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We present a case-study of a boy with Hunter's syndrome (mucopolysaccharidosis type II) and stridor in which a laryngeal mask airway (LMA) failed to secure airway control. A rigid bronchoscopy was performed and a polypoid formation discovered. We believe that the use of the LMA could explain the laryngeal obstruction in this child.
A congenital laryngotracheo-oesophageal cleft is a rare airway malformation which results from incomplete separation of the larynx and trachea from the hypopharynx and oesophagus. Patients usually present with stridor, aspiration and cyanosis associated with feeding. For early diagnosis, a high index of suspicion is needed. Unless an appropriate diagnostic approach is taken, the diagnosis can be missed. The successful ventilation of a neonate with the Laryngeal Mask Airway is described.
A 3-week-old full-term female neonate was admitted with a 4-day history of episodic stridor, desaturations and difficult feeding. Initial assessment using fluoroscopy suggested distal tracheomalacia. Inhalational induction for examination under anaesthesia of the upper airway at 4 weeks of age caused almost complete airway obstruction due to severe anterior, or epiglottic, laryngomalacia. This airway obstruction was unresponsive to continuous positive airway pressure, the use of an oropharyngeal airway and hand ventilation and required urgent tracheal intubation using suxamethonium. Epiglottopexy, a relatively unknown procedure, was performed uneventfully 2 days later, with complete relief of the respiratory compromise. However, the infant remained desaturated postoperatively. A ventilation perfusion scan subsequently revealed multiple pulmonary arteriovenous malformations, unsuitable for embolization and requiring nocturnal home oxygen therapy. Review at 3 months of age found a thriving infant with no airway obstruction and good epiglottic positioning on examination under anaesthesia. Although the patient's oxygen requirements had diminished, the long-term outcome remains uncertain.
A 5-year-old boy with cerebral palsy and severe learning difficulties developed massive tongue swelling of sudden onset following the use of synthetic saliva. Acute airway obstruction and severe stridor ensued which required tracheal intubation and transfer to paediatric intensive care. The child was treated with intravenous steroids, antihistamines and epinephrine. With cessation of synthetic saliva, the swelling gradually resolved and the child was extubated on day 5.
Compression of the paediatric airway is a relatively common and often unrecognized complication of congenital cardiac and aortic arch anomalies. Airway obstruction may be the result of an anomalous relationship between the tracheobronchial tree and vascular structures (producing a vascular ring) or the result of extrinsic compression caused by dilated pulmonary arteries, left atrial enlargement, massive cardiomegaly, or intraluminal bronchial obstruction. A high index of suspicion of mechanical airway compression should be maintained in infants and children with recurrent respiratory difficulties, stridor, wheezing, dysphagia, or apnoea unexplained by other causes. Prompt diagnosis is required to avoid death and minimize airway damage. In addition to plain chest radiography and echocardiography, diagnostic investigations may consist of barium oesophagography, magnetic resonance imaging (MRI), computed tomography, cardiac catheterization and bronchoscopy. The most important recent advance is MRI, which can produce high quality three-dimensional reconstruction of all anatomic elements allowing for precise anatomic delineation and improved surgical planning. Anaesthetic technique will depend on the type of vascular ring and the presence of any congenital heart disease or intrinsic lesions of the tracheobronchial tree. Vascular rings may be repaired through a conventional posterolateral thoracotomy, or utilizing video-assisted thoracoscopic surgery (VATS) or robotic endoscopic surgery. Persistent airway obstruction following surgical repair may be due to residual compression, secondary airway wall instability (malacia), or intrinsic lesions of the airway. Simultaneous repair of cardiac defects and vascular tracheobronchial compression carries a higher risk of morbidity and mortality.
Anomalies of venous return due to absence of the ductus venosus have been described in the literature. This is a report of the prenatal diagnosis of two cases of isolated ductus venosus agenesis occurring at 20 and 37 weeks' gestation, confirmed postnatally by color-coded Doppler sonography. In both cases the hepatic veins assumed the function of the ductus venosus without compromising fetal hemodynamics or causing hydrops. In the first case, a healthy female infant was delivered at term. In the second case, a 46,XY,dup(8) (q21.1q22)[35]/47,idem, + r(8)[15] karyotype was diagnosed by amniocentesis. The male newborn showed facial anomalies, a bell-shaped thorax and increased intermamillary distance. There was muscular hypotonia, delayed psychomotor development and an inspiratory stridor leading to obstructive nocturnal dyspnea. On the basis of our observations in these two cases and previous reports from animal studies, we conclude that absence of the ductus venosus may be compatible with normal fetal development without relevant disturbance of circulation and oxygenation.
