Jones J R, Kemmann E
Obstet Gynecol Annu. 1976;5:443-66.
The clinical syndrome of OGD in the female is displayed as hypogonadism and most commonly type I hyposmia ("anosmia"). The main pathologic findings are absence of the olfactory bulbs and tracts, hypoplasia of the hypothalamus, a normal pituitary gland, and normal appearing, although unstimulated, ovaries. The syndrome of OGD may be considered as an attenuated form of holoprosencephaly. Various facial abnormalities have been observed in patients with OGD and their families. Thorough neurologic examination may reveal other abnormalities. In the majority of cases the etiology of OGD is not known; however, among about one-fourth of the females, other members of the family exhibit either OGD or anosmia, implying a genetic basis. In this familial form of OGD some pedigrees suggest an X-chromosomal pattern and others, an autosomal inheritance pattern. Although hypogonadotropic hypogonadism is usually considered the only endocrinologic abnormality, stimulatory test of pituitary and hypothalamic function may reveal poor responses of growth hormone. ACTH, prolactin, and possibly MSH. The administration of LRH has shown varying pituitary gonadotropin responses, implying, in some instances, an associated pituitary malfunction. However, these observations may be the result of variations in technic and, therefore, further data are necessary to clarify this issue. Cyclic estrogen and progestin administration stimulates secondary sexual sex characteristics. Exogenous gonadotropins are capable of stimulating ovarian steroidogenesis and, in most patients, inducing ovulation. Thre pregnancies have been reported.
女性嗅觉丧失-性腺功能减退综合征的临床症状表现为性腺功能减退,最常见的是I型嗅觉减退(“嗅觉丧失”)。主要病理发现为嗅球和嗅束缺如、下丘脑发育不全、垂体正常以及卵巢外观正常但未受刺激。嗅觉丧失-性腺功能减退综合征可被视为全前脑畸形的一种轻型形式。在嗅觉丧失-性腺功能减退综合征患者及其家族中观察到了各种面部异常。全面的神经系统检查可能会发现其他异常。在大多数情况下,嗅觉丧失-性腺功能减退综合征的病因尚不清楚;然而,在约四分之一的女性中,家族中的其他成员表现出嗅觉丧失-性腺功能减退综合征或嗅觉丧失,这意味着存在遗传基础。在这种家族性嗅觉丧失-性腺功能减退综合征中,一些家系提示为X染色体模式,而另一些则提示为常染色体遗传模式。虽然低促性腺激素性性腺功能减退通常被认为是唯一的内分泌异常,但垂体和下丘脑功能的刺激试验可能显示生长激素、促肾上腺皮质激素、催乳素以及可能的促黑素细胞激素反应不佳。促性腺激素释放激素的给药显示垂体促性腺激素反应各异,这在某些情况下意味着存在相关的垂体功能障碍。然而,这些观察结果可能是技术差异的结果,因此需要更多数据来阐明这个问题。周期性给予雌激素和孕激素可刺激第二性征发育。外源性促性腺激素能够刺激卵巢甾体生成,并且在大多数患者中可诱导排卵。已有3例妊娠报道。