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Artificial nail aspiration masquerading as refractory croup.

Infants with an aspirated foreign body who present with acute onset of obstructed airflow should be readily diagnosed. When aspirated foreign body is overlooked, patients may present with recurrent inspiratory, biphasic, or expiratory stridor. We report an infant who repeatedly presented with varying degrees of inspiratory stridor. The diagnosis of aspirated foreign body was hampered by the presence of respiratory syncytial virus infection with the initial episode.

Cough↗

Hypocalcemic laryngospasm and tetany in a child with renal dysplasia.

Stridor is a common presenting sign of respiratory illness in the pediatric population-especially in the winter. Infrequently, tetany as a chief complaint may be seen with tetanus, seizures, and dystonic reactions to medications. There are few medical conditions that present with both symptoms. This is a case of a patient who presented to our emergency department in early winter with both stridor and tetany. The child was diagnosed with hypocalcemia resulting from a previously undiagnosed renal dysplasia and his symptoms resolved with the administration of IV calcium.

Humans↗

Aggravation of laryngeal rheumatoid arthritis after use of a laryngeal mask airway.

We report a case of aggravation of laryngeal rheumatoid arthritis (RA) after the use of a laryngeal mask airway (LMA). The patient was a 55-year-old woman with severe RA who underwent wrist arthrodesis under general anesthesia using a size 3 LMA. A few days after the operation, she reported hoarseness. Inflammation of the arytenoid region and vocal cord immobility were observed by fiberoptic nasolaryngoscopy. Vocal function returned to normal by postoperative day 42 in response to prednisolone therapy. In this case, stridor was misdiagnosed preoperatively, and postoperative symptoms and local findings were probably caused by aggravation of laryngeal RA resulting from substantial compression of the arytenoid region by the LMA. For patients with RA referred for surgery, a careful search for cricoarytenoid arthritis is essential, particularly in those with laryngeal stridor, because it may be aggravated after general anesthesia.

Arthritis, Rheumatoid↗

Fronto-metaphyseal dysplasia. Further delineation of the clinical syndrome.

Fronto-metaphyseal dysplasia is a rare but clinically striking condition characterized by skeletal and other abnormalities. Typical clinical and radiological features of the disease were present in four individuals in the family in this report. One of these, a male, had an obstructive uropathy at birth, making this the fourth male reported with fronto-metaphyseal dysplasia and an associated renal abnormality. In addition, he had severe congenital stridor associated with sub-glottic narrowing of the trachea. His affected sister also had severe congenital stridor associated with sub-glottic stenosis and a tracheal web. Both children had recurrent respiratory tract infection requiring frequent hospital admission. Respiratory problems have been reported in several other individuals with fronto-metaphyseal dysplasia and it would appear that renal and respiratory tract abnormalities are part of the syndrome. Although sex-linked inheritance has been considered the most likely mode of transmission of the disease, the segregation and clinical severity of the condition in this family could be equally well explained on the basis of dominant inheritance with varying expressivity.

Abnormalities, Multiple↗

Angioedema from angiotensin-converting enzyme (ACE) inhibitor treated with complement 1 (C1) inhibitor concentrate.

BACKGROUND: Up to seven in every 1000 patients experience angioedema from angiotensin-converting enzyme (ACE) inhibitors, even after many years of use. In 2003, every 20th Norwegian used an ACE inhibitor. CASE REPORT: A 61-year-old woman with chronic obstructive pulmonary disease and a past acute myocardial infarction had used 7.5 mg of ramipril daily for the past 7 years. She also used acetylsalicylic acid, simvastatin, theophylline and salmeterol. One night she woke up with edema of the tongue. On hospital arrival, 250 mg of hydrocortisone and 5 mg of dexchlorpheniramine were given intravenously (i.v.) and 0.3 mg of epinephrine was given subcutaneously (s.c.). The edema of the tongue progressed over the next 8 h and made the tongue protrude. Fiberscopy revealed glassy edema of the arytenoids. Inspiratory stridor was heard and the patient could not speak. She became increasingly uneasy and restless. Berinert complement 1 (C1) inhibitor concentrate (1500 units) was administered i.v. Over the following 20 min, stridor gradually subsided, the patient calmed and she was able to talk. DISCUSSION: ACE inhibitor-provoked angioedema shares many clinical features with hereditary angioedema (HAE), including a limited effect of steroids, antihistamines and epinephrine. HAE, caused by excess bradykinin formation as a result of C1 inhibitor deficiency, usually has its laryngeal edema effectively reversed by C1 inhibitor in less than 0.5 h. Although patients experiencing ACE inhibitor-provoked angioedema have normal C1 inhibitor values, as in our patient, excess bradykinin is probably important as ACE breaks down bradykinin. It is unknown why ACE inhibitor-provoked angioedema appears in some and sometimes after many years of use. CONCLUSION: We believe that C1 inhibitor was effective in reversing the ACE inhibitor-induced angioedema in our patient.

