Behbehani A W, Gahr M, Schröter W
Monatsschr Kinderheilkd. 1983 Nov;131(11):784-7.
We report the first case of lysinuric protein intolerance observed in Germany. A girl of Turkish ancestry suffered from severe dehydration at the age of 6 months after changing from breast milk to cow milk. Because of a microcytic hypochromic anaemia and erythrophagocytosis in the bone marrow a hematologic disease (Farquar's disease) was suspected. The definite diagnosis of lysinuric protein intolerance was eventually clarified by the following laboratory and clinical data: increased urinary excretion and low plasma concentration of lysine, arginine and ornithine, apathy, vomiting, diarrhea and hyperammonemia after an oral protein load, high serum LDH-activity, ferritinemia, and increased urinary excretion of orotic acid. Under therapy with citrullin and a low-protein diet the metabolic situation remained stable, even during infections. The bone marrow findings have been reported only in one further case of lysinuric protein intolerance.
我们报告了在德国观察到的首例赖氨酸尿性蛋白不耐受病例。一名具有土耳其血统的女孩在6个月大时从母乳喂养改为牛奶喂养后出现严重脱水。由于存在小细胞低色素性贫血以及骨髓中的红细胞吞噬现象,怀疑患有血液系统疾病(法夸尔病)。赖氨酸尿性蛋白不耐受的明确诊断最终通过以下实验室和临床数据得以明确:赖氨酸、精氨酸和鸟氨酸的尿排泄增加以及血浆浓度降低,口服蛋白质负荷后出现冷漠、呕吐、腹泻和高氨血症,血清乳酸脱氢酶活性升高、铁蛋白血症以及乳清酸尿排泄增加。在使用瓜氨酸和低蛋白饮食治疗期间,即使在感染期间,代谢状况也保持稳定。骨髓检查结果仅在另外一例赖氨酸尿性蛋白不耐受病例中被报道过。