Subscribe to RSS
DOI: 10.1055/s-0041-1739417
Serial Inhibin B Measurements in Boys with Congenital Monorchism Indicate Compensatory Testicular Hypertrophy in Early Infancy
Funding This work was supported by the Novo Nordisk Foundation, grant number NNF SA170030576.Abstract
Aim Congenital monorchism is considered a condition in which an initially normal testis has existed but subsequently atrophied and disappeared due to a third trimester catastrophe (presumably torsion). Since inhibin B concentrations appear related to Sertoli and germ cells number, we evaluated pre- and postoperative inhibin B of boys with congenital monorchism to determine whether the well-known hypertrophy of the contralateral testis was reflected in inhibin B concentrations.
Materials and Methods Twenty-seven boys consecutively diagnosed with congenital monorchism (median age 12 months) underwent follow-up with reproductive hormones 1 year postoperatively (median age 25 months). The results were compared with inhibin B of 225 boys with congenital nonsyndromic unilateral cryptorchidism, by converting values to multiple of the median (MoM) for age in normal boys.
Results Ten boys (37%) had blind-ending vessels and ductus deferens (vanished testis) and the remaining (63%) had testicular remnants. At the time of diagnostic procedure, monorchid boys did not have significantly lower inhibin B (median 114, range 20–208) than unilateral cryptorchid boys (136, 47–393) (p = 0.27). During follow-up, MoM values of inhibin B increased in monorchid boys (median 0.59 to 0.98) and in unilateral cryptorchid boys (0.69 to 0.89) (both p < 0.0001). Compared with the concentration at surgery, an additional 44% monorchid boys had inhibin B MoM values higher than 1.0, whereas only additional 23% of unilateral cryptorchid boys exhibited such values (p = 0.04).
Conclusion Generally, inhibin B MoM values were normalized during follow-up in boys with congenital monorchism, reflecting compensatory hypertrophy within the first 2.5 years of life. The compensatory capacity to increase was better in monorchism than in unilateral cryptorchidism.
Keywords
monorchism - vanished testis - testicular regression syndrome - inhibin B - compensatory testicular hypertrophyPublication History
Received: 17 July 2021
Accepted: 06 September 2021
Article published online:
30 November 2021
© 2021. Thieme. All rights reserved.
Georg Thieme Verlag KG
Rüdigerstraße 14, 70469 Stuttgart, Germany
-
References
- 1 Pirgon Ö, Dündar BN. Vanishing testes: a literature review. J Clin Res Pediatr Endocrinol 2012; 4 (03) 116-120
- 2 Smith NM, Byard RW, Bourne AJ. Testicular regression syndrome–a pathological study of 77 cases. Histopathology 1991; 19 (03) 269-272
- 3 Law H, Mushtaq I, Wingrove K, Malone M, Sebire NJ. Histopathological features of testicular regression syndrome: relation to patient age and implications for management. Fetal Pediatr Pathol 2006; 25 (02) 119-129
- 4 Cortes D, Thorup JM, Lenz K, Beck BL, Nielsen OH. Laparoscopy in 100 consecutive patients with 128 impalpable testes. Br J Urol 1995; 75 (03) 281-287
- 5 Kirsch AJ, Escala J, Duckett JW. et al. Surgical management of the nonpalpable testis: the Children's Hospital of Philadelphia experience. J Urol 1998; 159 (04) 1340-1343
- 6 Kumar R, Mandal KC, Halder P, Hadiuzzaman M, Mukhopadhyay M, Mukhopadhyay B. Laparoscopy in the evaluation of impalpable testes and its short-term outcomes: a 7 years' experience. J Indian Assoc Pediatr Surg 2017; 22 (04) 232-236
- 7 Cisek LJ, Peters CA, Atala A, Bauer SB, Diamond DA, Retik AB. Current findings in diagnostic laparoscopic evaluation of the nonpalpable testis. J Urol 1998;160(3 Pt 2):1145–1149, discussion 1150
- 8 Braga LH, Kim S, Farrokhyar F, Lorenzo AJ. Is there an optimal contralateral testicular cut-off size that predicts monorchism in boys with nonpalpable testicles?. J Pediatr Urol 2014; 10 (04) 693-698
