Sävendahl L, Davenport M L
Department of Woman and Child Health, Pediatric Endocrinology Unit, Karolinska Institute, Stockholm, Sweden.
J Pediatr. 2000 Oct;137(4):455-9. doi: 10.1067/mpd.2000.107390.
To measure the delays in diagnosis of Turner's syndrome (TS) and to propose strategies for earlier screening and diagnosis.
The medical records of 81 girls with TS were reviewed for age at diagnosis, reason(s) for karyotype analysis, and clinical features including growth failure. Delay in diagnosis was calculated as equal to age at diagnosis for children born with lymphedema and/or 2 or more of the following dysmorphic features: webbed neck, nail dysplasia, high palate, and short fourth metacarpal. For all others, delay in diagnosis was calculated as the difference between the age at which height fell below the 5th percentile and the age at which the diagnosis of TS was made.
Lymphedema was the key to diagnosis in 97% of the girls diagnosed with TS in infancy, and short stature was the key to diagnosis for 82% of the girls diagnosed in childhood or adolescence. For girls diagnosed in childhood or adolescence, the delay in diagnosis averaged 7.7 +/- 5.4 years. Many had dysmorphic features and/or a history of lymphedema at birth, and diagnosis was made an average of 5.3 years after patients had fallen below the 5th percentile for height. By the time of diagnosis, patients were very short, averaging -2.9 SD in height.
The diagnosis of TS is often delayed. We recommend karyotype analysis for all girls with unexplained short stature, delayed puberty, webbed neck, lymphedema, or coarctation of the aorta. Furthermore, karyotype analysis should be strongly considered for those who remain above the 5th percentile for height but have 2 or more features of TS, including high palate, nail dysplasia, short fourth metacarpal, and strabismus.
测量特纳综合征(TS)的诊断延迟情况,并提出早期筛查和诊断策略。
回顾81例TS女童的病历,记录诊断时的年龄、进行核型分析的原因以及包括生长发育迟缓在内的临床特征。对于出生时伴有淋巴水肿和/或具有以下2种或更多畸形特征的患儿:蹼颈、指甲发育异常、高腭弓和第四掌骨短小,诊断延迟时间等于诊断时的年龄。对于其他所有患儿,诊断延迟时间计算为身高低于第5百分位数的年龄与确诊TS的年龄之差。
淋巴水肿是97%在婴儿期被诊断为TS的女童的诊断关键,身材矮小是82%在儿童期或青春期被诊断的女童的诊断关键。对于在儿童期或青春期被诊断的女童,诊断延迟平均为7.7±5.4年。许多患儿出生时具有畸形特征和/或淋巴水肿病史,在身高低于第5百分位数后平均5.3年才得以诊断。到诊断时,患儿身高非常矮小,平均身高标准差为-2.9。
TS的诊断常常延迟。我们建议对所有原因不明的身材矮小、青春期延迟、蹼颈、淋巴水肿或主动脉缩窄的女童进行核型分析。此外,对于身高仍高于第5百分位数但具有2种或更多TS特征(包括高腭弓、指甲发育异常、第四掌骨短小和斜视)的女童,应强烈考虑进行核型分析。