Skip to main content

Advertisement

Log in

Presence of viable germ cells in testicular regression syndrome remnants: Is routine excision indicated? A systematic review

  • Original Article
  • Published:
Pediatric Surgery International Aims and scope Submit manuscript

Abstract

There is no consensus in the literature about the necessity for excision of testicular remnants in the context of surgery for an impalpable testis and testicular regression syndrome (TRS). The incidence of germ cells (GCs) within these nubbins varies between 0 and 16% in previously published series. There is a hypothetical potential future malignancy risk, although there has been only one previously described isolated report of intratubular germ-cell neoplasia. Our aim was to ascertain an accurate incidence of GCs and seminiferous tubules (SNTs) within excised nubbins and hence guide evidence-based practice. The systematic review protocol was designed according to the PRISMA guidelines, and subsequently published by the PROSPERO database after review (CRD42013006034). The primary outcome measure was the incidence of GCs and the secondary outcome was the incidence of SNTs. The comprehensive systematic review included articles published between 1980 and 2016 in all the relevant databases using specific search parameters and terms. Strict inclusion and exclusion criteria were ultilised to identify articles relevant to the review questions. Twenty-nine paediatric studies with a total of 1455 specimens were included in the systematic review. The mean age of the patients undergoing nubbin resection was 33 months and the TRS specimen was more commonly excised from the left (68%). The incidence of SNTs was 10.7% (156/1455) and the incidence of GCs, 5.3% (77/1455). Histological analysis excluding the presence of either SNTs or GCs was consistent with TRS, fibrosis, calcification or haemosiderin deposits. There is limited evidence on subset analysis that GCs and SNTs may persist with increasing patient age. This systematic review has identified that 1 in 20 of resected testicular remnants has viable GCs and 1 in 10 has SNTs present. There is insufficiently strong evidence for the persistence of GCs and SNTs with time or future malignant potential. Intra-abdominal TRS specimens may contain more elements and, therefore, require excision, although this is based on limited evidence. However, there is no available strong evidence to determine that a TRS specimen requires routine excision in an inguinal or scrotal position.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Fig. 1
Fig. 2

Similar content being viewed by others

References

  1. Vikraman J, Hutson JM, Li R, Thorup J (2016) The undescended testis: Clinical management and scientific advances. Semin Pediatr Surg 25:241–248

    Article  PubMed  Google Scholar 

  2. Hegarty PK, Mushtaq I, Sebire NJ (2007) Natural history of testicular regression syndrome and consequences for clinical management. J Pediatr Urol 3:206–208

    Article  PubMed  Google Scholar 

  3. Law H, Mushtaq I, Wingrove K, Malone M, Sebire NJ (2006) Histopathological features of testicular regression syndrome: relation to patient age and implications for management. Fetal Pediatr Pathol 25:119–129

    Article  CAS  PubMed  Google Scholar 

  4. Rozanski TA, Wojno KJ, Bloom DA (1996) The remnant orchiectomy. J Urol 155:712–713 (discussion 4)

    Article  CAS  PubMed  Google Scholar 

  5. Storm D, Redden T, Aguiar M, Wilkerson M, Jordan G, Sumfest J (2007) Histologic evaluation of the testicular remnant associated with the vanishing testes syndrome: is surgical management necessary? Urology 70:1204–1206

    Article  PubMed  Google Scholar 

  6. Ueda N, Shiroyanagi Y, Suzuki H, Kim WJ, Yamazaki Y, Tanaka Y (2013) The value of finding a closed internal ring on laparoscopy in unilateral nonpalpable testis. J Pediatr Surg 48:542–546

    Article  PubMed  Google Scholar 

  7. Renzulli Ii JF, Shetty R, Mangray S, Anderson KR, Weiss RM, Caldamone AA (2005) Clinical and histological significance of the testicular remnant found on inguinal exploration after diagnostic laparoscopy in the absence of a patent processus vaginalis. J Urol 174:1584–1586

    Article  Google Scholar 

  8. Emir H, Ayik B, Elicevik M, Buyukunal C, Danismend N, Dervisoglu S et al (2007) Histological evaluation of the testicular nubbins in patients with nonpalpable testis: assessment of etiology and surgical approach. Pediatr Surg Int 23:41–44

    Article  PubMed  Google Scholar 

  9. Bader MI, Peeraully R, Ba’ath M, McPartland J, Baillie C (2011) The testicular regression syndrome–do remnants require routine excision? J Pediatr Surg 46:384–386

    Article  PubMed  Google Scholar 

  10. Belman AB, Rushton HG (2001) Is the vanished testis always a scrotal event? BJU Int 87:480–483

    Article  CAS  PubMed  Google Scholar 

  11. Cendron M, Schned AR, Ellsworth PI (1998) Histological evaluation of the testicular nubbin in the vanishing testis syndrome. J Urol 160:1161–1162 (discussion 3)

    Article  CAS  PubMed  Google Scholar 

  12. Chandrasekharam VV (2005) Laparoscopy vs inguinal exploration for nonpalpable undescended testis. Indian J Pediatr 72:1021–1023

