A prospective study on the long‐term outcome of prepubertal and pubertal boys undergoing testicular biopsy for fertility preservation prior to hematologic stem cell transplantation

Background Few studies have reported the long‐term outcomes of prepubertal and pubertal boys undergoing testicular biopsy for fertility preservation (FP). Procedure This prospective longitudinal study examined 21 boys (aged 1.5‐14.5 years) who underwent testicular biopsy for FP prior to allogeneic (...

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Veröffentlicht in:Pediatric blood & cancer 2020-09, Vol.67 (9), p.e28507-n/a, Article 28507
Hauptverfasser: Borgström, Birgit, Fridström, Margareta, Gustafsson, Britt, Ljungman, Per, Rodriguez‐Wallberg, Kenny A.
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container_issue 9
container_start_page e28507
container_title Pediatric blood & cancer
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creator Borgström, Birgit
Fridström, Margareta
Gustafsson, Britt
Ljungman, Per
Rodriguez‐Wallberg, Kenny A.
description Background Few studies have reported the long‐term outcomes of prepubertal and pubertal boys undergoing testicular biopsy for fertility preservation (FP). Procedure This prospective longitudinal study examined 21 boys (aged 1.5‐14.5 years) who underwent testicular biopsy for FP prior to allogeneic (n = 20) or autologous (n = 1) hematological stem cell transplantation (HSCT) between 2003 and 2010. During counseling, pubertal boys were encouraged to produce a sperm sample by masturbation , while prepubertal boys were presented with surgical testicular tissue retrieval as an option for experimental FP. Clinical outcomes included postoperative complications, pubertal development, and sex‐hormone levels. Survivors approaching adulthood were encouraged to provide semen samples. Results Twenty boys, including 14 in prepuberty and six in early puberty (Tanner stage 2‐3), underwent open testicular biopsies. Two pubertal biopsies contained mature sperms, which were cryopreserved. Testicular tissue was vitrified in the remaining 18 cases. One pubertal boy (Tanner stage 4) underwent percutaneous testicular sperm aspiration and sperms obtained were cryopreserved. Postoperative complications (hematoma or infection) were rare. Overall, 14 boys survived >5 years (mean follow‐up after HSCT, 7.2 years) and 11 showed advanced puberty. Semen samples were provided by five boys and obtained sperm were cryopreserved from two. Individuals at adulthood had normal testosterone levels but subnormal testicular size, high follicle stimulating hormone, and low inhibin B and anti‐Müllerian hormone levels. Conclusion No long‐term risks were detected during continuous clinical follow‐up. Experimental testicular biopsies for FP were well accepted by the patients and families, despite the absence of methods to use prepubertal tissue for fertility treatment.
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Procedure This prospective longitudinal study examined 21 boys (aged 1.5‐14.5 years) who underwent testicular biopsy for FP prior to allogeneic (n = 20) or autologous (n = 1) hematological stem cell transplantation (HSCT) between 2003 and 2010. During counseling, pubertal boys were encouraged to produce a sperm sample by masturbation , while prepubertal boys were presented with surgical testicular tissue retrieval as an option for experimental FP. Clinical outcomes included postoperative complications, pubertal development, and sex‐hormone levels. Survivors approaching adulthood were encouraged to provide semen samples. Results Twenty boys, including 14 in prepuberty and six in early puberty (Tanner stage 2‐3), underwent open testicular biopsies. Two pubertal biopsies contained mature sperms, which were cryopreserved. Testicular tissue was vitrified in the remaining 18 cases. One pubertal boy (Tanner stage 4) underwent percutaneous testicular sperm aspiration and sperms obtained were cryopreserved. Postoperative complications (hematoma or infection) were rare. Overall, 14 boys survived &gt;5 years (mean follow‐up after HSCT, 7.2 years) and 11 showed advanced puberty. Semen samples were provided by five boys and obtained sperm were cryopreserved from two. Individuals at adulthood had normal testosterone levels but subnormal testicular size, high follicle stimulating hormone, and low inhibin B and anti‐Müllerian hormone levels. Conclusion No long‐term risks were detected during continuous clinical follow‐up. Experimental testicular biopsies for FP were well accepted by the patients and families, despite the absence of methods to use prepubertal tissue for fertility treatment.