Fetal thoracoamniotic shunting as the only treatment for pulmonary sequestration with hydrops: favorable long-term outcome without postnatal surgery.
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Although it is established that bronchopulmonary sequestration complicated by fetal hydrops carries a high risk of perinatal mortality, prenatal management remains controversial. Therapeutic options include immediate delivery, medical therapy based on an inotropic regimen, alcohol ablation of the vascular pedicle, open fetal surgery, or thoracoamniotic shunting of fetal pleural effusions. We report a case of pulmonary sequestration with hydrothorax and fetal hydrops which was successfully treated by a single fetal thoracoamniotic catheter drainage. Following emergency catheter placement, hydrothorax and hydrops decreased dramatically. The sequestration gradually disappeared postnatally, and long-term outcome remained normal without any postnatal therapy. Based on this observation, the natural history of pulmonary sequestration and prenatal management options are reviewed. (+info)
Congenital cystic adenomatoid malformation (CCAM) and extralobar pulmonary sequestration (EPS) are 2 rare types of bronchopulmonary malformations that typically appear in infancy and childhood. A CCAM is a hamartomatous lesion of the lung, thought to be the result of the cessation of bronchiolar maturation with overgrowth of mesenchymal elements. An EPS is a discrete mass of lung tissues separate from the tracheobronchial tree and is supplied by a systemic vascular source (usually aortic). As well, it forms outside the normal pleural investment. Conran and Stocker analyzed 50 cases of EPS and found that 50% (23 of 46) were associated with a coexistent CCAM. Rare cases of intra-abdominal EPS (IEPS) have been reported in the literature, and in these cases, CCAMs commonly occur within the pulmonary sequesterant. We present an interesting case of CCAM manifesting within the liver of a patient treated prenatally for a large pulmonary type II CCAM and review the reported cases of IEPS with CCAM. (+info)
Prenatal sonographic features of intralobar bronchopulmonary sequestration.
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Congenital lung lesions that can be diagnosed on the basis of prenatal sonography include cystic adenomatoid malformations and extrapulmonary bronchopulmonary sequestrations. Intralobar bronchopulmonary sequestration is a rare congenital malformation of the lung. The prenatal sonographic features of this condition are reported here. (+info)
Adherent neutrophils mediate permeability after atelectasis.
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Re-expansion of atelectatic lung is associated with increased permeability. This study tests whether neutrophils mediate this event. Right middle lobar atelectasis was induced in anesthesized rabbits (n = 18) by intraluminal obstruction of the bronchus after a 20-minute ventilation with 100% O2. After 1 hour of bronchial obstruction and 20 minutes after lobar re-expansion, leukopenia was noted, 2870 +/- 210 white blood cells (WBC)/mm3, relative to control animals treated with a noninflated balloon catheter, 6500 +/- 410 WBC/mm3 (p less than 0.05). Three hours after re-expansion, neutrophils were sequestered in the previously atelectatic region 78 +/- 7 polymorphonuclear leukocytes (PMN)/10 high-power field (HPF), as well as in nonatelectatic areas, 40 +/- 3 PMN/10 HPF, higher than control values of 26 +/- 3 PMN/10 HPF (p less than 0.05). In the atelectatic region, neutrophil sequestration was associated with increased protein concentration in lobar bronchoalveolar lavage (BAL) of 1370 +/- 100 micrograms/mL, higher than control values of 270 +/- 20 micrograms/mL (p less than 0.05). Reexpansion also induced increases in lung wet-to-dry weight ratio (W/d) of 6.2 +/- 0.2, higher than control values of 4.3 +/- 0.1 (p less than 0.05). Rendering rabbits neutropenic (n = 18) (0 to 4 PMN/mm3) limited the atelectasis-induced protein accumulations in BAL (520 +/- 60 micrograms/mL) and increase in lung W/d (5.2 +/- 0.1) (both p less than 0.05). Intravenous (I.V.; treatment of another group (n = 18) with an anti-CD 18 monoclonal antibody (R 15.7, 1 mg/kg) before balloon deflation prevented leukopenia (6550 +/- 560 WBC/mm3), minimized neutrophil sequestration (36 +/- 2 PMN/10 HPF), and attenuated protein leak (710 +/- 95 micrograms/mL) and the increased lung W/d (5.6 +/- 0.1) (all p less than 0.05). A final atelectatic group (n = 9) was treated I.V. with the anti-intercellular adhesion molecule-1 monoclonal antibody (RR 1/1, 1 mg/kg), which also prevented leukopenia and showed similar protection of microvascular barrier function. These data indicate that adherent neutrophils in large part mediate lung permeability and edema after atelectasis and re-expansion. Adhesion receptors of both neutrophils and endothelial cells regulate this event. (+info)
Foetal intralobar lung sequestration: antenatal diagnosis and management.
