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患有性别焦虑症、多发性硬化症和易栓症的卡尔曼综合征患者。

Kallmann syndrome patient with gender dysphoria, multiple sclerosis, and thrombophilia.

作者信息

Renukanthan Aniruthan, Quinton Richard, Turner Benjamin, MacCallum Peter, Seal Leighton, Davies Andrew, Green Richard, Evanson Jane, Korbonits Márta

机构信息

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.

出版信息

Endocrine. 2015 Nov;50(2):496-503. doi: 10.1007/s12020-015-0562-5. Epub 2015 Mar 5.

DOI:10.1007/s12020-015-0562-5
PMID:25739677
Abstract

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.

摘要

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。

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本文引用的文献

1
Brain changes in Kallmann syndrome.卡尔曼综合征中的脑部变化。
AJNR Am J Neuroradiol. 2014 Sep;35(9):1700-6. doi: 10.3174/ajnr.A3946. Epub 2014 Apr 30.
2
Genetics of congenital hypogonadotropic hypogonadism in Denmark.丹麦先天性低促性腺激素性性腺功能减退的遗传学研究
Eur J Med Genet. 2014 Jul;57(7):345-8. doi: 10.1016/j.ejmg.2014.04.002. Epub 2014 Apr 13.
3
Two years outcome of isolated distal deep vein thrombosis.孤立性远端深静脉血栓形成的两年结局
全外显子组测序在跨性别个体队列中识别罕见遗传变异的应用。
Sci Rep. 2019 Dec 27;9(1):20099. doi: 10.1038/s41598-019-53500-y.
4
ANOS1: a unified nomenclature for Kallmann syndrome 1 gene (KAL1) and anosmin-1.ANO1S:卡尔曼综合征 1 基因(KAL1)和 anosmin-1 的统一命名法。
Brief Funct Genomics. 2017 Jul 1;16(4):205-210. doi: 10.1093/bfgp/elw037.
Thromb Res. 2014 Jul;134(1):36-40. doi: 10.1016/j.thromres.2014.03.033. Epub 2014 Mar 22.
4
Neuroprotective effects of testosterone treatment in men with multiple sclerosis.睾酮治疗对多发性硬化症男性患者的神经保护作用。
Neuroimage Clin. 2014 Mar 6;4:454-60. doi: 10.1016/j.nicl.2014.03.001. eCollection 2014.
5
Reversal and relapse of hypogonadotropic hypogonadism: resilience and fragility of the reproductive neuroendocrine system.低促性腺激素性性腺功能减退症的逆转和复发:生殖神经内分泌系统的弹性和脆弱性。
J Clin Endocrinol Metab. 2014 Mar;99(3):861-70. doi: 10.1210/jc.2013-2809. Epub 2013 Jan 1.
6
Multiple sclerosis.多发性硬化症
Glob Adv Health Med. 2013 Jan;2(1):50-6. doi: 10.7453/gahmj.2013.2.1.009.
7
Incidence and prevalence of multiple sclerosis in the UK 1990-2010: a descriptive study in the General Practice Research Database.1990-2010 年英国多发性硬化症的发病率和患病率:一般实践研究数据库中的描述性研究。
J Neurol Neurosurg Psychiatry. 2014 Jan;85(1):76-84. doi: 10.1136/jnnp-2013-305450. Epub 2013 Sep 19.
8
Olfactory system and demyelination.嗅觉系统与脱髓鞘。
Anat Rec (Hoboken). 2013 Sep;296(9):1424-34. doi: 10.1002/ar.22736. Epub 2013 Jul 31.
9
Mutations in FGF17, IL17RD, DUSP6, SPRY4, and FLRT3 are identified in individuals with congenital hypogonadotropic hypogonadism.在先天性低促性腺激素性性腺功能减退症患者中鉴定出 FGF17、IL17RD、DUSP6、SPRY4 和 FLRT3 的突变。
Am J Hum Genet. 2013 May 2;92(5):725-43. doi: 10.1016/j.ajhg.2013.04.008.
10
Evidence that FGFR1 loss-of-function mutations may cause variable skeletal malformations in patients with Kallmann syndrome.有证据表明,FGFR1 功能丧失性突变可能导致 Kallmann 综合征患者出现多种骨骼畸形。
Adv Med Sci. 2012;57(2):314-21. doi: 10.2478/v10039-012-0036-4.