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• W2079457666 abstract "Primary congenital abdominal aortic aneurysm is an extremely rare entity, with only 15 patients reported in the literature. Options for repair are often limited secondary to branch vessel size and other anatomic limitations. We present a neonate diagnosed with an abdominal aortic aneurysm on prenatal ultrasound. A postpartum computed tomography angiogram revealed an extensive type IV thoracoabdominal aortic aneurysm extending to the aortic bifurcation and resulting in bilateral renal artery stenosis. The unique features of this patient and challenges in management are discussed. Primary congenital abdominal aortic aneurysm is an extremely rare entity, with only 15 patients reported in the literature. Options for repair are often limited secondary to branch vessel size and other anatomic limitations. We present a neonate diagnosed with an abdominal aortic aneurysm on prenatal ultrasound. A postpartum computed tomography angiogram revealed an extensive type IV thoracoabdominal aortic aneurysm extending to the aortic bifurcation and resulting in bilateral renal artery stenosis. The unique features of this patient and challenges in management are discussed. Abdominal aortic aneurysm (AAA) in neonates is rare and occurs most commonly secondary to other causes such as connective tissue disease, infection, vasculitis, and umbilical artery catheterization.1Mendeloff J. Stallion A. Hutton M. Goldstone J. Aortic aneurysm resulting from umbilical artery catheterization: case report, literature review, and management algorithm.J Vasc Surg. 2001; 33: 419-424Abstract Full Text Full Text PDF PubMed Scopus (54) Google Scholar Primary congenital AAA is very uncommon, with only 15 such aneurysms diagnosed in infancy having been reported thus far.2Bell P. Mantor C. Jacocks M.A. Congenital abdominal aortic aneurysm: a case report.J Vasc Surg. 2003; 38: 190-193Abstract Full Text Full Text PDF PubMed Scopus (23) Google Scholar, 3Buddingh K.T. Zeebregts C.J. Tilanus M.E. Roofthooft M.T. Broens P.M. Large neonatal thoracoabdominal aneurysm: case report and review of the literature.J Pediatr Surg. 2008; 43: 1361-1364Abstract Full Text Full Text PDF PubMed Scopus (9) Google Scholar, 4Cantinotti M. Luchi C. Assanta N. Prenatal diagnosis of abdominal aortic aneurysm.Pediatr Cardiol. 2010; 31: 155-156Crossref PubMed Scopus (4) Google Scholar, 5Cheung S.C. Khong P.L. Chiu W. Metreweli C. Congenital abdominal aortic aneurysm and renal dysplasia.Pediatr Radiol. 2004; 34: 827-830PubMed Google Scholar, 6Howorth M.B. Aneurysm of abdominal aorta in the newborn infant Report of case.N Engl J Med. 1967; 276: 1133-1134Crossref PubMed Scopus (36) Google Scholar, 7Kim E.S. Caiati J.M. Tu J. Nowygrod R. Stolar C.J. Congenital abdominal aortic aneurysm causing renovascular hypertension, cardiomyopathy, and death in a 19-day-old neonate.J Pediatr Surg. 2001; 36: 1445-1449Abstract Full Text Full Text PDF PubMed Scopus (26) Google Scholar, 8Kim J.I. Lee W. Kim S.J. Seo J.W. Chung J.W. Park J.H. Primary congenital abdominal aortic aneurysm: a case report with perinatal serial follow-up imaging.Pediatr Radiol. 2008; 38: 1249-1252Crossref PubMed Scopus (11) Google Scholar, 9Laing A.J. Hussey A.J. Joyce M.R. Salmo E. Cassidy M. Flynn J. Ruptured abdominal aortic aneurysm in a 12-month-old boy.J Pediatr Surg. 2002; 37: 1234-1235Abstract Full Text Full Text PDF PubMed Scopus (8) Google Scholar, 10Latter D. Béland M.J. Batten A. Tchervenkov C.I. Dobell A.R. Congenital abdominal aortic aneurysm.Can J Surg. 1989; 32: 135-138PubMed Google Scholar, 11Malee M.P. Carr S. Rubin L.P. Johal J. Feit L. Prenatal ultrasound diagnosis of abdominal aortic aneurysm with fibrotic occlusion in aortic branch vessels.Prenat Diagn. 