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1/32. Solitary renal myofibromatosis: an unusual cause of infantile hypertension.

    INTRODUCTION: Renovascular disease accounts for the vast majority of cases of infantile hypertension with complications resulting from umbilical arterial catheterization predominating in the neonatal period and fibrodysplastic lesions of the renal artery predominating outside the neonatal period. We report a previously undescribed cause of renovascular hypertension: solitary renal myofibromatosis. CASE REPORT: A 9-month-old male infant was transported to the intensive care unit at Children's Hospital in Denver, colorado, for evaluation and treatment of a dilated cardiomyopathy and severe systemic hypertension. The child was full-term with no perinatal problems. Specifically, the child never required umbilical arterial catheterization. He was well until 6 months of age when his parents noted poor weight gain. At 9 months of age, he was evaluated at the referral hospital for failure to thrive. On examination he was noted to have a blood pressure of 170/110 mm Hg, but no other abnormalities. A chest radiograph showed cardiomegaly. Laboratory studies demonstrated normal electrolytes, blood urea nitrogen, and creatinine. However, urinalysis demonstrated 4 protein without red blood cells. An echocardiogram showed severe left ventricular dilatation with an ejection fraction of 16%. On admission the child was noted to be cachectic. His vital signs, including blood pressure, were normal for age. The physical examination was unremarkable. serum electrolytes, blood urea nitrogen, and creatinine were normal. Echocardiographic studies suggested a dilated hypertrophic cardiomyopathy. He was started on digoxin and captopril. Subsequently, he demonstrated episodic hypertension ranging from 170/90 to 220/130 mm Hg. A repeat echocardiogram 24 hours after admission demonstrated a purely hypertrophic cardiomyopathy. verapamil and nifedipine were added to the treatment regimen in an effort to better control the blood pressure without success. urine and blood for catecholamines and plasma renin activity, respectively, were sent and treatment with phentolamine instituted because of a possible pheochromocytoma. A spiral abdominal computerized tomographic scan revealed a markedly abnormal right kidney with linear streaky areas of calcification around the hilum and also an area of nonenhancement in the posterior upper pole. The adrenals and the left kidney were normal. Doppler ultrasound revealed a decrease in right renal arterial flow. The urinary catecholamines were normal and surgery was scheduled after the blood pressure was brought under control by medical treatment. At surgery, tumorous tissue and thrombosis of the renal artery were found in the right upper pole. A right nephrectomy was performed. Pathologic examination of the kidney showed the presence of a diffuse spindle cell proliferation in the interstitium of the kidney. The angiogenic/angiocentric character of the proliferation was demonstrated in several large renal vessels. The lumen of most vessels was narrowed and some vessels were totally occluded with recanalization and dystrophic calcifications observed. Immunostaining of the tumor demonstrated strong desmin and vimentin positivity and minimal actin positivity in the spindle cells. Mitotic activity was not noted in the spindle cell process. These pathologic changes were consistent with a diagnosis of infantile myofibromatosis (IM). The child's preoperative plasma renin activity was 50 712 ng/dL/h (reference range, 235-3700 ng/dL/h). DISCUSSION: The causes of systemic hypertension in infancy are many although renal causes are by far the most common. Renal arterial stenosis or thrombosis accounts for 10% to 24% of cases of infantile hypertension. renal artery thrombosis is usually a consequence of umbilical arterial catheterization, which can also lead to embolization of the renal artery. renal artery stenosis may result from fibrodysplastic lesions (74%), abdominal aortitis (9%), a complication of renal transplantation (5%), and ren
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2/32. Hypertension and pseudoaneurism on the renal artery following retrograde endopyelotomy (Acucise).

    Acucise endopyelotomy has gained widespread use in the treatment of ureteropelvic junction obstruction. Acute postoperative bleeding is a well-known complication. We report one case with a delayed postoperative formation of pseudoaneurism, and one case which developed arterial hypertension postoperatively.
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3/32. Prerenal hypertension caused by a postoperative A-V fistula.

    The A-V fistula in the renal pedicle with functioning renal parenchyma leads to a prerenal hypertension by the blood-steal effect from the renal artery through the fistula into the vein, according the pressure gradient over the fistula. The etiology of these A-V fistulas is pointed out. A case corrected by surgical removal of the fistula is reported.
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4/32. renal artery stenosis and pheochromocytoma: coexistence and treatment.

