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Innominate artery compression of the trachea. Treatment by reimplantation of the innominate artery.

The standard treatment of innominate artery compression of the trachea has been innominate artery suspension, but the syndrome can recur when this technique is used. From October 1985 until March 1991, 29 infants and children were treated for this syndrome by innominate artery reimplantation. In all patients severe anterior vascular compression of the trachea was demonstrated preoperatively by bronchoscopy and in 17 the diagnosis was confirmed by magnetic resonance imaging scanning. Indications for operation included apneic episodes in 17 (58.6%), severe stridor in seven (24.1%), recurrent bronchopulmonary infections in four (13.8%), and exercise-induced stridor in one (3.4%). Through a median sternotomy, the innominate artery was reimplanted more proximally on the ascending aorta and to the right of the trachea. In this new position, the innominate artery no longer crosses the trachea anteriorly, and the anatomic cause of tracheal obstruction is completely eliminated. There were no early or late deaths, and 27 of the 29 patients (93%) had complete resolution of symptoms. Partial resolution of symptoms was seen in two patients (7%). Reimplantation of the innominate artery offers a unique method by which the anatomic cause of tracheal compression is eliminated. It is a safe and effective means of treating symptomatic compression of the trachea by the innominate artery.

Adolescent↗

[Laryngomalacia in a follow-up of a child development cohort for antecedents of perinatal encephalopathy. Implications for nosologic conceptualization].

We conducted a retrospective analysis of 10 cases of congenital laryngeal stridor. Reports of laryngeal endoscopy and diagnosis define laryngomalacia as laryngeal flaccidity and stridor. Some authors postulate that in addition to immaturity of cartilage, there exist the possibility of laryngeal uncoordination and dyskinesia. They support this idea in cases of late presentation, neurological damage, and atypical cases related with functional state or anesthesia. Laryngeal endoscopies were carried out in 10 cases included in a cohort of subjects from a longitudinal follow-up diagnosed with neurologica damage of perinatal origin. One case was diagnosed with postoperative unilateral paralysis of vocal chord and another identified vascular ring. The eight remaining cases fulfilled laryngomalacia criteria of diagnosis, but because of their characteristics origin is not an anatomic alteration but a functional hypotonia. The need to carry out an integral study to describe co-morbidity is emphasized.

Brain Diseases↗

Outcome of vocal cord paralysis in infants.

Although laryngomalacia is the leading cause of stridor in infancy, vocal cord paralysis, despite its low incidence, is still the second most common cause. However, the etiology of infant vocal cord paralysis is different from that of adults, and the management protocol is controversial. Therefore, we conducted this study to better characterize the cause and outcome of vocal cord paralysis in infants. From January 1997 to December 2003, we treated thirteen infants younger than one year for vocal cord paralysis. Seven infants were idiopathic (idiopathic group), two might be caused by prior surgery (iatrogenic group), two might be caused by central neuropathy (neurological group), and two were born after difficult delivery (obstetrical group). In the idiopathic group, six infants spontaneously recovered and one infant had right-side recovery, but the left side was still paralytic. All infants in the iatrogenic and obstetrical groups spontaneously recovered. However, no infant in the neurological group recovered. Spontaneous recovery occurred in 76.9% of affected infants. More than half (70%) of these spontaneous recoveries occurred within 6 months. In our experience, direct flexible laryngoscopy is mandatory for all infants younger than one year of age presenting with stridor. Except for extreme infants (e. g. bilateral vocal cord paralysis with severe respiratory distress and central neuropathy) who require a temporary tracheotomy to relieve the airway obstruction, we recommend waiting for at least 6 months before proceeding to invasive surgical interventions.

Dyspnea↗

Post-transplant lymphoproliferative disorder with supraglottic involvement.