1997; 17: 479-482Crossref PubMed Scopus (10) Google Scholar, 12Malikov S. Delarue A. Fais P.O. Keshelava G. Anatomical repair of a congenital aneurysm of the distal abdominal aorta in a newborn.J Vasc Surg. 2009; 50: 1181-1184Abstract Full Text Full Text PDF PubMed Scopus (8) Google Scholar, 13Mehall J.R. Saltzman D.A. Chandler J.C. Kidd J.N. Wells T. Smith S.D. Congenital abdominal aortic aneurysm in the infant: case report and review of the literature.J Pediatr Surg. 2001; 36: 657-658Abstract Full Text Full Text PDF PubMed Scopus (22) Google Scholar, 14Meyers R.L. Lowichik A. Kraiss L.W. Hawkins J.A. Aortoiliac reconstruction in infants and toddlers: replacement with decellularized branched pulmonary artery allograft.J Pediatr Surg. 2006; 41: 226-229Abstract Full Text Full Text PDF PubMed Scopus (17) Google Scholar, 15Saad S.A. May A. Abdominal aortic aneurysm in a neonate.J Pediatr Surg. 1991; 26: 1423-1424Abstract Full Text PDF PubMed Scopus (22) Google Scholar, 16Tsunematsu R. Shinozaki T. Fukushima K. Yumoto Y. Hidaka N. Morokuma S. et al.Congenital abdominal aortic aneurysm with porencephaly: a case report.Fetal Diagn Ther. 2011; 29: 248-252Crossref PubMed Scopus (4) Google Scholar The paucity of reported cases precludes a standardized approach to management of these aneurysms. Although repair has been successful in several patients,2Bell P. Mantor C. Jacocks M.A. Congenital abdominal aortic aneurysm: a case report.J Vasc Surg. 2003; 38: 190-193Abstract Full Text Full Text PDF PubMed Scopus (23) Google Scholar, 8Kim J.I. Lee W. Kim S.J. Seo J.W. Chung J.W. Park J.H. Primary congenital abdominal aortic aneurysm: a case report with perinatal serial follow-up imaging.Pediatr Radiol. 2008; 38: 1249-1252Crossref PubMed Scopus (11) Google Scholar, 10Latter D. Béland M.J. Batten A. Tchervenkov C.I. Dobell A.R. Congenital abdominal aortic aneurysm.Can J Surg. 1989; 32: 135-138PubMed Google Scholar, 12Malikov S. Delarue A. Fais P.O. Keshelava G. Anatomical repair of a congenital aneurysm of the distal abdominal aorta in a newborn.J Vasc Surg. 2009; 50: 1181-1184Abstract Full Text Full Text PDF PubMed Scopus (8) Google Scholar, 13Mehall J.R. Saltzman D.A. Chandler J.C. Kidd J.N. Wells T. Smith S.D. Congenital abdominal aortic aneurysm in the infant: case report and review of the literature.J Pediatr Surg. 2001; 36: 657-658Abstract Full Text Full Text PDF PubMed Scopus (22) Google Scholar, 14Meyers R.L. Lowichik A. Kraiss L.W. Hawkins J.A. Aortoiliac reconstruction in infants and toddlers: replacement with decellularized branched pulmonary artery allograft.J Pediatr Surg. 2006; 41: 226-229Abstract Full Text Full Text PDF PubMed Scopus (17) Google Scholar, 15Saad S.A. May A. Abdominal aortic aneurysm in a neonate.J Pediatr Surg. 1991; 26: 1423-1424Abstract Full Text PDF PubMed Scopus (22) Google Scholar aneurysm extent and anatomy often prohibit repair in neonates. In addition, concomitant renovascular hypertension can further complicate management in these patients.5Cheung S.C. Khong P.L. Chiu W. Metreweli C. Congenital abdominal aortic aneurysm and renal dysplasia.Pediatr Radiol. 2004; 34: 827-830PubMed Google Scholar, 7Kim E.S. Caiati J.M. Tu J. Nowygrod R. Stolar C.J. Congenital abdominal aortic aneurysm causing renovascular hypertension, cardiomyopathy, and death in a 19-day-old neonate.J Pediatr Surg. 2001; 36: 1445-1449Abstract Full Text Full Text PDF PubMed Scopus (26) Google Scholar, 11Malee M.P. Carr S. Rubin L.P. Johal J. Feit L. Prenatal ultrasound diagnosis of abdominal aortic aneurysm with fibrotic occlusion in aortic branch vessels.Prenat Diagn. 