Rappeport J M, Ginns E I
N Engl J Med. 1984 Jul 12;311(2):84-8. doi: 10.1056/NEJM198407123110203.
We performed allogeneic bone-marrow transplantation of normal cells in an eight-year-old patient with Type 3 Gaucher's disease in an attempt to alter his progressive deterioration. The procedure resulted in complete engraftment of the enzymatically normal donor cells. Donor monocyte precursors were present in the circulation of the recipient by 28 days after transplantation. Post-transplantation beta-glucocerebrosidase activity in mononuclear white cells in peripheral blood exceeded normal levels. Despite these early results no change in Gaucher's-cell infiltration of the bone marrow was noted for at least 111 days, and Gaucher's cells persisted in the marrow for at least 274 days. The plasma glucocerebroside concentration ultimately returned to normal. During the post-transplantation course, until the patient died from an episode of sepsis, there was no important change in his clinical status. However, this case demonstrates that the enzymatic abnormality in hematopoietic cells in Gaucher's disease is correctable by bone-marrow transplantation, leading to normalization of plasma levels of glucocerebroside. We observed the long survival of Gaucher's cells, which was consistent with the lack of clinical improvement. Intervention might have been more effective earlier in the course of the disease.
我们对一名患有3型戈谢病的8岁患者进行了正常细胞的异基因骨髓移植,试图改变其病情的逐渐恶化。该手术导致酶活性正常的供体细胞完全植入。移植后28天,受体循环中出现了供体单核细胞前体。移植后外周血单核白细胞中的β-葡萄糖脑苷脂酶活性超过正常水平。尽管有这些早期结果,但至少111天内未观察到骨髓中戈谢细胞浸润的变化,且戈谢细胞在骨髓中持续存在至少274天。血浆葡萄糖脑苷脂浓度最终恢复正常。在移植后的病程中,直到患者死于败血症发作,其临床状况没有重要变化。然而,该病例表明,戈谢病造血细胞中的酶异常可通过骨髓移植得到纠正,从而使血浆葡萄糖脑苷脂水平恢复正常。我们观察到戈谢细胞的长期存活,这与临床改善的缺乏相一致。在疾病过程中更早进行干预可能会更有效。