</description><identifier>ISSN: 1545-5009</identifier><identifier>ISSN: 1545-5017</identifier><identifier>EISSN: 1545-5017</identifier><identifier>DOI: 10.1002/pbc.28507</identifier><identifier>PMID: 32649054</identifier><language>eng</language><publisher>HOBOKEN: Wiley</publisher><subject>Adolescent ; Autografts ; Biopsy ; Boys ; Child ; Child, Preschool ; Cryopreservation ; Fertility ; fertility preservation ; Fertility Preservation - methods ; Follow-Up Studies ; Hematologic Neoplasms - pathology ; Hematologic Neoplasms - surgery ; Hematology ; Hematoma ; Hematopoietic Stem Cell Transplantation ; Humans ; Infant ; Inhibin ; Life Sciences &amp; Biomedicine ; Longitudinal Studies ; Male ; Medicin och hälsovetenskap ; Oncology ; pediatric neoplasm ; Pediatrics ; prepubertal children ; Preservation ; Prognosis ; Prospective Studies ; Puberty ; Science &amp; Technology ; Semen ; Sperm ; sperm banking ; Stem cell transplantation ; Stem cells ; Testes ; testicular tissue cryopreservation ; Testis - surgery ; Testosterone</subject><ispartof>Pediatric blood &amp; cancer, 2020-09, Vol.67 (9), p.e28507-n/a, Article 28507</ispartof><rights>2020 The Authors. published by Wiley Periodicals LLC</rights><rights>2020 The Authors. Pediatric Blood &amp; Cancer published by Wiley Periodicals LLC.</rights><rights>2020. This article is published under http://creativecommons.org/licenses/by-nc/4.0/ (the “License”). 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Procedure This prospective longitudinal study examined 21 boys (aged 1.5‐14.5 years) who underwent testicular biopsy for FP prior to allogeneic (n = 20) or autologous (n = 1) hematological stem cell transplantation (HSCT) between 2003 and 2010. During counseling, pubertal boys were encouraged to produce a sperm sample by masturbation , while prepubertal boys were presented with surgical testicular tissue retrieval as an option for experimental FP. Clinical outcomes included postoperative complications, pubertal development, and sex‐hormone levels. Survivors approaching adulthood were encouraged to provide semen samples. Results Twenty boys, including 14 in prepuberty and six in early puberty (Tanner stage 2‐3), underwent open testicular biopsies. Two pubertal biopsies contained mature sperms, which were cryopreserved. Testicular tissue was vitrified in the remaining 18 cases. One pubertal boy (Tanner stage 4) underwent percutaneous testicular sperm aspiration and sperms obtained were cryopreserved. Postoperative complications (hematoma or infection) were rare. Overall, 14 boys survived &gt;5 years (mean follow‐up after HSCT, 7.2 years) and 11 showed advanced puberty. Semen samples were provided by five boys and obtained sperm were cryopreserved from two. Individuals at adulthood had normal testosterone levels but subnormal testicular size, high follicle stimulating hormone, and low inhibin B and anti‐Müllerian hormone levels. Conclusion No long‐term risks were detected during continuous clinical follow‐up. Experimental testicular biopsies for FP were well accepted by the patients and families, despite the absence of methods to use prepubertal tissue for fertility treatment.</description><subject>Adolescent</subject><subject>Autografts</subject><subject>Biopsy</subject><subject>Boys</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Cryopreservation</subject><subject>Fertility</subject><subject>fertility preservation</subject><subject>Fertility Preservation - methods</subject><subject>Follow-Up Studies</subject><subject>Hematologic Neoplasms - pathology</subject><subject>Hematologic Neoplasms - surgery</subject><subject>Hematology</subject><subject>Hematoma</subject><subject>Hematopoietic Stem Cell Transplantation</subject><subject>Humans</subject><subject>Infant</subject><subject>Inhibin</subject><subject>Life Sciences &amp; Biomedicine</subject><subject>Longitudinal Studies</subject><subject>Male</subject><subject>Medicin och hälsovetenskap</subject><subject>Oncology</subject><subject>pediatric neoplasm</subject><subject>Pediatrics</subject><subject>prepubertal children</subject><subject>Preservation</subject><subject>Prognosis</subject><subject>Prospective Studies</subject><subject>Puberty</subject><subject>Science &amp; Technology</subject><subject>Semen</subject><subject>Sperm</subject><subject>sperm