Briscioli V, Floridia G, Rossi E, Selicorni A, Lalatta F, Zuffardi O
Clinica Pediatrica II De Marchi, Milano, Italy.
J Med Genet. 1993 Jul;30(7):601-3. doi: 10.1136/jmg.30.7.601.
We report a boy with multiple congenital anomalies compatible with trisomy for the distal region of the long arm of chromosome 10 and a male karyotype with one 18p+. In situ hybridisation with a cDNA for ornithine aminotransferase (OAT), whose locus maps to 10q26, confirmed the clinical suspicion of distal trisomy 10q. Subterminal localisation of the labelling signals on chromosome 10 and on the der(18) indicated the localisation of the OAT locus in the proximal part of 10q26. Two clusters of labelling signals were also found on the pericentromeric and proximal portion of the X chromosome short arm, thus confirming the presence in this region of two non-adjacent OAT pseudogenes. The phenotypic similarities of this patient to previously reported cases provide further support for the delineation of trisomy 10qter as a specific, clinically recognisable syndrome.
我们报告了一名患有多种先天性异常的男孩,这些异常与10号染色体长臂远端三体综合征相符,其男性核型为一个18p+。用鸟氨酸转氨酶(OAT)的cDNA进行原位杂交,该基因座定位于10q26,证实了临床对远端10q三体综合征的怀疑。10号染色体和der(18)上标记信号的亚末端定位表明OAT基因座位于10q26的近端部分。在X染色体短臂的着丝粒周围和近端部分也发现了两簇标记信号,从而证实了该区域存在两个不相邻的OAT假基因。该患者与先前报道病例的表型相似性为将10qter三体综合征描述为一种特定的、临床上可识别的综合征提供了进一步支持。