OBJECTIVE: Transplant patients with primary Epstein-Barr virus (EBV) infection may develop post-transplant lymphoproliferative disorder (PTLD). Since many infants are seronegative at the time of transplantation, PTLD is a major concern for paediatric transplant centres. First manifestations of PTLD are frequently observed in the ENT area with adenoidal and/or tonsillar involvement. DESIGN: Retrospective study of two cases of PTLD with confirmed supraglottic involvement, their management and outcome. Only patients with pathologically and immunologically demonstrated B-cell proliferation were diagnosed as PTLD. RESULT: Two infants developed an acute stridor during PTLD respectively 8 and 10 months after orthotopic liver transplantation (OLT). These infants were seronegative for EBV at the time of transplantation. IgM anti-EBV and/or detection of EBV genome by polymerase chain reaction were positive. Laryngeal examination revealed hypopharyngeal and/or supraglottic mucosal hyperplasia. Immunostaining of laryngeal biopsy was positive for latent membrane protein-1 (LMP1). Patients were treated by a reduction in immunosuppression as far as tolerated with the intent to recover natural immune response by the patient over the proliferation of EBV-infected cells. Complete remission of PTLD was observed in these two cases. CONCLUSION: Tonsillar hypertrophy and adenoid enlargement are the most encountered features of PTLD in OLT occurring in the ENT area. Acute stridor with supraglottic involvement may also be observed in PTLD and must be promptly diagnosed as the prognosis of this disorder is related to rapid reduction in immunosuppression and consequently to the recovering of a natural immune response against the EBV infection.

Female↗

[HTLV-I associated myelopathy with bilateral abductor vocal cord paralysis--case report].

We have reported a 50-year-old woman with HTLV-I associated myelopathy (HAM) who had bilateral abductor vocal cord paralysis. The symptoms and signs were slowly progressive spastic paraplegia, dysuria, inspiratory stridor, and snoring during sleep. She had no hoarseness. Titers of anti-HTLV-I antibody were elevated in both the serum and cerebrospinal fluid. FEV1.0% on the spirogram was reduced to 66%. The fiberscopic examination demonstrated the abductor limitation of the vocal cords during the inspiratory phase. During induced sleep after the intravenous administration of thiopental sodium, this abductor paralysis was worsened, producing a high pitched inspiratory stridor. The adduction was not disturbed at all. Needle electromyogram of the posterior crico-arytenoid muscle which is a sole abductor muscle revealed the high amplitude up to approximately 1.0 mV (normal less than 0.8 mV) with poor interference pattern, indicating neurogenic changes. After 2 months course of prednisolone (60 mg/alternative day), FEV1.0% was recovered to be 92% with the improvement of the gait disturbance, which suggests the abductor vocal cord paralysis is related to HAM. The abductor vocal cord paralysis in HAM would require a careful follow-up observation to protect the respiratory failure in the advanced stage.

Electromyography↗

Radiologic management of impacted coin in the oesophagus--a case report.

The incidence of swallowed foreign body is high in children and young adults. The common age of occurrence is below 10 years of age. It is a well known paediatric emergency often requiring urgent oesophagoscopy. Majority of swallowed foreign bodies (FBs) are impacted at sites known conventionally as constrictions. The commonest FB swallowed by children is coins; by adults - bones, fish bones and large bolus of meat, and in the older age group - dentures. The most common presenting symptoms are drooling of saliva, dysphagia and odynophagia. The anatomic proximity of the upper airway and oesophagus permit the development of respiratory symptoms like cough and stridor. Long standing foreign body impaction with weight loss, consolidated lungs and failure to thrive are documented presentations of FB in the oesophagus. We present a case of a 20 year old male who inadvertently swallowed a coin which got impacted at the thoracic inlet - one of the conventional areas of constriction. He presented late with cough, stridor, odynophagia and weight loss. The presentation of weight loss that could arise from unduely prolonged odynophagia rather than from complications like fistula, empyema thoracis or ominous predisposing lesions like malignancy was noted. The case highlighted the oddity of an adult swallowing a coin, its impaction in the, oesophagus of an apparently healthy adult and the non-surgical retrieval of the FB by fluoroscopic guidance.

Adult↗

[Malignant tracheal tumor--differential diagnosis in bronchial asthma].

Primary tumours of the trachea are rare and most of the symptoms relate to obstruction of the air passage leading to inspiratory and expiratory stridor. Unfortunately, the inclusion of malignant tracheal neoplasms in the differential diagnosis of onset of wheeze in adults is even more rare than the tumours themselves. We report a case where a tracheal adenoid cystic carcinoma presented symptoms suggestive of bronchial asthma. It is emphasized that delay by the physician in diagnosing tracheal tumours is the principal problem, and that adult patients who experience onset of stridor and dyspnoe and do not respond to accepted medical treatment deserve systematic radiographic and endoscopic evaluation of the entire air passage.

Aged↗

Obstructive subglottic granuloma after removal of a minitracheostomy tube.