1997; 17: 479-482Crossref PubMed Scopus (10) Google Scholar Here we describe a neonate diagnosed prenatally with what appears to be the most extensive AAA yet reported. The obstacles presented in association with nonoperative management in the setting of renovascular disease are discussed. A girl, weighing 2472 grams, was born by elective Cesarean section at 34-4/7 weeks' gestation. She was twin B in a dichorionic-diamniotic twin pregnancy to a 24-year-old G4 P0030 mother. An ultrasound examination at 32 weeks' gestation had revealed an AAA with possible dissection flap. No abnormal findings were noted in the sibling. Apgar scores were 7 at 1 minute and 8 at 5 minutes. She required blow-by oxygen briefly after birth and was transferred to a tertiary children's hospital for further evaluation of the AAA. A computed tomography angiogram revealed a fusiform aneurysm measuring 2.3 cm in maximum diameter and extending from just above the diaphragm to the aortic bifurcation (Fig 1). There were luminal folds distally but no true dissection. All visceral vessels originated from the aneurysm with evidence of ostial stenosis. The renal arteries were very small, and no obvious connection with the aortic lumen was seen on computed tomography angiogram. The right renal artery appeared to be fed primarily through phrenic artery collaterals, and both kidneys showed minimal contrast enhancement (Fig 2). Abdominal ultrasound imaging revealed patent renal and mesenteric vessels but with flow patterns consistent with ostial effacement. A prenatal and two postnatal echocardiograms demonstrated mild right ventricular hypertrophy and no evidence of aortic dissection.Fig 2An axial cut of a computed tomography angiogram shows the patient's abdominal aortic aneurysm at the level of the renal arteries (arrows), illustrating poor bilateral renal perfusion and no clear connection of the renal arteries with the aortic lumen.View Large Image Figure ViewerDownload Hi-res image Download (PPT) After multidisciplinary discussion involving neonatology, surgery, genetics, and nephrology, it was determined that repair would require complete replacement of the abdominal aorta and reimplantation of all branch vessels. Given the patient's size relative to the aneurysm and the congenital stenosis of the branch vessels, we elected to manage nonoperatively with aggressive blood pressure control and defer surgical intervention. Upon presentation to our hospital she had a mean arterial pressure (MAP) of ≥90 mm Hg. She was initially started on an esmolol drip to control her hypertension. The decision was made to avoid angiotensin-converting enzyme inhibitor or angiotensin receptor blocker therapy in the setting of severely impaired renal perfusion on imaging. The patient was transitioned to isradipine and weaned off esmolol once MAPs were maintaining near 70 mm Hg. During her admission, a discussion was held with the parents regarding the complexity of the aneurysm and the dire consequences of rupture. She was discharged on isradipine for blood pressure control (goal MAP <70 mm Hg) to reduce the risk of aneurysm expansion and rupture. Transforming growth factor-β receptor 1 and 2 DNA sequencing revealed no mutations, thus excluding Loeys-Dietz syndrome as a cause of the patient's aneurysm. The plan was to closely monitor blood pressure and renal perfusion as an outpatient with a plan for reimaging and possible repair at age 