    PURPOSE: The aim of this study was to develop an optimal treatment plan for coexisting pheochromocytoma and renal artery stenosis. methods: A retrospective analysis of the evaluation and management of a hypertensive 14-year-old boy was conducted. RESULTS: Secondary causes of hypertension were excluded initially including intracranial, cardiac, and endocrine abnormalities. A renal scan showed right renal function at 39%. angiography confirmed a 90% subsegmental stenosis, as well as noting a blush suggesting a tumor. A computerized tomography (CT) scan of the abdomen showed a right adrenal mass. serum epinephrine was 3,006 pg/mL (normal, <1,009 pg/mL) and 24-hour urinary norepinephrine was 2,001 microg (normal, <105 microg/24 h), suggesting the presence of a pheochromocytoma. During the operation for resection, a right subsegmental renal artery was found to be draped around a renal hilar mass; the adrenal gland was normal. The tumor was resected, and pathology confirmed a pheochromocytoma. Postoperatively, the patient had some mild hypertension that gradually resolved. CONCLUSIONS: Extraadrenal pheochromocytomas (paragangliomas) occur more frequently in children and are most commonly located in the renal hilum. In this location, they may present initially as renal artery stenosis as a result of direct arterial compression, fibrous bands, or catecholamine-induced vasospasm. Our experience supports tumor resection of extraadrenal pheochromocytomas as the definitive treatment for both conditions.
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5/32. Midaortic syndrome in childhood associated with a ruptured cerebral aneurysm: a case report.

    BACKGROUND: We present a patient with a midaortic syndrome who presented with subarachnoid hemorrhage caused by rupture of an anterior communicating artery aneurysm.CASE DESCRIPTION: A 14-year-old boy with midaortic syndrome was admitted to our hospital because of subarachnoid hemorrhage due to rupture of an anterior communicating artery aneurysm. He also developed acute renal failure due to previously controlled hypotension. After blood dialysis, successful clipping of the aneurysm was performed. The postoperative course was complicated by malignant renovascular hypertension due to midaortic syndrome. Medical treatment failed to control his hypertension; left primary nephrectomy improved his condition.CONCLUSION: Although midaortic syndrome is rare, it may be significant as a cause of cerebral hemorrhage in childhood.
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6/32. Middle aortic syndrome: distal thoracic and abdominal coarctation, a disorder with multiple etiologies.

    BACKGROUND: Congenital coarctation of the thoracic aorta at the ligamentum arteriosum or the aortic arch is well recognized. But a much less common variety (0.5% to 2.0%) of aortic coarctation is located in the distal thoracic aorta, or abdominal aorta, or both and is often called "middle aortic syndrome" or "mid-aortic dysplastic syndrome." These types of aortic coarctation are most often secondary to a form of granulomatis vasculitis commonly known as Takayasu's disease in this country or aortitis syndrome in japan. No single genesis explains every case and beside vasculitis as a cause, some are thought to be congenital in origin and others are associated with von Recklinghausen's disease. STUDY DESIGN: Eight patients with severe hypertension or claudication secondary to middle aortic coarctation were studied with aortograms and subsequently treated by vascular reconstruction procedures. RESULTS: Vascular reconstructions consisted of aortoaortic bypass, aortic resection with interposed grafting, reanastomotic resection of renal arteries into prosthetic grafts, and renal artery bypass with autogenous material. All eight patients' grafts have remained patent, with followups of 4 to 9 years, with relief of hypertension and claudication. Although Takayasu's disease can be progressive, aggressive surgical treatment in eight patients followed for 4 to 8 years postoperatively demonstrates that severe hypertension, claudication, or both are important indications for revascularization. CONCLUSIONS: Whatever the cause, assuming that active aortic inflammation has been medically treated and is in a burned-out state, patients with abdominal coarctation who have symptomatic renovascular hypertension, claudication, or both are good candidates for revascularization. Although surgical repair is more difficult than with congenital thoracic coarctation, because aortic walls are fibrotic and often also involve the renals, all eight of our patients had successful longterm correction of their hypertension and coarctation.
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7/32. An infant with systemic hypertension, renal artery stenosis, and neuroblastoma.

