Intratesticular xenografting of Klinefelter pre-pubertal testis tissue as potential model to study testicular fibrosis

Willems, Margo; Seβenhausen, Pia; Gies, Inge; Vloeberghs, Veerle; Tournaye, Herman; Saen, Dorien Van; Goossens, Ellen

doi:10.1016/j.rbmo.2022.01.009

Cited by 4 publications

(3 citation statements)

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“…Recently xenografting of human pre-pubertal KS testes tissue to the immunodeficient mouse testes has been offered as a model to study the fibrotic process in Klinefelter testis. 28 Tissue injury in other organs results in inflammation and apoptosis regulated by mast cells and macrophages, making them an important potential contributor to the KS testicular pathology. D'Aurora and colleagues identified several genes involved in the BTB, antigen presentation, the immune response, and apoptotic pathways, thus proposing that the interplay between dysregulation of the BTB, increased inflammatory and immune response, and pro-apoptotic genes were the genetic foundation of fibrosis and hyperplasia in KS testes.…”

Section: Discussionmentioning

confidence: 99%

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Examining potential mechanisms of testicular fibrosis in Klinefelter Syndrome: A review of current understanding

Bradshaw

Deebel

et al. 2022

Andrology

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Background Men with Klinefelter Syndrome develop some degree of seminiferous tubule degeneration, hyalinization, and fibrosis by adulthood. However, the pathophysiology surrounding testicular fibrosis in Klinefelter Syndrome patients remains incompletely understood. Objectives To perform a systematic review of literature studying the mechanisms of fibrosis initiation or propagation in Klinefelter Syndrome testes. Materials/methods PubMed was searched systematically for articles specific to Klinefelter Syndrome and the process of fibrosis. Articles that did not contain original data or specifically addressed the target material were excluded. Additional references were extracted when pertinent from the reference lists of included studies. Results Primary search yielded 139 articles for abstract review, which was narrowed to 16 for full‐text review. Following full‐text review, eight contained original data and met topic criteria, with one paper added from reference review for a total of nine papers. Discussion The date range for included papers was 1992–2022. The proposed mechanisms of fibrosis mainly were centered around the impact of altered Sertoli cells on germ cells, the hormonal impact on Leydig cells, the inflammation mediated by mast cells, or the fibrous extracellular matrix deposition by peritubular myoid cells. Additionally, discussions of the role of the altered microvasculature and the specific proteins involved in the blood‐testis barrier or the seminiferous tubule architecture are reviewed. Recent papers have incorporated advanced sequencing and offer future directions for targeted gene expression analysis. Still, much of the published data consists solely of immunohistological assessment by age range, creating difficulties in extrapolating causality. Conclusion The specific initiating factors of fibrosis of the seminiferous tubules and the propagation mechanisms unique to Klinefelter Syndrome remain incompletely understood with a relative paucity of data. Nonetheless, academic interest is increasing in this field as it may further elucidate the pathophysiology behind Klinefelter syndrome.

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Section: Discussionmentioning

confidence: 99%

“…Pursuing other initiating mechanisms for testicular fibrosis would likely be a more fruitful research direction. Recently xenografting of human pre‐pubertal KS testes tissue to the immunodeficient mouse testes has been offered as a model to study the fibrotic process in Klinefelter testis 28 …”

Section: Discussionmentioning

confidence: 99%

Examining potential mechanisms of testicular fibrosis in Klinefelter Syndrome: A review of current understanding

Bradshaw

Deebel

et al. 2022

Andrology

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“…Whether the altered vascularisation also has influence on the occurrence of fibrosis in the KS testes is currently investigated. Recent research is focused on a role of mast cells (MCs) that has been reported in increased numbers in biopsies from infertile patients [ 45 , 46 ].…”

Section: Discussionmentioning

confidence: 99%

Testicular microlithiasis in paediatric patients with Klinefelter syndrome from infancy till adolescence: early start of degenerative process in the testes—preliminary results

2022

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To present the results of testicular ultrasonography supported by clinical and hormonal aspects in paediatric patients with Klinefelter syndrome (KS). Prospective analysis of medical files of 20 patients diagnosed with KS between 2016 and 2022. Assessed data included analysis of causes of referral, ultrasound, and clinical characterisation with hormonal evaluation of serum FSH, LH, testosterone, inhibin B, and anti-Müllerian hormone. Non-mosaic Klinefelter syndrome (47, XXY) was diagnosed in 65% of cases (13/20) by the geneticist (including 7 cases prenatally), in 25% (5/20) by the endocrinologist and in 10% (2/20) by the hematologist. Ultrasound assessment revealed bilateral testicular microlithiasis (TM) in all patients. The youngest KS patient with TM was 3 months old. TM patterns have not changed during follow-ups of up to 6 years in any of the patients. In all KS patients markedly reduced echogenicity and in pubertal KS patients, also irregular echostructure of the testes was observed. The hormonal patterns observed in the study group were typical for those already described in KS. Sertoli and Leydig cell function was intact in prepubertal patients and deteriorated after the start of puberty.Conclusion: Although the degenerative process in the testicular tissue starts very early in the testes in KS and is reflected in morphological changes seen in ultrasonography, Sertoli and Leydig cell hormonal function is normal in prepubertal KS patients. What is Known:• So far, normal Leydig and Sertoli cell function was observed in infants and prepubertal KS patients. What is New:• The morphological changes in the testes in KS may already be seen in early infancy.

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Testicular fibrosis pathology, diagnosis, pathogenesis, and treatment: A perspective on related diseases

Xu,

Hu,

Chen

et al. 2024

Andrology

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Testicular fibrosis is a chronic and progressive condition characterized by the excessive deposition of extracellular matrix proteins. This process leads to fibrotic remodeling, damage to testicular tissue, and the irreversible loss of male reproductive function. However, there is currently a lack of comprehensive reviews systematically elucidating the pathology, diagnosis, pathogenesis, and treatment of testicular fibrosis from the perspectives of different related diseases. This review addresses these aspects of testicular fibrosis, with a particular emphasis on elucidating the underlying mechanisms of testicular cells. It provides insights that can be relevant for future research and clinical interventions.

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Intratesticular xenografting of Klinefelter pre-pubertal testis tissue as potential model to study testicular fibrosis

Cited by 4 publications

References 53 publications

Examining potential mechanisms of testicular fibrosis in Klinefelter Syndrome: A review of current understanding

Examining potential mechanisms of testicular fibrosis in Klinefelter Syndrome: A review of current understanding

Testicular microlithiasis in paediatric patients with Klinefelter syndrome from infancy till adolescence: early start of degenerative process in the testes—preliminary results

Testicular fibrosis pathology, diagnosis, pathogenesis, and treatment: A perspective on related diseases

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