6 months, when the patient's growth might make her vascular anatomy more amenable to operative intervention. Before discharge, the family decided to make the patient do not resuscitate. She was noted to have good blood pressure control at home and at her nephrology follow-up appointment. At age 4 weeks, a few hours after a follow-up appointment with her pediatrician, the patient presented to another emergency department with a firmly distended abdomen and unrecoverable blood pressure. She ultimately died, with the clinical diagnosis of a ruptured aneurysm. Since the first case report describing a ruptured AAA in infancy,6Howorth M.B. Aneurysm of abdominal aorta in the newborn infant Report of case.N Engl J Med. 1967; 276: 1133-1134Crossref PubMed Scopus (36) Google Scholar only 14 other patients have been reported, most of which were infrarenal.2Bell P. Mantor C. Jacocks M.A. Congenital abdominal aortic aneurysm: a case report.J Vasc Surg. 2003; 38: 190-193Abstract Full Text Full Text PDF PubMed Scopus (23) Google Scholar, 6Howorth M.B. Aneurysm of abdominal aorta in the newborn infant Report of case.N Engl J Med. 1967; 276: 1133-1134Crossref PubMed Scopus (36) Google Scholar, 8Kim J.I. Lee W. Kim S.J. Seo J.W. Chung J.W. Park J.H. Primary congenital abdominal aortic aneurysm: a case report with perinatal serial follow-up imaging.Pediatr Radiol. 2008; 38: 1249-1252Crossref PubMed Scopus (11) Google Scholar, 9Laing A.J. Hussey A.J. Joyce M.R. Salmo E. Cassidy M. Flynn J. Ruptured abdominal aortic aneurysm in a 12-month-old boy.J Pediatr Surg. 2002; 37: 1234-1235Abstract Full Text Full Text PDF PubMed Scopus (8) Google Scholar, 10Latter D. Béland M.J. Batten A. Tchervenkov C.I. Dobell A.R. Congenital abdominal aortic aneurysm.Can J Surg. 1989; 32: 135-138PubMed Google Scholar, 12Malikov S. Delarue A. Fais P.O. Keshelava G. Anatomical repair of a congenital aneurysm of the distal abdominal aorta in a newborn.J Vasc Surg. 2009; 50: 1181-1184Abstract Full Text Full Text PDF PubMed Scopus (8) Google Scholar, 13Mehall J.R. Saltzman D.A. Chandler J.C. Kidd J.N. Wells T. Smith S.D. Congenital abdominal aortic aneurysm in the infant: case report and review of the literature.J Pediatr Surg. 2001; 36: 657-658Abstract Full Text Full Text PDF PubMed Scopus (22) Google Scholar, 14Meyers R.L. Lowichik A. Kraiss L.W. Hawkins J.A. Aortoiliac reconstruction in infants and toddlers: replacement with decellularized branched pulmonary artery allograft.J Pediatr Surg. 2006; 41: 226-229Abstract Full Text Full Text PDF PubMed Scopus (17) Google Scholar, 15Saad S.A. May A. Abdominal aortic aneurysm in a neonate.J Pediatr Surg. 1991; 26: 1423-1424Abstract Full Text PDF PubMed Scopus (22) Google Scholar, 16Tsunematsu R. Shinozaki T. Fukushima K. Yumoto Y. Hidaka N. Morokuma S. et al.Congenital abdominal aortic aneurysm with porencephaly: a case report.Fetal Diagn Ther. 2011; 29: 248-252Crossref PubMed Scopus (4) Google Scholar This is only the second report of a congenital type IV thoracoabdominal aneurysm. Buddingh et al3Buddingh K.T. Zeebregts C.J. Tilanus M.E. Roofthooft M.T. Broens P.M. Large neonatal thoracoabdominal aneurysm: case report and review of the literature.J Pediatr Surg. 2008; 43: 1361-1364Abstract Full Text Full Text PDF PubMed Scopus (9) Google Scholar described a neonate with several saccular aneurysms combining to form a large thoracoabdominal aneurysm, but to our knowledge, the present patient represents the most extensive continuous congenital AAA yet reported and the first with severe visceral and renal artery stenosis producing renovascular hypertension. Although