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PubMed1996

[A case of X-linked bulbospinal muscular atrophy with bilateral abductor vocal cord paralysis].

We report a 54-year-old man with X-linked bulbospinal muscular atrophy (BSMA) with bilateral abductor vocal cord paralysis. He noticed distal weakness in the lower limbs at age 20. In the following 18 years the weakness and atrophy of his leg muscles increased gradually. He has complained of stridors during respiratory tract infection and snored heavily during sleep since his age of 50. He was admitted to our hospital for the progressive stridors during meals. His two brothers were said to have similar complaints. Physical examination showed gynecomastia, hypertension and inspiratory stridor. Neurologic examination revealed distal muscular atrophy in his four extremities, especially more severe in bilateral lower limbs. Deep tendon reflexes were absent in all extremities. His tongue was slightly atrophic with fasciculation. Neurological diagnosis was made by family history, neurological findings, electromyography and a CAG repeat expansion in the androgen receptor gene. Lungs and diaphragm were normal on the chest radiograph. Cranial MRI including brain stem was also normal. Direct laryngoscopy showed a complete paralysis of both vocal cords in paramedian position. Tracheostomy was done right away; his respiratory distress showed prompt improvement after the tracheostomy. No previous report of bilateral vocal cord paralysis in BSMA has been found. Life expectancy in BSMA patients with vocal cord paralysis may be shortened because of respiratory distress or asphyxia. Of clinical importance is a careful assessment of vocal cord function in BSMA patients.

Endoscopy↗
PubMed1996

Vocal cord paralysis as an initial sign of multiple system atrophy in the central nervous system.

Multiple system atrophy (MSA) of the central nervous system has been recognized as a rare cause of stridor secondary to vocal cord paralysis. This respiratory problem usually occurs in the later stages of the disease. We report the case of a 53-year-old man who presented with bilateral vocal cord paralysis accompanied by stridor and sleep apnea, which were relieved by a tracheostomy. Two years after the onset of stridor, he developed extrapyramidal and cerebellar signs combined with severe autonomic failure. Magnetic resonance imaging of the brain revealed atrophy of the brainstem and cerebellum. This clinical picture is consistent with the diagnosis of MSA. Vocal cord paralysis preceding any other neurologic and autonomic manifestations has been infrequently described. This case should remind clinicians that MSA should be considered in the differential diagnosis of vocal cord palsy of undetermined origin.

Atrophy↗
PubMed2026

Congenital Laryngomalacia: Pathophysiology, Clinical Spectrum, and Holistic Management.

Congenital laryngomalacia is the most common cause of infant stridor and arises from interacting structural, neuromuscular, and inflammatory mechanisms that produce dynamic supraglottic collapse. Disease severity spans mild stridor to significant obstruction, aspiration, and failure to thrive, often influenced by comorbid medical conditions. Diagnosis relies on flexible laryngoscopy supported by instrumental swallowing studies and microdirect laryngoscopy and bronchoscopy when indicated. Most infants respond to conservative management, particularly targeted feeding modifications, while acid suppression offers benefit in selective cases. Supraglottoplasty provides effective, durable improvement for severe disease and significantly enhances infant outcomes and family quality of life.

Humans↗
PubMed1998

Full polysomnographic evaluation of the infant airway.

