Retroperitoneal Arteriovenous Malformation: A Rare Cause of Pulmonary Hypertension in a Premature Infant

Zagura, Maksim; Konik, Heleri; Padari, Helgi; Metsvaht, Tuuli; Hermlin, Toomas; Salumae, Liis; Tomberg, Tiiu

doi:10.14740/ijcp214w

Cited by 2 publications

(3 citation statements)

References 8 publications

(11 reference statements)

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“…These lesions can occur anywhere in the body and clinical symptoms and signs are related to both the location and dimensions of the AVM [5]. Cases of massive aortic AVMs giving rise to suprasystemic pulmonary hypertension are sparsely reported in the literature [4]. Most documented cases of symptomatic AVMs refer to those occurring within the cerebral or hepatic circulations.…”

Section: Discussionmentioning

confidence: 99%

“…There are few instances where increased pulmonary blood flow (PBF) result in neonatal hypertension, one of which is the presence of a massive AVM [7]. There have been multiple case reports in the literature of neonatal pulmonary hypertension secondary to cerebral, hepatic, and pulmonary AVMs, but only one of neonatal suprasystemic pulmonary hypertension due to a massive retroperitoneal aortocaval AVM [4]. The supportive management of pulmonary hypertension in this case included NIV CPAP, furosemide (1mg/kg twice daily), and spironolactone (1mg/kg twice daily) to aid in reducing circulatory overload.…”

Section: Discussionmentioning

confidence: 99%

“…The most common site for neonatal vascular malformations is the liver, followed by the brain and lung [3]. Diagnosis of AVMs can be difficult in the neonatal period as their clinical presentation is very variable [4]. As AVMs are extremely rare, they are far down the list of potential causes of severe pulmonary hypertension in the neonatal period.…”

Section: Introductionmentioning

confidence: 99%

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A Massive Arteriovenous Malformation arising from the Aorta causing Severe Pulmonary Hypertension

Kennelly

Rea²,

Crowe

2018

IPCC

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Aortic arteriovenous malformations (AVMs) are rare vascular anomalies where the aorta communicates with adjacent venous structures, bypassing the capillary system. We report a case of a neonate born at 34 weeks gestation who presented at 3 weeks of life in respiratory distress. Echocardiography demonstrated severe pulmonary hypertension with right ventricular dilatation but an otherwise structurally normal heart. Clinical examination revealed a pulsatile abdominal mass with an audible bruit. Abdominal ultrasound was performed and it demonstrated a large cystic mass arising from the inferior vena cava (IVC) with both arterial and venous vascular flow within it. Further evaluation with magnetic resonance angiography (MRA) showed a massive arteriovenous malformation from both left and right iliac arteries, and lumbar arteries communicating with the IVC. A decision was made with interventional radiology to attempt coiling of the vascular lesion. Following this procedure his pulmonary hypertension worsened in severity, culminating in acute heart failure and multi-organ dysfunction. A subsequent interventional radiology procedure found that there was no blood flow to his bowel or abdominal viscera. Cardiac arrest followed with an unsuccessful attempt at resuscitation. This case describes a presentation of severe pulmonary hypertension initially considered to be secondary to either congenital heart disease or intrinsic pulmonary disease, but which was found to be as a result of a very rarely occurring massive aortic AVM.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%