Airway Obstruction↗

Brief upper airway (laryngeal) dysfunction.

We describe in six men, recurrent episodes recurring over months or years, of sudden, brief complete obstruction to respiration followed by dyspnoea with loud inspiratory stridor lasting two to five minutes. Attacks occurred during wakefulness and/or sleep. In one patient an episode was witnessed endoscopically: the initial obstruction was seen to be caused by complete laryngeal closure. The false vocal cords then opened, but the vocal cords remained adducted and caused inspiratory stridor. The similarity of the attacks described by the other patients suggests that they were all caused by laryngeal closure. Furthermore, they could simulate the episodes by voluntarily adducting their vocal cords. The symptoms were usually preceded by a sensation of throat irritation and in four cases symptoms of upper respiratory infection were present. Associated features present in some of the patients included post-nasal discharge, snoring, sleep apnoea and gastro-oesophageal reflux. None was hypocalcaemic. Although stimulation of laryngeal receptors is known to produce reflex laryngeal closure, cough is the usual response during wakefulness. Treatment aimed at reducing upper airway irritation and voluntary inhibition of coughing appeared successful in reducing the incidence and severity of the episodes. Recognition of the condition is important as it may be confused with other causes of acute dyspnoea and it appears to respond to specific management.

Adult↗

Intramuscular versus oral dexamethasone for the treatment of moderate-to-severe croup: a randomized, double-blind trial.

UNLABELLED: Glucocorticoids are an effective treatment for croup, although the most beneficial route of administration remains unclear. Recent studies have concluded that both intramuscular dexamethasone and oral dexamethasone are effective treatments, but there are few data directly comparing the two for moderate-to-severe croup. OBJECTIVES: The authors' primary objective was to determine whether there is a difference in proportion of children with resolution of symptoms attributable to croup at 24 hours, when treated with oral or intramuscular dexamethasone. Secondarily, the authors sought to estimate whether there is a difference in proportion of children with resolution of symptoms attributable to croup at 10 days and to estimate the interval to complete resolution of symptoms between these two routes. METHODS: The authors performed a prospective, randomized, double-blind trial involving children aged 3-84 months with moderate-to-severe croup, presenting to a suburban teaching emergency department (ED). Patients were eligible for enrollment if they had inspiratory stridor or a barky cough and a croup score of 2 or greater after 10-15 minutes of cool mist therapy. The patients were randomized to one of two intervention groups. In both groups, the parents were not present in the treatment room during study drug administration. One group received 0.6 mg/kg of intramuscular dexamethasone and an oral placebo, while the other group received 0.6 mg/kg of oral dexamethasone and direct pressure on their thigh with the hub of a syringe. A nurse placed a Band-Aid on the site of the real or mock injection. Parents were contacted by telephone approximately 1 and 10 days after the index visit to ask about their child's symptoms using a standardized questionnaire. Data were analyzed using an intention-to-treat approach. RESULTS: Of 126 patients eligible, 96 were recruited, with complete follow-up on 95. The groups were similar in all baseline characteristics, treatments received in the ED, and disposition. At 24 hours and 10 days after the visit, there were no statistical differences between the groups for the proportion with stridor, expiratory sounds, barky cough, sleep pattern, the degree of improvement, or the proportion with complete resolution of symptoms at one day. CONCLUSIONS: No statistical differences for any parameters were observed between intramuscular and oral dexamethasone treatments for children with moderate-to-severe croup at 24 hours or at any time the week after treatment. The durations of symptoms were similar between the treatment groups.