BACKGROUND: There is little information regarding full polysomnographic examination of infants for the evaluation of stridor or stertor. OBJECTIVE: To determine the usefulness of polysomnographic examination in the evaluation of airway disorders in infants. DESIGN: Case review series. SETTING: Tertiary pediatric care center and sleep disorders center. PATIENTS: Younger than 10 months. INTERVENTION: Full polysomnography and treatment with a positive-pressure assistive device or surgery if indicated. MAIN OUTCOME MEASURES: Electroencephalographic findings, amount and length of apnea, percentage of desaturation and carbon dioxide retention, sleep architecture, amount of hypopnea as well as sleep arousals, episodes of gastroesophageal reflux, and clinical follow-up. RESULTS: Of 60 full pediatric polysomnograms performed at our institution each year, only 17 were obtained in children younger than 10 months for determination of symptoms of stridor or stertor over the past 8 years. In several cases, interpretation of 4-channel studies led to the wrong conclusions and inadequate treatment. Diagnoses made using full polysomnography included 3 children with seizure disorders, 1 with disorganized brain activity, 2 with gastroesophageal reflux, and 7 with anatomical obstructions, 3 of whom were successfully treated with a positive-pressure assistive device and 4 of whom were treated surgically. CONCLUSIONS: Full polysomnography provides physiological data that complement anatomical data obtained via endoscopy and is a useful tool for evaluating the significance of airway disorders in infants.

Bronchoscopy↗
PubMed2000

Use of internal bioabsorbable PLGA "finger-type" stents in a rabbit tracheal reconstruction model.

OBJECTIVES: To design and develop a biodegradable tracheal stent that can be used internally to stabilize and support surgically reconstructed airways. DESIGN: In vitro mechanical and degradative properties of 80:20 poly(D,L-lactide-co-glycolide) (PLGA) "finger-like" stents were determined. The stents were then tested in vivo in rabbits that underwent anterior patch tracheoplasties with fascia lata grafts. Comparisons were made between a control group and an internal stent group for stridor development, overall group mortality, reconstructed airway lumen size, and histological findings. SUBJECTS: Twenty-five New Zealand white rabbits. RESULTS: The average dry modulus for the internal stents was 6800 kPa. All of the internal stents cracked by 4 weeks in buffer solution. Significant mass loss was not noted in vitro until after 5 weeks in buffer solution. By 14 weeks, the stents were nearly 100% degraded. The attrition rate for the control group was 23% compared with 17% for the experimental group. The stridor rate for the control group was also higher at 38% compared with 17% for the stented group. The stented rabbits had a significantly smaller average stenosis (23%) across the entire reconstruction site than the control group (34%) (P<.05). CONCLUSION: Biodegradable PLGA stents degrade in a predictable fashion and have a statistically significant effect in augmenting anterior patch tracheoplasties with fascia lata grafts in rabbits.

Animals↗
PubMed2002

Bilateral vocal fold paresis and multiple system atrophy.

OBJECTIVE: To review a case series of patients with systemic neurodegenerative disease presenting to a laryngologist for workup of dysphonia and found to have bilateral vocal fold paresis. DESIGN: Case series. SETTING: Tertiary care voice center. PATIENTS: Series of patients with neurodegenerative disorders examined for dysphonia. MAIN OUTCOME MEASURES: History and physical examination including fiberoptic laryngoscopy were performed on all patients. Some patients underwent polysomnography. RESULTS: Seven patients during a 2-year period were noted to have bilateral abductor vocal fold paresis. Five of 7 (71%) had the diagnosis of multiple system atrophy proposed by the laryngologist. All 7 patients described sleep-disordered breathing with stridor. CONCLUSIONS: Patients with systemic neurodegenerative disorders such as Parkinson disease should be examined for multiple system atrophy and for evidence of bilateral vocal fold paresis. Workup for stridor should include polysomnography. Treatment of glottic obstruction in these patients includes constant positive airway pressure at night or tracheotomy. The finding of bilateral vocal fold paresis can be life threatening.

Aged↗
PubMed2003

Orthotopic tracheal allografts undergo reepithelialization with recipient-derived epithelium.

