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PubMed2001

[Autonomic dysfunction with nocturnal dyspnea (Gerhardt-syndrome) in a patient with multiple system atrophy].

FINDINGS: This case involves a 56-year-old man with a 3-year case history, starting with vertigo, cerebellar symptoms, followed by inspiratory stridor, especially during the night, and autonomic failure with recurrent syncopes. Magnetic resonance imaging confirmed the diagnosis of olivopontocerebellar atrophy and tilting table tests with plasma catecholamine measurement revealed an extensive drop of blood pressure with no adequate change in the heart rate or plasma catecholamine, leading to the diagnosis of multiple system atrophy (MSA). In addition, bronchoscopy revealed that the inspiratory stridor was caused by paradoxical movement of the vocal cords (sometimes known as Gerhardt's syndrome). TREATMENT AND COURSE: The patient needed a tracheotomy and treatment of the autonomic symptoms. He could be discharged in better psychic condition, without syncopes or respiratory problems.

Diagnosis, Differential↗
PubMed2002

[Clinical manifestations of diffuse idiopathic skeletal hyperostosis (DISH)].

BACKGROUND: Diffuse idiopathic skeletal hyperostosis (DISH, Forestier disease) is in most cases a harmless, asymptomatic disease characterized by a massive, noninflammatory ossification with intensive formation of osteophytes affecting ligaments, tendons, and fasciae, especially of the spinal column. If the disease becomes symptomatic at all, the leading complaints will usually be dysphagia in the sense of a lump in the throat and difficulty in swallowing. However, in rare cases the osteophytes may influence the laryngeal function ranging from hoarseness and immobility of the vocal cord to life-threatening inspiratory stridor. Such laryngeal manifestations are almost unknown. PATIENTS AND METHOD: We report on three patients, two female and one male, suffering not only from chronic dysphagia but also from increasing inspiratory stridor and difficult breathing. RESULTS: Especially one case illustrates how difficult it can be to establish the etiological relationship between a cancer-like ulcer in the postcricoid region and a bilateral inactivity of the vocal cord on the one hand and DISH on the other hand. Only the resistance of the ulceration to any therapy as well as rare case reports in the literature confirmed our suspicion that the ulcer and disturbance of laryngeal function were caused by chronic pressure exerted by the huge vertebral osteophytes. In all three patients surgical resection of the osteophytes resulted in complete relief of complaints. CONCLUSION: Uni- or bilateral immobility of the arytenoids, possibly associated with chronic inflammatory hyperplasia of the tissue of the arytenoids and the postcricoid region, may be a symptom of an unusual manifestation of DISH.

Aged↗
PubMed2003

Chronic subglottic and tracheal stenosis: endoscopic management vs. surgical reconstruction.

Laryngotracheal stenosis has been and remains one of the most vexing problems in the field of head and neck surgery. Two treatment modalities prevail, endoscopic and external. The indication for each modality is not yet clearly defined. This undefined situation motivated our current work, and we decided to assess laser-assisted endoscopy (with or without stenting) vs. open surgery for treating chronic laryngotracheal stenosis. Our study included 28 cases of chronic laryngotracheal stenosis that were classified according to treatment in two main groups: group I included 13 patients who were endoscopically treated and group II included 15 patients with surgical reconstruction. The mean follow-up period was 12.58 months for group I and 27.43 months for group II. Respiratory function tests (RFT) were carried out preoperatively, 1-month postoperatively and on completion of follow-up. Except for age, which was significantly higher in group I (P<0.001), there was no significant difference between both groups. Although the incidence of complications was higher in group I (69%) than in group II (47%), it was nonsignificant. The postoperative RFT improved significantly in both groups. Although the improvement was higher in group I than group II, the difference was nonsignificant. The correlation between preoperative stridor and all other variables demonstrated that preoperative stridor correlated with the diameter of the stenosed segment (rs=-0.631, P<0.001) and the peak expiratory flow rate (PEFR) (rs=-0.488, P=0.030). Our results indicate that open surgery is the treatment of choice. Compared with endoscopic treatment, it provides a higher success rate and better functional results, especially long term. However, if contraindications to open surgery exist, whether local or general, laser-assisted endoscopy with stenting can offer good palliative results.

