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PubMed1990

Thoracoabdominal asynchrony in acute upper airway obstruction in small children.

The assessment of the severity and response to therapy of acute upper airway obstruction (UAO) in small children relies on subjective parameters. Using a noncalibrated respiratory inductance plethysmograph (RIP), we quantitated the rib cage (RC) to abdominal (AB) asynchrony and the lag phase in chest wall expansion by the phase angle from the RC versus AB signal curve. Phase angles were obtained in 17 children aged 1 to 50 months with acute UAO and 30 normal control subjects. The phase angle in UAO (16 to 165 degrees; mean = 83.6 degrees) was significantly higher than in control subjects (3 to 25 degrees; mean = 11.5 degrees), p less than 0.001. Following 29 episodes of inhalation treatment with 0.03 ml/kg of racemic epinephrine, the phase angle in the UAO group decreased to 7 to 160 degrees (mean = 38.3; p = 0.001) as the shape of the RC versus AB loop became narrower. In response to the treatment, the clinical severity of UAO decreased and the tidal breathing flow-volume loop improved. A high association was observed between the phase angle and the degree of stridor (p less than 0.005 Fisher's exact test), and in 90% (26 of 29) the changes in the phase angle and in the degree of stridor were in agreement. We conclude that the RC-AB asynchrony in acute UAO can be objectively quantitated by phase-angle measurement from a noncalibrated RIP and is thus suitable for use in infants and small children. The phase angle may be used to assess objectively the response of UAO to therapy.

Abdomen↗

PubMed1993

Klippel-Feil sequence and sleep-disordered breathing in two children.

We report two children with severe sleep-disordered breathing associated with Klippel-Feil sequence. In both patients, minor vertebral anomalies were associated with a major hindbrain anomaly. In one child, the Klippel-Feil sequence had been diagnosed previously, but the hindbrain anomaly was not recognized. Two years later, this child developed fatal obstructive sleep apnea. In the other child, neither the Klippel-Feil sequence nor hindbrain anomaly had been identified before the child's presentation with sleep-disordered breathing characterized by bradypnea and stridor. Because many of the complications of hindbrain anomalies may be amenable to neurosurgical treatment, we recommend that patients with Klippel-Feil sequence be followed for the development of sleep-disordered breathing. Sleep complaints need prompt evaluation with polysomnography, whereas neurologic signs require imaging with attention to the cervicomedullary junction. Unsuspected CNS disorders must be considered in children who present with stridor or serious respiratory disturbances during sleep.

Adolescent↗

PubMed2000

Laser partial epiglottidectomy as a treatment for obstructive sleep apnea and laryngomalacia.

Obstructive sleep apnea (OSA) and laryngomalacia are two different entities. Occasionally, they may have a common etiology: an elongated, flaccid, and lax epiglottis that is displaced posteriorly during inspiration causing airway obstruction. Twenty-seven adults with a diagnosis of airway obstruction or OSA of various degrees, and 12 infants with severe stridor associated with frequent apneas due to laryngomalacia, who on fiberoptic examination were found to have a posteriorly displaced epiglottis, underwent partial epiglottidectomy with a CO2 laser. Their postoperative recovery was uneventful. Polysomnographic studies performed after operation in the adult patients demonstrated statistically significant improvement in 85% of the patients. In all the cases of laryngomalacia, stridor ceased permanently after surgery, together with complete cessation of the apneic episodes. This study demonstrates that similar pathophysiological mechanisms may be involved in both laryngomalacia and in OSA. Effective and relatively safe treatment can be achieved by partial resection of the epiglottis with a microlaryngoscopic CO2 laser.

Adult↗

PubMed2001

Bifid epiglottis associated with Joubert's syndrome.

This report presents the case of a 2-year-old boy who had a bifid epiglottis associated with Joubert's syndrome. He had episodic tachypnea and apnea, inspiratory stridor, aspiration, and growth and mental retardation. Direct laryngoscopy demonstrated agenesis of the right half of the epiglottis and hypertrophied mucosa over the arytenoid cartilage. After the hypertrophied mucosa was partially vaporized with a CO2 laser, the inspiratory stridor soon improved and the aspiration was alleviated. Brain magnetic resonance imaging showed cerebellar vermian agenesis and enlargement of the fourth ventricle.

