Horseshoe lung with left lung hypoplasia and critical pulmonary venous stenosis

Lutterman, Joel; Jedeikin, Roy; Cleveland, David C.

doi:10.1016/s0003-4975(03)01213-x

Cited by 17 publications

(25 citation statements)

References 6 publications

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“…19,20 None of the four previously reported cases of horseshoe lung with left lung hypoplasia had malformations consistent with a diagnosis of FAV sequence. 17,20,22,23 The development of horseshoe lung and other horseshoe organs is thought to be associated with the inappropriate fusion of splanchic mesoderm, potentially secondary to deficiency of a mesodermal organizer. 18,20,22 Pulmonary agenesis is thought to be related to a vascular disruption, and Cunningham and Mann hypothesized that pulmonary agenesis could lead to ipsilateral vessel maldevelopment and subsequent structural malformations.…”

Section: Discussionmentioning

confidence: 99%

Horseshoe lung and facio-auriculo-vertebral sequence: A previously unreported association

et al. 2006

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We describe a case of horseshoe lung in an infant with facio-auriculo-vertebral (FAV) sequence that included mild hemifacial microsomia, ear anomalies, a missing left rib, left hemivertebrae (T2-T4), and complex congenital heart disease. Of the approximately 40 cases of horseshoe lung described since 1962, most are reported in association with scimitar syndrome, and only four reported cases were associated with left lung hypoplasia. None of these cases included malformations consistent with a diagnosis of FAV sequence.

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Section: Discussionmentioning

confidence: 99%

Horseshoe lung and facio-auriculo-vertebral sequence: A previously unreported association

et al. 2006

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“…The involvement of the right lung with hypoplasia is more common and in most cases, it is associated with the scimitar syndrome. The involvement of the left lung is exceptional and related to worse outcomes, as well as the presence of associated cardiac malformations and pulmonary hypertension 1,2 .…”

Section: Introductionmentioning

confidence: 99%

Hipertensão pulmonar em lactente associada a pulmão em ferradura: relato de caso

Neves¹,

Arrieta²,

Cavalcanti³

et al. 2011

Arq. Bras. Cardiol.

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“…Although previous strategies including surgical relief, balloon angioplasty and stenting have provided little improvement, new therapies such as lung transplantation and the use of drug-eluting stents, together with proper preoperative diagnosis, might be potentially promising treatment strategies for PVS/PVO. 9,10 In summary, we describe a rare case of congenital PVS/PVO associated with PAPVC, which was diagnosed preoperatively.…”

Section: Discussionmentioning

confidence: 94%

“…In the present case, although the patient died from pneumonia, we had been able to take a surgical approach to the lesion on the basis of proper preoperative diagnosis. Although previous strategies including surgical relief, balloon angioplasty and stenting have provided little improvement, new therapies such as lung transplantation and the use of drug‐eluting stents, together with proper preoperative diagnosis, might be potentially promising treatment strategies for PVS/PVO 9,10 …”

Section: Discussionmentioning

confidence: 99%

Persistent lung shadow in an infant with ventricular septal defect and partial anomalous pulmonary venous connection associated with pulmonary venous obstruction

Iwasa

Mitani

Sawada

et al. 2008

Pediatrics International

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Congenital pulmonary vein obstruction (PVO) is a life-threatening lesion that is frequently associated with total anomalous pulmonary vein (PV) connection, but rarely with hypoplastic left heart syndrome, transposition of the great artery, or cor triatriatum. 1 Preoperative diagnosis of this combination is sometimes challenging: previous cases are reported in the postoperative or postmortem studies. We here report on an infant with a rare association of congenital PVO, ventricular septal defect (VSD), and partial anomalous pulmonary venous connection (PAPVC), which was diagnosed preoperatively in the work-up of failure to thrive and persistent infi ltrated lung shadow. Case reportThe patient was a 9-month-old boy. He had failure to gain weight at 4 -7 months of age. Chest X-ray showed infiltrated shadow in the lower lobe of the right lung and cardiomegaly ( Fig. 1 ). At 8 months of age he had tachypnea and subcostal retraction, accompanied by the persistent infi ltrated shadow on the chest X-ray. At 9 months of age he was referred to Mie University Hospital for subsequent examination.Physical examination on admission showed indicated low bodyweight for age (6.2 kg), tachypnea (respiratory rate, 60/ min) and subcostal retraction. No obvious heart murmur was heard with increased II p component of the second heart sound. Electrocardiography indicated right ventricular hypertrophy with no left ventricular or atrial hypertrophy. Echocardiography indicated dilatation of the right atrium and ventricle, perimembranous VSD with bilateral shunting, severe pulmonary hypertension, atrial septal defect (ASD); and right upper lobe PV drain to the superior vena cava (SVC; Fig. 2a ). The right lower lobe PV was not found on repeat echocardiogram. On pulsed Doppler echocardiography the peak velocity in the left upper lobe PV was 1.62 m/s and that of the right upper lobe PV was 2.54 m/s ( Fig. 2a,b ). Computed tomography (CT) indicated that the right lower lobe PV was interrupted behind the left atrium, that the orifi ce of the left upper -middle lobe PV seemed stenotic ( Fig. 2c ), and that the left lower lobe PV was dilated ( Fig. 2d ). The right upper lobe PV was unclear. Pulmonary sequestration was excluded. Technetium-99 m lung perfusion scans showed absent fl ow to the right lower lobe and decreased fl ow to the bilateral upper lobes ( Fig. 3a,b ).Cardiac catheterization was performed, in which mean pulmonary arterial pressure was 59 mmHg, with mean aortic pressure of 66 mmHg. Pulmonary arterial wedge pressure was 6 mmHg in the right upper pulmonary artery (PA), 6 mmHg in the right middle PA, 23 mmHg in right lower PA and 4 mmHg in the left lower PA. The venous phase of the right pulmonary angiogram indicated obstruction of the right lower lobe PV, and stenosis of right upper lobe PV draining to SVC ( Fig. 3c ). Oxygen challenge test was good: pulmonary arterial diastolic pressure decreased from 38 mmHg to 27 mmHg, negating the possibility of Eisenmenger physiology. Accordingly, the preoperative diagnosis was PAPVC (stenoti...

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Horseshoe lung with left lung hypoplasia and critical pulmonary venous stenosis

Cited by 17 publications

References 6 publications

Horseshoe lung and facio-auriculo-vertebral sequence: A previously unreported association

Horseshoe lung and facio-auriculo-vertebral sequence: A previously unreported association

Hipertensão pulmonar em lactente associada a pulmão em ferradura: relato de caso

Persistent lung shadow in an infant with ventricular septal defect and partial anomalous pulmonary venous connection associated with pulmonary venous obstruction

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