Adult↗
PubMed2005

Growth hormone abuse and bodybuilding as aetiological factors in the development of bilateral internal laryngocoeles. A case report.

A 36-year-old man presented with hoarseness and stridor. He was an elite professional bodybuilder and admitted to having abusing anabolic steroids and growth hormone in the recent past. A CT scan showed bilateral laryngocoeles. The patient was initially managed with intravenous corticosteroids and broad-spectrum antibiotics, and the stridor resolved sufficiently to permit discharge from the hospital. He proceeded to undergo endoscopic marsupialisation of his laryngocoeles and to date has made a full recovery. This is the first reported case where anabolic steroid and growth hormone abuse combined with an elite bodybuilder's exercise regime has been implicated in the aetiology of bilateral laryngocoeles.

Adult↗
PubMed2007

Surgical treatment of exercise-induced laryngeal dysfunction.

A method for combined ergo-spirometry and continuous laryngeal inspection during exercise, entitled continuous laryngoscopy exercise test (CLE-test) has been developed in order to study airway obstruction at the laryngeal level during exercise. The aim of the study was to apply the CLE-test on patients experiencing respiratory distress during exercise in order to reveal the usefulness of the CLE-test both as a diagnostic tool in the selection of patients for surgery and in evaluation of treatment effects postoperatively. Until now, 81 patients with a history of exercise-induced stridor have undergone the CLE-test. Ten of these patients were selected for surgical treatment based on the severity of symptoms and their motivation for treatment. All ten patients underwent endoscopic supraglottoplasty (ES), with laser incision in both aryepiglottic folds anterior to the cuneiform cartilages and removal of the mucosa around the top of the tubercles. Each patient was examined by the CLE-test before and 3 months after surgery. Eight patients felt subjectively that their breathing capacity during exercise was improved. When pre- and postoperative ergo-spirometry evaluations were compared, increased peak oxygen consumption was observed in four out of ten patients and better maximal minute ventilation in seven out of ten. Postoperative evaluation of the laryngeal images showed less prominent aryepiglottic folds. The typical adduction of the supraglottic structures concomitant with inspiratory stridor found preoperatively was not present in any of the patients during exercise postoperatively. The ES procedure is an efficient surgical treatment for exercise-induced laryngeal supraglottic obstruction and the CLE-test eases the selection of patients for surgery and facilitates the evaluation of treatment effects.

Adolescent↗
PubMed2005

Vascular rings--presentation, investigation and outcome.

UNLABELLED: Our aim was to determine the presentation of patients with vascular rings and evaluate the effectiveness of investigations. Surgical outcomes and respiratory sequelae were also examined. The design was a retrospective case note study over a 13-year period set in a tertiary children's hospital. Children below the age of 16 years presenting with a vascular ring to the Royal Hospital for Sick Children, Glasgow were studied. Demographic data at presentation, including symptoms, were recorded. The ability of diagnostic investigations to identify the presence of a vascular ring was evaluated. Surgical outcomes were determined by measuring surgical complications and mortality. Respiratory sequelae were recorded by the presence of persistent symptoms or the need for tracheostomy or long-term ventilation following surgery. A total of 24 patients were identified with a median age at presentation of 4.5 months. Stridor was the commonest presenting symptom (14/24). Angiography, chest CT scanning and MRI were the most accurate imaging modalities (accurate in 100% of cases used). Chest X-ray films and echocardiography had the lowest detection rates. Surgical complications (4/24) and mortality (1/24) were low. A substantial number of patients available to follow-up (7/20) were still experiencing stridor 3 months post-operatively. CONCLUSION: Vascular rings are rare, however, often present with common symptoms. Most children present in early infancy, but a minority presents much later. The investigation of choice is a barium swallow followed by high-resolution computed tomography. Surgery is safe although a number of patients will have persisting symptoms.

Aorta, Thoracic↗
PubMed2005

Sleep disorders in multiple system atrophy.