BACKGROUND: While the rejection of heterotopic tracheal allografts is characterized by complete airway obliteration, the rejection of orthotopic allografts leads to airway edema and cellular infiltrate of the lamina propria, but is not associated with obliteration. We hypothesized that orthotopic tracheal allografts undergo reepithelialization with recipient-derived mucosa and that this process prevents airway obliteration. METHODS: Thirty mice were randomly assigned to 6 experimental groups. BALB/c donor tracheal segments were transplanted orthotopically or heterotopically into syngeneic BALB/c or major histocompatability mismatched allogeneic C57BL/6 recipients. Recipients of allogeneic grafts were divided into a nonimmunosuppression group and an immunosuppression group (cyclosporine, 7 mg/kg per day). Twenty-one days after transplantation, histological assessment, immunohistochemistry for CD4 and CD8 lymphocyte infiltration and major histocompatibility-specific immunohistochemistry were performed on the grafts to assess rejection and donor or recipient origin of tissue. RESULTS: Untreated heterotopic allografts underwent complete airway obliteration by day 21. This response was prevented with cyclosporine immunosuppression. Untreated orthotopic allografts, however, demonstrated edema and lymphocytic infiltrate of the lamina propria resulting in clinical stridor without airway obliteration. Immunosuppressed orthotopic allografts did not develop edema or infiltrate of the lamina propria and consequently stridor did not occur. Immunohistochemical analysis demonstrated migration of recipient-derived mucosa into the donor allograft segment in both the untreated and treated orthotopic groups. CONCLUSIONS: Airway obliteration characteristic of rejecting heterotopic tracheal allografts does not occur in the orthotopic allografts. Migration of recipient mucosa into the donor allograft appears to prevent airway obliteration in the orthotopic allografts. These findings suggest that the orthotopic tracheal transplantation model more accurately represents the biological behavior of clinical tracheal allografts than the traditional heterotopic model.

Airway Obstruction↗
PubMed1997

Anterior cricoid split. Use of hyoid as autologous grafting material.

OBJECTIVE: To examine the use of hyoid as a readily available autologous grafting material for the anterior cricoid split (ACS) procedure. DESIGN: Prospective analysis of 20 patients undergoing ACS with hyoid interposition grafting for subglottic stenosis over a 3-year period. The patients received at least 1 year of follow-up after surgery. SETTING: Tertiary care children's hospital. PATIENTS: Twenty infants (age range, 2-9 months) with endoscopically confirmed acquired and congenital subglottic stenosis. Presenting symptoms included stridor, failure to extubate, and recurrent atypical croup. All 20 children underwent ACS with hyoid interposition grafting. RESULTS: All 20 patients exhibited improvement in their symptoms of airway obstruction. All 12 patients in whom extubation had previously failed subsequently underwent successful extubation. The 8 patients with symptoms of stridor and atypical croup showed marked improvement in their symptoms. Serial bronchoscopy revealed mucosal healing and incorporation of the hyoid grafts. CONCLUSION: Hyoid provides a readily available and reliable grafting material for interposition grafting in the ACS procedure for neonates and infants.

Airway Obstruction↗
PubMed1984

Radiolucent intratracheal foreign body mistaken for croup in a 9-year-old boy.

Stridor was present for at least three months in a 9-year-old boy whose cervical trachea contained granulation tissue in association with a chronically enlodged plastic wheel, 1.8 cm in diameter. Roentgenograms revealed irregularly lobulated narrowing of the cervical trachea. Our clinical impression was croup until bronchoscopic examination led to removal of the toy. We could elicit no history of aspiration. Chronic stridor in association with a radiolucent intratracheal foreign body is unusual in the absence of a history of aspiration.

Bronchoscopy↗
PubMed1984

Caustic ingestions. Symptoms as predictors of esophageal injury.

The ingestion of a caustic substance can lead to severe damage to the esophagus. Currently, esophagoscopy is recommended for all patients with a history of caustic substance ingestion because clinical criteria have not proved to be reliable predictors of esophageal injury. The records of 79 consecutive patients younger than 20 years who were first seen with a history of corrosive ingestion were reviewed. The presence or absence of three serious signs and symptoms--vomiting, drooling, and stridor--as well as the presence and location of oropharyngeal burns were compared with the findings on subsequent esophagoscopy. Fifty percent (7/14) of the patients with two or more of these serious signs and symptoms (vomiting, drooling, and stridor) had serious esophageal injury as compared with no positive endoscopic results in the group with none or only one of these clinical findings. The presence of oropharyngeal burns did not identify patients with serious esophageal injury. These results suggest that the presence of two or more signs or symptoms in patients with a history of caustic substance ingestion may be a reliable predictor of esophageal injury.

