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PubMed2006

Dysphagia-gastroesophageal reflux complex: complications due to dysfunction of solitary tract nucleus-mediated vago-vagal reflex.

We report on the complication of gastroesophageal reflux (GER) in four patients with lower brainstem dysfunction. These patients suffered from perinatal asphyxia, cerebellar hemorrhage, or congenital dysphagia of unknown origin and showed facial nerve palsy, inspiratory stridor due to vocal cord paralysis, central sleep apnea, and dysphagia, in various combinations. Naso-intestinal tube feeding was introduced in all of the patients due to recurrent vomiting and aspiration pneumonia resulting from GER. T2-weighted magnetic resonance (MR) imaging revealed symmetrical high intensity lesions in the tegmentum of the lower pons and the medulla oblongata in two of the patients, and pontomedullary atrophy in another patient. In normal subjects, lower esophageal sphincter contraction is provoked by distension of the gastric wall, through a vago-vagal reflex. Since this reflex arc involves the solitary tract nucleus, where the swallowing center is located, the association of dysphagia and GER in the present patients is thought to result from the lesions in the tegmentum of medulla oblongata. We propose the term "dysphagia-GER complex" to describe the disturbed motility of the upper digestive tract due to lower brainstem involvement. In children with brainstem lesions, neurological assessment of GER is warranted, in addition to the examination of other signs of brainstem dysfunction, including dysphagia and respiratory disturbance.

Child, Preschool↗
PubMed2006

[Negative-pressure pulmonary edema (NPPE)].

Negative-pressure pulmonary edema (NPPE) is a clinical entity of anaesthesiologic relevance, perioperatively caused by obstruction of the conductive airways (upper airway obstruction, UAO) due to laryngospasm in approx. 50% of the cases, its early recognition and treatment by the anaesthesist is mandatory. NPPE, also addressed as post-obstructive pulmonary edema (POPE) presents in most cases as a complex of symptoms with rapid onset, consisting of acute respiratory failure with dyspnea, tachypnea, and strained respiratory efforts. Additional signs are paradoxe ventilation, pink frothy sputum, stridor, and severe agitation. UAO produce extreme reduction of intrathoracic pressure during spontaneous ventilation, consecutively causing increase in venous return to the right ventricle and in intrathoracic blood volume, resulting in elevated hydrostatic pressures and interstitial transudation of fluids. Partially due to largely differing criteria used for diagnosis, opinions about incidence and prevalence of NPPE are unhomogenous in medical literature. It has been shown that generation of NPPE is not only limited to patients being intubated and ventilated, but occurs also in patients requiring higher fractions of oxygen.

Adolescent↗
PubMed2006

Evaluation of respiratory syncytial virus detection by rapid antigen tests in childhood.

BACKGROUND: Rapid and reliable diagnosis is crucial for clinical management of respiratory syncytial virus infection in childhood. We assessed the performance characteristics of respiratory syncytial virus antigen immunoassays in children hospitalized for respiratory infection. METHOD: A total of 1600 children up to three years of age hospitalized for diseases potentially caused by RSV were included in the study. Nasopharyngeal secretions were obtained in a standardized manner in the first 24 hours after hospital admission and tested in parallel by PCR and rapid antigen tests for RSV. The following parameters were recorded: recruitment center, gender, age, presence of fever, rhinitis, stridor, barking cough, cough, wheezing, vomiting, disturbed eating or sleep, tachypnea, tachycardia, increased body temperature, decreased oxygen saturation, C-reactive protein, erythrocyte sedimentation rate and chest X-ray results. RESULTS: Considering PCR testing as gold standard, rapid antigen testing had a specificity of 89.9% and a sensitivity of 66.2% for all samples tested. Logistic regression analysis revealed age and recruitment center as the only parameters influencing sensitivity whereas no such influence on specificity was found. Positive likelihood ratios ranged from 4,9 to 6.9 in different age groups. Negative likelihood ratio was 0.24 (95% CI: 0.18-0.42) in children aged up to 3 months but 0.67 (95% CI: 0.53-0.84) for children older than 2 years. CONCLUSION: Rapid detection of RSV antigen in this study was useful in detection of RSV mediated disease in younger infants but shows decreasing sensitivity in children older than three months.