Abnormalities, Multiple↗

PubMed2003

Involuntary expiratory phonation as a dose-related consequence of L-dopa therapy in a patient with Parkinson's disease.

We report a case of involuntary phonation caused by abnormal vocal cord movements during expiration in a patient with Parkinson's disease. A 60-year-old woman had been treated for parkinsonism at the outpatient clinic of the Department of Neurology since August 1999. She began to groan involuntarily in the daytime in September 2001. She could not eat well while groaning. Stridor was not noted during sleep at night. Endoscopic examination of the larynx revealed insufficient abduction of the bilateral vocal cords, although the glottis was not so small as to cause stridor during inspiration. During expiration, however, the vocal cords adducted, resulting in the involuntary production of voice. Electromyography showed an increase in the activity of the thyroarytenoid and lateral cricoarytenoid muscles. This muscle activity was further enhanced during inspiration. The involuntary phonation disappeared when the patient's dose of L-dopa was decreased, although she had a decrease in her systemic mobility as well. When the dose of L-dopa was increased to the therapeutic level, involuntary phonation recurred, and her voluntary systemic activity improved. In the present case, it was considered that excessive dopaminergic denervation occurred in the nerve innervating the laryngeal adductors. Involuntary voice appeared to be produced by hypertonus of the laryngeal adductors because of a lowering in the threshold level for L-dopa, even though the drug was administered at the usual dose.

Antiparkinson Agents↗

PubMedDate not supplied

Management of the cleft larynx and tracheoesophageal clefts.

The anomaly of posterior laryngeal cleft and the more extensive laryngotracheoesophageal cleft is extremely rare. Twenty-one cases of cleft larynx are reviewed. A new, simpler clinically oriented classification of the clefts is proposed, and the difficulty in establishing the precise diagnosis is stressed. Stridor is the commonest presenting symptom in type 1 clefts, and aspiration on feeding, recurrent pneumonia, and abnormalities of cry suggest type 2 clefts. Type 3 clefts present with severe aspiration cyanosis and incipient cardiorespiratory failure. Aspiration reflux and persistent stridor are extremely common postoperative problems and are responsible for the delays in decannulation in spite of apparently successful operative closure of the clefts.

Esophagus↗

PubMedDate not supplied

Epiglottoplasty for the treatment of laryngomalacia.

Laryngomalacia is the most common congenital laryngeal anomaly. Patients present with different degrees of stridor and feeding problems that usually resolve by 18 months of age. A small number present with severe feeding problems, failure to thrive, stridor with cyanosis, and apnea, which may result in cardiopulmonary disease. These infants require surgical intervention, usually a hyomandibulopexy or tracheotomy. We present a new procedure, epiglottoplasty, that is performed endoscopically and involves excision of redundant mucosa over the lateral edges of the epiglottis, aryepiglottic folds, arytenoids, and corniculate cartilages. Ten patients have undergone this procedure with good results. Epiglottoplasty represents an alternative to tracheotomy in severe laryngomalacia. Indications, techniques, postoperative management, and complications are presented.

Child↗

PubMedDate not supplied

Synchronous airway lesions in infancy.

Stridor in the young infant is evaluated by careful laryngoscopy. When a lesion of the larynx is diagnosed, the necessity and risk of bronchoscopy are challenged. To assess the need for careful examination of both the upper and lower respiratory tract, a 2-year retrospective study was performed at the Children's Hospital of Pittsburgh. Of 103 infants who underwent diagnostic laryngoscopy and bronchoscopy for airway obstruction, stridor, or both, 18 (17.5%) had two or more synchronous airway lesions detected. Laryngoscopy alone, without further workup of the entire respiratory tract (ie, bronchoscopy, radiographic studies) may fail to detect concurrent disorders in infants with airway obstruction.

Airway Obstruction↗

PubMed1991

Tracheomalacia and bronchopulmonary dysplasia.