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OBJECTIVES: The objectives of this study are to discuss the use of ultrasonography for the diagnosis of foetal intralobar sequestration (FILS) antenatally and the management options available for these pregnancies. METHODS: This is a retrospective review of six cases of FILS diagnosed antenatally by two dimensional (2D) and colour Doppler ultrasonography out of a total of 31,508 deliveries over a two-year period at the KK Women's and Children's Hospital. RESULTS: The incidence of FILS in this hospital was 1 in 5,251 deliveries. 2D ultrasonography showed an echogenic lung in all cases. FILS was confirmed by the demonstration of a systemic vessel leading to the affected lung on colour Doppler examination. After counselling, four terminated their pregnancies during mid-trimester, while two continued their pregnancies to term. Confirmation of the terminated cases was by post-mortem. In the two pregnancies that continued, regular growth scans were done to monitor the progression of the condition. Computed tomography confirmed the diagnosis post-delivery. Both were well but one had a resection of the sequestrated lung although he was asymptomatic. Histology also confirmed the diagnosis. CONCLUSION: FILS is a rare anomaly. 2D and colour Doppler ultrasonography are used to diagnose the condition antenatally. Termination of the pregnancy is not always indicated, as there are favourable outcomes from FILS. (+info)
Localised fibrous mesothelioma arising in an intralobar pulmonary sequestration.
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A localised fibrous mesothelioma arising from an intralobar lung sequestration occurred in a 64 year old Turkish woman. This appears to be the first report of a mesothelioma occurring within a pulmonary sequestration. (+info)
Spontaneous improvement of intrathoracic masses diagnosed in utero.
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The diagnosis and pregnancy outcome for 14 fetuses with sonographically detected chest masses were reviewed retrospectively. Six lesions became smaller or less apparent during gestation or resolved between antenatal and perinatal imaging studies; these included all three types of cystic adenomatoid malformation (CAM), as well as one case of pulmonary sequestration (PS). This information is extremely important in counseling patients in the second trimester who are considering termination of pregnancy. The poor prognosis traditionally assigned to type II and type III CAM needs changing to reflect the phenomenon of improvement with excellent long-term outcomes. Sonographic indicators of poor outcome were polyhydramnios, hydrops or marked cardiac deviation. (+info)
Volume contrast imaging: A new approach to identify fetal thoracic structures.
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OBJECTIVE: To assess the potential of volume contrast imaging for evaluation of fetal intrathoracic structures. METHODS: Volume contrast imaging is a new ultrasonographic method that increases the contrast between tissues. It consists of a 5- to 10-mm-thick slice-shaped volume image projected on a 2-dimensional screen. The rendering process applied on the slice smoothens the speckle pattern of the image by filling up the gaps with tissue information from the adjacent layers. To evaluate the potential of volume contrast imaging for enhancing the contrast between fetal lungs and surrounding tissues, we compared the ability of volume contrast imaging and conventional ultrasonography to image the fetal thymus in 50 controls. We also applied volume contrast imaging to prenatal imaging of 6 thoracic abnormalities (2 left congenital diaphragmatic hernias, 1 right diaphragmatic hernia, 2 congenital adenomatoid lung malformations, and 1 lung sequestration). RESULTS: In controls, the thymus was identified in all cases by volume contrast imaging and in 42 cases (84%) by conventional 2-dimensional ultrasonography. Clear images of macrocystic and microcystic congenital adenomatoid malformations were obtained by volume contrast imaging, which provided precise contouring of the lesions. In cases with congenital diaphragmatic hernias, volume contrast imaging provided clear images of the limits of the lungs ipsilateral to the hernia. CONCLUSIONS: Volume contrast imaging may enhance the contrast between fetal lungs and surrounding organs and can be applied to prenatal imaging of intrathoracic structures in cases with thoracic fetal abnormalities. (+info)