{"title":"11名男孩在婴儿期接受重组LH和FSH持续皮下输注(CSGI)治疗的青春期早期评估。","authors":"Anne-Sophie Lambert, Trouvin Marie-Agathe, Khadidja Fouatih, Adrien Lecoeuvre, Cecile Thomas-Teinturier, Anya Rothenbuhler, Jerome Bouligand, Séverine Trabado, Agnès Linglart, Claire Bouvattier","doi":"10.1530/EC-24-0082","DOIUrl":null,"url":null,"abstract":"<p><strong>Introduction: </strong>We previously observed that continuous subcutaneous gonadotropin infusion (CSGI) in infants with congenital hypogonadotropic hypogonadism (CHH) can mimic minipuberty.</p><p><strong>Objective: </strong>to describe the early adolescence outcome of boys treated during the first year of life.</p><p><strong>Methods: </strong>In this retrospective cohort study, we describe 11 CHH boys aged 12 years [11.5-14.6] treated at the age of 4.5 months [2.0-11] with CSGI. To caompare we report testicular function of 12 untreated CHH boys aged 12 years [12-15.9].</p><p><strong>Results: </strong>In response to CSGI, serum testosterone and inhibinB levels increased from 0.03 ng/mL [0-0.07] to 2.25 ng/mL [1.12-3.86] and from 73 [11-173] to 401 [185-727] pg/mL, respectively. Testicular volume increased from 0.50 mL [0.5-1] to 1.50 mL [0.7-3]. Between end of CSGI and early adolescence, testicular volume in the treated group decreased from 1.5 mL [0.7-3] to 1.05 ml [0.7-2.36] (p=0.024) and differed from that in untreated boys (0.3 mL [0.13-1.3]). Hormone levels were higher in the treated group : serum AMH and inhibin B levels in treated patients decreased from 1028 pmol/l [550-1750] and 356 [185-727] pg/mL at neonatal period to 331 pmol/l [85-479] and 68 pg/ml [19-239] respectively at early adolescence and differed from those in untreated patients (57.5 [30-169] and 8 pg/ml [<5-37] (p<0.001)).</p><p><strong>Conclusion: </strong>We report the first long-term follow-up of CHH boys treated with CSGI in infancy. Our results shown that the CSGI treatment resulted in higher inhibin B, AMH levels and testicular volume at early adolescence age. Follow-up should be continued until the end of puberty to assess spermatogenesis.</p>","PeriodicalId":11634,"journal":{"name":"Endocrine Connections","volume":" ","pages":""},"PeriodicalIF":2.6000,"publicationDate":"2025-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11915493/pdf/","citationCount":"0","resultStr":"{\"title\":\"Early adolescence assessment of eleven boys treated with continuous subcutaneous infusion (CSGI) of recombinant LH and FSH during infancy.\",\"authors\":\"Anne-Sophie Lambert, Trouvin Marie-Agathe, Khadidja Fouatih, Adrien Lecoeuvre, Cecile Thomas-Teinturier, Anya Rothenbuhler, Jerome Bouligand, Séverine Trabado, Agnès Linglart, Claire Bouvattier\",\"doi\":\"10.1530/EC-24-0082\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><strong>Introduction: </strong>We previously observed that continuous subcutaneous gonadotropin infusion (CSGI) in infants with congenital hypogonadotropic hypogonadism (CHH) can mimic minipuberty.</p><p><strong>Objective: </strong>to describe the early adolescence outcome of boys treated during the first year of life.</p><p><strong>Methods: </strong>In this retrospective cohort study, we describe 11 CHH boys aged 12 years [11.5-14.6] treated at the age of 4.5 months [2.0-11] with CSGI. To caompare we report testicular function of 12 untreated CHH boys aged 12 years [12-15.9].