- 9 Laron Z, Dickerman Z, Ritterman I, Kaufman H. Follow-up of boys with unilateral compensatory testicular hypertrophy. Fertil Steril 1980; 33 (03) 297-301
- 10 Huff DS, Snyder III HM, Hadziselimovic F, Blyth B, Duckett JW. An absent testis is associated with contralateral testicular hypertrophy. J Urol 1992; 148 (2 Pt 2): 627-628
- 11 Snodgrass WT, Yucel S, Ziada A. Scrotal exploration for unilateral nonpalpable testis. J Urol 2007; 178 (4 Pt 2): 1718-1721
- 12 Koff SA. Does compensatory testicular enlargement predict monorchism?. J Urol 1991; 146 (2 (Pt 2)): 632-633
- 13 Hutson JM, Southwell BR, Li R. et al. The regulation of testicular descent and the effects of cryptorchidism. Endocr Rev 2013; 34 (05) 725-752
- 14 Hildorf S, Dong L, Thorup J, Clasen-Linde E, Yding Andersen C, Cortes D. Sertoli cell number correlates with serum inhibin B in infant cryptorchid boys. Sex Dev 2019; 13 (02) 74-82
- 15 Orth JM, Gunsalus GL, Lamperti AA. Evidence from Sertoli cell-depleted rats indicates that spermatid number in adults depends on numbers of Sertoli cells produced during perinatal development. Endocrinology 1988; 122 (03) 787-794
- 16 Main KM, Toppari J, Suomi AM. et al. Larger testes and higher inhibin B levels in Finnish than in Danish newborn boys. J Clin Endocrinol Metab 2006; 91 (07) 2732-2737
- 17 Jørgensen N, Liu F, Andersson AM. et al. Serum inhibin-b in fertile men is strongly correlated with low but not high sperm counts: a coordinated study of 1,797 European and US men. Fertil Steril 2010; 94 (06) 2128-2134
- 18 Hegarty PK, Mushtaq I, Sebire NJ. Natural history of testicular regression syndrome and consequences for clinical management. J Pediatr Urol 2007; 3 (03) 206-208
- 19 Kraft KH, Bhargava N, Schast AW, Canning DA, Kolon TF. Histological examination of solitary contralateral descended testis in congenital absence of testis. J Urol 2012; 187 (02) 676-680
- 20 Thorup J, Petersen BL, Kvist K, Cortes D. Bilateral vanished testes diagnosed with a single blood sample showing very high gonadotropins (follicle-stimulating hormone and luteinizing hormone) and very low inhibin B. Scand J Urol Nephrol 2011; 45 (06) 425-431
- 21 Gerbo M, Crigger C, Samadi Y, Ost MC, Al-Omar O. Prenatally diagnosed testicular torsion: a rare condition that causes dilemma in management. Case Rep Pediatr 2021; 2021: 8825763
- 22 Hutson J, Thorup J, Beasley S. Descent of the Testis. 2nd ed.. Switzerland: Springer International Publishing; 2016
- 23 Thorup J, Kvist K, Clasen-Linde E, Hutson JM, Cortes D. Serum inhibin B values in boys with unilateral vanished testis or unilateral cryptorchidism. J Urol 2015; 193 (05) 1632-1636
- 24 Hildorf S, Cortes D, Clasen-Linde E, Fossum M, Thorup J. The impact of early and successful orchidopexy on hormonal follow-up for 208 boys with bilateral non-syndromic cryptorchidism. Pediatr Surg Int 2021; 37 (03) 339-345
- 25 Lee PA, O'Leary LA, Songer NJ, Coughlin MT, Bellinger MF, LaPorte RE. Paternity after unilateral cryptorchidism: a controlled study. Pediatrics 1996; 98 (4 Pt 1): 676-679
- 26 Hadziselimovic F, Verkauskas G, Vincel B, Krey G, Zachariou Z. Abnormal histology in testis from prepubertal boys with monorchidism. Basic Clin Androl 2020; 30: 11
- 27 Gaudino R, Cavarzere P, Camilot M, Teofoli F, Zampieri N, Tatò L. Prepubertal serum inhibin B in cryptorchid infants and in monorchid boys with compensatory testicular hypertrophy. Fertil Steril 2008; 90 (06) 2217-2221
- 28 Grinspon RP, Habib C, Bedecarrás P, Gottlieb S, Rey RA. Compensatory function of the remaining testis is dissociated in boys and adolescents with monorchidism. Eur J Endocrinol 2016; 174 (03) 399-407
- 29 Spires SE, Woolums CS, Pulito AR, Spires SM. Testicular regression syndrome: a clinical and pathologic study of 11 cases. Arch Pathol Lab Med 2000; 124 (05) 694-698
- 30 De Luna AM, Ortenberg J, Craver RD. Exploration for testicular remnants: implications of residual seminiferous tubules and crossed testicular ectopia. J Urol 2003; 169 (04) 1486-1489