Noordzij J G, Wulffraat N M, Haraldsson A, Meyts I, van't Veer L J, Hogervorst F B L, Warris A, Weemaes C M R
Department of Paediatrics, Reinier de Graaf Gasthuis, Delft, The Netherlands.
Arch Dis Child. 2009 Jun;94(6):448-9. doi: 10.1136/adc.2008.149351. Epub 2009 Feb 17.
Ataxia-telangiectasia (A-T) is characterised by progressive neurological abnormalities, oculocutaneous telangiectasias and immunodeficiency (decreased serum IgG subclass and/or IgA levels and lymphopenia). However, 10% of A-T patients present with decreased serum IgG and IgA with normal or raised IgM levels. As cerebellar ataxia and oculocutaneous telangiectasias are not present at very young age, these patients are often erroneously diagnosed as hyper IgM syndrome (HIGM). Eight patients with A-T, showing serum Ig levels suggestive of HIGM on first presentation, are described. All had decreased numbers of T lymphocytes, unusual in HIGM. The diagnosis A-T was confirmed by raised alpha-fetoprotein levels in all patients. To prevent mistaking A-T patients for HIGM it is proposed to add DNA repair disorders as a possible cause of HIGM.
共济失调毛细血管扩张症(A-T)的特征为进行性神经功能异常、眼皮肤毛细血管扩张和免疫缺陷(血清IgG亚类和/或IgA水平降低以及淋巴细胞减少)。然而,10%的A-T患者表现为血清IgG和IgA降低,IgM水平正常或升高。由于小脑共济失调和眼皮肤毛细血管扩张在非常年幼时并不存在,这些患者常被误诊为高IgM综合征(HIGM)。本文描述了8例A-T患者,他们首次就诊时血清Ig水平提示为HIGM。所有患者的T淋巴细胞数量均减少,这在HIGM中并不常见。所有患者甲胎蛋白水平升高,从而确诊为A-T。为防止将A-T患者误诊为HIGM,建议将DNA修复障碍作为HIGM的一种可能病因。