A woman with obstructive sleep apnea who was admitted to the sleep disorders laboratory at St George Hospital in Sydney, Australia, for assessment subsequently experienced respiratory arrest. She was successfully resuscitated and 10 days later was transferred from the critical care unit to the respiratory ward with a cuffed, fenestrated tracheostomy tube in situ. A hoarse voice, oxygen desaturation, and upper airway stridor when capping the tracheostomy alerted nursing staff to the existence of an upper airway obstruction. Repositioning of the tracheostomy tube alleviated some of the patient's difficulties, but further investigation revealed large vocal polyps as the underlying factor in her episode of respiratory failure. Removal of the polyps and inception of noninvasive positive pressure ventilation resulted in a range of benefits to the patient's health status, including resolution of her respiratory failure, better management of her diabetes, and a markedly improved quality of life.
Bilateral vocal cord immobility can be life threatening for some patients. Others, who have an open glottic chink, may have a breathy dysphonia, intermittent dyspnea, and stridor. These signs and symptoms may also be found in a number of other conditions that cause weakness or paradoxical motion of the vocal cords that mimics paralysis. These other conditions include central nervous system diseases, neuromuscular disorders, laryngospasm, and psychogenic disorders. In addition, patients with cricoarytenoid joint immobility or interarytenoid scar can also have similar symptoms at presentation. It is critical to consider the differential diagnosis of an assumed bilateral vocal cord paralysis and understand the management of paradoxical movement, weakness, joint fixation, interarytenoid scar, laryngospasm, and psychogenic disorders. The treatment for bilateral immobility should proceed only after a thorough evaluation, which might include electromyography and/or examination during general anesthesia under dense anesthetic paralysis. Reconstructive procedures are the treatments of choice, and destructive procedures should be chosen only as a last resort.
OBJECTIVE: To develop a technique to identify and localize the recurrent laryngeal nerve (RLN) during video-assisted thoracoscopic surgery (VATS) for patent ductus arteriosus. DESIGN: Prospective clinical study. SETTING: Children's hospital. PARTICIPANTS: Sixty infants and children scheduled for elective closure of patent ductus arteriosus. INTERVENTIONS: With parental informed consent, 60 infants and children undergoing elective VATS for patent ductus arteriosus were studied. A thin, pencil-point, Teflon-coated, stimulating probe allowed direct stimulation (<2 mA, 100-msec pulse width) of the left RLN inside the thorax. A commercially available 4-channel neurologic monitor recorded compound evoked electromyograms (EMGs) from the left RLN and right RLN (as control) by needle electrodes placed percutaneously in the neck. Hoarseness, stridor, feeding difficulties, and voice changes were assessed postoperatively. MEASUREMENTS AND MAIN RESULTS: Left RLN EMGs were easily obtained in 59 of the 60 patients. The surgeon correctly identified the RLN visually once in the first 7 patients; this ability subsequently improved. EMG localization of the location or course of the RLN altered dissection, clip size, or clip position in 37 of 59 patients. CONCLUSION: Intraoperative EMG to identify location and route of the RLN was easy to perform, was effective in identifying RLN position, and appeared to facilitate dissection and clipping of the ductus.
The clinical findings and management of five cats with abnormalities consistent with acromegaly were examined retrospectively. Growth hormone (GH) concentrations were elevated in four cats. In one, a minimal elevation of GH was accompanied by a marked elevation in insulin-like growth factor-1 (IGF-1). Insulin-like growth factor-1 concentrations supported the diagnosis in three of four cats measured, but was not elevated initially in one cat, despite a markedly elevated GH concentration. These findings suggest that elevated IGF-1 concentrations are a reliable indicator of acromegaly, but that values within the reference range do not exclude such a diagnosis. Clinical signs of acromegaly were similar to those previously reported, although upper respiratory stridor occurred in one cat, and insulin-resistant diabetes mellitus was not a consistent feature. Despite the lack of a widely available definitive treatment for acromegaly, good control of the clinical signs of diabetes mellitus can be achieved for long periods despite high doses of insulin often being required.