Wheezing in the infant with bronchopulmonary dysplasia results from various pathophysiologic mechanisms, including upper or central airway obstruction. We report an infant with bronchopulmonary dysplasia who presented with episodic wheezing associated with hypoxia and bradycardia without evidence of stridor. Bronchoscopy revealed the presence of tracheomalacia. Upper or central airway obstruction, if undiagnosed, may be life-threatening. We suggest that wheezing with hypoxia and bradycardia in neonates should alert the physician to the possibility of tracheomalacia, even in the absence of audible stridor. Infants presenting with these symptoms should undergo bronchoscopy to confirm the diagnosis.

Adult↗

PubMed1995

Innominate artery compression of the trachea: diagnosis and treatment by anterior suspension. A 25-year experience.

Suspension of the innominate artery to the sternum has been a widely accepted therapy for the relief of tracheal compression. Recently, reimplantation of the innominate artery has been advocated as a superior operative procedure. While generally successful, arterial transfer carries the risk of early bleeding and stroke, and the potential for late stenosis at the anastomotic site. Between 1969 and 1994, 25 infants and children at our institution received diagnoses of innominate artery compression and were treated by anterior suspension. All presented with stridor and one third had a history of suspected or proven apnea. Twenty-four children had excellent results, while 1 required resuspension after stridor returned. There were no major complications. Our series strongly supports the belief that anterior suspension of the innominate artery is a successful and reliable operation with minimal morbidity and mortality. More complex procedures are rarely indicated.

Apnea↗

PubMed1997

State-dependent laryngomalacia.

We have observed 5 infants who demonstrate normal breathing when awake, but develop stridor while asleep. Flexible laryngoscopy in the awake state reveals either a normal larynx or redundancy of the aryepiglottic folds or arytenoid soft tissue without prolapse into the laryngeal inlet. When these children are sedated, however, the classic signs of laryngomalacia appear. Wet inspiratory stridor with concomitant supraglottic prolapse can be demonstrated by flexible videolaryngoscopy in this state. As these findings vary with level of consciousness, we have dubbed this condition "state-dependent" laryngomalacia. We believe the appearance and disappearance of classic laryngomalacia with changes in level of consciousness adds credence to the neurogenic theory of laryngomalacia.

Consciousness↗

PubMed1992

Laser therapy for endobronchial tumours.

Endobronchial laser therapy has been performed at Knightswood Hospital, Glasgow since 1983. During the period 1983 to 1990, 62 patients underwent a total of 149 laser treatments. The principal indications for therapy were tracheo-carinal stridor (24%), dyspnoea due to bronchial occlusion (60%) and haemoptysis (13%). Squamous carcinoma accounted for 80% of the lesions. Over 75% of patients had already received some form of prior therapy (radiotherapy 71%, chemotherapy 8%, surgical resection 11%). Laser therapy reduced stridor in 67% of patients with tracheal and carinal tumours and produced symptomatic improvement in 72% of patients with bronchial obstruction but without evidence of lobar collapse. Haemoptysis was controlled in all but one of patients treated. Two patients (3.2%) died during laser treatment following severe haemorrhage.

Adult↗

PubMedDate not supplied

Bronchogenic cysts: a case report.

The otolaryngologist has a reference frame for congenital stridor that rarely includes diagnosis of a bronchogenic cyst. The life-threatening potential of this lesion makes consideration and recognition imperative. Representing less than 5% of the mediastinal childhood masses in the infant, respiratory distress most often initiates diagnostic studies leading to identification and extirpation. The case presentation highlights the clinical course. The diagnostic hallmark of this case was the delayed onset of stridor with subsequent progression. Thereafter, a chest film and barium swallow suggested the diagnosis. In newborns, however, such cysts may not be evident on routine chest films and, nonetheless, cause significant respiratory distress from airway compression. Surgical extirpation should be affected as soon as possible after the diagnosis is entertained in order to insure against a sudden respiratory death.

Bronchi↗

PubMed2002

Bilateral vocal cord palsy following interscalene brachial plexus nerve block.