Administration, Oral↗

Autonomic dysfunction in pathologically confirmed multiple system atrophy and idiopathic Parkinson's disease--a retrospective comparison.

INTRODUCTION: Autonomic dysfunction (AD) can be a feature of both multiple system atrophy (MSA) and idiopathic Parkinson's disease (IPD), conditions that are frequently misdiagnosed in life. Most studies on AD in MSA and IPD are based on clinical cases without pathological verification. MATERIAL AND METHODS: We retrospectively analysed AD in 135 pathologically confirmed cases of IPD and in 33 of MSA from the UK PD Society Brain Bank. RESULTS: MSA started at a younger age than IPD (54.4 +/- 10.7 yrs versus 60.6 +/- 10.8 yrs), and AD began earlier in the course of the illness All MSA patients had some degree of AD in life whereas AD was absent in 24% of IPD patients. Although each of five autonomic domains was affected in variable numbers of IPD patients, AD in MSA generally involved more autonomic domains than in IPD, and to a more severe degree, in particular with regard to inspiratory stridor. CONCLUSIONS: These results indicate that the presence of autonomic disturbance alone does not distinguish between MSA and IPD in individual cases. However, the presence of severe AD, of AD preceding parkinsonism, or of inspiratory stridor, are all individually suggestive of MSA.

Adult↗

Radioactive iodine treatment of a functional thyroid carcinoma producing hyperthyroidism in a dog.

Radioactive iodine (131I) was used in the treatment of a 12-year-old female dog with hyperthyroidism resulting from a large, unresectable (and metastatic) thyroid carcinoma associated with signs of severe inspiratory stridor and dyspnea. Hyperthyroidism was diagnosed on the basis of clinical signs (polyuria, polydipsia, polyphagia, weight loss, nervousness) and high basal serum thyroxine (T4) concentrations, as well as thyroid radioiodine kinetic studies that showed a high radioiodine uptake into the thyroid (% thyroid uptake) and markedly increased serum concentrations of protein-bound iodine-131 (PB131I) after 131I tracer injection. Thyroid imaging revealed diffuse radionuclide accumulation by the tumor, which involved both thyroid lobes. The dog was treated with three large doses of radioiodine (131I), ranging from 60 to 75 mCi, given at intervals of 5 to 7 months. The dog became euthyroid, and the size of the tumor decreased by approximately 25% after each 131I treatment, improving the severe inspiratory stridor and dyspnea, but both the hyperthyroid state and breathing difficulty recurred within a few months of each treatment. The dog was euthanatized 5 months after the last treatment because of progressive tracheal compression and pulmonary metastasis.

Animals↗

Laryngomalacia and inspiratory obstruction in later childhood.

Pulmonary function and symptoms were reviewed in 20 children in whom laryngomalacia had been diagnosed by direct laryngoscopy in infancy. Most children developed stridor in the first 2 weeks, but 3 children did not develop it until age 3 months. The mean duration of stridor was 4 years 2 months, with a range of 4 months to 12 years 7 months. Six children had marked posseting as infants. Airway dynamics were assessed by flow volume loops. All except one had normal expiratory flow volume curves. Inspiratory abnormalities were detected in 7 children; 6 were assessed as having variable extrathoracic inspiratory obstructions and 1 had a fixed obstruction consistent with subglottic stenosis. The mean ratios of maximal inspiratory flow at 50% of vital capacity divided by forced vital capacity in the laryngomalacia and control groups differed significantly, as did the mean ratios of maximal expiratory flow to maximal inspiratory flow at 50% of forced vital capacity. Laryngomalacia is not necessarily a benign disorder of limited duration; there may be persisting inspiratory obstruction in later childhood.

