Papers

Testicular neoplasia in cryptorchid boys at primary surgery: case series

Dina Cortes, registrar ^a,  Jakob Visfeldt, professor ^b,  Henrik Møller, head ^c,  Jørgen Thorup, consultant paediatric surgeon ^a. 

^a Department of Paediatric Surgery, 4072, Rigshospitalet, DK-2100 Copenhagen Ø, Denmark, ^b Department of Pathology, Rigshospitalet, ^c Centre for Research in Health and Social Statistics, Danish National Research Foundation, DK-2100 Copenhagen Ø, Denmark

Correspondence to: J Thorup J-Thorup{at}rh.dk

Cryptorchidism is associated with testicular cancer; the lifetime risk of 2-3% is about four times higher than in the general population. ^{1 2} Some groups of cryptorchid patients may have an especially high risk of testicular cancer.³ Testicular carcinoma in situ is a well described histological pattern that precedes germ cell tumours. ^{1 4} We investigated whether it is possible at primary surgery to identify cryptorchid boys who have testicular neoplasia and therefore are at high risk of testicular cancer.

	Method and results

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We examined 1535 consecutive specimens of testicular tissue that were obtained from undescended testes at surgery for cryptorchidism in 1249 boys between 1971 and 1998. Previous reports have described 1026 of the biopsies in detail. ^{2 4} No patient had fallopian tubes or a uterus.

The table shows the total occurrence of testicular neoplasia at surgery for cryptorchidism. There was one case of invasive germ cell tumour, six cases of testicular carcinoma in situ, and one Sertoli cell tumour. Of the eight testes with neoplasia from seven patients, three neoplasms were diagnosed in intra-abdominal testes (cases 1-3), four occurred in three boys with abnormal external genitalia other than cryptorchidism (cases 4-6), and two were diagnosed in boys with known abnormal karyotype (cases 3 and 7).

All the case reports were reviewed. In the 97 boys (124 specimens) with intra-abdominal testes, three had known abnormal karyotype; 46,XY/47,XYY (case 3), 46,XYdel(11p), and 46,XY,13/20 unbalanced translocation; five had abnormal external genitalia, two with hypospadias, one with epispadias, and two with small penis and scrotum. Furthermore, 28 patients (38 specimens) had abnormal external genitalia but no intra-abdominal testes: 14 with hypospadias, two with epispadias, two with some ambiguity of the external genitalia (cases 4 and 5), two with hypoplastic scrotum, and eight with small penis and scrotum, of whom four had Kallmann's syndrome and one had testicular neoplasia (case 6). Moreover, 10 patients (14 specimens) had known abnormal karyotype: seven with 47,XXY; one with 45,X/46,XY (case 7); one with 46,XX; and one with 47,XYY.

At surgery for cryptorchidism the risk of testicular neoplasia was 7/135 (5.2%) in patients with intra-abdominal testis, abnormal external genitalia other than cryptorchidism, or diagnosed abnormal karyotype. In contrast, no case of testicular neoplasia occurred in 1114 patients without these characteristics. The figures are significantly different (Fischer's exact test, P<0.001).

At surgery the risk of testicular neoplasia was 4/286 (1.4%) in the 286 patients operated on for bilateral cryptorchidism, which is not significantly different from 3/963 (0.3%) in the 963 patients with unilateral cryptorchidism (P=0.10). However, intra-abdominal testis, abnormal external genitalia, or abnormal karyotype were reported in 42 (14.7%) patients operated on for bilateral cryptorchidism, compared with 93 (9.7%) patients with unilateral cryptorchidism (P<0.05).

	Comment

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All seven boys with testicular neoplasia at surgery for cryptorchidism had intra-abdominal testis, abnormal external genitalia, or known abnormal karyotype. No case of testicular neoplasia was found in patients without these characteristics. To our knowledge, this information is new, but it is not inconsistent with the literature. ^{1 4 5} At operation for cryptorchidism the surgeon may elect to request a biopsy of testes with a high risk of neoplasia. This bias may explain the conflicting interpretation of our previously published results² and the results published by Swerdlow et al.³ In clinical practice, we recommend a testicular biopsy at surgery for cryptorchidism if the boy has intra-abdominal testis, abnormal external genitalia, or known abnormal karyotype. These abnormalities were more common in patients with bilateral than with unilateral cryptorchism. The suggested procedure may diagnose testicular neoplasia at surgery for cryptorchidism and some patients may be treated for testicular neoplasia before invasive cancer develops.

	Acknowledgments

   Contributors: JV verified the pathological data and contributed to interpretation and reporting. HM contributed to interpretation and reporting. JT verified the clinical data and contributed to study design, interpretation, and reporting. DC took the initiative in this study, verified the pathological and clinical data, and was responsible for interpreting the results and drafting the paper. All authors are guarantors.

	Footnotes

Funding: No specific funding.

Competing interests: None declared.

	References

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1.	Cortes D. Cryptorchidismaspects of pathogenesis, histology and treatment. Scand J Urol Nephrol 1998; 32(suppl 196): 1-54.
2.	Møller H, Cortes D, Engholm G, Thorup J. Risk of testicular cancer with cryptorchidism and with testicular biopsy: cohort study. BMJ 1998; 317: 729[Free Full Text].
3.	Swerdlow AJ, Higgins CD, Pike MC. Risk of cancer in cohort of boys with cryptorchidism. BMJ 1997; 314: 1507-1511[Abstract/Free Full Text].
4.	Cortes D, Thorup J, Frisch M, Møller H, Jacobsen GK, Beck BL. Examination for intratubular germ cell neoplasia (ITGCN) at operation for undescended testis in boys. A follow-up study. J Urol 1994; 151: 722-725[Medline].
5.	Müller J, Skakkebæk NE, Ritzén M, Plöen L, Petersen KE. Carcinoma in situ of the testis in children with 45,X/46,XY gonadal dysgenesis. J Pediatr 1985; 106: 431-436[Medline].

(Accepted 12 May 1999)