Quinn Charles T, Rogers Zora R, Buchanan George R
University of Texas Southwestern Medical Center, 5323 Harry Hines Blvd, Dallas, TX 75390, USA.
Blood. 2004 Jun 1;103(11):4023-7. doi: 10.1182/blood-2003-11-3758. Epub 2004 Feb 5.
Contemporary survival data are not available for children with sickle cell disease (SCD). The few previous childhood SCD cohort studies do not reflect the benefits of modern therapy. We defined an inception cohort of newborns with sickle cell anemia (SS), sickle-beta degrees -thalassemia (S beta degrees ), sickle-hemoglobin C disease (SC), or sickle-beta(+)-thalassemia (Sbeta(+)) who were identified by newborn screening and followed for up to 18 years. The incidence of death and stroke were calculated. Overall survival, SCD-related survival (considering only SCD-related deaths), and strokefree survival were determined. The 711 subjects provided 5648 patient-years of observation. Twenty-five subjects died; mean age at death was 5.6 years. Five patients died from infection. Thirty had at least one stroke. Among SS and Sbeta degrees subjects (n = 448), the overall rates of death and stroke were 0.59 and 0.85/100 patient-years. Survival analysis of SS and Sbeta degrees subjects predicted the cumulative overall, SCD-related, and stroke-free survival to be 85.6%, 93.6%, and 88.5% by 18 years of age. No SCD-related deaths or strokes occurred in SC or Sbeta(+) subjects (n = 263). Childhood mortality from SCD is decreasing, the mean age at death is increasing, and a smaller proportion of deaths are from infection.
目前尚无镰状细胞病(SCD)患儿的当代生存数据。此前为数不多的儿童SCD队列研究并未反映出现代疗法的益处。我们定义了一个起始队列,纳入通过新生儿筛查确定的患有镰状细胞贫血(SS)、镰状-β0-地中海贫血(Sβ0)、镰状-血红蛋白C病(SC)或镰状-β+-地中海贫血(Sβ+)的新生儿,并对其进行长达18年的随访。计算死亡和中风的发生率。确定总生存率、SCD相关生存率(仅考虑与SCD相关的死亡)和无中风生存率。711名受试者提供了5648人年的观察数据。25名受试者死亡;死亡时的平均年龄为5.6岁。5名患者死于感染。30名患者至少发生过一次中风。在SS和Sβ0受试者(n = 448)中,死亡和中风的总发生率分别为0.59和0.85/100人年。对SS和Sβ0受试者的生存分析预测,到18岁时,累积总生存率、SCD相关生存率和无中风生存率分别为85.6%、93.6%和88.5%。SC或Sβ+受试者(n = 263)未发生与SCD相关的死亡或中风。SCD导致的儿童死亡率正在下降,死亡的平均年龄在增加,死于感染的比例也在减小。