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1/30. Acute respiratory failure associated with intrathoracic masses in neonates.

    BACKGROUND: Intrathoracic masses are uncommon in children. Occasionally, they present with acute respiratory failure in the neonatal period. Although emergency resection usually is the treatment of choice, other modalities are sometimes necessary to stabilize the patient. methods: Seven neonates with intrathoracic masses were treated. Five had congenital cystic adenomatoid malformations (CCAM), 1 had a mediastinal teratoma, and 1 had a pneumatocele. These cases were reviewed retrospectively. RESULTS: Four of the 7 infants had respiratory failure in the neonatal period. A patient with a large mediastinal teratoma and 1 with a CCAM that increased rapidly after presentation underwent emergency operation, relieving respiratory distress. The other 2 large CCAMs presented with severe respiratory distress immediately after birth because of pulmonary hypoplasia. One neonate with a Stocker-I CCAM died after emergency resection. One more recent patient with a Stocker-III CCAM survived after successful treatment with delayed resection, performed 3 days after birth. nitric oxide (NO), and extracorporeal membrane oxygenation (ECMO) were instituted as supportive care because of profound persistent fetal circulation (PFC). CONCLUSIONS: Acute respiratory failure associated with intrathoracic masses in neonates may be managed in 1 of 2 ways. A small mass that increases rapidly should be resected soon after presentation. In neonates with large masses with associated PFC, surgery can be delayed until the patient is stable. ECMO, NO, and high-frequency oscillation (HFO) can be used aggressively for stabilizing such neonates.
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2/30. Hereditary surfactant protein B deficiency resulting from a novel mutation.

    Hereditary surfactant protein B (SP-B) deficiency is an autosomal recessive disease in which affected infants are unable to produce normally functional surfactant, resulting in neonatal respiratory failure and death within the first year of life. The most common cause of SP-B deficiency is a frameshift mutation in exon 4 (121ins2) of the SP-B gene. We report a newborn infant who had onset of respiratory distress during the first days, was unresponsive to exogenous surfactant, corticosteroids, prostacyclin, high frequency oscillatory ventilation and inhaled nitric oxide, and died after 27 days. Immunostaining of lung tissue obtained at biopsy demonstrated absent staining for SP-B, and robust extracellular staining for proSP-C, findings characteristic for SP-B deficiency. dna analysis revealed the 121ins2 mutation on one of her SP-B alleles and a novel mutation, 122delC, on her other SP-B allele. The proximity of the novel mutation in exon 4 allele found in this infant to the 121ins2 supports the notion that this region may represent a "hot spot" for SP-B gene mutations and confirms the heterogeneity of mechanisms which lead to SP-B deficiency. Hereditary SP-B deficiency is a rare, newly diagnosable and probably under-recognized disease, which should be suspected in term newborn infants with unexplained respiratory failure.
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3/30. Congenital misalignment of pulmonary vessels and alveolar capillary dysplasia: how to manage a neonatal irreversible lung disease?

    Congenital misalignment of pulmonary vessels (MPV) with alveolar capillary dysplasia is a rare condition consisting of anomalous veins in bronchovascular bundles, a decreased number of alveolar capillaries, and increased muscularization of pulmonary arterioles. In the literature, infants reported as having such a malformation developed respiratory distress with persistent pulmonary hypertension and ultimately died. We report the case of an infant with MPV and alveolar capillary dysplasia who was unresponsive to maximal cardiorespiratory support, including high-frequency oscillatory ventilation and inhaled nitric oxide; the infant died of pulmonary hemorrhage after 19 days, during venoarterial extracorporeal membrane oxygenation bypass. We conclude that the diagnosis of MPV and alveolar capillary dysplasia should be considered during autopsy of infants who have died of irreversible persistent pulmonary hypertension. If a lung biopsy in infants with prolonged refractory hypoxemia confirms such diagnosis before death, expensive and invasive treatments such as extracorporeal membrane oxygenation could be avoided.
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4/30. One-sided high-frequency oscillatory ventilation in the management of an acquired neonatal lobar emphysema: a case report and review.

    We describe a premature infant (gestational age 28 weeks and birth weight 1280 g) with a left-sided acquired lobar emphysema (ALPE). Left lateral decubitus positioning, right-sided conventional ventilation (CV), tracheal high-frequency oscillatory ventilation (HFOV), and dexamethasone administration were subsequently used in the treatment without success. The emphysema was resolved and the patient was extubated after selective intubation and HFOV of the right unaffected lung. We also review the reported cases of ALPE in neonates that were treated by one-sided high-frequency ventilation (HFV).
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5/30. A case of pneumomediastinum in paediatric ARDS: to oscillate or not?

