No approved-drug candidate for sertoli cell-only syndrome is corroborated in the literature DeepSearch retrieved. Some conditions are managed with non-pharmacological care — a device, surgery or physical therapy — rather than a medicine; that may be the case here, or the literature we found may simply be too sparse yet to support a drug-repurposing angle.
A 2022 systematic review of monogenic mutations in Sertoli cell-only syndrome (SCOS) identified 15 eligible studies published between 2005 and 2020, covering 1513 cases from seven countries. Only 13% of the studies were published before 2012; 87% appeared after that date. Most studies (66.7%) were on Asian populations, primarily Japanese. All patients had normal karyotypes and no Y-chromosome microdeletions. Across the 15 studies, approximately 51 variations in 13 genes (USP26, RAD21L, SEPTIN12, PAPOLB, PLK4, H3t, ETV5, GILZ, SIN3A, CUL4B, DMRT1, SPATA17, LRWD1) were identified, but only seven variations showed a statistically significant association with SCOS. The review concludes that comprehensive large-scale studies on monogenic causes of SCOS are still lacking.
Earlier work on Sertoli cells has focused on their biology and potential therapeutic use outside of SCOS. A 1989 minireview on Sertoli cell culture cautioned that in vitro results should not be extrapolated to the intact animal without in vivo validation. A 2018 review described Sertoli cells as delivering immunomodulatory and trophic factors, and noted their experimental use in cell transplantation for diabetes mellitus, Laron dwarfism, Duchenne muscular dystrophy, and prevention of skin allograft rejection. In a 2012 rat model of renal acute rejection, cotransplanted allogeneic Sertoli cells (2 × 10⁶ cells) increased survival duration to 19.50 ± 4.3 days, compared with 14 ± 3.1 days in controls; the cyclosporine group survived 21.50 ± 5.9 days. Serum creatinine was significantly higher in both the Sertoli cell and cyclosporine groups than in controls.
Separate literature concerns Sertoli cell tumours, not SCOS. A 2020 meta-analysis of 435 patients with Sertoli cell tumours found that fewer than 1% showed local recurrence after testis-sparing surgery. Fifty patients presented with metastases; median time to recurrence was 12 months, and median life expectancy after metastasis was 20 months. Metastasectomy produced complete remission in six patients, but systemic treatment alone did not result in long-term remission. A 1995 case report described a sclerosing Sertoli cell tumour subtype and noted that the recognised variants differ in malignant potential.
What is still missing: large-scale genetic studies with whole-exome or whole-genome sequencing in SCOS cohorts that exclude known chromosomal causes; functional validation of the seven variants that reached statistical significance; and any clinical trial testing Sertoli cell transplantation or other interventions specifically for SCOS in humans.
Retrieved by DeepSearch across 234,678,978 indexed works and resolved on OpenAlex — ranked by citations, including the results that did not work.
Biology of Reproduction · 1989 · 58 citations
Sertoli Cells in Culture: Views from the Perspectives of an in Vivoist and an in Vitroist
AbstractCulturing Sertoli cells has proven to be a valuable technique to study the responses of these cells under a variety of conditions. This minireview focuses on the culture system and how it has contributed to our knowledge of the capabilities of isolated Sertoli cells. In addition, it focuses on the limitations of the system and the gaps in our knowledge about Sertoli cells in culture and what the results of in vitro studies mean in the intact animal. Caution is urged in interpreting in vitro results to the intact animal without their validation in vivo.
https://doi.org/10.1095/biolreprod41.4.571Andrology · 2018 · 56 citations · open access
Sertoli cells for cell transplantation: pre‐clinical studies and future perspectives
AbstractSertoli cells are located in the testes where they control several key functions in spermatogenesis. Over the past 30 years, Sertoli cells have been upgraded from a simple scaffold-like structural system to a dynamic functional system of intercellular support that delivers potent immunomodulatory and trophic factors. Since the discovery of new Sertoli cell secretory products, these cells have been utilized in experimental cell transplantation and co-transplantation protocols aimed at treating both chronic inflammatory and degenerative disorders. For these reasons, this work reviews the application of both naked and microencapsulated Sertoli cells used in cell transplantation studies of several chronic or autoimmune diseases such as diabetes mellitus, Laron dwarfism, and Duchenne muscular dystrophy and in studies aimed at the prevention of skin allograft rejection.