Antigens, Viral↗
PubMed1984

Innominate artery compression of the trachea in infancy and childhood: is surgical therapy justified?

We reviewed innominate artery compression of the trachea in 10 medically treated patients (group I) and 6 children who underwent innominate arteriopexy (group II). The symptoms were as follows: (1) stridor (80% in group I, and 66% in group II), (2) recurrent bronchopulmonary infections (40% in group I, 66% in group II) and (3) apneic attacks (10% in group I and 100% in group II). Diagnosis was made in all cases by tracheobronchoscopy . It revealed severe tracheal stenosis (narrowing more than 2 thirds) in all children treated by surgery. In group I the tracheal stenosis was mild (narrowing less than one third) in 5 cases, moderate (narrowing from one third to 2 thirds) in 4 cases and severe in only one case. The indication for surgery was based on a history of apneic attacks. In this study innominate arteriopexy has proven to be a reliable and low risk procedure which relieved reflex apnea and recurrent bronchopulmonary infections in all 6 children operated upon.

Apnea↗
PubMed1998

[Right ventricular thrombus after pacemaker implantation in a patient with secondary antiphospholipid syndrome].

HISTORY AND ADMISSION FINDINGS: An 85-year-old woman was admitted because of frequent syncopes. She also reported slight weight loss, cough and dyspnoea. Chest auscultation revealed slight stridor and a cardiac arrhythmia, with an irregular ventricular rate between 120 and 140 beats/min. INVESTIGATIONS: She had a thrombocytopenia (96 platelets/nl), and the ECG and long-term monitoring showed a tachyarrhythmia with atrial fibrillation, a bifascicular block (left anterior hemiblock and right bundle branch block), as well typical signs of sick-sinus syndrome with short periods of bradycardic sinus rhythm and pauses of up to 6 s on rhythm change. Echocardiography indicated moderately reduced left ventricular function. Chest radiogram revealed tracheal narrowing by a retrosternal goitre. No evidence of tumour was found on bronchoscopy. DIAGNOSIS, TREATMENT AND COURSE: A VVI pacemaker was implanted. When the platelet count dropped to 30/nl idiopathic thrombocytopenic purpura was suspected, but administration of high doses of corticoids and immunoglobulin was without effect. Another echocardiogram, performed because of chest pain suspicious of pulmonary embolism, revealed a large bowl-shaped right ventricular thrombus with floating parts. Demonstration of anticardiolipin antibodies established the diagnosis of antiphospholipid syndrome (APLS), thought to be secondary to thyroid cancer suspected from the computed tomography. The patient died 2 months later from recurrent pulmonary embolism and progressive liver failure. Autopsy revealed a not previously diagnosed tracheal carcinoma with metastases to the thyroid, as well as haematogenous metastatic foci within the right ventricular thrombus. INTERPRETATION: In case of thrombocytopenia of uncertain aetiology APLS should be included in the differential diagnosis, even in the absence of any early or acute thrombosis. If anticardiolipin antibodies and/or lupus anticoagulant are demonstrated, malignant neoplasm should be considered in addition to autoimmune disease.

Aged↗
PubMed1998

[Secondary obstructive sleep apnea syndrome in a patient with tracheal stenosis and bilateral recurrent paresis. Successful treatment with nasal continuous positive airway pressure therapy].

