient setting between January 2005 and December 2020. Data regarding the patient's characteristics, indications, and periprocedural complications were extracted. RESULTS: In total, we included 231 patients with a total of 308 FEP: 36% biopsy, 20% hyaluronic acid injection (including 3.5% at the level of the cavum), 20% injection of other substances (in descending order: botulinum toxin, cidofovir, physiological serum, cortisone), 20% exploration for an occult tumor, 3% samples for microbiological analysis, 1% other procedures. Of the 308 FEP included in this study, 24 patients (10.3%) had complications corresponding to 7.8% of the procedures performed. During the procedures, reported complications include minor laryngeal bleeding (n = 5), vasovagal syncope (n = 5), laryngospasm (n = 1) or nausea (n = 3), dysphagia (n = 3), and voice disorders (n = 3). Post-procedural complications were hypertensive crisis (n = 1), asthma attack (n = 1), pneumonia (n = 1), laryngitis (n = 1). Using the Clavien-Dindo classification system, these complications could be defined as grade I (laryngeal bleeding, vasovagal syncope, laryngospasm, dysphagia, nausea, voice disorders, and laryngitis) and grade II (hypertensive crisis, asthma attack, pneumonia) in 9.1% and 1.2% of cases, respectively. Most of these complications were self-limiting, while asthma attacks, pneumonia, laryngitis, and voice disorders required a medical intervention. All complications were managed without sequelae. There was no serious complication grade (no grade III, IV or V). CONCLUSIONS: FEP, which is now well standardized in our institution, makes it possible to carry out a wide range of interventions with little morbidity. These results are in line with those of literature but this technique remains out of nomenclature in France. Our experience led to the development of an evidence-based standard of care that can serve as a framework for practitioners on a nationwide level, while the work to establish official guidelines by the French society of phoniatrics and laryngology is in progress.

Paraneoplastic jaw dystonia and laryngospasm with antineuronal nuclear autoantibody type 2 (anti-Ri).

BACKGROUND: Opsoclonus-myoclonus syndrome and breast carcinoma were initially described as neurologic and oncologic accompaniments of antineuronal nuclear autoantibody type 2 (ANNA-2, also known as anti-Ri). However, the neurologic spectrum of ANNA-2 autoimmunity is broader, includes a syndrome of jaw dystonia and laryngospasm, and can be accompanied by lung carcinoma. OBJECTIVE: To describe clinically (with a video) ANNA-2-associated jaw dystonia and laryngospasm, its pathologic correlates, and therapeutic outcomes. DESIGN: Retrospective case series with prospective clinical follow-up. SETTING: Mayo Clinic's Neuroimmunology Laboratory, Rochester, Minnesota. PATIENTS: Consecutive patients with ANNA-2 seropositivity identified since January 1, 1990. MAIN OUTCOME METHODS: Clinical (in 9 patients) and neuropathologic (in 2 patients) findings were reviewed. RESULTS: Of 48 patients with ANNA-2 seropositivity, 9 (19%) had multifocal neurologic manifestations that included jaw dystonia and laryngospasm. Among 6 patients with jaw dystonia, 5 had severely impaired nutrition, causing profound weight loss. Five patients had documented laryngospasm, which contributed to 1 patient's death. Neuropathologic examination revealed diffuse infiltration by CD8(+) T lymphocytes, with axonal loss and gliosis in brainstem and descending spinal cord tracts. Some patients improved symptomatically after immunosuppressant or cytotoxic therapies; 1 patient improved after treatment with botulinum toxin. One patient who underwent tracheostomy because of recurrent laryngospasm was alive and well longer than 3 years after symptom onset. CONCLUSIONS: Jaw dystonia and laryngospasm are common accompaniments of ANNA-2 autoimmunity and are associated with significant morbidity. We propose that selective damage to antigen-containing inhibitory fibers innervating bulbar motor nuclei by CD8(+) T lymphocytes (histopathologically observed infiltrating brainstem reticular formation) is the proximal cause of this syndrome. Early and aggressive therapy offers the prospect of neurologic improvement or stabilization.

Sudden death in a toddler with laryngotracheitis caused by human parainfluenza virus-1.

