Demarchi I, Genoni G, Prodam F, Petri A, Busti A, Cortese L, Negro M, Bellone S, Acucella G, Bona G
Dipartimento di Scienze Mediche, Università del Piemonte Orientale Amedeo Avogadro, Novara, Italia.
Minerva Pediatr. 2011 Aug;63(4):335-9.
Neurofibromatosis type 1 (NF1) is an autosomal dominant disorder characterized by cafe-au-lait spots, axillary and inguinal freckling, cutaneous neurofibromas with a variable clinical expression, iris Lisch nodules, and multiple tumors, in particular optic nerve and other central nervous system gliomas. About 6% of patients develop hypertension due to renovascular diseases, mid-aortic syndrome, or pheochromocytoma. We present a case of a 8 year old girl with primary diagnosis of NF1suffering of skin and encefalic neurofibromas, inguinal freckling, café-au-lait spots, optic nerve glioma, headache, facial flushing. The 24-h ambulatory blood pressure revealed hypertension without paroximal attacks. Urinary metanephrines, serum aldosteron, renin and kalemia were constantly normal. Magnetic resonance imaging (MRI) and angioMRI excluded stenoses of the renal arteries or adrenal masses. Standard 2D echocardiography was normal. The antihypertensive medication controlled pressure values. We concluded for hypertension due to a low-grade vasculopathy. The periodic monitoring of blood pressure in NF1 patients, accompanied by appropriate diagnostic modalities and physical examination, is essential to precociously diagnose hypertension and avoid life-threatening organ damages and increased mortality.
1型神经纤维瘤病(NF1)是一种常染色体显性疾病,其特征为咖啡牛奶斑、腋窝和腹股沟雀斑、临床表现各异的皮肤神经纤维瘤、虹膜Lisch结节以及多种肿瘤,尤其是视神经和其他中枢神经系统胶质瘤。约6%的患者因肾血管疾病、主动脉中段综合征或嗜铬细胞瘤而出现高血压。我们报告一例8岁女孩,初步诊断为NF1,患有皮肤和脑内神经纤维瘤、腹股沟雀斑、咖啡牛奶斑、视神经胶质瘤、头痛和面部潮红。24小时动态血压显示高血压,无阵发性发作。尿间甲肾上腺素、血清醛固酮、肾素和血钾一直正常。磁共振成像(MRI)和血管造影MRI排除了肾动脉狭窄或肾上腺肿块。标准二维超声心动图正常。抗高血压药物控制了血压值。我们得出结论,高血压是由低度血管病变引起的。对NF1患者进行定期血压监测,并辅以适当的诊断方法和体格检查,对于早期诊断高血压、避免危及生命的器官损害和降低死亡率至关重要。