HISTORY AND ADMISSION FINDINGS: A 67-year-old woman complained of marked daytime sleepiness, as well as loud snoring and apnoeas during sleep. She was known to have had 3 thyroidectomies for goitre 41, 23 and 12 years ago, with known tracheal stenosis and recurrent nerve palsy for 11 years. Physical examination revealed marked stridor, hoarse voice and slightly enlarged and palpable recurrent right thyroid. INVESTIGATIONS: Polysomnography demonstrated a clearly elevated obstructive sleep apnoea activity (apnoea index: 34/h, apnoea-hypopnea index: 40/h, desaturation index: 31/h, minimal saturation: 63%). Selective tracheal imaging showed subglottic tracheal stenosis with an inspiratory luminal diameter of 4 mm and an expiratory luminal diameter of 8 mm. Lung function analysis revealed marked flattening of the flow-volume curve as sign of a functionally effective tracheal stenosis. These findings indicated a secondary obstructive sleep apnoea (OSA) due to tracheal stenosis and bilateral recurrent nerve palsy. The patient declined further studies, such as bronchoscopy. TREATMENT AND COURSE: As the patient did not want any surgical treatment, nasal continuous positive airway pressure therapy (CPAP) was instituted as a trial. No apnoea occurred at a pressure of 12 mm H2O and this was well tolerated. She has now continued CPAP at home for 12 months and her vigilance was markedly improved. CONCLUSIONS: Tracheal stenosis or recurrent nerve palsy is a rare cause of OSA which can be effectively treated by nasal CPAP.

Aged↗
PubMed1998

[Primary pulmonary plasmacytoma. Case report of a rare lung tumor].

HISTORY AND CLINICAL FINDINGS: A 45-year old man, suffering from dry cough without haemoptysis, was admitted for evaluation of pulmonary changes of uncertain aetiology. A chest radiograph and computed tomography (CT) had shown a large space-occupying lesion in the left hilus. His general condition was poor and he had resting dyspnoea and stridor. On auscultation of the lung there were decreased breath sounds and dry rales over the left and vesicular breathing over the right lung. INVESTIGATIONS: CT revealed a 7.0 x 6.0 cm space-occupying lesion in the left hilus and ipsilaterally a tumour in the dorsolateral lower lobe surrounding the left lower lobe bronchus. Initial bronchoscopy showed this bronchus to be involved in the tumour process. Biopsy demonstrated monomorphic proliferating plasma cells which on immunochemical testing showed selective staining to lambda-light chains and immunoglobulin G. The first serum electrophoresis was unremarkable, but a later repeat discovered a small amount of paraprotein for IgG-lambda type. Bence-Jones protein was demonstrated in the urine. On the basis of these findings an extramedullary plasmacytoma of the lung (PPP) was diagnosed. TREATMENT AND COURSE: At first the patient was given i.v. chemotherapy, and to prevent a threatening bronchial occlusion oral dexamethasone was given. Pericardial effusion and recurrent retention pneumonia developed. 6 month after onset of symptoms radiotherapy was begun and the tumour became markedly smaller. CONCLUSION: PPP should be included in the differential diagnosis of pulmonary space-occupying lesions even when initial serum protein electrophoresis is negative for monoclonal paraproteinaemia. Pericardial effusion may be caused by pulmonary involvement of the plasma cell tumours.

Biopsy↗
PubMed1992

Food-dependent exercise-induced anaphylaxis: report of two cases.

Exercise-induced anaphylaxis (EIAn) is a rare condition characterized by giant urticaria, angioedema and acute gastrointestinal symptoms that develops on exertion. In the most severe forms it may be associated with acute cardiorespiratory symptoms (laringeal stridor, wheezing), profound hypotension or syncope. In some individuals, EIAn characteristically occurs after a meal suggesting that the anaphylactic reaction is provoked by both exercise and ingestion of a foodstuff to which the patient has become sensitized. Two representative cases of severe food-dependent EIAn are described, which emphasize the need of performing a careful allergological evaluation in sportsmen with unexplained cardiovascular and/or respiratory symptoms during effort, especially when associated with other allergic manifestations and/or occurring in the post-prandial period.

Adult↗
PubMedDate not supplied

[Secretory immunoglobulin A in childhood: does the saliva value reflect the bronchial value?].