    Article  CAS  PubMed  Google Scholar 

  13. De Luna AM, Ortenberg J, Craver RD (2003) Exploration for testicular remnants: implications of residual seminiferous tubules and crossed testicular ectopia. J Urol 169:1486–1489

    Article  PubMed  Google Scholar 

  14. Froeling FM, Sorber MJ, de la Rosette JJ, de Vries JD (1994) The nonpalpable testis and the changing role of laparoscopy. Urology 43:222–227

    Article  CAS  PubMed  Google Scholar 

  15. Gatti JM, Cooper CS, Kirsch AJ (2003) Bimanual digital rectal examination for the evaluation of the nonpalpable testis. J Urol 170:207–210

    Article  PubMed  Google Scholar 

  16. Godbole PP, Morecroft JA, Mackinnon AE (1997) Laparoscopy for the impalpable testis. Br J Surg 84:1430–1432

    Article  CAS  PubMed  Google Scholar 

  17. Grady RW, Mitchell ME, Carr MC (1998) Laparoscopic and histologic evaluation of the inguinal vanishing testis. Urology 52:866–869

    Article  CAS  PubMed  Google Scholar 

  18. Humphrey GM, Najmaldin AS, Thomas DF (1998) Laparoscopy in the management of the impalpable undescended testis. Br J Surg 85:983–985

    Article  CAS  PubMed  Google Scholar 

  19. Kojima Y, Mizuno K, Imura M, Kato T, Kohri K, Hayashi Y (2009) Laparoscopic orchiectomy and subsequent internal ring closure for extra-abdominal testicular nubbin in children. Urology 73:515–519 (discussion 9–20)

    Article  PubMed  Google Scholar 

  20. Mizuno K, Kojima Y, Kamisawa H, Kurokawa S, Moritoki Y, Nishio H et al (2012) Feasible etiology of vanishing testis regarding disturbance of testicular development: histopathological and immunohistochemical evaluation of testicular nubbins. Int J Urol Off J Jpn Urol Assoc 19:450–456

    CAS  Google Scholar 

  21. Naslund MJ, Gearhart JP, Jeffs RD (1989) Laparoscopy: its selected use in patients with unilateral nonpalpable testis after human chorionic gonadotropin stimulation. J Urol 142:108–110

    Article  CAS  PubMed  Google Scholar 

  22. Nataraja RM, Asher CM, Nash R, Murphy FL (2015) Is routine excision of testicular remnants in testicular regression syndrome indicated? J Pediatr Urol 11:151 e1–5

    Article  PubMed  Google Scholar 

  23. Papparella A, Romano M, Noviello C, Cobellis G, Nino F, Del Monaco C et al (2010) The value of laparoscopy in the management of non-palpable testis. J Pediatr Urol 6:550–554

    Article  CAS  PubMed  Google Scholar 

  24. Singh RR, Rajimwale A, Nour S (2011) Laparoscopic management of impalpable testes: comparison of different techniques. Pediatr Surg Int 27:1327–1330

    Article  PubMed  Google Scholar 

  25. Spires SE, Woolums CS, Pulito AR, Spires SM (2000) Testicular regression syndrome: a clinical and pathologic study of 11 cases. Arch Pathol Lab Med 124:694–698

    CAS  PubMed  Google Scholar 

  26. Sutcliffe JR, Wilson-Storey D, Smith NM (1996) Ante-natal testicular torsion: only one cause of the testicular regression syndrome? J R Coll Surg Edinb 41:99–101

    CAS  PubMed  Google Scholar 

  27. Tekant G, Emir H, Froglu E, Akman M, Buyukunal C, Danismend N et al (2001) Experience with laparoscopy in nonpalpable testis. Horm Res 55:52

    CAS  PubMed  Google Scholar 

  28. Van Savage JG (2001) Avoidance of inguinal incision in laparoscopically confirmed vanishing testis syndrome. J Urol 166:1421–1424

    Article  PubMed  Google Scholar 

  29. Antic T, Hyjek EM, Taxy JB (2011) The vanishing testis: a histomorphologic and clinical assessment. Am J Clin Pathol 136:872–880

    Article  PubMed  Google Scholar 

  30. Thorup J, Kvist K, Clasen-Linde E, Hutson JM, Cortes D (2015) Serum inhibin B values in boys with unilateral vanished testis or unilateral cryptorchidism. J Urol 193:1632–1636

    Article  CAS  PubMed  Google Scholar 

  31. Snodgrass W, Chen K, Harrison C (2004) Initial scrotal incision for unilateral nonpalpable testis. J Urol 172:1742–1745 (discussion 5)

    Article  PubMed  Google Scholar 

  32. Liberati A, Altman DG, Tetzlaff J, Mulrow C, Gotzsche PC, Ioannidis JP et al (2009) The PRISMA statement for reporting systematic reviews and meta-analyses of studies that evaluate healthcare interventions: explanation and elaboration. Bmj 339:b2700

    Article  PubMed  PubMed Central  Google Scholar 

  33. PROSPERO (2012) International prospective register of systematic reviews. https://www.crd.york.ac.uk/prospero/. Accessed 1 July 2016