We report herein a patient with subglottic granuloma after removal of a minitracheostomy tube (Minitrach II, SIMS Portex Inc., Hythe, Kent, UK). The patient underwent pulmonary resection for lung cancer followed by insertion of the minitracheostomy tube for prevention of sputum retention. The tube was removed 4 days after insertion. Twelve weeks later, the patient developed severe dyspnea and stridor. Bronchoscopy showed an obstructive subglottic granuloma arising from the anterior wall. The granuloma was removed by coring out using a conventional tracheal tube, followed by local injection of methylprednisolone acetate. The patient is now asymptomatic without regrowth of the granulation tissue 12 weeks after the treatment. With complication in mind, attention should be paid to patients suffering dyspnea or stridor after removal of a minitracheostomy tube.

Airway Obstruction↗

Vocal cord dysfunction presenting as asthma.

A 14 year old boy presented with deteriorating asthma and marked stridor. Neither asthma nor stridor responded to an increase in anti-asthma medication, including high dose oral steroids. Indirect laryngoscopy revealed adduction of the vocal cords throughout the respiratory cycle, a phenomenon previously identified as a psychosomatic conversion reaction. When gently confronted with these findings and offered psychological assistance the boy's symptoms abated totally. After two sessions of hypnotherapy he has had better control of both his physical (asthma) and psychological problems.

Adolescent↗

[High-grade tracheomalacia and tracheal stenosis in congenital esophageal atresia with lower esophagotracheal fistula (Type III b)].

Stenosis and malacia of the trachea wall can provoke chronic stridor and/or chronic bronchitis, but usually stenosis and malacia only exist separately. The finding of an infant born with atresia of the oesophagus and a lower tracheoesophageal fistula which was cured by surgery on the 1st day of life are discussed. During the following 8 months we observed persistent stridor, chronic cough and (4-times) relapsing episodes of respiratory insufficiency ("nearly-sudden-infant-death-syndrome"/NSIDS) due to gastrooesophageal reflux (GER with aspiration) and severe tracheomalacia combined with tracheostenosis and bacterial infections (Pseudomonas aeruginosa). The strategy of therapy for GER and for the tracheal abnormality are discussed.

Esophageal Atresia↗

[Bilateral abductor vocal cord paralysis (Gerhardt syndrome) in the Shy-Drager syndrome].

Ten consecutive patients with a progressive pan-autonomic failure of the Shy-Drager syndrome were investigated. Movement disorders of the vocal cords were examined with a fiber-optic laryngoscope as well as a video-recorder. Moderate to severe vocal cord paralysis was present in five of ten patients. The vocal cords were almost immobile during inspiration, while there was no limitation of the adduction during phonation. In two cases, grade of vocal cord paralysis was asymmetric. One patient developed peculiar twisting-like dystonic movements of the vocal cord. Polygraphic studies revealed that SaO2 was lowered in spite of tachypnea during sleep. In two cases, the expiratory flow volume curve in effort-dependent portion near TLC showed a plateau and the inspiratory part of the curve also showed a plateau indicating constant flow. These functional disorders suggest an upper airway obstruction probably due to the vocal cord dysfunction. There was no vocal cord paralysis in two patients who had neither snore nor stridor. Development of a severe vocal cord dysfunction usually manifested itself clinically as stridor, snore or respiratory failure requiring tracheostomy. There was little information on the pathology of the vagal nerves and nuclei supplying motor control to the laryngeal muscles. The mechanism of the selective involvement of abductor muscle (posterior muscle) of the vocal cord (Gerhardt syndrome) remains unsolved. Vocal cord paralysis should be looked for since it can result in respiratory failure leading to death.

Adult↗

[Extrapulmonary respiratory sounds in newborns: classification and iconographic contribution].

The extra-pulmonary stridors of the newborn represent a frequent and difficult diagnosis pathology for the radiologists and the neonatologist. After an introduction about the most important anatomical dates, A. reviews the reasons of the neonatologic stridor, and emphasizes the vascular congenital anomalies, most important for a early differential diagnosis. Concluding, are reported radiologic findings of this shifty pathology.

Anesthesia↗

Transendoscopic neodymium:yttrium aluminum garnet laser irradiation in horses.

