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        "resumen" => "Hypertension affect about 1&#37; of patients with neurofibromatosis type 1 &#40;NF1&#41;&#46; Major causes are concomitant pheocromocytoma in adults and renovascular hypertension in children&#46; In most cases&#44; NF1 is associated with renal artery stenosis&#44; smooth cell proliferation and advential fibrosis&#46; We describe a 16 year old girl with hypertension complicating NF1 secondary to severe coarctation of abdominal aorta and tight stenosis of right renal artery&#44; a very uncommon case&#46; She was first diagnosed when she was 3-years-old and managed with antihypertensive drugs &#40;atenolol&#44; hidralazine and nifedipine&#41;&#59; she expirienced progressive uncontrollable hypertension but no symptoms&#44; thus she was admitted to repeat studies&#46; Laboratory evaluation &#40;including creatinine&#44; serum electrolites&#44; urinalysis&#44; urine catecholamines and creatinine clearance&#41; was normal Percutanaous transfemoral magnetic resonance angiography disclosed severe coarctation of abdomina aorta&#44; functional oclussion of superior mesenteric artery and tight stenosis of right renal artery with postestenotic dilatation&#46; Patient underwent surgery with aorto-aortic by-pass and right kidney artery reimplantation&#46; Periodical controls confirmed no hypertension&#44; even four years after surgery and normal flow patterns in Doppler ultrasonography&#46; Patients with NF1 must be screened for pheochromoctyoma and renovascular hypertension&#46; If hypertension appears&#44; careful management is mandatory&#44; as periodical follow-up even after surgey&#44; since the long-term recurrence rate of renovascular lessions is not well established&#46;"
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High blood pressure due to aortic coarctation and renal artery stenosis in a teenager with type 1 neurofibromatosis
Hipertensión secundaria a coartación de aorta y estenosis de arteria renal en adolescente con neurofibromatosis tipo 1
R.. Pardoa, L.. Somaloa, Serafín Málaga Guerreroa, F.. Santosa
a Sección de Nefrología Pediátrica. Departamento de Pediatría, Hospital Universitario Central de Asturias, Asturias, Asturias, España,
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    "textoCompleto" => "<p class="elsevierStylePara"><span class="elsevierStyleBold">INTRODUCTION</span></p><p class="elsevierStylePara">The incidence of high blood pressure in patients with NF1 is around 1&#37;&#44;<span class="elsevierStyleSup">1</span> mainly associated to pheochromocytomas in adulthood&#46; During childhood&#44; most cases are secondary to renal artery stenosis and rarely to coarctation of the abdominal aorta<span class="elsevierStyleSup">2</span>&#46;</p><p class="elsevierStylePara">We present a 16-year old female with NF1&#44; who had been controlled since she was 3 year old because of hypertension secondary to abdominal coarctation&#44; which was angiographically confirmed&#46; She had no symptoms but the blood pressure could not be controlled with drugs &#40;atenolol&#44; hydralazine&#44; nifedipine&#41;&#46;</p><p class="elsevierStylePara">On physical exam multiple &#171;caf&#233; au lait&#187; spots could be seen&#44; as well as freckles in axillary areas&#44; with no cutaneous neurofibromas&#46; A murmur could be heard on right superior quadrant of the abdomen&#44; and peripheral pulses were palpable and symmetrical&#46; Neurological examination was normal&#46; During the follow-up&#44; the blood pressure measurements were between 150&#47;110 and 140&#47;100 mmHg&#46; These values were always over the 95th percentile for sex&#44; age and height&#46;</p><p class="elsevierStylePara">Further investigations&#58; renal function and urine catecholamines levels were within the normal range&#46; The funduscopic examination revealed grade I hypertensive retinopathy&#46; The echocardiography showed mild left ventricle hypertrophy&#46; The cranial MRI disclosed a glioma involving the optic chiasm and nerve&#46; An ambulatory blood pressure monitoring was performed&#58; During the day high blood pressure was detected with nocturnal dip&#46; On renal Doppler ultrasound the renal flow was diminished in the right kidney&#44; which suggested renal artery stenosis&#46; The abdominal angioresonance showed severe coarctation of the abdominal aorta with a lumen reduction of 85&#37; and right renal artery stenosis&#46; The same could be seen on percutaneous femoral angiography &#40;fig&#46; 1&#41;&#46;</p><p class="elsevierStylePara">With these findings surgical intervention was decided consisting on aorto-aortic bypass and right renal artery reimplantation&#46; A control angiography was performed that confirmed the reestablishment of the circulatory pattern &#40;fig&#46; 2&#41;&#46; Pharmacological therapy was required associated to oral nifedipine during four months&#46; Four years after the intervention renal function was normal&#44; Doppler ultrasounds performed during the follow-up showed preserved flows&#44; and the arterial pressure was normal&#46;</p><p class="elsevierStylePara">The frequency of high blood pressure in patients with NF1 is around 1&#37; and can be due to renal artery stenosis&#44; pheochromocytomas&#44; coarctation of the abdominal aorta&#44; or cerebral tumors&#46;<span class="elsevierStyleSup">1</span> During childhood&#44; the most frequent cause is renal artery stenosis&#44; while in adults it is pheochromocytoma&#46;<span class="elsevierStyleSup">3</span> The association between renal artery stenosis and aortic coarctation in patients with neurofibromatosis is exceptional<span