Hashimoto H, Maruyama H, Koshida R, Okuda N, Murayama K, Katsumi T, Watanabe K, Sato T
Department of Pediatrics, Kanazawa National Hospital, Japan.
Arch Dis Child. 1997 Mar;76(3):268-71. doi: 10.1136/adc.76.3.268.
A case is reported of dysgenetic male pseudohermaphroditism (DMPH) having Turner stigmata and 45,X/46,X+mar karyotype. The marker chromosome of this patient consisted of most if not all of the short arm, including the sex determining region of the Y chromosome. Although this karyotype is relatively common in Turner's syndrome and occasionally observed in mixed gonadal dysgenesis, DMPH is usually exemplified by a 46,XY karyotype except for one patient reported with 45,X/46,XY mosaicism. Turner stigmata have not previously been reported in DMPH. The present patient is an intermediate case between mixed gonodal dysgenesis and typical DMPH, and this indicates that 45,X/ 46,X +mar karyotype abnormality can result in a wide range of phenotype such as DMPH, mixed gonodal dysgenesis and Turner's syndrome.
报告了一例具有特纳氏体征和45,X/46,X+mar核型的发育不全性男性假两性畸形(DMPH)病例。该患者的标记染色体由大部分(如果不是全部)短臂组成,包括Y染色体的性别决定区域。虽然这种核型在特纳综合征中相对常见,在混合性性腺发育不全中偶尔也会观察到,但除了一例报告为45,X/46,XY嵌合体的患者外,DMPH通常以46,XY核型为典型。此前尚未有在DMPH中出现特纳氏体征的报道。该患者是混合性性腺发育不全和典型DMPH之间的中间病例,这表明45,X/46,X +mar核型异常可导致广泛的表型,如DMPH、混合性性腺发育不全和特纳综合征。