Adolescent↗
PubMed1999

Vallecular cysts in newborns and young infants.

Vallecular cyst is a rare cause of stridor and respiratory distress in infancy and has been associated with sudden airway obstruction resulting in death. In a retrospective review of eight cases over a 20-year period all infants developed symptoms during the first week of life. All had stridor and feeding difficulties and two required endotracheal intubation. Other common findings included signs of increased work of breathing and episodes of cyanosis. Failure to thrive was present in five patients diagnosed later than the first week of life. An abnormal or a hoarse cry was present in only two patients. Diagnosis in all cases was made by endoscopy. Where a VC is clinically suspected, it is important to stress the need to visualize the base of the tongue during any diagnostic endoscopic procedure. At endoscopy, a smooth localized mass arising from and distorting the lingual surface of the epiglottis was identified. Histologically, the cysts contained respiratory epithelium with mucous glands with an external lining of squamous epithelium. Treatment by cyst marsupialization is safe and definitive, in particular when performed by CO2 laser.

Airway Obstruction↗
PubMed2000

Intravenous dexamethasone for extubation of newborn infants.

BACKGROUND: Endotracheal tubes are foreign bodies that may injure the upper airway causing laryngeal edema. This in turn may result in failure of extubation in preterm infants. Corticosteroids have been used prophylactically to reduce upper airway obstruction and facilitate extubation. OBJECTIVES: In newborn infants having their endotracheal tube removed following a period of intermittent positive pressure ventilation (IPPV), what are the effects of intravenous corticosteroids on the incidence of endotracheal reintubation, stridor, atelectasis and adverse side effects? SEARCH STRATEGY: Searches were made of the Oxford Database of Perinatal Trials, Medline (MeSH search terms "dexamethasone", "extubat*" and "exp infant, newborn"), previous reviews including cross references, abstracts of conferences and symposia proceedings, expert informants, journal handsearching mainly in the English language and expert informant searches in the Japanese language by Prof. Y. Ogawa. SELECTION CRITERIA: Trials were included which used random or quasi-random patient allocation, and which compared intravenous steroids given immediately prior to a planned extubation with placebo. DATA COLLECTION AND ANALYSIS: Data were extracted independently by the two authors and analysed in Revman for all trials. Prespecified subgroup analyses were performed to examine differences in response between infants at high risk for upper airway edema and those receiving routine prophylaxis prior to extubation. MAIN RESULTS: Administration of dexamethasone prior to extubation significantly reduced the need for reintubation of the trachea. This result applies to both the high risk group and to the total population of infants enrolled. However, the incidence of extubation failure was zero in the trial that attempted to exclude infants at high risk of airway edema. The side effects of higher blood sugar levels and glycosuria were found in the 2 trials where these were sought. REVIEWER'S CONCLUSIONS: Implications for practice Dexamethasone reduces the need for endotracheal reintubation of neonates after a period of IPPV. In view of the lack of effect in low risk infants and the documented and potential side effects, it appears reasonable to restrict its use to infants at increased risk for airway edema and obstruction, such as those who have received repeated or prolonged intubations. Implications for research Issues of dosage and applicability to the extremely low birthweight population could be addressed in future trials. Longer term outcomes such as chronic lung disease, duration of assisted ventilation and length of hospital stay should also be examined.

Anti-Inflammatory Agents↗
PubMed1988

G syndrome: an unusual family.

