Yavuz Halûk, Ozel Ahmet, Christensen Mette, Christensen Ernst, Schwartz Marianne, Elmaci Mithat, Vissing John
Department of Pediatrics, Meram Medical Faculty, Selcuk University, Konya, Turkey.
Arch Neurol. 2007 Mar;64(3):435-8. doi: 10.1001/archneur.64.3.435.
To study the effect of continuous ambulatory peritoneal dialysis on nucleoside levels and clinical course in a patient with mitochondrial neurogastrointestinal encephalomyopathy (MNGIE). Patient We studied a patient with genetically verified MNGIE, who prior to treatment had lost weight progressively, developed amenorrhea, vomited multiple times daily, and had abdominal pain. Intervention The patient was treated with peritoneal dialysis for 3 years, and the effect on symptoms and plasma concentrations of thymidine and deoxyuridine were monitored.
Dialysis stopped vomiting and reduced abdominal pain, and the patient gained 5 kg in weight and started to menstruate again. Symptoms returned if dialysis was paused. Dialysis did not affect plasma nucleoside levels.
This study shows an unambiguous clinical benefit of peritoneal dialysis on gastrointestinal symptoms in MNGIE. Dialysis did not affect nucleoside levels, indicating elevated thymidine and deoxyuridine levels are not solely responsible for the pathogenesis of MNGIE.
研究持续性非卧床腹膜透析对线粒体神经胃肠性脑肌病(MNGIE)患者核苷水平及临床病程的影响。
我们研究了一名经基因验证为MNGIE的患者,该患者在治疗前体重逐渐减轻,出现闭经,每天呕吐多次,并伴有腹痛。
该患者接受了3年的腹膜透析治疗,并监测了其对症状以及血浆胸苷和脱氧尿苷浓度的影响。
透析停止了呕吐并减轻了腹痛,患者体重增加了5千克,月经也再次来潮。如果暂停透析,症状会再次出现。透析未影响血浆核苷水平。
本研究表明腹膜透析对MNGIE患者的胃肠道症状具有明确的临床益处。透析未影响核苷水平,表明胸苷和脱氧尿苷水平升高并非MNGIE发病机制的唯一原因。