it is generally accepted that early elective repair is preferred to prevent rupture, no standard operative approach has been established. Multiple repair methods have been reported, including aneurysmorrhaphy,15Saad S.A. May A. Abdominal aortic aneurysm in a neonate.J Pediatr Surg. 1991; 26: 1423-1424Abstract Full Text PDF PubMed Scopus (22) Google Scholar repair with native vessels,12Malikov S. Delarue A. Fais P.O. Keshelava G. Anatomical repair of a congenital aneurysm of the distal abdominal aorta in a newborn.J Vasc Surg. 2009; 50: 1181-1184Abstract Full Text Full Text PDF PubMed Scopus (8) Google Scholar cryopreserved allograft,2Bell P. Mantor C. Jacocks M.A. Congenital abdominal aortic aneurysm: a case report.J Vasc Surg. 2003; 38: 190-193Abstract Full Text Full Text PDF PubMed Scopus (23) Google Scholar, 14Meyers R.L. Lowichik A. Kraiss L.W. Hawkins J.A. Aortoiliac reconstruction in infants and toddlers: replacement with decellularized branched pulmonary artery allograft.J Pediatr Surg. 2006; 41: 226-229Abstract Full Text Full Text PDF PubMed Scopus (17) Google Scholar and various synthetic grafts.8Kim J.I. Lee W. Kim S.J. Seo J.W. Chung J.W. Park J.H. Primary congenital abdominal aortic aneurysm: a case report with perinatal serial follow-up imaging.Pediatr Radiol. 2008; 38: 1249-1252Crossref PubMed Scopus (11) Google Scholar, 10Latter D. Béland M.J. Batten A. Tchervenkov C.I. Dobell A.R. Congenital abdominal aortic aneurysm.Can J Surg. 1989; 32: 135-138PubMed Google Scholar, 13Mehall J.R. Saltzman D.A. Chandler J.C. Kidd J.N. Wells T. Smith S.D. Congenital abdominal aortic aneurysm in the infant: case report and review of the literature.J Pediatr Surg. 2001; 36: 657-658Abstract Full Text Full Text PDF PubMed Scopus (22) Google Scholar Timing of these repairs range from the neonatal period up to age 15 months, but all were undertaken in patients whose AAAs were confined to the infrarenal aorta. No repair was attempted in several patients due to aneurysm extent and prohibitively small vessel size3Buddingh K.T. Zeebregts C.J. Tilanus M.E. Roofthooft M.T. Broens P.M. Large neonatal thoracoabdominal aneurysm: case report and review of the literature.J Pediatr Surg. 2008; 43: 1361-1364Abstract Full Text Full Text PDF PubMed Scopus (9) Google Scholar, 5Cheung S.C. Khong P.L. Chiu W. Metreweli C. Congenital abdominal aortic aneurysm and renal dysplasia.Pediatr Radiol. 2004; 34: 827-830PubMed Google Scholar, 7Kim E.S. Caiati J.M. Tu J. Nowygrod R. Stolar C.J. Congenital abdominal aortic aneurysm causing renovascular hypertension, cardiomyopathy, and death in a 19-day-old neonate.J Pediatr Surg. 2001; 36: 1445-1449Abstract Full Text Full Text PDF PubMed Scopus (26) Google Scholar, 11Malee M.P. Carr S. Rubin L.P. Johal J. Feit L. Prenatal ultrasound diagnosis of abdominal aortic aneurysm with fibrotic occlusion in aortic branch vessels.Prenat Diagn. 1997; 17: 479-482Crossref PubMed Scopus (10) Google Scholar, 16Tsunematsu R. Shinozaki T. Fukushima K. Yumoto Y. Hidaka N. Morokuma S. et al.Congenital abdominal aortic aneurysm with porencephaly: a case report.Fetal Diagn Ther. 2011; 29: 248-252Crossref PubMed Scopus (4) Google Scholar as well as hemodynamic instability from concomitant medical issues.7Kim E.S. Caiati J.M. Tu J. Nowygrod R. Stolar C.J. Congenital abdominal aortic aneurysm causing renovascular hypertension, cardiomyopathy, and death in a 19-day-old neonate.J Pediatr Surg. 2001; 36: 1445-1449Abstract Full Text Full Text PDF PubMed Scopus (26) Google Scholar, 11Malee M.P. Carr S. Rubin L.P. Johal J. Feit L. Prenatal ultrasound diagnosis of abdominal aortic aneurysm with fibrotic occlusion in aortic branch vessels.Prenat Diagn. 