    An 18-month-old was transferred (intubated and ventilated) to our hospital with staphylococcal tracheitis, which progressed to a necrotizing pneumonitis, complicated by surgical emphysema and pneumomediastinum. Maximum conventional ventilation on a Servo 300 failed. Treatment with high frequency oscillatory ventilation (for 10 days) with a permissive hypercarbia and hypoxaemia strategy to limit mean airway pressure facilitated recovery in our patient.
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keywords = high frequency, frequency
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6/30. Selective fiberoptic left main-stem intubation for severe unilateral barotrauma in a 24-week premature infant.

    A 24-week premature infant developed severe right-sided pulmonary barotrauma secondary to mechanical ventilation for respiratory distress syndrome (RDS). High-frequency oscillatory ventilation and permissive hypercapnia were initiated. A chest tube was placed to relieve a pneumothorax, and a catheter was inserted into an air-filled cyst for drainage. These maneuvers failed to improve the child's respiratory status. The child's left main-stem bronchus was then successfully fiberoptically intubated for single-lung ventilation in order to reduce the unilateral barotrauma. Single-lung ventilation was effectively and safely continued for 5 days, with complete resolution of the pulmonary barotrauma.
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7/30. Neonatal pneumatocele as a complication of nasal continuous positive airway pressure.

    A preterm infant with mild respiratory insufficiency resulting from respiratory distress syndrome developed a pneumatocele after the start of nasal continuous positive airway pressure. pneumonia was excluded by sputum and blood cultures. Treatment with high frequency oscillation ventilation resulted in complete recovery.
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keywords = high frequency, frequency
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8/30. survival of a 300-g infant ventilated by high-frequency oscillatory ventilation for respiratory distress syndrome.

    We report a case of an infant delivered before the completion of 26 wks' gestation, weighing 300 g at birth, and treated with high-frequency oscillatory ventilation for respiratory distress syndrome. Her neurologic examination at 18 months of age is compatible with mild delay in gross and fine motor skills.
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9/30. Misalignment of lung vessels: diagnostic role of conventional histology and immunohistochemistry.

    Misalignment of lung vessels represents a rare congenital anomaly that may cause respiratory failure in the newborn. It is characterized by abnormal position of pulmonary veins and venules that lie adjacent to arteries and bronchi, and it is usually associated with a decreased number of alveolar capillaries (i.e., alveolar capillary dysplasia), although these two conditions have been separately described. awareness of this anomaly is required by pathologists because it can be easily overlooked on lung biopsy or autopsy, and because definite diagnosis relies on histology. We report the case of a newborn male baby who developed respiratory distress 18 h after an uncomplicated delivery. The patient died on the 7th day, after high frequency oscillatory ventilation, nitric oxide inhalation and extracorporeal membrane oxygenation were unsuccessful. On autopsy, histology and immunohistochemistry demonstrated diffuse changes, fulfilling diagnostic criteria of misalignment of lung vessels and of alveolar capillary dysplasia in both lungs, with muscularization of very peripheral pulmonary arteries and a prominent interstitial and periadventitial fibrosis. Diffuse distribution of vessel misalignment could explain the rapid onset of respiratory failure, and the presence of diffuse fibrosis might have contributed to irreversible respiratory dysfunction by impairment of lung parenchyma extensibility.
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ranking = 231.71372029191
keywords = high frequency, frequency
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10/30. High-frequency flow interruption in the rescue of preterm infants with severe RDS.

    High-frequency flow interruption (HFFI) was used successfully to rescue three preterm infants with severe respiratory distress syndrome (RDS) whose clinical condition continued to deteriorate while on the conventional mechanical ventilation. Had the HFFI not been used, the survival chances might have been 25 per cent for Case 1 and 2, and 45.5 per cent for Case 3. A dramatic, immediate, and sustained improvement in ventilation and oxygenation was demonstrated once the critical frequency and amplitude of HFFI were established. bronchopulmonary dysplasia which was already evidenced in one infant before the HFFI attempt was detected in two infants. This study demonstrates that HFFI is capable of achieving adequate gas exchange and improving survival in infants with severe RDS.
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