Immunoglobulin A (IgA) can be found in different body secretions and plays a major role in the local immune response. It inhibits bacterial adherence, neutralizes toxins and protects the mucosa from penetrating antigens and allergens. Whereas measurement of IgA in saliva mostly does not show any problems, difficulties in assessing IgA in bronchial fluids often occur mainly due to variable dilutional effects. Aim of the present study was to find out whether saliva IgA predicts bronchial IgA. In 15 children aged 4 months to 14 years (mean 53.5 months; SEM 12.3) with chronic cough (n = 10), mediastinal mass (n = 1), recurrent airways obstructions (n = 2) and inspiratory stridor (n = 2) we performed a diagnostic rigid bronchoscopy and assessed IgA by means of bronchial lavage (BL). We attempted to control for uncertain dilution by the use of albumin as a denominator and to present our data as ratios of IgA to albumin. As various disease states alter the integrity of the alveolar-capillary membrane and influence the concentration of albumin in the epithelial lining fluid we developed an optical score to describe the state of the bronchial mucosa. Measurement of saliva IgA is easy and can be done without dilutional effects. The mean value of IgA in saliva was 65.49 mg/l (SEM 14.75; range 3.5-227), the one of IgA in bronchial lavage fluid 30.75 mg/l (SEM 7.11; range 3.5-100). IgA-albumin ratio ranged from 0.006 to 1.46 (mean 0.36, SEM 0.12). Saliva IgA did neither significantly correlate with bronchial IgA nor with bronchial IgA-albumin ratio.(ABSTRACT TRUNCATED AT 250 WORDS)

Adolescent↗
PubMed1999

Clinical spectrum and diagnostic difficulties of infantile ponto-cerebellar hypoplasia type 1.

We present the clinical and histopathological features and the diagnostic difficulties encountered in five children affected by a motor neuron disorder other than spinal muscular atrophy. Investigations performed suggested the diagnosis of ponto-cerebellar hypoplasia type 1 (PCH-1). Severe respiratory difficulty was present at birth in two of these children; hypotonia, arthrogryposis, microcephaly and nystagmus were present in all. Early and progressive bulbar involvement with swallowing difficulties and stridor was also a common feature in these infants. Severe cognitive delay was invariably present. Brain magnetic resonance imaging showed ponto-cerebellar hypoplasia in four children while striking atrophy of the cerebellar vermis and cerebellar hemispheres were present in the fifth child. Electrophysiological and pathological investigations of proximal muscles performed at presentation in all these children were not conclusive, while the post-mortem studies, or the study of distal muscles during life, showed a clear neurogenic picture. Genetic studies excluded involvement of the SMN gene, or of other genes located on chromosome 5q, confirming that ponto-cerebellar hypoplasia type 1 is a different entity from typical proximal spinal muscular atrophy.

Abnormalities, Multiple↗
PubMed1997

[Diagnosis and therapy of tracheal rupture after blunt thoracic trauma].

Tracheobronchial ruptures are rare but potentially lifethreatening events. We report on the case of a 34-year-old suicidal unrestrained car driver, who developed subcutaneous and mediastinal emphysema and right-sided haematothorax following blunt thoracic trauma. Fibreoptical inspection of the tracheobronchial system revealed a rupture (approximately 2 cm in length) of the pars membranacea of the trachea ending shortly above the carina. CT-scan confirmed the diagnosis of mediastinal emphysema, tracheal rupture and, in addition, left-sided pulmonary contusion. A repair of the tracheal tear was performed by right-sided thoracotomy using a double-lumen tube. The left-sided double-lumen tube was used postoperatively to achieve respirator ventilation with low pressure on the tracheal lumen and on the suture of the tracheal tear. On the other hand, sufficient airway pressure with PEEP for the left lung showing contusion could be provided, using the endobronchial tube. The postperative course was without complications. The patient was on respiratory support for three days due to his-pulmonary contusion. Following final endoscopic control of the trachea he was discharged from the ICU one week after the trauma. The clinical and radiological signs of tracheobronchial ruptures are discussed (respiratory distress, haemoptysis, cyanosis, localised pain, hoarseness, coughing, dysphagia, stridor, subcutaneous emphysema and pneumothorax, tension pneumothorax, mediastinal emphysema). Fibreoptic bronchoscopy is the present gold standard for confirming the diagnosis. The surgical and anaesthesiological approach to the management of tracheobronchial ruptures is described reviewing the current literature.

Adult↗
PubMed1996

[Intubation trauma of the larynx--a literature review with special reference to arytenoid cartilage dislocation].

