Maruyama K, Ikeda H, Koizumi T, Tsuchida Y
Department of Neonatology, Gunma Children's Medical Center, Japan.
Pediatr Int. 1999 Feb;41(1):82-9. doi: 10.1046/j.1442-200x.1999.01022.x.
Hepatoblastoma in children of very low birthweight (< 1500 g) is increasing in Japan and this has suggested the presence of either a genetic or environmental etiology. This study was aimed at revealing common prenatal and postnatal histories, including family history of hepatoblastomas in children of very low birthweight.
The medical records of 15 patients, nine boys and six girls, were reviewed. The patients were diagnosed at the age of 6-77 months (median 16 months). Their birthweight ranged from 560 to 1380 g (median 826 g) and the gestational age was 23-33 weeks (median 25 weeks). No parents were exposed to any occupational risk factors and there were no characteristic features in the parents' history or the maternal reproductive history, although one patient was born to a mother who had taken a contraceptive before she got pregnant with the patient as a result of in vitro fertilization. A ventricular septal defect and an atresia of the external auditory canal were congenital anomalies seen in the patients, but congenital anomalies associated with hepatoblastoma were not seen. Early postnatal illnesses included respiratory distress syndrome in six patients, symptomatic patent ductus arteriosus in three patients, chronic lung disease in seven patients, cytomegalovirus hepatitis in one patient and cholelithiasis in one patient. Oxygen therapy was given to 13 patients for a period of 4-508 days (median 112 days) and lengths of oxygen therapy and assisted ventilation were significantly longer in patients with a stage IIIB or IV tumor than those with a stage II or IIIA tumor (P = 0.0040 and 0.0190, respectively). Furosemide was used in 13 patients for a period of 6-460 days (median 88 days) and the length of the treatment was also significantly longer in patients with advanced tumors (P = 0.0420). Among the patients at 23-25 weeks of gestation, these treatments tended to be longer in patients with a stage IIIB or IV tumor than those with a stage II or IIIA tumor.
These results suggest the presence of an environmental etiology, rather than a genetic one, which is responsible for the development of hepatoblastoma in children of very low birthweight. Close monitoring of the children after being discharged from the neonatal intensive care unit is essential and a case-control study is necessary to identify risk factors for hepatoblastoma in children of very low birthweight.
在日本，极低出生体重（<1500g）儿童的肝母细胞瘤发病率正在上升，这提示可能存在遗传或环境病因。本研究旨在揭示极低出生体重儿童肝母细胞瘤常见的产前和产后病史，包括家族史。
回顾了15例患者的病历，其中9例男孩，6例女孩。患者诊断时年龄为6 - 77个月（中位年龄16个月）。他们的出生体重在560至1380g之间（中位体重826g），孕周为23 - 33周（中位孕周25周）。父母均未接触任何职业危险因素，父母病史或母亲生殖史也无特征性表现，不过有1例患者的母亲在通过体外受精怀上该患者之前服用过避孕药。患者中可见室间隔缺损和外耳道闭锁等先天性异常，但未见与肝母细胞瘤相关的先天性异常。出生后早期疾病包括6例呼吸窘迫综合征、3例有症状的动脉导管未闭、7例慢性肺病、1例巨细胞病毒性肝炎和1例胆结石。13例患者接受了吸氧治疗，时间为4 - 508天（中位时间112天），且III B期或IV期肿瘤患者的吸氧治疗时间和辅助通气时间显著长于II期或III A期肿瘤患者（P值分别为0.0040和0.0190）。13例患者使用了速尿，时间为6 - 460天（中位时间88天），晚期肿瘤患者的治疗时间也显著更长（P = 0.0420）。在孕周为23 - 25周的患者中，III B期或IV期肿瘤患者的这些治疗时间往往长于II期或III A期肿瘤患者。
这些结果提示，极低出生体重儿童肝母细胞瘤的发生可能是由环境病因而非遗传病因导致的。新生儿重症监护病房出院后对儿童进行密切监测至关重要，且有必要开展病例对照研究以确定极低出生体重儿童肝母细胞瘤的危险因素。