Age Factors↗

Retropharyngeal abscess.

Of 31 children with retropharyngeal abscess treated at this hospital between 1954 and 1990, 17 (55%) were 12 months old or less and 10 (32%) less than 6 months. Three of these 10 children were neonates, only one of whom had a predisposing congenital lesion. Fourteen children (45%) had a preceding upper respiratory illness and four (13%) had a prior history of pharyngeal trauma or ingestion of a foreign body. In children less than 1 year old the clinical presentation was usually classical with fever, neck swelling, stridor, and pharyngeal swelling. Significantly fewer children over 1 year had neck swelling and no child over 3 years old had stridor. A lateral radiograph of the neck, when performed, had a sensitivity of 88% in diagnosis. Bacteria isolated included pure growths of Staphylococcus aureus (25%), klebsiella species (13%), group A streptococcus (8%), and a mixture of Gram negative and anaerobic organisms (38%). There were two deaths. In six cases (24%) the abscess recurred necessitating further surgical drainage.

Abscess↗

Corticosteroids for croup.

Croup (laryngotracheobronchitis) is a common childhood respiratory condition, characterised by a harsh, barking cough, a hoarse voice, stridor and variable respiratory distress. Corticosteroids are often used to treat the condition. Four years ago, we concluded that nebulised budesonide improved croup symptoms of stridor and cough (but not necessarily dyspnoea) but that more trials were needed to clarify its role. In this article, we reconsider the place of corticosteroids in croup and assess whether different products offer particular advantages.

Croup↗

Neurological abnormalities in the 'cri-du-chat' syndrome.

An unusual case of the cri-du-chat syndrome is described in a 6½ year old boy, who, as well as attacks of stridor and choking, showed disorders of spatial perception and cerebellar signs in the form of nystagmus, clumsiness of the hands, and ataxia. Pyramidal signs were also present. He was only mildly retarded mentally. Psychological testing showed that he had a severe deficit for number processing, and also constructional apraxia. Surprisingly, his vocabulary was quite good, as was his reading capacity. Chromosome analysis showed a very small deletion of the short arm of the group B chromosome. In infancy this diagnosis may be suspected because of the high-pitched cry and attacks of stridor and choking. In late childhood, when the signs may be only of a neurological disorder, its recognition may be difficult without confirmation from chromosome studies. The neurological features of this disease are reviewed.

Cerebrospinal Fluid Proteins↗

Histiocytosis X involving the thyroid and hypothalamus.

A case of histiocytosis X involving the thyroid and hypothalamus is reported. A 16 year old female presented with amenorrhoea and diabetes insipidus. She subsequently developed a painful goitre with biochemical hypothyroidism, and stridor. The stridor and goitre responded to cyclophosphamide. Previous publications on the use of cytotoxics in histiocytosis X involving the thyroid are reviewed. We describe for the first time both the ultrasound appearances of the thyroid in this condition and the use of serial volumetric measurements to monitor therapy.

Adolescent↗

[Contribution of flexible bronchoscopy to the diagnosis of upper airway alterations].

INTRODUCTION: Flexible bronchoscopy (FB) is becoming an increasingly common diagnostic and therapeutic technique and is currently an essential procedure in pediatric pulmonology departments. Because the procedure is easy to perform under sedation and topical anesthesia, avoiding surgery with general anesthesia, which is required for rigid bronchoscopy, it has allowed the use of airway endoscopy in infants and children to be increased and has reduced patient risks. OBJECTIVE: To analyze the contribution of FB to the diagnosis of upper airway alterations and to characterize the frequency and type of upper airway anomalies. PATIENTS AND METHODS: We retrospectively reviewed the FB performed in our center between January 1993 and March 2003 in children aged less than 14 years old. RESULTS: A total of 456 FB were performed in 378 children. One hundred eleven FB (24.5 % of all endoscopies) were required for suspected upper airway anomalies and 55.8 % corresponded to stridor. Upper airway inspection revealed abnormality in 30.7 % (140 FB). There were no severe complications. CONCLUSION: FB is useful in the diagnosis of upper airway abnormalities. The main indication for FB is stridor and the most common abnormal finding of the upper airway is laryngomalacia.