banking</subject><subject>Stem cell transplantation</subject><subject>Stem cells</subject><subject>Testes</subject><subject>testicular tissue cryopreservation</subject><subject>Testis - surgery</subject><subject>Testosterone</subject><issn>1545-5009</issn><issn>1545-5017</issn><issn>1545-5017</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2020</creationdate><recordtype>article</recordtype><sourceid>24P</sourceid><sourceid>WIN</sourceid><sourceid>AOWDO</sourceid><sourceid>EIF</sourceid><sourceid>D8T</sourceid><recordid>eNqNkstu1TAQhiMEoqWw4AWQJTYgdFrbsRNnWY64SZVgAevIcSanLokdbKdVdjwCT8MD8SRMmtODhITEyuPk-8f_XLLsKaOnjFJ-NjbmlCtJy3vZMZNCbiRl5f1DTKuj7FGMV4gWVKqH2VHOC1FRKY6zn-dkDD6OYJK9BhLT1M7EO5IugfTe7X59_5EgDMRPyfgBiO-Qh3FqICTdE-1acrg0fo5kci2EnbduRxLEZM3U60Aa68c4k84H0iFse5vmJVGEcK2TxQfHYPFn8uQSBp1873fWoB0YiIG-JyloF8deu3SLP84edLqP8GR_nmRf3r75vH2_ufj47sP2_GJjpOLlpqSlaSWvGgGNzNuyYEWlBGcyVznW3-lCVy1XoikqYUzHmOnKXDaUUyhVp0x-km3WvPFmKbpGl4MOc-21rfefvmIEtVgSU-Srf_LY5_aP6E7IhKAlFZyj9sWqRfDbhL2rBxuX4rUDP8WaC55TWag8R_T5X-iVn4LDTiyUwjkXVCD1cqUMTjgG6A52GK2XxalxcerbxUH22T7j1AzQHsi7TUHg1QrcQOO7aCw4AweMUmSKQjGBEWVIq_-nt3Yd6tZPLqH0bC-1Pcz_tlx_er1dvf8Goqv0Aw</recordid><startdate>202009</startdate><enddate>202009</enddate><creator>Borgström, Birgit</creator><creator>Fridström, Margareta</creator><creator>Gustafsson, Britt</creator><creator>Ljungman, Per</creator><creator>Rodriguez‐Wallberg, Kenny A.</creator><general>Wiley</general><general>Wiley Subscription Services, Inc</general><scope>24P</scope><scope>WIN</scope><scope>AOWDO</scope><scope>BLEPL</scope><scope>DTL</scope><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7T5</scope><scope>7TK</scope><scope>7TO</scope><scope>8FD</scope><scope>FR3</scope><scope>H94</scope><scope>K9.</scope><scope>P64</scope><scope>RC3</scope><scope>7X8</scope><scope>ADTPV</scope><scope>AOWAS</scope><scope>D8T</scope><scope>ZZAVC</scope><orcidid>https://orcid.org/0000-0003-4378-6181</orcidid></search><sort><creationdate>202009</creationdate><title>A prospective study on the long‐term outcome of prepubertal and pubertal boys undergoing testicular biopsy for fertility preservation prior to hematologic stem cell transplantation</title><author>Borgström, Birgit ; Fridström, Margareta ; Gustafsson, Britt ; Ljungman, Per ; Rodriguez‐Wallberg, Kenny A.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c5827-707cd529b4eb53d7616984215383905fa6a9d284b694ccf11cf735b020e78f8c3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2020</creationdate><topic>Adolescent</topic><topic>Autografts</topic><topic>Biopsy</topic><topic>Boys</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Cryopreservation</topic><topic>Fertility</topic><topic>fertility preservation</topic><topic>Fertility Preservation - methods</topic><topic>Follow-Up Studies</topic><topic>Hematologic Neoplasms - pathology</topic><topic>Hematologic Neoplasms - surgery</topic><topic>Hematology</topic><topic>Hematoma</topic><topic>Hematopoietic Stem Cell Transplantation</topic><topic>Humans</topic><topic>Infant</topic><topic>Inhibin</topic><topic>Life Sciences &amp; Biomedicine</topic><topic>Longitudinal Studies</topic><topic>Male</topic><topic>Medicin och hälsovetenskap</topic><topic>Oncology</topic><topic>pediatric neoplasm</topic><topic>Pediatrics</topic><topic>prepubertal children</topic><topic>Preservation</topic><topic>Prognosis</topic><topic>Prospective Studies</topic><topic>Puberty</topic><topic>Science &amp; Technology</topic><topic>Semen</topic><topic>Sperm</topic><topic>sperm banking</topic><topic>Stem cell transplantation</topic><topic>Stem cells</topic><topic>Testes</topic><topic>testicular tissue cryopreservation</topic><topic>Testis - surgery</topic><topic>Testosterone</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Borgström, Birgit</creatorcontrib><creatorcontrib>Fridström, Margareta</creatorcontrib><creatorcontrib>Gustafsson, Britt</creatorcontrib><creatorcontrib>Ljungman, Per</creatorcontrib><creatorcontrib>Rodriguez‐Wallberg, Kenny A.