  34. Slim K, Nini E, Forestier D, Kwiatkowski F, Panis Y, Chipponi J (2003) Methodological index for non-randomized studies (minors): development and validation of a new instrument. ANZ J Surg 73:712–716

    Article  PubMed  Google Scholar 

  35. Rowe N (2011) 327 Pathological analysis of testicular remants in nonpalpable undescended testis. J Urol 185:132

    Article  Google Scholar 

  36. Pettersson A, Richiardi L, Nordenskjold A, Kaijser M, Akre O (2007) Age at surgery for undescended testis and risk of testicular cancer. N Engl J Med 356:1835–1841

    Article  CAS  PubMed  Google Scholar 

  37. Hutson JM (2013) Journal of pediatric surgery-sponsored Fred McLoed lecture. Undescended testis: the underlying mechanisms and the effects on germ cells that cause infertility and cancer. J Pediatr Surg 48:903–908

    Article  PubMed  Google Scholar 

  38. Wright JE (1986) Impalpable testes: a review of 100 boys. J Pediatr Surg 21:151–153

    Article  CAS  PubMed  Google Scholar 

  39. Smith NM, Byard RW, Bourne AJ (1991) Testicular regression syndrome–a pathological study of 77 cases. Histopathology 19:269–272

    Article  CAS  PubMed  Google Scholar 

  40. Manivel JC, Simonton S, Wold LE, Dehner LP (1988) Absence of intratubular germ cell neoplasia in testicular yolk sac tumors in children. A histochemical and immunohistochemical study. Arch Pathol Lab Med 112:641–645

    CAS  PubMed  Google Scholar 

  41. Klein FA, Melamed MR, Whitmore WF (1985) Jr. Intratubular malignant germ cells (carcinoma in situ) accompanying invasive testicular germ cell tumors. J Urol 133:413–415

    Article  CAS  PubMed  Google Scholar 

  42. Sharma P, Dhillon J, Sexton WJ (2015) Intratubular germ cell neoplasia of the testis, bilateral testicular cancer, and aberrant histologies. Urol Clin North Am 42:277–275

    Article  PubMed  Google Scholar 

  43. Elder JS (1994) Laparoscopy for impalpable testes: significance of the patent processus vaginalis. J Urol 152:776–778

    Article  CAS  PubMed  Google Scholar 

  44. Snodgrass WT, Yucel S, Ziada A (2007) Scrotal exploration for unilateral nonpalpable testis. J Urol 178:1718–1721

    Article  PubMed  Google Scholar 

  45. Rajimwale A, Brant WO, Koyle MA (2004) High scrotal (Bianchi) single-incision orchidopexy: a “tailored” approach to the palpable undescended testis. Pediatr Surg Int 20:618 622

    Article  Google Scholar 

  46. Samuel DG, Izzidien AY (2008) Bianchi high scrotal approach revisited. Pediatr Surg Int 24:741–744

    Article  PubMed  Google Scholar 

  47. Gordon M, Cervellione RM, Morabito A, Bianchi A (2010) 20 years of transcrotal orchidopexy for undescended testis: results and outcomes. J Pediatr Urol 6:506–512

    Article  PubMed  Google Scholar 

  48. Esposito C, Iacobelli S, Farina A, Perricone F, Savanelli A, Settimi A (2010) Exploration of inguinal canal is mandatory in cases of non palpable testis if laparoscopy shows elements entering a closed inguinal ring. Eur J Pediatr Surg 20:138–139

    Article  CAS  PubMed  Google Scholar 

  49. Gonzalez KW, Dalton BG, Snyder CL, Leys CM, St Peter SD, Ostlie DJ (2016) The anatomic findings during operative exploration for non-palpable testes: a prospective evaluation. J Pediatr Surg 51:128–130

    Article  PubMed  Google Scholar 

  50. Tasian GE, Hittelman AB, Kim GE, DiSandro MJ, Baskin LS (2009) Age at orchiopexy and testis palpability predict germ and Leydig cell loss: clinical predictors of adverse histological features of cryptorchidism. J Urol 182:704–709

    Article  PubMed  Google Scholar 

  51. Park KH, Lee JH, Han JJ, Lee SD, Song SY (2007) Histological evidences suggest recommending orchiopexy within the first year of life for children with unilateral inguinal cryptorchid testis. Int J Urol Off J Jpn Urol Assoc 14:616–621

    Google Scholar 

  52. Mengel W, Hienz HA, Sippe WG, Hecker WC (1974) Studies on cryptorchidism: a comparison of histological findings in the germinative epithelium before and after the second year of life. J Pediatr Surg 9:445–450

    Article  CAS  PubMed  Google Scholar 

Download references

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Ramesh Mark Nataraja.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Nataraja, R.M., Yeap, E., Healy, C.J. et al. Presence of viable germ cells in testicular regression syndrome remnants: Is routine excision indicated? A systematic review. Pediatr Surg Int 34, 353–361 (2018). https://doi.org/10.1007/s00383-017-4206-0

Download citation

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00383-017-4206-0

Keywords

Navigation