Adolescent↗

Complications of airway management in very-low-birth-weight infants.

OBJECTIVE: To define the incidence of complications of endotracheal intubation and the factors associated with these complications. STUDY DESIGN: During a 22-month period, 227 intubated infants weighing <1,501 g were followed prospectively in a neonatal intensive care unit. Detailed records of events associated with airway management were kept after every intubation, in addition to clinical data. RESULTS: Eleven infants (4.8%) developed respiratory stridor after extubation which was treated with either systemic corticosteroids, racemic epinephrine and/or reintubation for respiratory failure. Four infants were submitted for bronchoscopy, mild subglottic stenosis with tracheal edema was found in 1 patient, granulation tissue and airway edema were noted in 3 infants. Traumatic intubation, prolonged ventilation, multiple intubations and bacterial colonization of the endotracheal tube were the factors associated with postextubation stridor. CONCLUSIONS: Subglottic stenosis is an infrequent complication of endotracheal intubation with current airway management of very-low-birth-weight infants. Less severe complications are still common, but they are usually amenable to clinical treatment. Bronchoscopy should be performed selectively only in infants with clinical evidence of airway obstruction after extubation.

Adrenal Cortex Hormones↗

Innominate artery compression syndrome. Presentation of 16 cases.

A series of 16 patients with tracheal compression by the innominate artery is presented. In more than half of the patients the symptoms began during the first few days of life with stridor varying severity, recurrent tracheobronchitis and apneic spells. In six cases requiring surgical treatment the immediate result was good. In ten mild cases treatment consisted of medical management and follow-up. The present study confirms that the great arteries can exert compression on the trachea, which can always be relieved by means of an operation which moves the arteries forward. Bronchoscopy is considered to be the only reliable means of diagnosis. This entity should be considered more often in the differential diagnosis in children less than 3 years of age with stridor, breathing difficulties, recurrent infections, apneic spells, and asthma.

Apnea↗

Functional approach to infants and young children with noisy breathing: validation of pneumotachography by blinded comparison with bronchoscopy.

Flow-volume loop evaluation yields considerable diagnostic information about adult patients with upper airway obstruction. No conclusive data support the reliability of this method in young children with noisy breathing. We used analysis of flow-volume loops at tidal breathing (TB-FV) as a first diagnostic approach to young children presenting with persistent noisy breathing (chronic stridor and/or wheezing). Flexible fiberoptic bronchoscopy was performed to establish a conclusive diagnosis and was used to verify the accuracy of the preliminary functional localization of the airway obstruction causing noisy breathing. The physician conducting pneumotachography was blinded to the bronchoscopic findings in the study, and the investigators conducting bronchoscopy were blinded to the pneumotachographic findings. Through a 6-yr period, 113 consecutive young children (ranging in age from 15 to 48 mo) with noisy breathing were enrolled in the study. Three morphologically abnormal TB-FV patterns, as compared with the normal round-shaped TB-FV loops obtained with 15 healthy children, were identified in 110 patients. A TB-FV pattern of inspiratory fluttering was found in 26 subjects and in the first 3 yr of the study was always associated with an endoscopic diagnosis of isolated laryngomalacia. Subsequently, this pattern was used to diagnose isolated laryngomalacia in 18 other infants, in whom endoscopy was avoided. Of infants with endoscopic evidence of airway obstruction ranging from the glottis to the mainstem bronchi (49 subjects), all but three showed a TB-FV loop pattern characterized by expiratory-limb flattening. A concave expiratory loop, with early expiratory peak flow and low flow at low volume, was invariably associated with peripheral bronchoconstriction, without endoscopic evidence of anatomic abnormalities (20 cases). In conclusion, TB- FV loop analysis is a noninvasive, accurate method of establishing the site of airway obstruction in young children with recurrent stridor and/or wheezing. Clinical use of this method may provide interesting pathophysiologic information and may be useful in addressing the diagnostic management of such children.

Airway Obstruction↗