Prenatal Testicular Torsion: Principles of Management

0022-534 7/92/14 73-0670$03.00 /0 Vol. 147, 670-672, March 1992

THE JOURNAL OF UROLOGY

Printed in U.S.A.

Copyright© 1992 by AMERICAN UROLOGICAL ASSOCIATION, INC.

PRENATAL TESTICULAR TORSION: PRINCIPLES OF MANAGEMENT M. T. BRANDT, C. A. SHELDON, J. WACKSMAN

AND

P. MATTHEWS

From the Division of Pediatric Urology, Children's Hospital Medical Center, Cincinnati, Ohio

ABSTRACT

To assess our experience with in utero testicular torsion, we retrospectively studied all newborns presenting in the newborn nursery with a diagnosis of neonatal testicular torsion. A total of 25 consecutive cases of torsion of the spermatic cord in 23 patients was identified and explored on an emergency basis. To our knowledge this represents the largest single institution series and 40% of all of the confirmed cases reported in the literature. No viable testicles were found. We strongly believe that torsion of the testis presenting at birth represents an irreversible intrauterine event best treated in an early elective setting. We suggest an inguinal approach with contralateral orchiopexy in all patients who present with signs and symptoms of prenatal (in utero) testicular torsion. KEY

WORDS: prenatal diagnosis, spermatic cord torsion

Neonatal torsion was first described by Taylor in 1897 to explain the rare occurrence of testicular torsion in the perinatal period. 1 Nearly 100 cases with surgical confirmation have been reported since that time,2 and recently our understanding of this condition has expanded to include 2 separate entities: 1) prenatal (in utero) torsion and 2) postnatal torsion. In prior series this distinction was often overlooked and unappreciated. Our conclusions are based on emergency exploration correlated with pathological evaluation and long-term followup. Recommendations are proposed to minimize the potential for loss of testicular tissue and perioperative complications.

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To assess our experience with in utero testicular torsion we retrospectively studied all patients who presented to the neonatal nursery at our medical center with a diagnosis of testicular torsion between 1976 and 1990. Newborns discharged from the hospital who later returned with torsion were excluded, as were any who had findings of torsion during the first days of life with a normal scrotal examination at birth. Data from 23 patients representing 25 torsed testes were collected. Birth weight, gestational age, maternal parity, laterality, surgical findings and fate of the contralateral testis were tabulated. All newborns underwent immediate exploration when medically stable. Long-term followup was obtained for 20 of 23 patients, and the results were compared to the original surgical findings and treatment.

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Every patient in the series had a documented abnormal scrotal examination at birth. Emergency exploration was performed in 23 patients with 25 torsed testes and the patient age at exploration is shown in figure 1. Of 23 patients 19 were explored within hours of arrival at our hospital and 4 required stabilization in the neonatal intensive care unit from 12 to 168 hours. No viable testes were found and no testes were salvaged as a result of our efforts. Of the testes 17 were clearly necrotic and removed, with pathological findings confirming complete necrosis. Six testes were replaced in the scrotum: 2 (33%) had necrotic intraoperative biopsies and the remaining 4 were described as grossly necrotic by the surgeon. All 6 patients returned for followup and had severe atrophy. A predominance for torsion on the left side was noted (60 to 32%) and 2 patients (8%) had bilateral torsion. In only 1 instance was torsion intravaginal (fig. 2).

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FIG. 2. Frequency distribution of anatomical subsets of prenatal testis torsion.

Accepted for publication August 23, 1991. Read at annual meeting of American Urological Association, Dallas, Texas, May 7-11, 1989. 670

The gestational age by examination and dates was recorded and all neonates were term deliveries except for 1 born at 37 weeks. Birth weights were exceptionally high, with a mean of 3,797 ± 90 gm. (standard error). The range of birth weights for male term neonates is 3,200 to 3,700 gm. A total of 14 patients (60%) was above the 90th percentile in birth weight.

PRENATAL TESTICULAR TORSION

A strong correlation with multiparity was noted in the study group. The mean maternal parity was 3.0 ± 0.3, with parity ranging from 1 to 5. In 5 mothers parity was 5, while 4 were primigravida. Only 1 birth was by cesarean section. Of the 23 newborns 20 were followed by serial physical examinations and 3 were lost to followup. Figure 3 summarizes the data for each subject during the study period. The fate of the sole remaining testis appeared to be unrelated to intervention. Of 13 patients undergoing orchiopexy 12 returned and all (100%) have had normal testicular development. Two patients underwent hydrocele repair during the followup. Of 8 patients undergoing neither contralateral exploration nor orchiopexy 6 returned and all (100%) had normal examinations as well. A progressive tendency to perform contralateral orchiopexy was noted (fig. 4). DISCUSSION

The imprecision of the terms neonatal and perinatal testicular torsion has done much to obfuscate logical treatment of prenatal (in utero) torsion. As a result, only 67 verifiable cases of in utero torsion of the spermatic cord have been reported in the literature. Our experience with 25 consecutive cases of prenatal torsion represents the largest single institution series using the more accurate definition of in utero testicular torsion. Only approximately two-thirds of the prior reports of neonatal

