Agbo-kpati K-P, Condor R, Hollenberg H, Chalvon Demersay A, Cuisset L, Quartier P
Service de pédiatrie, centre hospitalier de Marne-La-Vallée, 2-4, cours de La-Gondoire, 77600 Jossigny, France.
Service de pédiatrie, centre hospitalier de Marne-La-Vallée, 2-4, cours de La-Gondoire, 77600 Jossigny, France.
Arch Pediatr. 2014 Jul;21(7):765-7. doi: 10.1016/j.arcped.2014.04.024. Epub 2014 Jun 13.
We report the cases of two sisters born of parents who were first-degree cousins, who started recurrent fever with lymph node and digestive tract involvement at the age of 2 years. There was no mutation of the familial Mediterranean fever gene and a diagnosis of partial mevalonate kinase (MVK) deficiency was made. However, immunoglobulin (Ig) D and A levels were normal. Elevated mevalonic acid in the patients' urine during an episode and MVK gene analysis provided the diagnosis. Clinical remission was obtained under anti-TNF-alpha treatment with etanercept. These observations and those of several previously reported patients, particularly in French and Dutch series, illustrate the importance of considering the diagnosis in a child with early-onset auto-inflammatory syndrome even in the absence of hyper-IgD or -IgA.
我们报告了一对父母为一级亲属的姐妹的病例,她们在2岁时开始反复发热,并伴有淋巴结和消化道受累。家族性地中海热基因无突变,诊断为部分甲羟戊酸激酶(MVK)缺乏症。然而,免疫球蛋白(Ig)D和A水平正常。发作期间患者尿液中甲羟戊酸升高以及MVK基因分析有助于确诊。使用依那西普进行抗TNF-α治疗后获得临床缓解。这些观察结果以及之前报道的一些患者的观察结果,尤其是法国和荷兰系列中的患者,表明即使在没有高IgD或高IgA的情况下,对于患有早发性自身炎症综合征的儿童考虑该诊断的重要性。