class="elsevierStyleSup">4</span>&#46; In fact&#44; there are approximately ten cases reported in the literature&#46;<span class="elsevierStyleSup">5-7</span></p><p class="elsevierStylePara">In spite of the diagnostic advances &#40;ABPM&#44; renal Doppler ultrasound&#44; angio-MRI&#41; conventional angiography still is the main diagnostic investigation&#44; especially in those cases that will be susceptible to surgical therapy&#46; The management of these patients must be individualized&#44; according to the underlying condition and age&#46; The surgical treatment is successful in 80&#37; of the cases&#46; For this reason it is currently the preferred therapeutic option&#44; and also taking into account that in children the results of the angioplasty are not satisfactory&#46;<span class="elsevierStyleSup">8</span> Anyway&#44; the follow-up of the patients in the intermediate and long term is very important&#44; as we do not know the relapse rate of these lesions&#46; <br></br></p>"
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        "resumen" => "La frecuencia estimada de la hipertensi&#243;n arterial en pacientes con neurofibromatosis tipo 1 &#40;NF1&#41; es de aproximadamente un 1&#37;&#44; habitualmente secundaria a feocromocitomas y estenosis de las arterias renales&#46; La coartaci&#243;n de aorta asociada en estos pacientes es una causa infrecuente de hipertensi&#243;n&#46; Presentamos el caso de una paciente con NF1 que padece hipertensi&#243;n desde los 3 a&#241;os de vida&#44; mal controlada con tratamiento farmacol&#243;gico&#46; Antecedentes familiares de NF1 &#40;madre&#41;&#46; A la exploraci&#243;n f&#237;sica presentaba m&#250;ltiples manchas caf&#233; con leche&#44; ef&#233;lides axilares y presi&#243;n arterial por encima de percentil 95 para edad y altura&#46; Las pruebas de laboratorio &#40;creatinina s&#233;rica&#44; electrolitos&#44; catecolaminas urinarias y aclaramiento de creatinina&#41; fueron normales&#46; La monitorizaci&#243;n ambulatoria de presi&#243;n arterial revel&#243; hipertensi&#243;n diurna con valores nocturnos normales&#46; La ecograf&#237;a Doppler renal mostr&#243; un patr&#243;n anormal de flujo en arteria renal derecha compatible con estenosis y la angiorresonancia magn&#233;tica&#58; coartaci&#243;n severa de aorta abdominal&#44; con oclusi&#243;n funcional de arteria mesent&#233;rica superior y estenosis moderada de arteria renal derecha con dilataci&#243;n postesten&#243;tica&#46; Ante estos hallazgos&#44; la paciente fue intervenida quir&#250;rgicamente&#44; con realizaci&#243;n de by-pass aorto-a&#243;rtico y reimplantaci&#243;n de arteria renal derecha&#46; Durante los cuatro meses siguientes a la intervenci&#243;n recibi&#243; tratamiento hipotensor con nifedipino oral que se suspendi&#243; ante la buena evoluci&#243;n cl&#237;nica&#46; Los controles peri&#243;dicos en la consulta durante los cuatro a&#241;os siguientes a la cirug&#237;a fueron satisfactorios&#44; con presi&#243;n arterial normal y funci&#243;n renal conservada&#46; Comentarios&#58; El control de la presi&#243;n arterial en pacientes con NF1 es aconsejable para detectar casos de hipertensi&#243;n arterial que&#44; en muchas ocasiones&#44; va a requerir tratamiento quir&#250;rgico&#46;"
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        "resumen" => "Hypertension affect about 1&#37; of patients with neurofibromatosis type 1 &#40;NF1&#41;&#46; Major causes are concomitant pheocromocytoma in adults and renovascular hypertension in children&#46; In most cases&#44; NF1 is associated with renal artery stenosis&#44; smooth cell proliferation and advential fibrosis&#46; We describe a 16 year old girl with hypertension complicating NF1 secondary to severe coarctation of abdominal aorta and tight stenosis of right renal artery&#44; a very uncommon case&#46; She was first diagnosed when she was 3-years-old and managed with antihypertensive drugs &#40;atenolol&#44; hidralazine and nifedipine&#41;&#59; she expirienced progressive uncontrollable hypertension but no symptoms&#44; thus she was admitted to repeat studies&#46; Laboratory evaluation &#40;including creatinine&#44; serum electrolites&#44; urinalysis&#44; urine catecholamines and creatinine clearance&#41; was normal Percutanaous transfemoral magnetic resonance angiography disclosed severe coarctation of abdomina aorta&#44; functional oclussion of superior mesenteric artery and tight stenosis of right renal artery with postestenotic dilatation&#46; Patient underwent surgery with aorto-aortic by-pass and right kidney artery reimplantation&#46; Periodical controls confirmed no hypertension&#44; even four years after surgery and normal flow patterns in Doppler ultrasonography&#46; Patients with NF1 must be screened for pheochromoctyoma and renovascular hypertension&#46; If hypertension appears&#44; careful management is mandatory&#44; as periodical follow-up even after surgey&#44; since the long-term recurrence rate of renovascular lessions is not well established&#46;"
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                0 => array:3 [
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                  "referenciaCompleta" => "Westenend PJ, Smedts F, de Jong MC, Lommers EJ, Assmann KJ. A 4- year old boy with neurofibromatosis and severe renovascular hypertension due to renal arterial dysplasia. Am J Surg Pathol 1994; 18: 512-6. <a href="http://www.ncbi.nlm.nih.gov/pubmed/8172325" target="_blank">[Pubmed]</a>"
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Article information
ISSN: 20132514
Original language: English
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