A neodymium:yttrium aluminum garnet (Nd:YAG) laser was used to study effects of applying laser irradiation transendoscopically to the corniculate process of the arytenoid cartilage in horses. Dosimetry was established initially in vitro in 10 corniculate cartilages that were irradiated and examined histologically to determine penetration depths at selected power settings. Eleven horses were given xylazine IV and butorphoral tartrate IV, and their left ventricle and corniculate process were irradiated. Six horses had left laryngeal hemiplegia and were euthanatized and necropsied 14 weeks after laser application and evaluation for upper airway stridor. Endoscopy was performed in the 5 other horses; they were euthanatized and necropsied at selected intervals to characterize the healing process. Healing was by second intention and was complete at 14 weeks. Two horses developed buds of granulation tissue along the laser incision, which resolved after a second laser application. Scar tissue formation resulted in left of midline displacement of the dorsal portion of the right corniculate process. The left ventricle healed without complications and was totally ablated. All horses had inspiratory stridor when exercised 14 weeks after laser irradiation.

Animals↗

[Mucopolysaccharidosis IV-A (Maroteaux-Lamy disease, severe form): incipient compressive myelopathy, cerebrospinal fluid fistula and tracheal stenosis in an adult patient].

A 25-year-old male with the severe form of Maroteaux-Lamy disease (mucopolysaccharidosis VI-A) developed rhinoliquorrhea of undetermined origin. The head was held permanently in extension, and there was both inspiratory and expiratory stridor. Flexion of the head worsened the stridor and caused dyspnea. Radiological examination showed a massive narrowing of the trachea and a marked retropharyngeal and retrotracheal swelling. Hyperreflexia and slight impairment of the sensation of the lower limbs were found on neurological examination. Computed tomography revealed hydrocephalus and extreme narrowing of the subarachnoid space in the region of the occipito-cervical junction, caused by marked epidural soft tissue thickening and a dysplastic arch of the atlas protruding dorsally into the foramen magnum, and displacement of the cervical spinal cord. At the age of 26 years rhinoliquorrhea suddenly stopped and the patient developed acute signs of occlusive hydrocephalus. Emergency ventriculo-peritoneal shunting was performed.

Adult↗

[Special infections of the respiratory tract in children].

Inspiratory or exspiratory stridor associated with dyspnea represents a common pediatric symptomatology compatible with various diseases which usually are of infectious etiology. The causative obstruction of the infantile airways may lead to life-threatening respiratory failure. A review is conducted of the most important emergencies characterized by stridor and dyspnea in early childhood: croup syndromes (acute viral laryngotracheitis, spasmodic croup, bacterial tracheitis), epiglottitis and bronchiolitis.

Bacterial Infections↗

Familial vocal cord dysfunction.

Vocal cord paralysis is a common cause of neonatal stridor. Familial vocal cord dysfunction, however, is unusual. All three siblings in one family had neonatal stridor. Vocal cord dysfunction was confirmed after endoscopic examination in two of the children; a temporary tracheotomy was required by one child. Results of evaluation, including pulmonary function tests, suggest discrete dysfunction localized to the neuromuscular pathway responsible for vocal cord abduction. Endoscopy is of prime importance in the diagnosis of vocal cord dysfunction. In considering therapy, the physician must weigh both the potentially life-threatening nature of vocal cord paralysis, as well as the likelihood of eventual spontaneous resolution of many familial and idiopathic cases.

Airway Obstruction↗

[The radiolucent esophageal foreign body in children (author's transl)].

The symptoms of radiolucent esophageal foreign bodies in small children may be predominantly respiratory with stridor and dyspnea. In a 1 3/12 years old girl who suffered from inspiratory stridor and only later from dysphagia as well this diagnosis was delayed for 2.5 months. Air in the upper part of the esophagus was the first striking roentgenologic symptom of the foreign body which was then clearly outlined by a barium swallow examination. The foreign body was a plastic lid of a candy tube.

Age Factors↗

[False appearances of aortic arch in laryngomalacia (author's transl)].

The authors report six cases of congenital laryngeal stridor in which appearances of oesophageal compression had led to a diagnosis of abnormalities of the aortic arches. Two children underwent surgery, thoracotomy failing to reveal any abnormality. The other four were investigated by endoscopy which made it possible to eliminate the diagnosis of tracheal compression. All of these children were suffering from laryngomalacia. These cases indicate the need for complete routine studies, in particular endoscopic, in all cases of stridor, as well as for careful definition of the radiological criteria of abnormality of the aortic arches. The pathogenesis of these false appearances of oesophageal compression in laryngomalacia is discussed.

Aorta, Thoracic↗