Renukanthan Aniruthan, Quinton Richard, Turner Benjamin, MacCallum Peter, Seal Leighton, Davies Andrew, Green Richard, Evanson Jane, Korbonits Márta
Department of Endocrinology, Barts and the London School of Medicine and Dentistry, Queen Mary University of London, Charterhouse Square, London, EC1M 6BQ, UK.
Institute of Genetic Medicine, Newcastle University, Newcastle-upon-Tyne, UK.
Endocrine. 2015 Nov;50(2):496-503. doi: 10.1007/s12020-015-0562-5. Epub 2015 Mar 5.
One of the challenging issues in patients with complex problems is that the various diseases and their treatment can influence each other and present unusual hurdles in management. We investigated one such complex case. A 34-year-old XY male presented with azoospermia, detected on semen analysis for pre-orchidectomy sperm banking. He had a 20-year history of gender dysphoria and bilateral breast swelling. The patient suffered a deep vein thrombosis at the age of 19 years. Examination confirmed clinical features of Kallmann syndrome including unilateral cryptorchidism, micropenis, congenital anosmia, and bimanual synkinesis (mirror movements), with reduced serum testosterone and normal gonadotropin levels demonstrating hypogonadotropic hypogonadism. MRI showed missing olfactory bulbs. Osteopenia and reduced vitamin D levels of 21 nmol/L were identified. He was found to harbor a heterozygous factor-V-Leiden mutation. The genetic basis of Kallmann syndrome remains unknown: his screening tests were negative for mutations in CHD7, FGF8, FGFR1, GNRH1, GNRHR, HS6ST1, KAL1, KISS1R, KISS1, NELF, PROK2, PROKR2, TAC3, and TACR3. The patient initially declined testosterone therapy with a view to undergo gender reassignment. Over the next 2 years, the patient experienced recurrent episodes of weakness and paresthesia, associated with classical MRI appearances of multiple sclerosis-related demyelination in the spinal cord and brain. Although it was difficult to elucidate an association between the patient's gender dysphoria and untreated congenital hypogonadism, his desire to become female together with his co-existing thrombophilia, presented challenges to the administration of hormone treatment. Furthermore, we have considered an association between multiple sclerosis and hypogonadotropic hypogonadism.
患有复杂问题的患者面临的一个具有挑战性的问题是,各种疾病及其治疗方法可能相互影响,并在管理中带来异常的障碍。我们调查了这样一个复杂病例。一名34岁的XY男性,在进行睾丸切除术前精子库的精液分析时被检测出无精子症。他有20年的性别焦虑症和双侧乳房肿胀病史。该患者在19岁时曾患深静脉血栓形成。检查证实了卡尔曼综合征的临床特征,包括单侧隐睾、小阴茎、先天性嗅觉缺失和双手联带运动(镜像运动),血清睾酮降低,促性腺激素水平正常,显示为低促性腺激素性性腺功能减退。MRI显示嗅球缺失。发现有骨质减少,维生素D水平降至21 nmol/L。他被发现携带杂合子因子V莱顿突变。卡尔曼综合征的遗传基础仍然未知:他的筛查试验在CHD7、FGF8、FGFR1、GNRH1、GNRHR、HS6ST1、KAL1、KISS1R、KISS1、NELF、PROK2、PROKR2、TAC3和TACR3的突变检测中呈阴性。患者最初拒绝睾酮治疗,希望进行性别重新分配。在接下来的2年里,患者经历了反复发作的虚弱和感觉异常,伴有脊髓和脑部与多发性硬化相关的脱髓鞘的典型MRI表现。尽管很难阐明患者的性别焦虑症与未经治疗的先天性性腺功能减退之间的关联,但他成为女性的愿望以及同时存在的血栓形成倾向,给激素治疗的管理带来了挑战。此外,我们考虑了多发性硬化与低促性腺激素性性腺功能减退之间的关联。