Total Anomalous Pulmonary Venous Connection with Severe Pulmonary Venous Obstruction

Hastreiter, Alois R.; Paul, Milton H.; Molthan, Marian E.; Miller, Robert A.

doi:10.1161/01.cir.25.6.916

Cited by 74 publications

(16 citation statements)

References 18 publications

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“…Although the anatomical variations of the anomalous major pulmonary veins have been reported in detail (Burroughs & Edwards 1960, Hastreiter et al 1962, Elliott & Edwards 1962, Delisle et ul. 1976)~ controversy still exists concerning the nature and importance of the intrapulmonary vascular lesions in TAPVC.…”

Section: Discussionmentioning

confidence: 99%

Pulmonary vascular disease in 57 necropsy cases of total anomalous pulmonary venous connection

Petersen

Edwards

1983

Histopathology

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In a semi-quantitative necropsy study, total anomalous pulmonary venous connection (TAPVC) was an isolated anomaly in 35 cases and was associated with asplenia and either pulmonary stenosis or pulmonary atresia in 22 cases. A comparison of the two groups showed pulmonary venous obstruction (26% vs. 4%), a small interatrial communication (51% vs. 4%), a patent ductus arteriosus or operative shunt (40% vs. 64%), medial hypertrophy of muscular pulmonary arteries (80 vs. 23%), muscularization of arterioles (80% vs. 23%), capillary engorgement (60% vs. 27%) alveolar wall thickening (29% vs. 0%), interlobular septal oedema (34% vs. 14%), dilated lymphatics (80% vs. 45%), arterialization of pulmonary veins (86% vs. 68%), medial hypertrophy of veins (26% vs. 0%), and venous dilatation (74% vs. 50%). Only one patient, a 5-year-old girl with isolated TAPVC, had severe plexogenic pulmonary arteriopathy. In the remainder, the pulmonary lesions were those generally associated with reversible pulmonary venous hypertension; they were most striking in patients with pulmonary venous obstruction, and were least prominent in patients with pulmonary stenosis or pulmonary atresia.

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Section: Discussionmentioning

confidence: 99%

Pulmonary vascular disease in 57 necropsy cases of total anomalous pulmonary venous connection

Petersen

Edwards

1983

Histopathology

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“…Certain variations exist and likewise combinations, but in general they follow the above patterns. 5 6 In all varieties except mixed drainage and some cases of total connection to the right atrium, there is a common pulmonary vein into which the individual pulmonary veins drain. This usually is in close proximity to the left atrium but occasionally it may be far away, as was seen in one of our cases.…”

Section: Discussionmentioning

confidence: 99%

Assessment of total anomalous pulmonary venous connection by two-dimensional echocardiography.

Smallhorn¹,

Sutherland²,

Tommasini³

et al. 1981

Heart

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Twenty-three patients with total anomalous pulmonary venous connection were studied by two-dimensional echocardiography. In all cases the diagnosis was made before invasive procedures, with surgical or angiocardiographic confirmation. Eleven patients had supracardiac drainage (three to the coronary sinus, two to the right atrium,) and seven had infracardiac drainage. In the majority of cases the precise pattern of drainage could be identified by combining suprasternal, praecordial, and subcostal views. In 12 cases where the suprasternal cut was used a pulmonary venous confluence could be identified, having a cross-like structure in nine, with three others appearing as a dilated channel behind and separate from the left atrium. Thus, two-dimensional echocardiography reliably makes the diagnosis of total anomalous pulmonary venous connection and in the majority the precise pattern of drainage can be determined.

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“…Lucas et al (1963) observed little change during the first month of life, though reporting the 'early' development of pulmonary arterial and venous medial hypertrophy in any cardiac malformation in which obstruction to pulmonary venous return is present at or soon after birth. 'Underdevelopment' of the left ventricle has been reported in total anomalous pulmonary venous return by some workers (Hastreiter et al, 1962), one of a normal size by others (Leachman et al, 1969).…”

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confidence: 90%

Structural study of pulmonary circulation and of heart in total anomalous pulmonary venous return in early infancy.

Haworth¹,

Reid²

1977

Heart

101

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Quantitative morphometric techniques have been applied to the injected and inflated lung and to the heart in 9 infants with total anomalous pulmonary venous return dying with obstruction to pulmonary venous return. In 5 (mean age at death 55 days) pulmonary venous blood drained to a supradiaphragmatic site; in the remaining 4 (mean age 20 days) to an infradiaphragmatic site.Structural changes were present in the pulmonary circulation in all patients, even in the youngest, an 8-day-old child. In both types of total anomalous pulmonary venous return increased arterial muscularity was severe, as shown by increase in wall thickness and by extension of muscle into smaller and more peripheral arteries than normal; these changes tended to increase with age. Where the pulmonary venous blood drained to a supradiaphragmatic site, the severity of arterial medial hypertrophy correlated inversely with the magnitude of the pulmonary: systemic flow ratio, increasing as the pulmonary blood flow fell. Vein wall thickness was increased and in all but the youngest child the veins were 'arterialised'. At the lung periphery the arteries and alveoli appeared to have multiplied normally. Arterial size varied according to whether pulmonary venous blood drained above or below the diaphragm; the diameter of pre-and intra-acinar arteries was increased only in cases where the pulmonary venous return drained to a supradiaphragmatic site, being normal when it drained to an infradiaphragmatic site.In the heart the left ventricle was of normal size in all but one case. Dilatation and severe hypertrophy of the right ventricle and septum were present only in cases of drainage to a supradiaphragmatic site.In the older patients with the latter anomaly dilatation of the pulmonary arteries and right ventricle suggested that a large left-to-right shunt had preceded the onset of obstruction to pulmonary venous return and that the more severe right ventricular and septal hypertrophy in these cases might be the result of a longer duration of pulmonary hypertension. In contrast, in total anomalous pulmonary venous return to an infradiaphragmatic site it appears that obstruction to pulmonary venous return develops soon after birth and prevents a large increase in pulmonary blood flow, and thus neither the pulmonary arteries nor the right ventricle become dilated. In infants with total anomalouspulmonary venous return and obstruction to pulmonary venous return, it is striking how rapidly the pulmonary circulation develops new muscle.Total anomalous pulmonary venous return is a Most infants with an infradiaphragmatic type of disease of infancy (Burroughs and Edwards, 1960; pulmonary venous connexion develop obstruction Bonham-Carter et al., 1969) since fewer than 25 to pulmonary venous return soon after birth, but per cent of patients with the uncomplicated form it is now recognised that also when the connexion survive infancy and these usually have a supra-is supradiaphragmatic, obstruction can develop diaphragmatic type of connexion. They develop at this ...

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Total Anomalous Pulmonary Venous Connection with Severe Pulmonary Venous Obstruction

Cited by 74 publications

References 18 publications

Pulmonary vascular disease in 57 necropsy cases of total anomalous pulmonary venous connection

Pulmonary vascular disease in 57 necropsy cases of total anomalous pulmonary venous connection

Assessment of total anomalous pulmonary venous connection by two-dimensional echocardiography.

Structural study of pulmonary circulation and of heart in total anomalous pulmonary venous return in early infancy.

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