https://doi.org/10.1111/andr.12484The Oncologist · 2020 · 43 citations · open access
Sertoli Cell Tumors of the Testes: Systematic Literature Review and Meta-Analysis of Outcomes in 435 Patients
AbstractBACKGROUND: Sertoli cell tumors (SCTs) of the testes are rare, and the literature provides only weak evidence concerning their clinical course and management. The objective of this study was to summarize evidence on SCTs' clinical presentation, clinicopathological risk factors for malignancy, treatment options, and oncological outcomes. MATERIALS AND METHODS: Data sources included Medline, Embase, Scopus, the Cochrane Database of Systematic Reviews, and Web of Science. Published case reports, case series, and cohorts were included. Data on clinicopathological variables, treatment of local or metastatic disease, site of metastasis, or survival were extracted from each study considered in this paper, and associations between clinicopathological variables and metastatic disease were analyzed. Whenever feasible, data on individual patients were collected. RESULTS: Of the 435 patients included, only one (<1%) showed local recurrence after testis-sparing surgery (TSS). Three patients underwent adjuvant retroperitoneal lymphadenectomy. Fifty patients presented with metastases, located in the retroperitoneal lymph nodes (76%), lungs (36%), and bones (16%); median time to recurrence was 12 months. Risk factors for metastatic disease included age, tumor size, necrosis, tumor extension to the spermatic cord, angiolymphatic invasion, and mitotic index. Patients with metastases had a median life expectancy of 20 months. In six patients, metastasectomy resulted in complete remission. CONCLUSION: Our findings suggest that few local recurrences result after TSS, and no adjuvant therapy can be regarded as a standard of care. Several risk factors are predictive of metastatic disease. Surgery leads to remission in metastatic disease, whereas systemic treatment alone does not result in long-term remission. IMPLICATIONS FOR PRACTICE: Testicular Sertoli cell tumors usually present without metastatic disease and show low local recurrence rates after testis-sparing surgery; no adjuvant therapy option can be regarded as a standard of care. Patients with risk factors should undergo staging investigations. Those with metastatic disease have poor prognoses, and metastasectomy may be offered in selected cases.
https://doi.org/10.1634/theoncologist.2019-0692The Journal of Urology · 1995 · 38 citations
Sclerosing Sertoli Cell Tumor of the Testis: A Distinct Histological Subtype
AbstractPURPOSE: A case of sclerosing Sertoli cell tumor of the testis is reported. The histological and clinical features are compared to those of other Sertoli cell tumor subtypes. MATERIALS AND METHODS: The current urological and pathological literature was reviewed. RESULTS: The tumor was identified incidentally in a 35-year-old white man. Pathological examination was performed and the patient was well at 9 months. CONCLUSIONS: Sertoli cell tumors are heterogeneous and not accurately considered a uniform entity. The currently recognized variants differ in apparent malignant potential as well as the association with disease processes in other unrelated organ systems.
