Ostergaard G Z, Nielsen H, Friis B
Department of Paediatrics, Roskilde County Hospital, Denmark.
Eur J Pediatr. 1992 Apr;151(4):291-4. doi: 10.1007/BF02072232.
We present a patient with Smith-Lemli-Opitz syndrome with immunodeficiency. The patient suffered numerous infectious episodes, atopic dermatitis and wheezing. Immunological investigations demonstrated severely reduced oxidative burst-responsiveness of the blood monocytes, whereas chemotaxis, phagocytosis and interleukin-1 production were normal. Tests of neutrophils and lymphocytes were normal excluding previously described immune deficiency disorders. The father proved to have diminished monocyte oxidative metabolism as well, whereas the mother had normal monocyte function. The genetic and immunological aspects are discussed in relation to the syndrome.
我们报告了一例患有免疫缺陷的史密斯-勒米-奥皮茨综合征患者。该患者经历了多次感染发作、特应性皮炎和喘息。免疫学检查显示血液单核细胞的氧化爆发反应性严重降低,而趋化性、吞噬作用和白细胞介素-1的产生正常。中性粒细胞和淋巴细胞检测正常,排除了先前描述的免疫缺陷疾病。事实证明,父亲的单核细胞氧化代谢也有所减弱,而母亲的单核细胞功能正常。本文结合该综合征对遗传和免疫学方面进行了讨论。