Laryngotracheitis caused by human parainfluenza virus (HPIV) and not complicated by bacterial superinfection rarely causes sudden unexpected death in infants and toddlers, especially in the absence of stridor and a barking cough. We therefore describe a 15-month-old white male who died suddenly and unexpectedly with clinical and pathological features of laryngotracheitis caused by culture-proven HPIV-1 infection. Given the presence of mucosal inflammation extending into the vocalis muscle of the larynx without associated significant narrowing of the laryngotracheal airway lumen, we propose his death was a result of a laryngospasm, perhaps mediated by immune responses.

Focal white matter changes in spasmodic dysphonia: a combined diffusion tensor imaging and neuropathological study.

Spasmodic dysphonia is a neurological disorder characterized by involuntary spasms in the laryngeal muscles during speech production. Although the clinical symptoms are well characterized, the pathophysiology of this voice disorder is unknown. We describe here, for the first time to our knowledge, disorder-specific brain abnormalities in these patients as determined by a combined approach of diffusion tensor imaging (DTI) and postmortem histopathology. We used DTI to identify brain changes and to target those brain regions for neuropathological examination. DTI showed right-sided decrease of fractional anisotropy in the genu of the internal capsule and bilateral increase of overall water diffusivity in the white matter along the corticobulbar/corticospinal tract in 20 spasmodic dysphonia patients compared to 20 healthy subjects. In addition, water diffusivity was bilaterally increased in the lentiform nucleus, ventral thalamus and cerebellar white and grey matter in the patients. These brain changes were substantiated with focal histopathological abnormalities presented as a loss of axonal density and myelin content in the right genu of the internal capsule and clusters of mineral depositions, containing calcium, phosphorus and iron, in the parenchyma and vessel walls of the posterior limb of the internal capsule, putamen, globus pallidus and cerebellum in the postmortem brain tissue from one patient compared to three controls. The specificity of these brain abnormalities is confirmed by their localization, limited only to the corticobulbar/corticospinal tract and its main input/output structures. We also found positive correlation between the diffusivity changes and clinical symptoms of spasmodic dysphonia (r = 0.509, P = 0.037). These brain abnormalities may alter the central control of voluntary voice production and, therefore, may underlie the pathophysiology of this disorder.

Variant Gaucher disease characterized by dysmorphic features, absence of cardiovascular involvement, laryngospasm, and compound heterozygosity for a novel mutation (D409H/C16S).

A 20-month-old girl with developmental delay, dysmorphic features, horizontal supranuclear gaze palsy, retrocollis, and episodes of laryngospasm was diagnosed with variant neuronopathic Gaucher disease. The diagnosis was made enzymatically. Mutation analysis showed compound heterozygosity for D409H and a previously unreported mutation C16S. The presence of dysmorphic features, laryngospasm, absent cardiac findings, and the severe clinical phenotype distinguishes our case from other cases of variant neuronopathic Gaucher disease. We therefore propose to extend the spectrum of variant Gaucher disease.

[Determining the type of drowning based on the levels of blood in the thoracic duct lymph]. / Obosnovanie tipa utopleniia po vyrazhennosti limfogemii v grudnom protoke.

The degree of lymphogemy in persons who died of different causes (including drowning) was studied by histologic analysis of the thoracic lymphatic ducts. It was found that quantitative estimation of the presented feature makes it possible to estimate the specific type of drowning which depends on the level of venous hypertension in the system of superior vena cava. This phenomena is strongly marked in spastic and mixed types of drowning and is absent in aspiration and reflex types.

Laryngospasm induced pulmonary edema.

Two case reports of laryngospasm-induced pulmonary edema following general anesthesia are presented. Therapy consisted of immediate reintubation, application of positive pressure ventilation, and diuresis. This phenomenon should be recognized rapidly with appropriate therapy instituted immediately to avoid other complications. Preventive measures are discussed and a modified protocol of the management is outlined.

[Tetanus. Apropos a case]. / Le tétanos. A propos d'un cas.

The authors report a case of tetanus in a 19 months infant who has only received BCG. After a four days period during which two laryngeal spasms occurred, the infant presents a generalized tetanus. The outcome was favorable with specific serotherapy myorelaxants and assisted ventilation of four weeks duration.