1997; 17: 479-482Crossref PubMed Scopus (10) Google Scholar Two patients presented with acute rupture and did not survive operative intervention6Howorth M.B. Aneurysm of abdominal aorta in the newborn infant Report of case.N Engl J Med. 1967; 276: 1133-1134Crossref PubMed Scopus (36) Google Scholar, 9Laing A.J. Hussey A.J. Joyce M.R. Salmo E. Cassidy M. Flynn J. Ruptured abdominal aortic aneurysm in a 12-month-old boy.J Pediatr Surg. 2002; 37: 1234-1235Abstract Full Text Full Text PDF PubMed Scopus (8) Google Scholar (Table). Our patient was hypertensive, and the extent of the aneurysm and small size of the splanchnic vessels made repair impractical. This further highlights that although prenatal diagnosis of AAAs is helpful in identifying patients who may benefit from early operative intervention,4Cantinotti M. Luchi C. Assanta N. Prenatal diagnosis of abdominal aortic aneurysm.Pediatr Cardiol. 2010; 31: 155-156Crossref PubMed Scopus (4) Google Scholar, 8Kim J.I. Lee W. Kim S.J. Seo J.W. Chung J.W. Park J.H. Primary congenital abdominal aortic aneurysm: a case report with perinatal serial follow-up imaging.Pediatr Radiol. 2008; 38: 1249-1252Crossref PubMed Scopus (11) Google Scholar, 11Malee M.P. Carr S. Rubin L.P. Johal J. Feit L. Prenatal ultrasound diagnosis of abdominal aortic aneurysm with fibrotic occlusion in aortic branch vessels.Prenat Diagn. 1997; 17: 479-482Crossref PubMed Scopus (10) Google Scholar, 16Tsunematsu R. Shinozaki T. Fukushima K. Yumoto Y. Hidaka N. Morokuma S. et al.Congenital abdominal aortic aneurysm with porencephaly: a case report.Fetal Diagn Ther. 2011; 29: 248-252Crossref PubMed Scopus (4) Google Scholar vascular anatomy may preclude such intervention.TablePreviously reported cases of congenital abdominal aortic aneurysm in infants and the current patientFirst authorAge at diagnosisMax diameterLocationRenovascular disease?RepairAge at repair or surgeryOutcome(cm)Bell2Bell P. Mantor C. Jacocks M.A. Congenital abdominal aortic aneurysm: a case report.J Vasc Surg. 2003; 38: 190-193Abstract Full Text Full Text PDF PubMed Scopus (23) Google Scholar1 day6.0InfrarenalNoCryopreserved allograft4 monthsUneventful post-op recoveryBuddingh3Buddingh K.T. Zeebregts C.J. Tilanus M.E. Roofthooft M.T. Broens P.M. Large neonatal thoracoabdominal aneurysm: case report and review of the literature.J Pediatr Surg. 2008; 43: 1361-1364Abstract Full Text Full Text PDF PubMed Scopus (9) Google Scholar1 day3.2ThoracoabdominalNoNoneN/AAlive at 8 months, aneurysm grown to 9.3 cm max diamCantinotti4Cantinotti M. Luchi C. Assanta N. Prenatal diagnosis of abdominal aortic aneurysm.Pediatr Cardiol. 2010; 31: 155-156Crossref PubMed Scopus (4) Google Scholar22 weeks' gestation2.6UnspecifiedNoNone reportedN/ANone reportedCheung5Cheung S.C. Khong P.L. Chiu W. Metreweli C. Congenital abdominal aortic aneurysm and renal dysplasia.Pediatr Radiol. 2004; 34: 827-830PubMed Google Scholar6 months3.0JuxtarenalNo, but with renal hypoplasiaNoneN/AAneurysm thrombosed on CT at 3 years, extensive collaterals, progressive renal dysfunctionHoworth6Howorth M.B. Aneurysm of abdominal aorta in the newborn infant Report of case.N Engl J Med. 1967; 276: 1133-1134Crossref PubMed Scopus (36) Google Scholar1 day11.0InfrarenalNoExploratory laparotomy1 dayPresented ruptured, OR deathKim7Kim E.S. Caiati J.M. Tu J. Nowygrod R. Stolar C.J. Congenital abdominal aortic aneurysm causing renovascular hypertension, cardiomyopathy, and death in a 19-day-old neonate.J Pediatr Surg. 2001; 36: 1445-1449Abstract Full Text Full Text PDF PubMed Scopus (26) Google Scholar7 days2.5JuxtarenalYes, renal artery thrombosisNoneNADied of progressive cardiac and renal failure at age 20 daysKim8Kim J.I. Lee W. Kim S.J. Seo J.W. Chung J.W. Park J.H. Primary congenital abdominal aortic aneurysm: a case report with perinatal serial follow-up imaging.Pediatr Radiol. 