</creatorcontrib><collection>Wiley Online Library (Open Access Collection)</collection><collection>Wiley Online Library (Open Access Collection)</collection><collection>Web of Science - Science Citation Index Expanded - 2020</collection><collection>Web of Science Core Collection</collection><collection>Science Citation Index Expanded</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>Immunology Abstracts</collection><collection>Neurosciences Abstracts</collection><collection>Oncogenes and Growth Factors Abstracts</collection><collection>Technology Research Database</collection><collection>Engineering Research Database</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>ProQuest Health &amp; Medical Complete (Alumni)</collection><collection>Biotechnology and BioEngineering Abstracts</collection><collection>Genetics Abstracts</collection><collection>MEDLINE - Academic</collection><collection>SwePub</collection><collection>SwePub Articles</collection><collection>SWEPUB Freely available online</collection><collection>SwePub Articles full text</collection><jtitle>Pediatric blood &amp; cancer</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Borgström, Birgit</au><au>Fridström, Margareta</au><au>Gustafsson, Britt</au><au>Ljungman, Per</au><au>Rodriguez‐Wallberg, Kenny A.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>A prospective study on the long‐term outcome of prepubertal and pubertal boys undergoing testicular biopsy for fertility preservation prior to hematologic stem cell transplantation</atitle><jtitle>Pediatric blood &amp; cancer</jtitle><stitle>PEDIATR BLOOD CANCER</stitle><addtitle>Pediatr Blood Cancer</addtitle><date>2020-09</date><risdate>2020</risdate><volume>67</volume><issue>9</issue><spage>e28507</spage><epage>n/a</epage><pages>e28507-n/a</pages><artnum>28507</artnum><issn>1545-5009</issn><issn>1545-5017</issn><eissn>1545-5017</eissn><abstract>Background Few studies have reported the long‐term outcomes of prepubertal and pubertal boys undergoing testicular biopsy for fertility preservation (FP). Procedure This prospective longitudinal study examined 21 boys (aged 1.5‐14.5 years) who underwent testicular biopsy for FP prior to allogeneic (n = 20) or autologous (n = 1) hematological stem cell transplantation (HSCT) between 2003 and 2010. During counseling, pubertal boys were encouraged to produce a sperm sample by masturbation , while prepubertal boys were presented with surgical testicular tissue retrieval as an option for experimental FP. Clinical outcomes included postoperative complications, pubertal development, and sex‐hormone levels. Survivors approaching adulthood were encouraged to provide semen samples. Results Twenty boys, including 14 in prepuberty and six in early puberty (Tanner stage 2‐3), underwent open testicular biopsies. Two pubertal biopsies contained mature sperms, which were cryopreserved. Testicular tissue was vitrified in the remaining 18 cases. One pubertal boy (Tanner stage 4) underwent percutaneous testicular sperm aspiration and sperms obtained were cryopreserved. Postoperative complications (hematoma or infection) were rare. Overall, 14 boys survived &gt;5 years (mean follow‐up after HSCT, 7.2 years) and 11 showed advanced puberty. Semen samples were provided by five boys and obtained sperm were cryopreserved from two. Individuals at adulthood had normal testosterone levels but subnormal testicular size, high follicle stimulating hormone, and low inhibin B and anti‐Müllerian hormone levels. Conclusion No long‐term risks were detected during continuous clinical follow‐up. Experimental testicular biopsies for FP were well accepted by the patients and families, despite the absence of methods to use prepubertal tissue for fertility treatment.</abstract><cop>HOBOKEN</cop><pub>Wiley</pub><pmid>32649054</pmid><doi>10.1002/pbc.28507</doi><tpages>11</tpages><orcidid>https://orcid.org/0000-0003-4378-6181</orcidid><oa>free_for_read</oa></addata></record>
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subjects Adolescent
Autografts
Biopsy
Boys
Child
Child, Preschool
Cryopreservation
Fertility
fertility preservation
Fertility Preservation - methods
Follow-Up Studies
Hematologic Neoplasms - pathology
Hematologic Neoplasms - surgery
Hematology
Hematoma
Hematopoietic Stem Cell Transplantation
Humans
Infant
Inhibin
Life Sciences & Biomedicine
Longitudinal Studies
Male
Medicin och hälsovetenskap
Oncology
pediatric neoplasm
Pediatrics
prepubertal children
Preservation
Prognosis
Prospective Studies
Puberty
Science & Technology
Semen
Sperm
sperm banking
Stem cell transplantation
Stem cells
Testes
testicular tissue cryopreservation
Testis - surgery
Testosterone
title A prospective study on the long‐term outcome of prepubertal and pubertal boys undergoing testicular biopsy for fertility preservation prior to hematologic stem cell transplantation
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