Saima S, Suzuki Y, Yoshimoto K, Nakamura Y, Munakata A
International Medical Center, Division of Nephrology, Tokyo, Japan.
Nihon Jinzo Gakkai Shi. 1996 Jun;38(6):278-84.
As long-term survival has become possible in patients with autosomal dominant polycystic kidney disease (ADPKD) with progress in hemodialysis (HD), complications by various extrarenal diseases has presented new problems. Recent experience of two rare cases of ADPKD ending fatally due to complications by polycystic liver is presented. Case 1: A 60-year-old female with a family history of ADPKD without a past history of liver disease, was diagnosed as ADPKD at the age of 45 years. Hemodialysis was started at the age of 58 years. From 6 months prior to her death, abdominal circumference increased (body height: 149 cm, abdominal circumference: 100 cm). Dyspnea, abdominal pain and anorexia appeared and she died of hepatic failure leading to cachexia. Case 2: A 76-year-old female with a family history of ADPKD without a past history of liver disease, was started on HD at the age of 73 years. Abdominal circumference was 84 cm (body height: 138 cm). She was repeatedly admitted to and discharged from the hospital due to febrile episodes. Infection of polycystic liver was complicated by DIC and she died of gastrointestinal hemorrhage. Autopsy revealed abscess in some of the cysts in the liver. Hepatic cysts most frequently complicating ADPKD so far have presented with scarcely any clinical problems. Recently, however, cases of infection of hepatic cysts, portal hypertension and hepatic insufficiency have been reported. The relationship between these hepatic diseases and the prognosis of ADPKD has received attention. Increase in the number of cases of complications similar to the present cases is anticipated.
随着血液透析(HD)技术的进步,常染色体显性遗传性多囊肾病(ADPKD)患者实现长期存活成为可能,各种肾外疾病引发的并发症带来了新问题。本文介绍了两例罕见的因多囊肝并发症导致死亡的ADPKD病例。病例1:一名60岁女性,有ADPKD家族史,无肝脏疾病史,45岁时被诊断为ADPKD。58岁开始血液透析。在她去世前6个月,腹围增大(身高149cm,腹围100cm)。出现呼吸困难、腹痛和厌食,最终死于肝衰竭导致的恶病质。病例2:一名76岁女性,有ADPKD家族史,无肝脏疾病史,73岁开始血液透析。腹围84cm(身高138cm)。因发热反复住院和出院。多囊肝感染并发弥散性血管内凝血(DIC),死于胃肠道出血。尸检发现肝脏部分囊肿有脓肿。迄今为止,ADPKD最常见的肝囊肿并发症几乎没有任何临床问题。然而,最近有肝囊肿感染、门静脉高压和肝功能不全的病例报道。这些肝脏疾病与ADPKD预后之间的关系受到关注。预计类似本文病例的并发症病例数将会增加。