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CLINICAL STUDIES: PEDIATRIC CARDIOLOGY

Isolated ductus arteriosus aneurysm in the fetus and infant: a multi-institutional experience

Umesh Dyamenahalli, MBBS, FRCPC^*, Jeffrey F. Smallhorn, MBBS, FRCPC^*, Tal Geva, MD, Jean-Claude Fouron, MD, FRCPC^||, Patricia Cairns, MD, FRCPC, Luc Jutras, MD, FRCPC^¶, Victoria Hughes, Marlene Rabinovitch, MD, FRCPC^* , Catherine A. E. Mason and Lisa K. Hornberger, MD, FACC^*

^* Department of Pediatrics, Division of Cardiology, the Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada
Cardiovascular Research, the Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada
MCTU Diagnostics Ltd., Toronto, Ontario, Canada
Department of Cardiology, Children’s Hospital and the Department of Pediatrics, Harvard Medical School, Boston, Massachusetts, USA
^|| Division of Cardiology, Sainte-Justine Hospital, Montreal University,Montreal, Canada
^¶ Division of Cardiology, Montreal Children’s Hospital, McGill University, Montreal, Canada

Manuscript received October 18, 1999; revised manuscript received January 18, 2000, accepted March 24, 2000.

Reprint requests and correspondence: Dr. Lisa K. Hornberger, Division of Cardiology, the Hospital for Sick Children, Toronto, Ontario, Canada, M5G 1X8
hornberg{at}sickkids.on.ca

OBJECTIVES

The purpose of this study was to describe the clinical characteristics and outcome and to elucidate the pathogenesis of ductus arteriosus aneurysm (DAA).

BACKGROUND

Ductus arteriosus aneurysm is a rare lesion that can be associated with severe complications including thromboembolism, rupture and death.

METHOD

We reviewed the clinical records, diagnostic imaging studies and available histology of 24 cases of DAA, diagnosed postnatally (PD) in 15 and antenatally (AD) in 9 encountered in five institutions.

RESULTS

Of PD cases, 13 presented at <2 months, and all AD cases were detected incidentally after 33 weeks of gestation during a late trimester fetal ultrasound study. Of the 24, only 4 had DAA-related symptoms and 6 had associated syndromes: Marfan, Smith-Lemli-Opitz, trisomies 21 and 13 and one possible Ehlers-Danlos. Three had complications related to the DAA: thrombus extension into the pulmonary artery, spontaneous rupture, and asymptomatic cerebral infarction. Six underwent uncomplicated DAA resection for ductal patency, DAA size or extension of thrombus. In the four examined, there was histologic evidence of reduced intimal cushions in two and abnormal elastin expression in two. Five of the 24 died, with only one death due to DAA. Of 19 survivors, all but one remain clinically asymptomatic at a median follow-up of 35 months; however, two have developed other cardiac lesions that suggest Marfan syndrome. A review of 200 consecutive third trimester fetal ultrasounds suggests an incidence of DAA of 1.5%.

CONCLUSIONS

Ductus arteriosus aneurysm likely develops in the third trimester perhaps due to abnormal intimal cushion formation or elastin expression. Although it can be associated with syndromes and severe complications, many affected infants have a benign course. Given the potential for development of other cardiac lesions associated with connective tissue disease, follow-up is warranted.

Abbreviations and Acronyms   CT = computerized tomography   DAA = ductus arteriosus aneurysm   MRI = magnetic resonance imaging   PDA = patent ductus arteriosus

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