Complaints about sleep disorders and excessive daytime sleepiness are common among patients with multiple system atrophy. The diffuse neurodegenerative process that encompasses the key structures involved in the regulation of the sleep/wake transition and respiratory function may account for these complaints and for the most frequent polysomnographic findings in MSA, i.e., sleep-related breathing disturbances and REM sleep behaviour disorder, which are both treatable conditions. Nocturnal stridor is an inspiratory sound produced by complex vocal cord muscle dysfunction. Often occurring with sleep apnoea, stridor is associated with decreased survival. REM sleep behaviour disorder, a parasomnia characterized by loss of normal skeletal muscle atonia during REM sleep with prominent motor activity, is detected in almost all patients. The pathophysiology of both disorders is partially elucidated but increasing evidence points to the role of basal ganglia dysfunction.

Animals↗
PubMed1994

Use of expandable wire stents for malignant airway obstruction.

The symptoms of progressive dyspnea and stridor in the setting of malignant airway obstruction are severe and distressing. Conservative nebulizer and oxygen therapy offer little relief, and conventional stenting with T tubes requires a tracheostomy. In this article, we describe our experience with stenting in the treatment of malignant mediastinal disease using the Gianturco expanding metal-wire stents. The technique of placement is simple and the procedure was successful in all 21 cases. Relief of stridor was immediate and the dyspnea usually abated. These benefits continued through the mean survival period after stenting of 134 days (range, 2 to 799 days). The patients required only brief hospitalization (2.83 days) before returning home or to the referring institution. It appears that expandable wire stents may offer a simple yet effective intervention in the palliative treatment of mediastinal malignancy.

Adult↗
PubMed1986

Pathophysiology of soman intoxication in primates.

Adult baboons were monitored during intravenous infusion of Soman (1,2,2-trimethylpropyl ester, phosphonofluoridate). Three groups of animals were studied. Two groups were anesthetized with sodium pentobarbital (initial dose, 20 mg/kg), instrumented for measurement of systemic blood pressure (BP), pulmonary artery pressure, cardiac output (CO), ECG, ventilatory flow, translaryngeal pressure (PTL), transdiaphragmatic pressure (Pdi), transpulmonary pressure (PTP), diaphragm EMG, and efferent phrenic nerve traffic (Eph). One group received no Soman and served as controls. In the other group, Soman was infused over 10 min at doses of 13.1, 8.21, 4.92, or 3.3 micrograms/kg. The onset of intoxication occurred within 7-8 min (before the end of the 10-min infusion), manifested by muscular fasciculations, stridorous breathing, copious secretions, and atrioventricular arrhythmias. Mean BP decreased to 30 mm Hg by the combination of decreased CO and decreased vascular resistance. There was a dose-related response in the onset and duration of these effects. Apnea occurred in most animals and coincided with cessation of the Eph signal. Stimulation of the diaphragm via the phrenic nerve following apnea yielded Pdi values unchanged from baseline, indicating an intact neuromuscular apparatus. All animals required ventilatory support. Some surviving animals exhibited severe behavior changes. The third group of animals was studied without anesthesia. Instrumentation was performed 3 days before using a tether system for the measurement of BP, CO, and ECG, and an arterial line for blood withdrawal. Soman was infused over 10 min at a dose of 13.1 micrograms/kg. The onset of intoxication occurred within 2-3 min, manifested by hyperactivity, severe muscle fasciculations which simulated grand-mal convulsions, stridorous respiratory sounds, copious secretions, and cardiac arrhythmias. Apnea and severe lactic acid acidosis developed in all animals and all required ventilatory assistance. None recovered spontaneous ventilation at the end of 4 hr.

Animals↗
PubMed1988

Bilateral abductor paresis masquerading as asthma.

Rare upper airway lesions may be mistaken for asthma. A 16-year-old Hispanic male athlete presented to our allergy clinic with a 4-month history of wheezing and snoring with hoarseness and progressive fatigue on exertion or during sleep. His mother taped periods of harsh stridor and sleep apnea. There was no family history of vocal cord abnormalities. A year before the onset of symptoms, he suffered injury to his oral cavity with a loss of consciousness during a wrestling match. He denied dysphagia or dysphonia. He failed to respond to bronchodilators, cromolyn, or prednisone therapy during 4 weeks. On referral to our clinic, his physical examination and tape recording were characterized by harsh inspiratory stridor. His pulmonary function tests were significant for peak flow depressed out of proportion to FEV1 with reduced FVC, no response to bronchodilator, and flattened inspiratory loop unresponsive to cough or panting. Fluoroscopy and endoscopy of the upper airway was consistent with "marked bilateral limitation of vocal cord abduction." Sleep study demonstrated desaturation with CO2s in the 60s during sleep. He was started on continuous positive airway pressure, 10 cm at night, with no desaturation or sleep disturbance on follow-up.