2008; 38: 1249-1252Crossref PubMed Scopus (11) Google Scholar21 weeks' gestation7.8InfrarenalNoAneurysmectomy, Dacron graft15 monthsUneventful post-op recoveryLaing9Laing A.J. Hussey A.J. Joyce M.R. Salmo E. Cassidy M. Flynn J. Ruptured abdominal aortic aneurysm in a 12-month-old boy.J Pediatr Surg. 2002; 37: 1234-1235Abstract Full Text Full Text PDF PubMed Scopus (8) Google Scholar12 months10.0InfrarenalNoExploratory laparotomy12 monthsPresented ruptured, OR deathLatter10Latter D. Béland M.J. Batten A. Tchervenkov C.I. Dobell A.R. Congenital abdominal aortic aneurysm.Can J Surg. 1989; 32: 135-138PubMed Google Scholar34 weeks' gestation6.0InfrarenalNoPTFE graft1 monthUneventful post-op recoveryMalee11Malee M.P. Carr S. Rubin L.P. Johal J. Feit L. Prenatal ultrasound diagnosis of abdominal aortic aneurysm with fibrotic occlusion in aortic branch vessels.Prenat Diagn. 1997; 17: 479-482Crossref PubMed Scopus (10) Google Scholar32 weeks' gestation3.2JuxtarenalYes, renal artery thrombosis, infarctsNoneN/ADied of acute pulmonary hypertension, acute right heart failure at age 9 daysMalikov12Malikov S. Delarue A. Fais P.O. Keshelava G. Anatomical repair of a congenital aneurysm of the distal abdominal aorta in a newborn.J Vasc Surg. 2009; 50: 1181-1184Abstract Full Text Full Text PDF PubMed Scopus (8) Google Scholar28 weeks' gestation3.3InfrarenalNoRepair using native iliac vessels10 daysUneventful post-op recoveryMehall13Mehall J.R. Saltzman D.A. Chandler J.C. Kidd J.N. Wells T. Smith S.D. Congenital abdominal aortic aneurysm in the infant: case report and review of the literature.J Pediatr Surg. 2001; 36: 657-658Abstract Full Text Full Text PDF PubMed Scopus (22) Google Scholar6 weeks6.0InfrarenalNoGore-TexaW. L. Gore and Associates, Flagstaff, Ariz. graft6 weeksUneventful post-op recoveryMeyers14Meyers R.L. Lowichik A. Kraiss L.W. Hawkins J.A. Aortoiliac reconstruction in infants and toddlers: replacement with decellularized branched pulmonary artery allograft.J Pediatr Surg. 2006; 41: 226-229Abstract Full Text Full Text PDF PubMed Scopus (17) Google ScholarNeonate, NOS3.7InfrarenalNoCryopreserved allograftNeonate NOSUneventful post-op recoverySaad15Saad S.A. May A. Abdominal aortic aneurysm in a neonate.J Pediatr Surg. 1991; 26: 1423-1424Abstract Full Text PDF PubMed Scopus (22) Google Scholar6 weeks4.5InfrarenalNoAneurysmorrhaphy6 weeksUneventful post-op recoveryTsunematsu16Tsunematsu R. Shinozaki T. Fukushima K. Yumoto Y. Hidaka N. Morokuma S. et al.Congenital abdominal aortic aneurysm with porencephaly: a case report.Fetal Diagn Ther. 2011; 29: 248-252Crossref PubMed Scopus (4) Google Scholar25 weeks' gestation2.0UnspecifiedNoNoneN/AStable aneurysm size at 6-month follow-upCurrent case32 weeks' gestation2.3ThoracoabdominalYesNoneN/ADied of rupture at age 28 daysCT, Computed tomography; N/A, not applicable; NOS, not otherwise specified; OR, operating room; PTFE, polytetrafluoroethylene.a W. L. Gore and Associates, Flagstaff, Ariz. Open table in a new tab CT, Computed tomography; N/A, not applicable; NOS, not otherwise specified; OR, operating room; PTFE, polytetrafluoroethylene. Nonoperative management must aim to minimize the risk of aneurysm expansion and rupture. Unfortunately, the discussion of nonoperative approaches in