A 19-year-old female with aplastic anemia who developed subglottal aspergillosis is reported. She presented with fever, cough and stridor. Inspiratory dyspnea progressed rapidly and emergent tracheostomy was performed, which confirmed the diagnosis. In spite of intensive anti-fungal treatment combined with adoptive immunotherapy, Aspergillus infection expanded and she died of pulmonary aspergillosis. Autopsy revealed the fungal mass obstructing the trachea and disseminated pulmonary aspergillosis. Difficulties in diagnosis and management of subglottal Aspergillus infection are discussed.
OBJECTIVES: To describe the clinical manifestations and the complications of cervical spine (C-spine) involvement in diffuse idiopathic skeletal hyperostosis (DISH). METHODS: Two patients, who presented with dysphagia resulting from large anterior osteophytes of the C-spine, were diagnosed as having DISH. A Medline search from 1964 to present, using the terms "diffuse idiopathic skeletal hyperostosis" and "cervical spine," identified several clinical manifestations associated with DISH. RESULTS: Two groups of conditions associated with DISH were found. 1. Spontaneous complications such as: dysphagia, being the commonest, dyspnea, stridor, myelopathy associated with ossification of the posterior longitudinal ligament (OPLL) or with atlanto-axial pseudoarthrosis or subluxation. Other rare events were aspiration pneumonia, sleep apnea and thoracic outlet syndrome. 2. Provoked complications such as endoscopic and intubation difficulties and fractures of the C-spine with frequent transverse shift of the fractured segment and resultant myelopathy. CONCLUSIONS: C-spine involvement in DISH is a recognized cause of various clinical manifestations involving the pharynx, larynx and the esophagus. Prior knowledge of the existence of cervical DISH should alert the clinicians for possible complications, at times severe, during invasive procedures in the neck region and as a consequence of trauma.
The lower cranial nerves (CN. IX-XII) are responsible for the nerve supply of the pharyngolaryngeal region. Loss of innervation is manifested by dysphagia, aspiration, altered speech production, dyspnea and/or stridor, and visible loss of movement. In general electrophysiological testing should be applied in every kind of paresis. Beside standard equipment for electromyography limited special equipment is necessary. Examination under local anaesthesia is possible and can serve as a basis for therapeutic consequences. In this article general aspects as seen from an oto-rhino-laryngological standpoint are presented.
More attention has been given to vocal cord dysfunction (VCD) in the past years. Even though the disease is known since 1983 and was brought to mind at least casuistically through the years, often VCD was not diagnosed as such, being mistaken for bronchial asthma - at times with grave consequences for the patient. VCD causes the acute onset of stridulous respiration with acute dyspnea making the differential diagnosis of asthma quite suggestive. The inspirational stridor and a peracute progression of the disease should cause doubts as to the diagnosis of asthma. We collected case reports and describe the symptoms and diagnostic approaches to VCD as well as showing discriminating findings towards bronchial asthma. We report on five children suffering from VCD (4 girls, 1 boy) aged from 2 to 13 years. It could be shown that VCD is caused by a multifactoral pathomechanism, which has both somatic and psychological aspects. A surprising find was that all of the children were diagnosed with a pathological gastroesophageal reflux, partly in serious dimensions, without showing the typical symptoms such as retrosternal pain, stomach-ache or recurrent vomiting (so called silent reflux). Since there is a multitude of pathomechanisms involved and the disease often coincides with allergic asthma, an individual therapy plan is needed for each patient.