Acute respiratory complications of an interscalene brachial plexus block include ipsilateral phrenic nerve and recurrent laryngeal nerve palsies. A 71-year-old woman who had undergone a total thyroidectomy for papillary carcinoma 35 years ago was administered a right interscalene brachial plexus block for a shoulder hemi-arthroplasty. Subsequently she developed acute respiratory distress associated with marked stridor secondary to an acute right vocal cord palsy, which was superimposed on what was assumed to be a preexisting left-sided vocal cord palsy. On extubation the patient was noted to develop stridor again necessitating reintubation and tracheostomy was performed two weeks later. The vocal cord palsies failed to resolve over the subsequent 18-month follow-up. We describe this case to highlight the significant risk of this procedure in patients with preexisting or suspected contralateral vocal cord palsy.

Aged↗

PubMed1997

Sleep-related laryngospasm.

The term "sleep-related laryngospasm" refers to episodic, abrupt interruption of sleep accompanied by feelings of acute suffocation followed by stridor. The condition is included in the diagnostic and coding manual of the American Sleep Disorders Association (ASDA), but there are few references in the peer-reviewed literature. Our description of the distinct clinical picture associated with this condition is based on an analysis of the histories of a series of 10 patients. The patients and their families gave precise, uniform accounts of the dramatic attacks. Diagnostic work-up included pulmonary and gastroenterological assessment. All patients reported sudden awakening from sleep due to feelings of acute suffocation, accompanied by intense fear. Apnoea lasting 5-45 s was followed by stridor. Breathing returned to normal within minutes. Patients were left exhausted by the attacks. Nine of our 10 patients had evidence of gastro-oesophageal reflux and six responded to antireflux therapy. We conclude that the nocturnal choking attacks (and the occasional daytime attacks experienced by some of the patients) are caused by laryngospasm. The pathogenesis of the apparent underlying laryngeal irritability is unknown. The condition may be related to a gastro-oesophageal reflux.

Adolescent↗

PubMed2004

Pseudosarcomatous fibrovascular proliferative tissue masquerading as a primary subglottic angiosarcoma.

A unique case of pseudosarcomatous fibrovascular proliferative tissue causing biphasic stridor and simulating a primary subglottic angiosarcoma is reported. The patient presented with a wheeze and was diagnosed initially with asthma. He subsequently developed worsening biphasic stridor. Flexible nasendoscopy revealed a subglottic mass obstructing the airway. The mass was removed with a carbon dioxide laser and the patient's condition improved dramatically. The specimen was difficult to interpret histologically and was thought at first to be a novel case of a primary subglottic angiosarcoma. However, the slides were reviewed by expert soft tissue pathologists in the UK and USA, and a final diagnosis of a pseudosarcomatous fibrovascular proliferative lesion was made. This case highlights the important principle of seeking additional opinions before making a malignant diagnosis in an atypical site. Our report also emphasizes the importance of good clinico-pathological liaison, especially in difficult cases.

Aged↗

PubMed2005

Aryepiglottoplasty for laryngomalacia: the Alder Hey experience.

Laryngomalacia is the most common cause of stridor in infants. Severely affected children are at risk of feeding difficulties, apnoeic episodes and cor pulmonale secondary to upper airway obstruction. The aim of this study was to assess the outcome of aryepiglottoplasty. This is a simple surgical procedure that relieves the obstruction by dividing the aryepiglottic folds. Thirty children had an aryepiglottoplasty at the Royal Liverpool Children's Hospital between January 1995 and June 2001. The case notes of all 30 children were reviewed for age, sex, age at operation, indications, operative technique, complications and long-term outcomes. Complete resolution of stridor was obtained in 83 per cent of patients, with an improvement in a further 7 per cent. Post-operative complications included lower respiratory tract infections (13 per cent) and vomiting (7 per cent). In conclusion, simple endoscopic aryepiglottoplasty remains an effective way of treating upper airway obstruction in children. Its high resolution and low complication rate make it a safe, first choice procedure for treatment of moderate to severe laryngomalacia.

Arytenoid Cartilage↗

PubMed1989

Sleep-related laryngeal obstruction presenting as snoring or sleep apnea.

The difference between snoring (with or without sleep apnea) and laryngeal stridor resulting from laryngeal dysfunction may not be readily apparent. Two cases of Shy-Drager syndrome and one undiagnosed case in which laryngeal dysfunction was exacerbated by sleep are reported. Such dysfunction might create life-threatening situations for which emergency tracheostomy should be considered. The importance of differentiating stridor from snoring is discussed.

Aged↗