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纸质出版:2011
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刘征,蒋利萍,刘玮,肖剑文,王世一,朱朝敏,戴碧涛,赵晓东.4例Chediak-Higashi综合征的临床特征和LYST基因分析[J].陆军军医大学学报(原第三军医大学学报),2011,33(17):1838-1842.
Liu Zheng,Jiang Liping,Liu Wei,Xiao Jianwen,Wang Shiyi,Zhu Chaomin,Dai Bitao,Zhao Xiaodong.Clinical features of Chediak-Higashi syndrome and LYST gene in 4 patients[J].J Amry Med Univ (J Third Mil Med Univ),2011,33(17):1838-1842.
刘征,蒋利萍,刘玮,肖剑文,王世一,朱朝敏,戴碧涛,赵晓东.4例Chediak-Higashi综合征的临床特征和LYST基因分析[J].陆军军医大学学报(原第三军医大学学报),2011,33(17):1838-1842. DOI:
Liu Zheng,Jiang Liping,Liu Wei,Xiao Jianwen,Wang Shiyi,Zhu Chaomin,Dai Bitao,Zhao Xiaodong.Clinical features of Chediak-Higashi syndrome and LYST gene in 4 patients[J].J Amry Med Univ (J Third Mil Med Univ),2011,33(17):1838-1842. DOI:
目的 探讨4例Chediak-Higashi综合征(Chediak-Higashi syndrome,CHS)的临床特征和LYST基因突变情况。 方法 分析4例CHS的临床资料和实验室检查结果;提取患儿及父母等亲属外周血DNA和RNA;采用DNA-PCR/RT-PCR直接测序法分析患儿的致病基因LYST;根据患儿突变点对亲属相应位点做PCR扩增和测序,确认携带者。 结果 4例CHS患儿均为女性,平均年龄3岁2个月。均有色素减退,双眼震颤,显微镜见头发黑色素减少为小点状分布。病例1~3还有发热、血细胞2系以上降低、骨髓有核细胞巨大包涵体等特征,诊断Chediak-Higashi综合征(加速期)。病例4年幼,表现不典型,临床诊断CHS。病例1~3存在LYST基因突变,各含2个突变位点,病例1 (c.3113CT,Q980X和c.6788AT,K2205X),病例2 (c.5327CT,R1718X和c.608CT,R145X),病例3(c.5411-5414 del TTTC,L1741fsX1758和c.7975 CT,R2596X)。6个突变均为新型突变。共确认8位携带者。病例4暂未发现LYST基因突变。 结论 Chediak-Higashi综合征具有色素减退、双眼震颤、头发黑色素减少为小点状分布和骨髓有核细胞巨大包涵体的临床特征,通过LYST基因分析,发现其中3例存在6个新型突变点。
Objective To study the clinical features of Chediak-Higashi syndrome (CHS) and lysosomal trafficking regulator (LYST) gene mutations in 4 patients. Methods Clinical and laboratory data about 4 female CHS children were analyzed. DNA and RNA were isolated from peripheral blood of the children and their parents. Pathogenic gene LYST was analyzed by DNA-PCR/RT-PCR and direct sequencing. Corresponding mutation points in the parents were amplified by PCR and sequenced to determine LYST gene mutation carriers according the mutation points in the children. Results The 4 CHS children were females with an average age of 38 months. All of them presented with hypopigmentation accompanying nystagmus. Their melanin was decreased in hair and its distribution was characterized by little spots under microscope. Fever occurred in patients 1-3 with decreased blood cell line 2 and nucleated gigantic inclusion bodies in bone marrow cells
thus CHS was diagnosed. The LYST gene mutations were found at c.3113CT
Q980X and c.6788AT
K2205X in patient 1
at c.5327CT
R1718X and c.608CT
R145X in patient 2
and at c.5411-5414 del TTTC
L1741fsX1758 and c.7975 CT
R2596X in patients 3. The 6 mutations were novel mutations and 8 LYST gene mutation carriers were identified. No LYST gene mutation was found in patient 4. Conclusion CHS is characterized by hypopigmentation
nystagmus
decreased hair melanin in hair with its distribution characterized by small spots
and nucleated gigantic inclusion bodies in bone marrow cells. Six novel LYST gene mutations have been found by LYST gene analysis.
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