Laparoscopic resection of intra-abdominal extralobar pulmonary sequestration

The intra-abdominal localization of extralobar pulmonary sequestration (EPS) is an uncommon entity, although there are an increasing number of publications in literature on EPS over recent years. There seems to be a predominance of left-sided suprarenal positioning of the sequester and so far resection has been undertaken by way of laparotomy. This paper describes the laparoscopic resection of EPS in two patients. In both instances the procedure was successful and the postoperative course was uneventful. It is concluded that EPS should be included in the differential diagnosis of suprarenal masses, particularly on the left side. Laparoscopic resection is the method of choice for EPS.     

Infradiaphragmatic extralobar pulmonary sequestration masquerading as an intra-abdominal, suprarenal mass

Three infants were found to have infradiaphragmatic masses by prenatal ultrasound. Postnatal imaging studies confirmed the presence of these masses, which were suspected of being intra-abdominal malignancies (neuroblastoma). The other principal differential diagnosis was extralobar pulmonary sequestration (EPS). Intraoperative findings were consistent with EPS, which was confirmed by histologic examination. We present these three infants, review the literature, and discuss the evaluation and treatment of infradiaphragmatic EPS.     

Intra-abdominal extralobar pulmonary sequestration

A left-upper-quadrant abdominal mass was identified in a routine ultrasound (US) examination in the 16th week of gestation. The sonographic features were those of a homogeneous, hyperechogenic lesion situated between the diaphragm and the left kidney. Other radiologic examinations (CT, MRI) confirmed the mass, but a presumptive diagnosis could not be made. The lesion was excised and histologic examination demonstrated an extralobar pulmonary sequestration. Although it is an uncommon type of congenital malformation, pulmonary sequestration should be included in the differential diagnosis when an echogenic intraabdominal mass is detected on antenatal US.     

Sclerosing haemangioma arising within extralobar pulmonary sequestration

Extralobar pulmonary sequestration is a rare anomaly of abnormal pulmonary tissue without any communication to the bronchial tree. Sclerosing haemangioma is a rare lung tumour, generally seen in middle-aged women. The combination of these two rare pathologies has not been previously reported. We describe the CT and CT angiographic findings of sclerosing haemangioma arising within an extralobar pulmonary sequestration in a 2-year-old girl.     

Infradiaphragmatic extralobar pulmonary sequestration in an infant

A rare case of infradiaphragmatic pulmonary sequestration with an antenatally diagnosed abdominal tumor is reported. Extirpation of the tumor was performed 2 months after delivery; the histological examination showed alveolar and bronchial tissue. Offprint requests to: M. Yagi     

Cystic sonographic appearance of extralobar pulmonary sequestration

A cystic fetal chest mass showing spontaneous improvement in utero is described. This mass was shown to be an extralobar pulmonary sequestration with associated cystic adenomatoid malformation type 2.     

Torsion and infarction of an extralobar pulmonary sequestration

An 8-year-old girl presented with rapid onset of haemorrhagic pleural effusion due to torsion and infarction of an extralobar sequestration of the lung. A literature survey has failed to reveal a similar case.     

Intra-abdominal extralobar pulmonary sequestration presenting antenatally as a suprarenal mass

Extralobar pulmonary sequestration is a rare pulmonary parenchymal anomaly which rarely may be present in an intra-abdominal location. The authors report a case of intra-abdominal extralobar pulmonary sequestration which presented to us as an antenatally diagnosed suprarenal mass and was worked up as such. The diagnosis was revealed only at laparotomy. Intra-abdominal extralobar pulmonary sequestration should also be kept in differential diagnosis in cases of masses in the suprarenal location, especially on the left side.     

Congenital cystic adenomatoid malformation and extralobar sequestration occurring independently in the ipsilateral hemithorax

We report a case of congenital cystic adenomatoid malformation (CCAM) and extralobar pulmonary sequestration (EPS) occurring independently in the ipsilateral hemithorax. A literature search using Medline, Winspirs 2.0, found 14 previously reported cases of CCAM within an EPS. There are no reports of each form of congenital pulmonary abnormality occurring separately in the same patient. Accepted: 17 December 1998     

Upper thoracic extralobar pulmonary sequestration presenting with respiratory distress in a newborn

An upper thoracic extralobar pulmonary sequestration in a newborn is presented. It was present on the initial chest radiograph taken because of respiratory distress. The sequestration was surgically removed and the infant's respiratory difficulties improved.     