Trauma to the cricoarytenoid joint represents a rare but serious complication of endotracheal intubation. Subluxation and luxation of the arytenoid cartilage may occur during difficult but also following uncomplicated intubation. Forces on the arytenoid cartilage exerted by the laryngoscope blade or by the distal part of the endotracheal tube may cause anterior and inferior displacement of the arytenoid cartilage. Due to the conventional intubation technique the left arytenoid cartilage is affected most frequently. Posterolateral subluxation is attributed to the pressure exerted on the posterior glottis by the convex part of the shaft of the tube. Systemic diseases (e.g. terminal renal insufficiency, bowel diseases, acromegaly) may cause degeneration of the cricoarytenoid ligaments, thus making the cricoarytenoid joint more susceptible to traumatic dislocation. Persisting alterations of voice, sore throat and pain on swallowing may hint to the diagnosis of arytenoid dislocation. However, stridor and shortness of breath have also been observed. If pharyngo-laryngeal complaints persist, evaluation by laryngologists is mandatory. In addition to indirect and direct laryngoscopy, computerised tomography and electromyography of the larynx play an important role in differentiating arytenoid dislocation from true vocal cord paralysis due to nerve damage. Early operative reposition results in fair prognosis, whereas delayed diagnosis may lead to ankylosis of the cricoarytenoid joint with permanent impairment of the voice and possibly compromised airway protection.

Anesthesia, Endotracheal↗
PubMed1999

[Rhinoscleroma: a case report].

BACKGROUND: Rhinoscleroma is a rare infectious disease of the upper respiratory tract caused by Klebsiella rhinoscleromatis. The nasal mucosa represents the primary region of occurrence which is most likely a result of respiratory transmission. Without adequate++ treatment, the disease can potentially spread to the rest of the upper and middle respiratory tract within a period of several years. Healing often occurs with extensive scarring and adhesions of the nose, palate and larynx. A life threatening late stage manifestation includes subglottic stenosis requiring immediate surgical intervention. Medical treatment primarily consists of a long-term course of antibiotics. Ciprofloxacin (Fluoroquinolon) has proved to be one of the most effective drugs. CASE REPORT: We report about a 33 year-old Egyptian male, who presented in our department with a 10 year history of a previously, only temporarily successfully treated rhinoscleroma. His main symptoms were nasal obstruction, epistaxis and inspiratory stridor. FINDINGS: We began treatment with Ciprofloxacin over a four week period, which lead to an improvement of his symptoms. The treatment was considered complete after several biopsies and smears were negative for live specimens. We then chose to reduce the scar tissue that was causing obstruction in the larynx and the nose. CONCLUSION: The rhinoscleroma is a rare disease in geographic areas with poor hygiene and can mimic several other infectious and malignant diseases. Treatment should include a long-term antimicrobial therapy and surgical intervention in cases with symptomatic obstruction.

Adult↗
PubMed1996

[Therapy of cystic lymphangioma in childhood. Report of 4 cases with manifestations in the area of the head-neck].

BACKGROUND: Lymphangiomas are localized in the head and neck area in about 75% of cases. About 75% of these cases are children less than one year old. Treating lymphangiomas with installation therapy to obliterate the cysts has been discussed, but surgery remains the treatment of choice. PATIENTS: Two lymphangiomas in newborns required resection due to the size of the lesions. The surgery involved monitoring of the facial and hypoglossal nerves. A third child had a large lesion with infiltration into the supraglottic space and the tongue requiring a tracheotomy. In a four-and-one-half-year-old child, a parapharyngeal lymphangioma caused stridor and had to be incised before it could be completely excised through an intraoral and extraoral approach. RESULTS: Lymphangiomas can be excised safely even in newborns. CONCLUSIONS: The use of neurologic monitoring is recommended for surgery of lymphangiomas in children since these lesions conceal neurovascular structures making them difficult to identify.

Child, Preschool↗
PubMed1989

Surgical Management and follow-up of vascular rings.