these patients in the literature is even sparser than that addressing operative management. Therapeutic decisions are largely extrapolated from the adult literature. The cornerstone of long-term management in adults is blood pressure control. The logic behind this approach is that aortic wall stress is higher in AAAs that progress to rupture, and that antihypertensive therapy decreases wall stress.17Truijers M. Pol J.A. Schultzekool L.J. van Sterkenburg S.M. Fillinger M.F. Blankensteijn J.D. Wall stress analysis in small asymptomatic, symptomatic and ruptured abdominal aortic aneurysms.Eur J Vasc Endovasc Surg. 2007; 33: 401-407Abstract Full Text Full Text PDF PubMed Scopus (106) Google Scholar, 18Fillinger M.F. Marra S.P. Raghavan M.L. Kennedy F.E. Prediction of rupture risk in abdominal aortic aneurysm during observation: wall stress versus diameter.J Vasc Surg. 2003; 37: 724-732Abstract Full Text Full Text PDF PubMed Scopus (568) Google Scholar Complicating the present case was that the patient had severely impaired renal perfusion contributing to her elevated MAPs. Renovascular hypertension has been reported in two other patients with congenital AAA, both of whom died of concomitant conditions within the first weeks of life.7Kim E.S. Caiati J.M. Tu J. Nowygrod R. Stolar C.J. Congenital abdominal aortic aneurysm causing renovascular hypertension, cardiomyopathy, and death in a 19-day-old neonate.J Pediatr Surg. 2001; 36: 1445-1449Abstract Full Text Full Text PDF PubMed Scopus (26) Google Scholar, 11Malee M.P. Carr S. Rubin L.P. Johal J. Feit L. Prenatal ultrasound diagnosis of abdominal aortic aneurysm with fibrotic occlusion in aortic branch vessels.Prenat Diagn. 1997; 17: 479-482Crossref PubMed Scopus (10) Google Scholar This patient required careful decision making regarding antihypertensive therapy to balance her risk of aneurysm expansion with her renovascular disease. Given the risk of progressive azotemia with angiotensin-converting enzyme inhibitor use in the setting of renal dysfunction,5Cheung S.C. Khong P.L. Chiu W. Metreweli C. Congenital abdominal aortic aneurysm and renal dysplasia.Pediatr Radiol. 2004; 34: 827-830PubMed Google Scholar the patient was started on esmolol and transitioned to isradipine. Calcium channel blockers were used in one other patient with congenital AAA5Cheung S.C. Khong P.L. Chiu W. Metreweli C. Congenital abdominal aortic aneurysm and renal dysplasia.Pediatr Radiol. 2004; 34: 827-830PubMed Google Scholar and showed at least some efficacy in the treatment of AAA in animal models,19Tomita N. Yamasaki K. Izawa K. Kunugiza Y. Osako M.K. Ogihara T. et al.Inhibition of experimental abdominal aortic aneurysm progression by nifedipine.Int J Mol Med. 2008; 21: 239-244PubMed Google Scholar although the benefits of specific drugs in pediatric AAA management has not been investigated. This patient presented with the most extensive continuous congenital AAA yet reported. The case highlights a number of important factors in the management of these aneurysms in neonates, especially with regard to anatomic considerations and hypertension management in patients with concomitant renovascular disease. It further stresses the importance of an individualized approach in treating neonates with this rare entity." @default.
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• W2079457666 date "2012-06-01" @default.
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• W2079457666 title "Extensive congenital abdominal aortic aneurysm and renovascular disease in the neonate" @default.
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