Genetic analysis of neonatal growth retardation, a lethal syndrome with multiple genes involving X-and Y-linked ones

大多数F_1-Dh/+雄虫，即近交系DDD雌虫与DH-Dh/+雄虫杂交，在新生儿期死亡。受影响的F_1-Dh/+男性表现出典型的生长迟缓症状，伴有排尿和排便问题。这是由直肠和膀胱之间形成的直肠膀胱瘘以及肛门不穿孔引起的。在DH-Dh/+的雌性和DDD的雄性之间的互反杂交中不发生致死表型。致死性基因座的遗传作图分析表明，致死性是三个独立基因座协同作用的结果；即Chr 1上DH位点的DH等位基因，X染色体上Grdhq1位点的DDD等位基因，以及一些自交系中假定的y连锁位点。通过建立Y染色体经济系，我证实了Y连锁基因的承诺。在研究过程中，我注意到Dh对男性生育能力有影响，并获得了一些初步结果，Dh可能与多个y连锁基因相互作用。

Most F_1-Dh/+ males, crosses between inbred DDD strain females and DH-Dh/+ strain males, die during the neonatal period. Affected F_1-Dh/+ males exhibit a typical symptom of growth retardation, with problems of urination and defecation. This is caused by a rectovesical fistula formed between the rectum and the urinary bladder in conjunction with an imperforate anus. The lethal phenotype does not occur in the reciprocal cross between DH-Dh/+ females and DDD males. Genetic mapping analysis of the loci underlying the lethality has revealed that the lethality is a consequence of synergism among three independent gene loci; that is, the DH allele at Dh locus on Chr 1, the DDD allele at the Grdhq1 locus on the X Chr, and a putative Y-linked locus in some inbred strains. By establishing Y chromosome consomic strains, I proved the commitment of Y-linked gene(s). During the course of the study, I noticed that Dh has effects on male fertility, and obtained some preliminary results that Dh may interacts with more than one Y-linked genes.