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Isolated necrotizing aortitis presenting as incidental thoracoabdominal aortic aneurysm: a case report.

ABSTRACT: Noninfectious aortitis is frequently asymptomatic yet often leads to the development of ascending aortic aneurysms requiring repair. We present the case of a 64-year-old Caucasian woman who presented to our medical center with an incidentally discovered thoracoabdominal aneurysm. She had previously been in good health and had not complained of chest pain or been otherwise symptomatic. At presentation to our clinic, her ascending aorta measured 5 cm at the sinotubular junction (STJ) with dilation of her descending thoracic aorta to 6 cm. Her coronary angiogram was normal. Cross-sectional imaging was notable for thickening of the aortic wall along its length. The entity of isolated noninfectious aortitis is increasingly being recognized as an identifiable factor leading to ascending aneurysmal disease. In reviewing this case, we outline current understanding and guidelines for management and follow-up of this pathology. Operative repair of the ascending arch was conducted using an interpositional graft. The aortic valve apparatus was normal and the proximal anastomosis was created at the STJ. Intraoperatively, an inflammatory obliteration of the aortopulmonary window was noted. It was not possible to completely excise the posterior aortic wall. Our patient had an uneventful recovery and final pathology revealed necrotizing aortitis (NA). She is currently undergoing routine surveillance of her descending thoracoabdominal aneurysm. Recent case series indicate that NA is a histologically distinct process, which is associated commonly with development of ascending aneurysm, and although it is most commonly an isolated finding, it may be associated with other vascular abnormalities including stenoses of branch vessels. There is a growing body of literature suggesting that NA represents a distinct clinical entity, associated with the development of ascending aortic aneurysms. Further research is required to determine the optimal follow-up and value of medical therapies.

SUBMITTER: Martin JT

PROVIDER: S-EPMC3331625 | biostudies-other | 2011 Mar

REPOSITORIES: biostudies-other

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Project description:Anomalous right coronary artery from pulmonary artery (ARCAPA) is one of four known anomalies of the coronary arteries originating from the pulmonary artery. ARCAPA is a rare congenital anomaly that is associated with possible long-term complications including myocardial ischemia and left ventricular dysfunction. Clinical presentation can vary from asymptomatic murmur to angina, dyspnea, fatigue, congestive heart failure, myocardial infarction and even cardiac arrest. We present a case of a 52-year-old male who presented to the hospital for intermittent chest pain. The patient was vitally stable and examination was unremarkable. Upon workup, electrocardiogram (EKG) on admission showed no acute ischemic changes and troponin were <0.03 ng/mL. The patient was evaluated by cardiology and had a dobutamine stress echocardiogram (ECHO) for ischemic evaluation. During the recovery phase of dobutamine stress ECHO, the patient reported having 10/10 chest pain with a drop in blood pressure to 90 mmHg systolic. He was subsequently given sublingual nitroglycerin and fluids. Pain did resolve and blood pressure improved after treatment. No wall motion abnormalities were noted on ECHO, but there were inferior ST depressions on EKG during the stress portion of the test. The patient had repeated blood work after the stress test which showed an elevated troponin level of 0.08 ng/mL. The patient was taken to the catheterization lab for diagnostic coronary angiogram. Cardiac catheterization showed a right coronary artery (RCA) arising from the pulmonary artery and diagnosis of isolated ARCAPA was made because of his intermittent chest pain. No intervention was performed as no significant lesions were found. The patient was started on medical therapy including aspirin, beta blocker and statin. The patient did not want to pursue surgery at that point and he did not want any further intervention. He was discharged home with instructions to follow up with cardiology in an outpatient setting to be referred for surgical repair. The purpose of writing this case is to increase awareness among physicians to be able to recognize possible anomalous coronary artery origins during cardiac catheterization and to understand the risk of long-term complications of ARCAPA and need for surgical repair.

Project description:To characterize the inflammatory and coagulopathic response after endovascular thoracoabdominal aortic aneurysm (TAAA) repair and to evaluate the effect of the response on postoperative renal function.From July 2005 to June 2008, 42 patients underwent elective endovascular repair of a TAAA using custom designed multi-branched stent-grafts at a single academic institution. Four patients were excluded from the analysis. White blood cell count (WBC), platelet count, prothrombin time (PT), and creatinine were measured in all patients. In the last nine patients, interleukin-6 (IL-6), protein C, Factor V, d-dimers, cystatin C, and neutrophil gelatinase-associated lipocalin (NGAL) levels were also measured. Change in lab values were expressed as a percentage of baseline values.The 30-day mortality rate was 5% (2/38). All patients (n = 38) had a higher WBC (mean +/- SD: 139 +/- 80%, P < .0001), lower platelet count (56 +/- 15%, P < .0001), and higher PT (median: 17%, Interquartile range (IQR) 12%-22%, P < .0001) after stent-graft insertion. Twelve of 38 patients (32%) developed postoperative acute renal insufficiency (>50% rise in creatinine). Patients with renal insufficiency had significantly larger changes in WBC (178 +/- 100% vs 121 +/- 64%, P = .04) and platelet count (64 +/- 17% vs 52 +/- 12%, P = .02) compared with those without renal insufficiency. All patients (n = 9) had significant increases in NGAL (182 +/- 115%, P = .008) after stent-graft insertion. Six of nine patients (67%) had increased cystatin C (35 +/- 43%, P = .04) after stent-graft insertion, with a greater rise in those with postoperative renal insufficiency (87 +/- 32% vs 8 +/- 13%, P = .02). IL-6 levels were markedly increased in all patients (n = 9) after repair (9840 +/- 6160%, P = .008). Protein C (35 +/- 10%, P = .008) and Factor V levels (28 +/- 20%, P = .008) were uniformly decreased, while d-dimers were elevated after repair in all patients (310 +/- 213%, P = .008).Leukocytosis and thrombocytopenia were uniform following endovascular TAAA repair, and the severity of the response correlated with post-operative renal dysfunction. Elevation of a sensitive marker of renal injury (NGAL) suggests that renal injury may occur in all patients after stent-graft insertion.

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Isolated necrotizing aortitis presenting as incidental thoracoabdominal aortic aneurysm: a case report.

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