Adolescent↗
PubMed1988

Late systemic-allergic reactions to inhalant allergen immunotherapy.

Late systemic-allergic reactions (defined as occurring between 30 minutes and 6 hours after injection) to inhalant allergen immunotherapy were prospectively studied in four allergy treatment centers; 35,674 injections were administered to 712 patients. Twenty-nine patients (4% of all patients) experienced 33 late reactions. Fifty-six injections (0.16% of all injections) were associated with the 33 late reactions. The most common clinical manifestation of a late reaction was urticaria. However, objective respiratory airway involvement, as manifested by wheezing and/or stridor, occurred in 10 (27%) of the late reactions. Delayed reactions involving wheezing and/or stridor were only related to injections from maintenance vials, and all these more severe reactions occurred no later than 60 minutes after injection. We conclude that late systemic-allergic reactions account for a significant percent of the total number of systemic-allergic reactions to inhalant allergen immunotherapy. It is our belief that a 60-minute postinjection waiting period for injections administered from maintenance vials of extract would enhance the safety of inhalant allergen immunotherapy.

Female↗
PubMed1982

Cry analysis in infants with infectious and congenital disorders of the larynx.

Thirty induced pain cries from the same number of infants with infectious or congenital disorders of the larynx (infectious laryngitis, laryngomalacia, paresis of the recurrent nerve and subglottic stricture) have been analyzed by means of sound spectrography. For each cry 21 phonetic features have been evaluated. This cry material was then compared with 120 pain cries from healthy infants. In the pathological group a significant increase could be noted in the occurrence of the following cry attributes: second pause, abnormal melody types (rising, falling-rising, flat and no melody types), instability of the fundamental frequency, bi-phonation, vibrato, half-voiced voice quality, noise concentration, and inspiratory stridor. Furthermore, a significant decrease could be noticed in the occurrence of voiceless cries, falling and rising-falling melody types, and glottal roll. These findings show that such spectrographic features as very high maximum and minimum pitch, bi-phonation, gliding, and abnormal melody type occur more often in cries of infants with central nervous system involvement. Moreover, such parameters as instability of the fundamental frequency and noise concentration as indicators of neurologic disorders need further consideration. Except features of inspiratory stridor, this study revealed no really clear parameters typical of peripheral diseases of the vocal tract.

Central Nervous System↗
PubMed1987

Pediatric bronchoscopy during a 17-year period.

From 1969 through 1985, 1032 pediatric bronchoscopies were performed on a total of 748 children under 16 years of age. Of the children 27.4% were under 6 months of age and the smallest patient weighed 600 g. Rigid bronchoscopes with a diameter ranging from 2.5 to 6 mm were used. A flexible fiberoptic bronchoscope with a diameter of 3.5 mm was used only occasionally, mostly for the diagnosis of laryngeal dynamics. All the endoscopies were performed under general anesthesia, except for some laryngoscopies performed with a flexible bronchoscope. The most common indications for bronchoscopy were suspected foreign body (16.7%), stridor (14.5%), recurrent respiratory infections (12.3%) and dyspnoe (9.6%). Laryngomalacia was observed in 13.8% of the children investigated and in patients with inspiratory stridor laryngomalacia was diagnosed in 39.8% of the cases. Subglottic stenosis was confirmed in 7.0% of the children. Tracheal compression was found in 10.8% and tracheal stenosis in 2.5% of the cases. Bronchiectasis was observed in 2.5% and tuberculosis in only 0.5% of the cases. Bronchoscopy was completely normal in only 10.6% of the patients investigated. The complications of pediatric bronchoscopy in this series were infrequent.

Adolescent↗
PubMed1992

Epipharyngeal teratoma in infancy.