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torsion clearly represented prenatal torsion, making reliable interpretation of many earlier reports and accumulated series difficult. 2 The terms prenatal and postnatal torsion of the spermatic cord should be used whenever possible in place of less precise terminology, since they represent distinct clinical and pathological entities. The presentation of prenatal (in utero) torsion often is enigmatic with minimal discomfort or localized findings. In contrast, most cases of postnatal torsion are marked by tenderness and variable amounts of swelling in the affected gonad of a previously normal scrotum. 3 The lack of fixation of the testicular tunics was first reported by Campbell and fixation is presumed to occur in normal newborns early in the postnatal period. 4 Prenatal torsion is almost exclusively extravaginal, while postnatal extravaginal torsion is extremely rare. Fixation may be delayed in premature infants, although the incidence of prenatal torsion appears to be unrelated to gestational age or low birth weight as our study confirms. 5 • 6 The birth weights of our patients were significantly above the mean expected for term neonates. Recent reviews have noted no association with prematurity or low birth weight. 5 • 6 An unusual association with high birth weight was first reported by Reeves et aF and has not been a subject of recent interest. Our mean weight of 3,797 gm. compares to 3,660 gm. found in 30 patients with neonatal torsion reported on by Burge8 and a mean birth weight for normal male neonates of only 3,400 gm. A plausible mechanism by which higher birth weight could affect the fetal testis has not been demonstrated, although higher pressures in the uterus or birth canal have been proposed. The tendency towards multiparity noted in our study would seem to argue against a relationship between increased stresses associated with labor and delivery and prenatal torsion. Our results with emergency exploration of presumed prenatal (in utero) torsion have yielded no salvaged testicles in 25 consecutive cases. This was true even though 14 patients in the series were less than 12 hours old and 3 were only 6 hours old. Patient age at exploration is more reflective of the referral process than any other single factor, since the majority of neonates were explored on arrival at our institution. Das and Singer recently reviewed the literature and concluded that the possibility of finding a viable testis when exploring in utero torsion is extremely remote. 2 Clearly, efforts should be directed at minimizing any potential perioperative complications by scheduling an elective operation after the neonate is medically stable. Prompt exploration, preferably through an inguinal incision, remains an important step in the management of presumed prenatal torsion. By avoiding undue delays exploration minimizes controversy associated with potential immunological effects of torsion on the contralateral testis. Exploration also establishes a diagnosis from a differential, which includes epididymyo-orchitis, hydrocele, meconium peritonitis, hernia, hematocele, torsion of the appendix testis or epididymis and benign or malignant neoplasms. Although benign processes are much more common than infantile scrotal neoplasms, the possibility of neonatal testicular9 • 10 or ectopic visceral 11 neoplasm exists warranting an inguinal approach . The loss of the sole remaining testis in a patient after testicular torsion is a catastrophic event from an endocrine and fertility standpoint. We currently perform contralateral orchiopexy in all patients, since our results show no deleterious effect of contralateral neonatal orchiopexy. Interestingly, the patients followed without contralateral orchiopexy in our study also have had normal testicular development but the presence of a contralateral anatomical abnormality cannot be ruled out. lntravaginal prenatal torsion certainly occurs 12 (we report 1 case) and the implication for the contralateral testis is well known. Bilateral synchronous as well as asynchronous extravaginal torsion has been clearly reported. 13 • 14 Of additional

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note, extravaginal torsion has been reported in male adolescents. 5· 15 Our study adds to the growing body of evidence indicating that prenatal testicular torsion represents an intrauterine event, and that the possibility of testicular salvage is negligible. Therapy should be directed toward prompt and safe establishment of the diagnosis, and protection of the remaining gonad. We are convinced that elective early inguinal exploration with orchiectomy and contralateral scrotal orchiopexy best accomplish these goals. REFERENCES

1. Taylor, M. R.: A case of testicle· strangulated at birth; castration; recovery. Brit. Med. J., 1: 458, 1897. 2. Das, S. and Singer, A.: Controversies of-peri-natal--torsion of the spermatic cord: a review, survey and recommendations. J. Urol., 143: 231, 1990. 3. Bret, A.: Infarction and torsion of the testicle in the newborn. Rev. Fr. Gynec. Obst., 63: 83, 1968. 4. Campbell, M. F.: The male genital tract and the female urethra. In: Urology, 3rd ed. Edited by M. F. Campbell and J. H. Harrison. Philadelphia: W. B. Saunders Co., vol. 2, sect. XII, chapt. 44, pp. 1834-1889, 1970.

5. Watson, R. A.: Torsion of spermatic cord in neonate. Urology, 5: 439, 1975. 6. Visani, S., Gentile, R. L. and Vijaya, L.: Perinatal torsion of spermatic cord. Urology, 6: 360, 1975. 7. Reeves, H. H., Sigler, R. M., Hahn, H. B., Jr. and Lynn, H. B.: Torsion of the spermatic cord in the newborn. Amer. J. Dis. Child., 110: 676, 1965. 8. Burge, D. M.: Neonatal testicular torsion and infarction: aetiology and management. Brit. J. Urol., 59: 70, 1987. 9. Uehling, D. T., Smith, J.E., Logan, R. and Hafez, G.-R.: Newborn granulosa cell tumor of the testis. J. Urol., 138: 385, 1987. 10. Lawrence, W. D., Young, R.H. and Scully, R. E.: Juvenile granulosa cell tumor of the infantile testis: a report of 14 cases. Amer. J. Surg. Path., 9: 87, 1985. 11. Mininberg, D. T. and Dattwyler, B.: Ectopic adrenal tumor presenting as torsion of the spermatic cord in a newborn infant. J. BroL, 109: 1037, 1973. 12. Jerkins, G. R., Noe, H. N., Hollabaugh, R. S. and Allen, R. G.: Spermatic cord torsion in the neonate. J. Urol., 129: 121, 1983. 13. LaQuaglia, M. P., Bauer, S. B., Eraklis, A., Feins, N. and Mandell, J.: Bilateral neonatal torsion. J. Urol., 138: 1051, 1987. 14. Kay, R., Strong, D. W. and Tank, E. S.: Bilateral spermatic cord torsion in the neonate. J. Urol., 123: 293, 1980. 15. Barker, K. and Raper, F. P.: Torsion of the testis. Brit. J. Urol., 36: 35, 1964.