Non-immune hydrops fetalis and bilateral pulmonary hypoplasia in a newborn infant with extralobar pulmonary sequestration

Extralobar pulmonary sequestration was found in a newborn premature infait that presented with non-immune hydrops fetalis, massive bilateral hydrothorax and polyhydramnios in utero. The baby died of severe respiratory insufficiency 15 h after birth. Postmortem examination revealed distended lymphatic vessels in the sequestered lung tissue probably due to impeded lymph drainage. We suggest that not extralobar pulmonary sequestration itself but a subsequent massive unilateral hydrothorax due to severe obstruction of lymph drainage was the cause of the non-immune hydrops fetalis, pulmonary hypoplasia and polyhydramnios. If these symptoms are diagnosed before delivery, a search for extralobar pulmonary sequestration is indicated.     

Prenatal ultrasound detection of intra-abdominal pulmonary sequestration with postnatal MRI correlation

Extralobar pulmonary sequestration is part of the spectrum of bronchopulmonary-foregut malformations. Intra-abdominal pulmonary sequestration has been described as a cause of a fetal abdominal mass [1–3]. We report a case of intra-abdominal pulmonary sequestration with associated elements of cystic adenomatoid malformation and a gastric duplication cyst detected on prenatal ultrasound with postnatal magnetic resonance imaging correlation.     

Complex bronchopulmonary foregut malformation: extralobar pulmonary sequestration associated with a duplication cyst of mixed bronchogenic and oesophageal type

We report a 13-year-old girl with an unusual, complex bronchopulmonary foregut malformation. The malformation included extralobar pulmonary sequestration and a duplication cyst of mixed bronchogenic and oesophageal type. Preoperative CT and MRI demonstrated the cystic and solid portions of the mass and indicated an aberrant vascular supply, suggesting the possibility of bronchopulmonary foregut malformation and several other differential diagnoses. A direct communication between the cyst and the bronchus of the sequestrated lung was found on pathological examination. This unusual combination of an extralobar pulmonary sequestration and a foregut cyst points to a common embryological pathogenesis. Received: 15 November 1999 Revised: 25 July 2000 Accepted: 16 October 2000     

Bronchial atresia is common to extralobar sequestration, intralobar sequestration, congenital cystic adenomatoid malformation, and lobar emphysema.

Congenital cystic adenomatoid malformation (CCAM), intralobar sequestration (ILS), extralobar sequestration (ELS), and lobar emphysema (LE) are well-accepted entities; however, certain findings are common to all, particularly the parenchymal maldevelopment characterizing CCAM. Isolated reports have described bronchial atresia (BA) in some specimens in all 4 entities, but this finding has not been evaluated in a prospective manner. With the aid of a dissecting microscope, we prospectively examined 47 lung specimens resected during the past 4 years and submitted with the clinical impression of ELS (n=11), ILS (n=11), CCAM (n=20), LE (n=4), and airway-esophageal communication (n=1). Most lesions were detected by prenatal ultrasound and were resected during infancy. The clinical impression and pathologic findings were compared. Pathologic examination revealed atresia of a lobar, segmental, or subsegmental bronchus in 100% of ELS, 82% of ILS, 70% of CCAM, and 50% of LE (those clinically recognized to have BA or minor CCAM) cases. Parenchymal maldevelopment that characterizes CCAM was present in 100% of CCAM cases (as expected by definition) as well as in 91% of ELS, 91% of ILS, and 50% of LE (those with BA) cases. Bronchial atresia is present in all ELS, most ILS and CCAM, and some LE cases, and its detection is greatly enhanced with the dissecting microscope. Bronchial atresia and CCAM nearly always coexist. It may be that both have the same etiopathogenesis with anatomic differences accounted for by aberrant genetic programs or other insults, perhaps modified by time of onset or duration.     

Coincidence of congenital cystic adenomatoid malformation and extralobar sequestration of the lung in a newborn

We present a rare case of coincidence of an extralobar sequestration with a congenital cystic adenomatoid malformation of the lung in a newborn. The symptoms, diagnostic features, and therapy are described and the etiology and classification are briefly discussed.     

MRI demonstration of pulmonary sequestration

The preoperative evaluation of pulmonary sequestration requires delineation of the arterial supply and venous drainage of both the sequestered segment and the neighboring lung. In this case magnetic resonance imaging was the sole modality which demonstrated all components of the arterial and venous circulations of a sequestration and the remainder of the ipsilateral lung.