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Background <p>Testicular regression syndrome (TRS), also known as vanishing testis syndrome, is a common cause of the non-palpable testis in children. Two major theories are proposed to explain it: a lack of blood supply after late birth or primary agenesis/atrophy in the development. The best way to determine these causes and how to operate is to examine the tissue removed during the surgery.</p> Objective <p>We reviewed the literature to estimate the number of cases of TRS presenting with evidence of previous injury to blood supplies in comparison with those with simple atrophy.</p> Methods <p>According to the PRISMA 2020 guidelines, PubMed, Web of Science, and Google Scholar were searched. Included were observational studies that reported on detailed histology of at least 10 excised TRS remnants that had data on fibrosis, dystrophic calcification, hemosiderin deposition, seminiferous tubules, and viable germ cells. We pooled proportions using 95% confidence intervals (CIs) with a DerSimonian-Laird random-effects model.</p> Results <p>Based on our strict selection criteria, ten retrospective cohorts evaluating 1,907 examined testicular remnants met our inclusion criteria. In 93.8% (95% CI 90.4–96.3%, I2 = 81%) of the cases, remnants displayed clear evidence of a vascular injury (fibrosis, dystrophic calcification, or hemosiderin). Primary atrophy with an absence of these vascular markers was seen in only 6.2% (95% CI 3.7–9.6%; I2 = 74) cases. Residual seminiferous tubules were seen in 8.9% (95% CI 6.1–12.4%), and viable germ cells were seen in 4.7% (95% CI 2.9–7.1%). Consistent secondary observations comprised a predominance of left side occurrences (68–80%) and a closed internal inguinal ring (&gt; 95%). Sensitivity analyses agreed with these findings.</p> Conclusion <p>The burden of histopathologic data suggests that a vascular event (e.g., antenatal torsion) is the primary cause of TRS in more than 93% of cases. Simple atrophy or agenesis is very rare. While the absolute risk of malignancy remains low, the identification of viable germ cells in nearly 5% of cases suggests that surgical excision of easily accessible remnants may be considered to minimize the long-term cancer risk.</p>

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Evidence for antenatal vascular insult in testicular regression syndrome: a systematic review and meta-analysis of histopathology in 1,907 patients

  • 期刊论文

  • 出版模式：

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  • 发表日期：

    2026年5月12日

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  • Madan Mohan Reddy Kambaiahgari,
  • Malarmannan Mathiyalagan,
  • Lavanya Selvaraj Kaveri,
  • Lakshmana Raman

摘要

Background

Testicular regression syndrome (TRS), also known as vanishing testis syndrome, is a common cause of the non-palpable testis in children. Two major theories are proposed to explain it: a lack of blood supply after late birth or primary agenesis/atrophy in the development. The best way to determine these causes and how to operate is to examine the tissue removed during the surgery.

Objective

We reviewed the literature to estimate the number of cases of TRS presenting with evidence of previous injury to blood supplies in comparison with those with simple atrophy.

Methods

According to the PRISMA 2020 guidelines, PubMed, Web of Science, and Google Scholar were searched. Included were observational studies that reported on detailed histology of at least 10 excised TRS remnants that had data on fibrosis, dystrophic calcification, hemosiderin deposition, seminiferous tubules, and viable germ cells. We pooled proportions using 95% confidence intervals (CIs) with a DerSimonian-Laird random-effects model.

Results

Based on our strict selection criteria, ten retrospective cohorts evaluating 1,907 examined testicular remnants met our inclusion criteria. In 93.8% (95% CI 90.4–96.3%, I2 = 81%) of the cases, remnants displayed clear evidence of a vascular injury (fibrosis, dystrophic calcification, or hemosiderin). Primary atrophy with an absence of these vascular markers was seen in only 6.2% (95% CI 3.7–9.6%; I2 = 74) cases. Residual seminiferous tubules were seen in 8.9% (95% CI 6.1–12.4%), and viable germ cells were seen in 4.7% (95% CI 2.9–7.1%). Consistent secondary observations comprised a predominance of left side occurrences (68–80%) and a closed internal inguinal ring (> 95%). Sensitivity analyses agreed with these findings.

Conclusion

The burden of histopathologic data suggests that a vascular event (e.g., antenatal torsion) is the primary cause of TRS in more than 93% of cases. Simple atrophy or agenesis is very rare. While the absolute risk of malignancy remains low, the identification of viable germ cells in nearly 5% of cases suggests that surgical excision of easily accessible remnants may be considered to minimize the long-term cancer risk.