The G syndrome is a multiple congenital anomaly (MCA) syndrome of hypertelorism, hypospadias, stridor, and swallowing difficulties. Cleft lip and palate, cardiac defects, cranial asymmetry, and bowel obstruction are occasional manifestations. Family data suggest autosomal dominant inheritance. Males appear to have more serious manifestations, whereas most females with the G syndrome have a benign course and are ascertained through affected male relatives. In the family reported here, the proposita was first seen at age 7 weeks because of swallowing difficulties, stridor, and unusual facial appearance, reminiscent of the G syndrome. Evaluation of the family showed striking facial changes in her father and all four of his sibs. These five individuals had megalencephaly, hypertelorism, and a broad prominent nasal root and bridge, reminiscent of the facial appearance in the BBB syndrome. There was no evidence for dysphagia, respiratory abnormality, or hoarse voice in any other relative. These individuals with the G syndrome display two unusual manifestations. There are no males with hypospadias; the proposita has the most severe laryngotracheoesophageal symptoms. Although hypospadias is not an invariable manifestation of the G syndrome and although six females with severe dysphagia or respiratory abnormalities have been reported previously, the manifestations in this family underline the variability of this condition and provide further support that the G syndrome can be expressed with equal severity in both males and females. Although phenotypic overlap between the BBB and G syndromes has long been recognized, many still consider them to be distinct nosologic entities. The occurrence of both BBB and G syndrome in different members of the same family has been observed previously.(ABSTRACT TRUNCATED AT 250 WORDS)

Abnormalities, Multiple↗
PubMed2005

Use of laryngeal mask airway in flexible bronchoscopy in infants and children.

In the past two decades, flexible bronchoscopy (FB) has gained increasing popularity among pediatric pulmonologists. The objective of this study was to review our experience with pediatric flexible bronchoscopy over the past 15 years, with special focus on route of bronchoscopy. This is a retrospective study. We reviewed our pediatric FB procedures performed at the University of Michigan, Mott Children's Hospital, from 1988-2003. The study included 1,947 procedures in 1,548 patients, with a mean of 1.3 procedures/patient. The male:female ratio was 1.66:1, and age was 4.9 +/- 5.6 years (mean +/- SD). Patients <2 years of age represented 46.6% of the study population. The laryngeal mask airway (LMA) was the most common route for flexible bronchoscopy in children 2 years of age and above. Complication rates were lower with the use of the LMA (1.9%) compared to the nasal route (3.5%). Stridor was the most common indication in the age group <2 years (20.8%), while persistent pulmonary infiltrates were more common in the older age groups (32.2-37%). Laryngomalacia was the most common finding in patients with stridor (31.5%), while inflammatory changes were more common with other indications. Procedure-related complications were reported in 2.3% of procedures. Bronchoalveolar lavage (BAL) samples were obtained and analyzed in 51.2% of FB procedures (n=1,000), of which 19.4% yielded positive microbiology cultures. In conclusion, FB is a safe procedure in pediatrics. Children less than 7 years of age represent the majority of FB subjects. The LMA offered a lower rate of procedure-related complications when compared to the nasal route or endotracheal tube. It also reduced procedure time and anesthesia time.

Adolescent↗
PubMedDate not supplied

Cricoarytenoid arthritis: a cause of acute upper airway obstruction in rheumatoid arthritis.

PURPOSE: To report acute upper airway obstruction due to cricoarytenoid arthritis, a well known but uncommon complication of rheumatoid arthritis. CLINICAL FEATURES: We report the case of a 70-yr-old female scheduled for a colostomy who had been suffering from rheumatoid arthritis for 17 years. Preoperative history and physical examination revealed no cardiopulmonary compromise. Anesthesia was induced while an assistant immobilized the cervical spine and an atraumatic intubation was performed. Surgery was uneventful. Muscle paralysis was reversed, demonstrated by normalization of the train-of-four response, and the patient was extubated awake. Shortly postextubation, the patient developed inspiratory stridor, which disappeared after a second dose of neostigmine. The patient was transported to the postanesthesia care unit. Just prior to arrival the patient once again developed inspiratory stridor, became distressed, and oxygen saturation decreased. Direct laryngoscopy followed by a nasal fibreoptic examination of the larynx was performed. Cricoarytenoid arthritis secondary to rheumatoid arthritis with airway compromise was diagnosed. An uneventful awake tracheostomy was performed. The patient was discharged on day ten with a colostomy and a tracheostomy in place. One month postdischarge the patient's trachea was decannulated. On follow-up, a normal voice and mobile cords were observed. CONCLUSION: Cricoarytenoid arthritis is an infrequent complication of rheumatoid arthritis. A thorough history and physical examination are necessary to recognize signs and symptoms of cricoarytenoid arthritis. Prompt recognition of airway obstruction due to cricoarytenoid arthritis is essential for appropriate management.