https://doi.org/10.1016/s0022-5347(01)66777-6Experimental and Clinical Transplantation · 2012 · 4 citations
Renoprotective Effects of Cotransplanted Allogeneic Testicular Sertoli Cells in a Renal Acute Rejection Model in Rats
AbstractOBJECTIVES: We sought to study the renoprotective effect of cotransplanted allogeneic testicular Sertoli cells on renal acute rejection in rats. MATERIALS AND METHODS: A renal acute rejection model using kidneys from Sprague-Dawley (n=30) transplanted into Wistar rats (n=30) was constructed. The rats were randomly divided into 3 groups: (1) the cyclosporine group, which was treated with daily hypodermic injections of cyclosporine (15 mg/kg) after transplant, (2) the Sertoli cells group with cell suspension (n = 2 × 10⁶ cells) into the subcapsular space of the renal graft, and (3) the control group, which received no posttransplant intervention. Graft function was measured based on serial serum creatinine. Graft histology was examined at 10 days posttransplant, and survival duration was recorded. RESULTS: Serum creatinine was significantly higher in the Sertoli cells and cyclosporine groups than in the controls. Survival duration was significantly longer in the Sertoli cells (19.50 ± 4.3 d) and cyclosporine groups (21.50 ± 5.9 d) than in the controls (14 ± 3.1 d). Allografts in the control group exhibited typical severe acute rejection, including widespread interstitial infiltration with tubulous, patchy necrosis and hemorrhage, severe glomerulitis with extensive capillary occlusion caused by endothelial swelling, and intimal arteritis in the cortex. Findings of acute rejection were less in the Sertoli cells and cyclosporine groups. CONCLUSIONS: Sertoli cell implantation is an effective method for increasing survival duration in rat renal transplant, and it has potential as a new alternative to cyclosporine immunosuppression.
https://doi.org/10.6002/ect.2012.0001The Journal of Sexual Medicine · 2022 · 0 citations
094 A Systematic Review of Monogenic Mutations Responsible of Sertoli Cell-Only Syndrome
AbstractABSTRACT Introduction Sertoli-cell only syndrome (SCOS) is the severe form of non-obstructive azoospermia, in which only the Sertoli cells line the wall of seminiferous tubules. Y chromosome microdeletions are the obvious cause of SCOS in men, but conclusive and summarized data is lacking on the monogenic mutations. Objective To systematically review published data on monogenic mutations causing SCOS. Methods Medline was searched without publication date restriction for all the genetic association studies on SCOS by using keywords: genetics of Sertoli cell-only syndrome, genetics of azoospermia secondary to SCOS and, genes of Sertoli cell-only syndrome. Quality evaluation and data extraction were done by two authors Studies included in the systematic review were presenting original clinical data, human male cases and patients with confirmed SCOS by histopathology or MD-TESE. The genetic evidence of SCOS was included even if the reported case and genetic testing were published separately. Non-English language studies, descriptive reviews, articles of gene expression in SCOS, and articles related to the other chromosomal abnormalities as a cause of SCOS were also excluded. Any dispute on exclusion and inclusion of a study was resolved by discussion and consensus between all authors. Results The initial database search identified 489 articles, after evaluation according to the defined criteria, only 15 studies matched the inclusion criteria for systematic review. Articles included in the systematic review were published from 2005 to 2020. Only 13% (n=2) of articles were published before 2012. From 2012 to 2020, there have been 87% (n=13) publications. Most of the studies (66.7%, n=10) were carried out on Asian populations (Japanese population) and the rest were on Caucasians. The 15 publications included a total of 1513 cases of SCOS, originating from 7 different countries. However, 151 cases appeared in 2 articles. Extracted data show that 40% of studies exclude patients with a history of infections, seminal tract obstruction, pituitary gland disorder or any other cause of testicular damage, and 27% of studies only recruit patients with no chromosomal abnormalities. Approximately 13% of eligible studies exclude the patients based on congenital abnormalities, past cryptorchidism, orchitis, cancer and other systemic illnesses. In addition, 13% of studies included patients who were diagnosed with SCOS. Only 7% of studies include the patient with unknown cause of male infertility. In all the studies only patients with normal karyotype and without Y-chromosome microdeletions were included. In all the studies semen analysis and confirmatory tests MD-TESE or histopathology were for SCOS. Most of the studies used the technique of direct sequencing and only one study performed whole-exome sequencing. Altogether approximately 51 variations in 13 genes (USP26, RAD21L, SEPTIN12, PAPOLB, PLK4, H3t, ETV5, GILZ, SIN3A, CUL4B, DMRT1, SPATA17 and, LRWD1) were identified from which only 7 variations showed a statistically significant association, but others showed rare or no association. Conclusion Comprehensive large-scale studies on monogenic causes of SCOS still lacking. Altogether approximately 51 variations in 13 genes were identified from which only 7 variations showed statistically significant association. Further studies are required to determine the monogenic causes of SCOS. Disclosure Work supported by industry: no.