    A newborn girl with neuroblastoma presented with hypertension (blood pressure 200/140 mm Hg). The concentration of active renin in the ipsilateral renal vein was exceedingly high compared with those in the other venous systems, and angiography results showed narrowing of the contralateral 2 renal arteries. The tumor regressed in size after chemotherapy, but the blood pressure remained high. Percutaneous transluminal angioplasty (PTA) for the left renal arteries was performed twice, the first one at 5 months of age, which achieved some success in the recovery of impaired kidney function. At 8 months of age, she underwent radical resection of the neuroblastoma and removal of the right kidney, and the blood pressure promptly returned to normal postoperatively. The current patient represents the second youngest, well-documented case of renovascular hypertension with neuroblastoma in early infancy.
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8/32. Hyponatremic hypertensive syndrome (HHS) in an 18-month old-child presenting as malignant hypertension: a case report.

    BACKGROUND: The combination of hyponatremia and renovascular hypertension is called hyponatremic hypertensive syndrome (HHS). Malignant hypertension as a presentation has been reported in adults with HHS but is rare in children. CASE PRESENTATION: An eighteen month-old male presented with drowsiness, sudden onset status epilepticus and blood pressure of 210/160. The electrolytes on admission revealed sodium of 120 mEq/L and potassium of 2.1 mEq/L. The peripheral renin activity (PRA) was 172 ng/ml/min (normal 3-11 ng/ml/min) and serum aldosterone level was 91 ng/dl (normal 4 to 16 ng/dl). Patient underwent angioplasty with no success, followed by surgical correction. Two years since the diagnosis, the blood pressure is controlled with labetolol and amlodipine (at less than sixth of the pre-operative dosages). The PRA is 2.4 ng/ml/min and aldosterone 15.5 ng/dl. The child not only had three renal arteries on left but all of them were stenosed which to best of our knowledge has not been described. CONCLUSION: As uncommon as HHS with malignant hypertension may be in adults it is under-reported in children and purpose of the case report is to raise its awareness.
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9/32. Management of severe hypertension in a child with tuberous sclerosis-related major vascular abnormalities.

    OBJECTIVE: We report the case of a 4-year-old girl with tuberous sclerosis who presented with severe renovascular hypertension secondary to compression of the renal arteries by a large infradiaphragmatic abdominal aortic aneurysm. DESIGN: Case report of a previously unreported case. RESULTS: Normotension could not be achieved despite conservative treatment with three antihypertensive drugs at high doses. An aortic graft was performed successfully but failed to correct the hypertension. Postoperatively, the child experienced acute renal failure after attempting treatment with additional enalapril. Further investigations revealed ischaemia of the left kidney and stenosis of the right renal artery. Subsequently, a combination of maximum dose minoxidil, a calcium antagonist, clonidine and peripheral alpha and beta-blockers, and diuretics was used, but her blood pressure control remained poor. A bypass between the hepatic artery and the right renal artery restenosed at the proximal anastomosis. Her renovascular hypertension was finally improved with an autotransplantation of her right kidney into the right iliac fossa. fibromuscular dysplasia and abundant collagen deposition were the predominant features seen on histology. She is currently normotensive on moderate doses of a beta-blocker, amlodipine and a diuretic. CONCLUSION: Hypertension in this case was impossible to control medically, and the surgical approach was compromised by numerous complications. This case illustrates the challenges in the management of renovascular hypertension in a child with disease-associated vascular malformations.
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10/32. Surgery for giant high-flow renal arteriovenous fistula: experience in one institution.

    OBJECTIVE: To present five cases of renal arteriovenous fistula (RAVF) seen at our institution, with an emphasis on surgical treatment. patients AND methods: We assessed five patients with giant high-flow RAVF. The affected kidney was evaluated functionally using (99m)Tc-diethylenetriamine pentaacetic acid renal scintigraphy. The size, location, involvement of branched arteries, and renal function dictated the choice of operative method. Four patients were treated with in situ ligation of the feeding artery for extrarenal RAVF, and the remaining patient had a nephrectomy of the nonfunctioning kidney. The clinical outcome was analysed by renal function, blood pressure control, radiographic cardiothoracic ratio (CTR), occlusion of RAVF, and renal artery patency. RESULTS: Of two patients with hypertension, the blood pressure was normalized in one. The renal function of the affected side was preserved in four patients. CTR was normalized or improved in all four patients with preoperative cardiomegaly. There was no recurrence of RAVF in any of the five patients and there was no major morbidity after surgery. CONCLUSIONS: Surgery for giant high-flow RAVF is safe and feasible from the perspective of complications and recurrence, even though transarterial embolization has become the first line of therapy for RAVF.
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