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Isolated hypogonadotropic hypogonadism in male identical twins.

IDIOPATHIC HYPOGONADOTROPIC HYPOGONADISM (IHH) is characterized by the failure of normal secondary sex characteristics to develop at the time of expected puberty. Anosmia is a common finding in patients with IHH (Kallmann's syndrome) and in their eugonadal relatives. Several reports have shown varied laboratory and clinical findings in patients with IHH suggesting a heterogeneous disorder.1'2 It is thought to be a genetic disorder, and both X-linked and autosomal inheritance have been postulated.3-5 Sporadic cases have also been reported.6 In patients with IHH, abnormalities of gonadotropin-releasing hormone (GnRH) secretion are discernible. These include a complete absence of gonadotropin secretion or abnormalities in the amplitude and frequency of pulsatile gonadotropin release and altered bioactivity of the secreted gonadotropin.7 There is a paucity of data regarding the sexual behavior of patients with IHH before treatment. The usual recommended treatment, especially for inducing fertility, is the intravenous administration ofhuman chorionic gonadotropin and human menopausal gonadotropin8 or the pulsatile administration ofGnRH.9 Herein we present the first report of sporadic idiopathic hypogonadotropic hypogonadism in identical twin brothers. Surprisingly, spermatogenesis occurred after long-term testosterone treatment in one ofthese patients.