Aged↗
PubMed2003

Bronchodilator premedication does not decrease respiratory adverse events in pediatric general anesthesia.

PURPOSE: Upper respiratory infections (URI) presage perioperative respiratory complications, but thresholds to cancel surgery vary widely. We hypothesized that autonomically-mediated complications seen during emergence from anesthesia would be predicted by capnometry and reduced with preoperative bronchodilator administration. METHODS: Afebrile outpatient tertiary-care children (age two months to 18 yr, n = 109) without lung disease or findings, having non-cavitary, non-airway surgery for under three hours, were randomized to bronchodilator premedication vs placebo and had preoperative capnometry. After halothane via mask, laryngeal mask airway, or endotracheal tube, and regional anesthesia as appropriate, patients recovered breathing room air while cough, wheeze, stridor, laryngospasm, and cumulative desaturations were recorded for 15 min. RESULTS: In this specific population, there was no association between adverse events and either URI within six weeks (n = 76) or URI within seven days (n = 21). Neither albuterol nor ipratropium premedication decreased adverse events. Endotracheal intubation was associated with increased emergence desaturations and placebo nebulized saline increased emergence coughing. Neither anesthesiologists nor preoperative capnometry predicted adverse events. CONCLUSIONS: Adverse events were neither predicted nor prevented. In afebrile outpatient ASA I and II children with no lung disease or findings, having non-cavitary, non-airway surgery for under three hours, there was no association between either recent URI or active URI and desaturation, wheeze, cough, stridor, or laryngospasm causing desaturation (all P > 0.05). In this highly selected population of afebrile patients, the results suggest that anesthesiologists may proceed with surgery using specific criteria in the presence of a URI.

Anesthesia, General↗
PubMed1998

[Successful staged repair of corrected transposition of great arteries and mitral atresia associated with double aortic arch].

We report a rare case of corrected transposition of great arteries and mitral atresia associated with double aoric arch. The patient showed polypnea and cyanosis soon after birth. She underwent balloon atrioseptostomy at the age of 18 days and pulmonary artery banding (PAB) at 24 days. But she showed severe persistent stridor after PAB. Esophagography and left counter-current aortography revealed double aortic arch. At the age of 49 days, division of the left lesser arch and ligation of patent ductus arteriosus were performed through left lateral thoracotomy. Stridor disappeared completely following operation. She underwent total cavopulmonary connection using a 18 mm Gore-Tex graft as intraatrial conduit concomitant with pacemaker implantation at the age of 8 years. Postoperative course was uneventful. To our knowledge, this is the first successful case report of surgical correction of corrected transposition of great arteries and mitral atresia associated with double aoric arch.

Aorta, Thoracic↗
PubMed1999

Intrathoracic retroesophageal goiter causing tracheal stenosis.

A 65-year-old woman presented with stridor revealed a mass on a chest X-ray on physical examination. A huge goiter arising in the left lobe of the thyroid had extended retroesophageally and across the midline to the right side of the posterior mediastinum far caudally down to the level of the carina. The trachea was remarkably compressed. Surgery was performed via a combined thoracic and cervical approach, and the tumor was completely removed with resultant relief from stridor. The patient is presently doing well at 1 year after the operation.

Aged↗