Epipharyngeal teratomas are rare congenital lesions that often cause respiratory distress, stridor at birth. The management of such anomalies should include establishment of secure upper airways, radiographic exclusion of midline CNS malformations and early surgical excision to prevent malignant transformation, asphyxia or permanent facial distortion. The diagnosis of congenital stridor, dyspnea and rhinitis should be performed very carefully.

Humans↗
PubMed1996

Innominate artery compression of the trachea in infants.

Compression of the airway by the innominate artery has been a controversial cause of stridor and apnea in infants. Magnetic resonance imaging has been applied to document the possible association of segmental tracheomalacia. Forty infants with symptoms of stridor were studied, 7 of 40 infants aged 3-21 months (mean 10 months) had innominate artery compression on the trachea. Five infants were treated conservatively and two infants underwent surgery. Flattening of the trachea was longer than the mass compression point by the innominate artery. Therefore, intrinsic abnormality of the trachea is the basis of the respiratory symptom.

Airway Obstruction↗
PubMed1994

Respiratory distress secondary to scalds in children.

Respiratory distress secondary to scalds in children is rare. We report 13 children (six girls and seven boys) with a mean age of 19 months who sustained this injury, who were admitted to a major referral hospital during a 5.5-year period. Associated scalds usually to the face were always present and the mean total burn surface area (TBSA) was 14.4 per cent (range 3-30 per cent). Stridor was the most common presenting symptom with a variable time of onset. Bronchoscopy was performed in 11 children and in nine the injury was confined to the supraglottic area. Five children were treated with epinephrine nebulization and the symptoms resolved in 3-4 days, one child had a prophylactic tracheostomy. Seven children required intubation and ventilation. Three children died, two of whom sustained burns to both the upper and lower respiratory tract. Respiratory distress secondary to scalds may not be recognized or the progressive nature of the injury not appreciated. In three-quarters of our patients the injury was confined mainly to above the glottis. Children with inspiratory stridor can be managed with epinephrine nebulization but more marked respiratory distress requires intubation and ventilation. Mortality was due to direct thermal injury to the respiratory tract and secondary bronchopneumonia.

Burns↗
PubMed1994

Racemic epinephrine in the treatment of laryngotracheitis: can we identify children for outpatient therapy?

The purpose of this study was to identify a cohort of children with laryngotracheitis (croup) who may be safely discharged from the emergency department (ED) after treatment with nebulized racemic epinephrine (RE), corticosteroids, and prolonged observation. Consecutive children (younger than 13 years of age) presenting to the ED with the diagnosis of laryngotracheitis were evaluated prospectively according to a croup scoring system. Sixty-one patients (4 to 108 months of age) with persistent inspiratory stridor at rest after 20 minutes of mist therapy who received nebulized RE (0.05) mL/kg of a 2.25% solution) and intramuscular dexamethasone (0.6 mg/kg) were enrolled in the study. Patients were observed in the ED while croup scores were assessed at 15, 60, 120, and 180 minutes. Croup scores were significantly improved (analysis of variance, P < .01) throughout the observation period in 31 patients (51%) who were discharged from the ED. Only one patient returned within 48 hours for further cool mist therapy. The maximum benefit from RE therapy was seen at 60 minutes. If a child had persistent resting stridor or a croup score greater than 2 at that time, hospitalization was inevitable. The 30 patients admitted to the hospital were younger (19.1 v 27.8 months) and had higher pretreatment croup scores (5.7 v 4.1). This was the first prospective study to identify a subset of children who have received RE to be safely discharged home after observation in the ED.

Administration, Inhalation↗
PubMed1983

Croup: pathogenesis and management.

Croup syndromes are common in children, most frequently being infectious in origin. Children present with a slow progression of inspiratory and expiratory stridor and a croupy, "barking seal" cough. Children are variably febrile and with progression of disease, exhaustion, agitation, cyanosis and air hunger may develop. The evaluation of the patient must focus on the degree of respiratory distress and associated findings. Epiglottitis and foreign body aspiration must be excluded. Management is primarily dependent upon administration of humidified air. Children with moderate to severe croup benefit from racemic epinephrine and steroids. Admission is indicated in children with stridor at rest, evidence of exhaustion, toxicity or respiratory distress. Active airway intervention is rarely required but may be life saving if obstruction develops.

Child↗