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两兄弟出现小头畸形、身材矮小及发育迟缓,伴有趋化缺陷和短暂性低丙种球蛋白血症。

Microcephaly, short stature, and developmental delay associated with a chemotactic defect and transient hypogammaglobulinaemia in two brothers.

作者信息

Say B, Barber N, Miller G C, Grogg S E

出版信息

J Med Genet. 1986 Aug;23(4):355-9. doi: 10.1136/jmg.23.4.355.

Abstract

Two brothers presented with unusual facial features, microcephaly, developmental delay, and severe postnatal growth retardation. They both developed eczema in infancy and have had recurrent infections. Additional physical findings in both boys included hypogonadism, flexion contractures, hypoplastic patellae, and scoliosis. Their facial similarity was striking with sloping foreheads, beaked noses, large, protruding ears, and micrognathia. Low levels of serum gammaglobulins and defective chemotaxis were present in both boys in infancy. The hypogammaglobulinaemia was transient and improved, reaching normal levels by 3 1/2 years and 15 months, respectively. Defective chemotaxis and recurrent infections have persisted to the present. Both parents were normal. The mode of inheritance was not clear, as both X linked and autosomal recessive patterns were possible. Although patients with congenital malformations who also had immunodeficiency have previously been reported, immune system abnormalities, especially those of a transient nature, may frequently go unrecognised.

摘要

两兄弟表现出异常的面部特征、小头畸形、发育迟缓以及严重的出生后生长发育迟缓。他们在婴儿期均患湿疹且反复感染。两个男孩的其他体格检查发现还包括性腺功能减退、屈曲挛缩、髌骨发育不全和脊柱侧弯。他们面部极为相似,前额倾斜、鼻子呈喙状、耳朵大且突出、下颌后缩。两个男孩在婴儿期血清γ球蛋白水平低且趋化性缺陷。低丙种球蛋白血症是暂时的,分别在3岁半和15个月时改善至正常水平。趋化性缺陷和反复感染一直持续至今。父母双方均正常。遗传模式不明确,因为X连锁和常染色体隐性模式均有可能。虽然此前曾报道过患有先天性畸形且伴有免疫缺陷的患者,但免疫系统异常,尤其是那些具有暂时性的异常,可能常常未被识别。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9f5d/1049705/d739870e16b6/jmedgene00090-0068-a.jpg

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