</p><p><strong>Results: </strong>In response to CSGI, serum testosterone and inhibinB levels increased from 0.03 ng/mL [0-0.07] to 2.25 ng/mL [1.12-3.86] and from 73 [11-173] to 401 [185-727] pg/mL, respectively. Testicular volume increased from 0.50 mL [0.5-1] to 1.50 mL [0.7-3]. Between end of CSGI and early adolescence, testicular volume in the treated group decreased from 1.5 mL [0.7-3] to 1.05 ml [0.7-2.36] (p=0.024) and differed from that in untreated boys (0.3 mL [0.13-1.3]). Hormone levels were higher in the treated group : serum AMH and inhibin B levels in treated patients decreased from 1028 pmol/l [550-1750] and 356 [185-727] pg/mL at neonatal period to 331 pmol/l [85-479] and 68 pg/ml [19-239] respectively at early adolescence and differed from those in untreated patients (57.5 [30-169] and 8 pg/ml [<5-37] (p<0.001)).</p><p><strong>Conclusion: </strong>We report the first long-term follow-up of CHH boys treated with CSGI in infancy. Our results shown that the CSGI treatment resulted in higher inhibin B, AMH levels and testicular volume at early adolescence age. Follow-up should be continued until the end of puberty to assess spermatogenesis.</p>\",\"PeriodicalId\":11634,\"journal\":{\"name\":\"Endocrine Connections\",\"volume\":\" \",\"pages\":\"\"},\"PeriodicalIF\":2.6000,\"publicationDate\":\"2025-02-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11915493/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Endocrine Connections\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1530/EC-24-0082\",\"RegionNum\":3,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q3\",\"JCRName\":\"ENDOCRINOLOGY & METABOLISM\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Endocrine Connections","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1530/EC-24-0082","RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"ENDOCRINOLOGY & METABOLISM","Score":null,"Total":0}
Early adolescence assessment of eleven boys treated with continuous subcutaneous infusion (CSGI) of recombinant LH and FSH during infancy.
Introduction: We previously observed that continuous subcutaneous gonadotropin infusion (CSGI) in infants with congenital hypogonadotropic hypogonadism (CHH) can mimic minipuberty.
Objective: to describe the early adolescence outcome of boys treated during the first year of life.
Methods: In this retrospective cohort study, we describe 11 CHH boys aged 12 years [11.5-14.6] treated at the age of 4.5 months [2.0-11] with CSGI. To caompare we report testicular function of 12 untreated CHH boys aged 12 years [12-15.9].
Results: In response to CSGI, serum testosterone and inhibinB levels increased from 0.03 ng/mL [0-0.07] to 2.25 ng/mL [1.12-3.86] and from 73 [11-173] to 401 [185-727] pg/mL, respectively. Testicular volume increased from 0.50 mL [0.5-1] to 1.50 mL [0.7-3]. Between end of CSGI and early adolescence, testicular volume in the treated group decreased from 1.5 mL [0.7-3] to 1.05 ml [0.7-2.36] (p=0.024) and differed from that in untreated boys (0.3 mL [0.13-1.3]). Hormone levels were higher in the treated group : serum AMH and inhibin B levels in treated patients decreased from 1028 pmol/l [550-1750] and 356 [185-727] pg/mL at neonatal period to 331 pmol/l [85-479] and 68 pg/ml [19-239] respectively at early adolescence and differed from those in untreated patients (57.5 [30-169] and 8 pg/ml [<5-37] (p<0.001)).
Conclusion: We report the first long-term follow-up of CHH boys treated with CSGI in infancy. Our results shown that the CSGI treatment resulted in higher inhibin B, AMH levels and testicular volume at early adolescence age. Follow-up should be continued until the end of puberty to assess spermatogenesis.
期刊介绍:
Endocrine Connections publishes original quality research and reviews in all areas of endocrinology, including papers that deal with non-classical tissues as source or targets of hormones and endocrine papers that have relevance to endocrine-related and intersecting disciplines and the wider biomedical community.