https://doi.org/10.1016/j.jsxm.2022.03.369Journal of the Endocrine Society · 2025 · 0 citations · open access
MON-194 A Case Of Sertoli Cell Tumor In A Patient With Familial Partial Lipodystrophy Type 2
AbstractAbstract Disclosure: V. Rivera-Hernandez: None. I. Singh: None. Familial partial lipodystrophies (incidence of 1 in 1 million people) are a group of disorders of selective adipose tissue loss that can be caused by Autosomal dominant or autosomal recessive inheritance. Familial partial lipodystrophy (FPL) type 2 (also called Dunnigan lipodystrophy) is the most common form and results from a missense mutation of Lamins A and C. Individuals have normal adipose tissue distribution during childhood, with gradual loss of adipose tissue that occurs at or after puberty at the arms, legs and trunk, and accumulation at the face, neck, and upper back. FPL is also characterized by the appearance of hypertrophic muscles and metabolic disorders including insulin resistance, hypertriglyceridemia, and hyperandrogenism. Sertoli cell tumors of the ovary (incidence of &lt; 0.5%) are a rare cause of hyperandrogenemia. There are no published case reports of Sertoli cell tumor of the ovary in an individual with FPL as we now describe. A 21-year-old female with history of polycystic ovarian syndrome (PCOS) presented for evaluation of PCOS and concern for excess cortisol levels. Diagnosed with PCOS 5 years earlier by Gyn. Reported excess hair growth at the chin, lower abdominal area, and upper back, anterior scalp hair thinning, round face, and “buffalo hump”. Menarche occurred at age 11, two years after which her periods became irregular, skipping 2-3 timer per year. She is of Polish and German descent and her sister also has a history of PCOS. PCOS was managed with OCP, spironolactone and metformin. On physical exam, was noted to have some facial rounding, hirsutism, hair thinning, mildly coarse facial features, and wide neck. No dorsocervical fad pad. She had markedly muscular extremities. Additional history revealed that her sister also has a similar muscular physique without exercise training. Blood work yielded triglycerides 314 mg/dL (&lt;150 mg/dL), total testosterone 552 ng/dL (2-45 ng/dL), free testosterone 149.0 pg/dL (0.1-6.4 pg/dL), sex-hormone binding globulin 11 nmol/L, insulin at 32.7 uIU/mL, and DHEA-s, 24 H urinary cortisol, IGF-1, TSH, Free T4, A1c and metabolic panel were normal. Transvaginal pelvic ultrasound revealed a mildly hyperechoic 3.2 cm circumscribed nodule in the left ovary suspicious for a Sertoli/Sertoli-Leydig cell tumor. MRI done for further evaluation showed a 3 cm lesion in the left ovary with isointense signal on T1, intermediate signal on T2; postcontrast images revealed a homogeneous enhancement. Patient was referred to gynecology oncology and underwent left salpingo-oophorectomy. Pathology showed a 2.9 cm well circumscribed Sertoli cell tumor. The possibility of adipose tissue re-distribution and increased musculature was evaluated further, and the patient has been diagnosed with FPL type 2.The case highlights a rare cause of hyperandrogenism in a patient with FPL type 2. It is unknown if there are any associations between these two rare diseases. Presentation: Monday, July 14, 2025
https://doi.org/10.1210/jendso/bvaf149.1925Disease module: DeepOracle (Open Targets). Structures: RDKit from PubChem SMILES. Literature: retrieved by DeepSearch across 234,678,978 indexed works (targeted per-candidate search), resolved on OpenAlex.
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