Thirty patients with aortic arch anomalies resulting in tracheo-oesophageal compression were treated during the period 1966 through 1987. These anomalies are important causes of upper respiratory and oesophageal obstruction in babies and small children. Although symptoms started within the first months of life in most cases, only 15 underwent surgery before six months and a delay of more than one year occurred in 5. Diagnosis was established by chest roentgenogram, barium oesophagogram and angiography. Thirteen (43%) patients had a double aortic arch, 9 (30%) cases had aberrant right subclavian artery and 6 (20%) patients had right aortic arch with ductus or ligamentum arteriosum. One (3.5%) patient had pulmonary artery sling and 1 (3.5%) case had right aortic arch and ductus arteriosum and aberrant right subclavian artery. Associated malformations were seen in 8 (27%) cases, (4 ventricular septal defects, 1 atrial septal defect, 1 coarctation of the aorta, 1 hypoplasia of left pulmonary artery, 1 left diaphragmatic eventration). Basic surgical procedure includes exposure through a left thoracotomy, complete identification of the anomaly and division of the constricting ring. 85% of the patients are asymptomatic and minimal to moderate stridor persists among the remainder. Severe tracheomalacia was responsible for the only two deaths in the series.

Aorta, Thoracic↗
PubMed1986

[Aberrant course of the left pulmonary artery--a contribution to a rare disease picture].

An anomalous left pulmonary artery obstructs the right main bronchus and/or the trachea. Depending on the severity of the obstruction, symptoms range from a more or less disturbing stridor over attacks of shortness of breath to a life-threatening or even lethal apnoea. An 18-month-old girl suffering from a mild form of this anomaly is presented. The diagnostic and therapeutic possibilities are discussed. After exact clarification of the characteristics of the pulmonary sling, exclusion of associated aberrations, and considering good experience with a former case a conservative approach is recommended if the patient is not substantially impaired.

Bronchial Diseases↗
PubMedDate not supplied

[Pulmonary sling or ring-sling complex? respiratory problems--diagnosis, therapy, prognosis].

PS is a rare congenital vascular anomaly in which the left pulmonary artery originates from the right pulmonary artery and courses between trachea and esophagus to the hilus of the left lung causing compression of the right mainstem bronchus and trachea. In about half of all cases this vessel anomaly is associated with tracheo-bronchial and/or cardiovascular malformations. We present 6 patients with PS concentrating on the accompanying malformations of the tracheobronchial tree. All patients had the typical symptoms of wheezing and stridor already in early infancy. 3 patients showed concomitant tracheobronchial anomalies. 2 patients had additional cardiovascular deformities. In 3 of the patients the positive clinical course allowed conservative therapy. In the case of 2 further patients the respiratory problems demanded surgery, one of this patients died. A further patient died after palliative surgery of cardiac anomaly.

Adolescent↗
PubMed1997

[The treatment of juvenile laryngeal papillomatosis with argon plasma coagulation].

HISTORY AND CLINICAL FINDINGS: At 3 years of age a girl known for one year to have progressive juvenile laryngeal papillomatosis with involvement of the lower respiratory tract was seen by her general practitioner because of increasing hoarseness. As about 20 sessions of CO2-laser treatment and adjuvant administration of interferon-alpha had failed to prevent increasing glottal stenosis and spread of the tracheal involvement, a tracheostomy had to be performed. Removal of the papilloma became progressively more difficult and at the age of 6 years she was admitted for further treatment. INVESTIGATIONS: She had marked inspiratory and expiratory stridor. Results of laboratory tests were unremarkable. Examination of the respiratory tract with a flexible endoscope revealed obstruction of the tracheal lumen by the papilloma. TREATMENT AND COURSE: The papilloma was removed by argon plasma coagulation (APC) under general anaesthesia during intermittent apnoeic phases. After four treatment sessions no papilloma could be seen endoscopically. There were no side effects or complications. The interval between treatments has become progressively longer. Further removal of papilloma is only rarely necessary nowadays. CONCLUSION: APC via a flexible endoscope is a promising method in the treatment of juvenile laryngeal papillomatosis involving the lower respiratory tract. It achieves precise, circumscribed tissue penetration without carbonisation or steaming-up, while bleeding can be controlled.

Child, Preschool↗