Unilateral recurrent nerve paralysis leads to glottic insufficiency, causing a significant lack of vocal ability. In contrast, bilateral palsies present with stridor on inspiration due to glottic stenosis. Most of the underlying lesions are iatrogenic, with thyroid surgery being the single most important causative factor. However, a variety of different reasons can lead to such a condition. Whenever aetiology is uncertain a complete diagnostic work-up is mandatory. Indirect laryngoscopy confirms the diagnosis. Laryngeal electromyography is of great value because it differentiates between paralysis and ankylosis of the cricoarytaenoid joint. Moreover, in many cases laryngeal electromyography provides a reliable prognosis of clinical outcome. While unfavorable results can be predicted with high accuracy, correct prognosis of complete recovery is more difficult. Speech therapy is the treatment of choice in case of unilateral recurrent nerve palsy. Only if a significant glottal gap persists medialization procedures may become useful for voice improvement. Endoscopic as well as open approaches are available for this purpose. Bilateral recurrent nerve palsies need to be addressed surgically in the vast majority of cases. Today, a variety of endoscopic techniques for widening the glottic airway are available. Compared to permanent tracheostomy these procedures have much less impact on the patient's quality of life and should be preferred whenever possible. Inevitably, voice quality is traded for airway normalisation. However, modern surgical techniques accomplish very tolerable phonatory results.
Relapsing polychondritis (RP) is a rare disease causing inflammation and destruction of cartilage and other connective tissues. Specific laboratory aberrations are lacking. Predominant clinical manifestations include auricular chondritis, polyarthritis, nasal chondritis, ocular inflammation, audiovestibular damage, and respiratory tract chondritis. A relapsing course is characteristic. Airways are involved in 50% of patients and may cause dyspnea, stridor, wheezing, hoarseness, aphonia, and laryneal or tracheal tenderness. Airflow obstruction may result from RP involving the tracheobronchial tree; there is no interstitial or pulmonary vascular component. Collapse or failure of the trachea to dilate during inspiration is a key feature. Fast computed tomographic (CT) scanners can visualize dynamic airway collapse. Randomized, controlled trials of therapy have not been done. Corticosteroids and nonsteroidal anti-inflammatory drugs (NSAIDs) are used most commonly, but optimal regimens and duration of therapy have not been elucidated. Endobronchial stents or tracheostomy may be required for severe stenoses refractory to medical therapy.
HISTORY: Three school children (2 girls and 1 boy, aged 9-15 years) complained about exercise-induced shortness of breath and stridor. The children were treated with inhalation steroids, one of them with systemic steroids, without a significant effect. INVESTIGATIONS: Because of lack of treatment response a chest X-ray was done. In all cases a mediastinal mass was visible. No radiological signs of an obstructive pulmonary disease was demonstrated. In none of the three cases did a pulmonary function tests demonstrate an obstructive pulmonary disease. DIAGNOSIS, TREATMENT, AND COURSE: Bone marrow aspiration in one girl showed an acute lymphoblastic leukemia of T-cell immunophenotype with a large mediastinal lymphoma. Lymph node biopsy in the boy demonstrated a nodular sclerosing subtype of Hodgkin's disease. In the other girl a ganglioneuroma was found. CONCLUSION: In children and adolescents a chest radiograph and pulmonary function tests should be performed if history and clinical signs are suggestive of bronchial asthma, before a specific inhalative or systemic treatment is started.
BACKGROUND AND STUDY AIMS: The need to administer procedural sedation to children has increased in recent years, as has experience in this field among nonanesthesiologists. Using propofol makes it easier to achieve sufficiently deep sedation. There is a considerable literature on the administration of propofol by nonanesthesiologists for gastroscopy in adults, but very few data are available on this issue in children. The aim of the present study was to assess the safety and efficacy of procedural sedation with propofol for gastroscopy in a pediatric ward with trained personnel and monitoring facilities. PATIENTS AND METHODS: A training protocol was developed to educate nurses and residents. Children requiring gastroscopy were included in the study prospectively and underwent procedural sedation with propofol administered by nonanesthesiologists. RESULTS: A total of 811 upper gastrointestinal endoscopies were carried out with procedural sedation. Sedation was achieved in all procedures, and all but three (0.4%) were conducted successfully. None of the patients required intubation. Stridor with signs of upper airway obstruction occurred in 14 of the 811 procedures (1.7%). Laryngoscopy was required to manage difficulties in introducing the gastroscope in 16 of the 811 procedures (2.0%). Major desaturation requiring a short course of ventilation occurred in six procedures (0.7%), and transient desaturation that resolved spontaneously occurred in 97 of the procedures (12%). CONCLUSIONS: Administration of propofol by nonanesthesiologists for gastroscopy examinations in children was successful in this study, but was associated with a small risk of potentially severe complications. Although the residents were generally able to administer procedural sedation alone, constant and immediate availability of anesthesiological support continues to be mandatory.