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Scand J Urol Nephrol25: 3 19-320, 199 1 LONG TERM COMPLICATION OF UNTREATED BILATERAL TESTICULAR TORSION IN THE NEWBORN Case Report Samson Liu, Simon Andrew Holmes and Nicholas Paul Cohen From the Department of Urology, King George V Block, St. Bartholomew’s Hospital, West Smithjeld, London, England (Submitted October 15, 1990. Accepted for publication October 19, 1990) Acute bilateral scrotal swelling of the neonate may be caused by a variety of conditions. Bilateral torsion of the testes is an uncommon but important cause of such swelling and must be suspected in all cases. A case of delayed complication of untreated bilateral testicular torsion in the newborn presenting in adoles- cence is reported and followed by discussion of the literature review. Key word: bilateral testicular torsion. CASE REPORT A full term newborn baby delivered at home was noticed to have bilateral scrotal swelling shortly after delivery and was admitted immediately to hospital under the care of the paediatrician. The scrotum was described as being red, angry looking and tender. Urine microscopy showed a raised leucocyte count and although the urine culture was sterile, a diagnosis of bilateral epididymo-orchitis was made. He was treated with antibiotics and was discharged home af- ter twenty days when the swelling had decreased in size and urine microscopy and culture were normal. At outpatient follow-up five weeks later the testes were noted to be enlarged but otherwise “normal”, and he was discharged from further follow-up. At the age of fifteen, the patient was referred by his general practitioner to a urologist with a diagnosis of bilateral undescended testes (after failing to attend at the age of ten for the same problem). Secondary sex- ual characteristics were absent and he was of short stature (151.5 cm or below the 3rd centile). Neither testis was palpable. The gonadotrophin levels were high (luteinising hormone at 55 units per litre and interstitial cell stimulating hormone at greater than 50 units per litre) confirming testicular failure. Bi- lateral groin exploration was then performed. At op- eration both spermatic cords with normal looking gonadal vessels and vasa deferentia were present and both ended blindly just distal to the external inguinal rings. Biopsies were taken from the ends of the cords and these showed no evidence of any testicular tissue. Testicular prostheses were inserted. Post-operatively the patient was treated with intramuscular testoster- one injections (1 ml of Sustanon 250) to induce pu- berty and the development of secondary sexual char- acteristics. The resultant growth spurt is shown in Fig. 1. DISCUSSION Bilateral neonatal testicular torsion is a rare condition and it may have a genetic predisposi- L J. SUSTANON 250 (1 ml/month) 4.1.67 started on 13 4 83 __-- a Start of Hormone 50 1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 I7 18 19 AGE (Years) Fig. 1. Height centile chart showing patient’s height prior to hormone therapy (well below 25th centile) and the effect of hormone treatment to induce puber- ty (around 40th centile at age 18). Scand J Urol Nephrol25 Scand J Urol Nephrol Downloaded from informahealthcare.com by University of North Carolina on 11/04/14 For personal use only.

Long Term Complication of Untreated Bilateral Testicular Torsion in the Newborn

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Scand J Urol Nephrol25: 3 19-320, 199 1

LONG TERM COMPLICATION OF UNTREATED BILATERAL TESTICULAR TORSION IN THE NEWBORN

Case Report

Samson Liu, Simon Andrew Holmes and Nicholas Paul Cohen

From the Department of Urology, King George V Block, St. Bartholomew’s Hospital, West Smithjeld, London, England

(Submitted October 15, 1990. Accepted for publication October 19, 1990)

Acute bilateral scrotal swelling of the neonate may be caused by a variety of conditions. Bilateral torsion of the testes is an uncommon but important cause of such swelling and must be suspected in all cases. A case of delayed complication of untreated bilateral testicular torsion in the newborn presenting in adoles- cence is reported and followed by discussion of the literature review. Key word: bilateral testicular torsion.

CASE REPORT A full term newborn baby delivered at home was noticed to have bilateral scrotal swelling shortly after delivery and was admitted immediately to hospital under the care of the paediatrician. The scrotum was described as being red, angry looking and tender. Urine microscopy showed a raised leucocyte count and although the urine culture was sterile, a diagnosis of bilateral epididymo-orchitis was made. He was treated with antibiotics and was discharged home af- ter twenty days when the swelling had decreased in size and urine microscopy and culture were normal. At outpatient follow-up five weeks later the testes were noted to be enlarged but otherwise “normal”, and he was discharged from further follow-up.

At the age of fifteen, the patient was referred by his general practitioner to a urologist with a diagnosis of bilateral undescended testes (after failing to attend at the age of ten for the same problem). Secondary sex- ual characteristics were absent and he was of short stature (151.5 cm or below the 3rd centile). Neither testis was palpable. The gonadotrophin levels were high (luteinising hormone at 55 units per litre and interstitial cell stimulating hormone at greater than 50 units per litre) confirming testicular failure. Bi- lateral groin exploration was then performed. At op- eration both spermatic cords with normal looking gonadal vessels and vasa deferentia were present and both ended blindly just distal to the external inguinal rings. Biopsies were taken from the ends of the cords and these showed no evidence of any testicular tissue. Testicular prostheses were inserted. Post-operatively

the patient was treated with intramuscular testoster- one injections (1 ml of Sustanon 250) to induce pu- berty and the development of secondary sexual char- acteristics. The resultant growth spurt is shown in Fig. 1.

DISCUSSION Bilateral neonatal testicular torsion is a rare condition and it may have a genetic predisposi-

L J. SUSTANON 250 ( 1 ml /mon th )

4.1.67 s t a r t e d on 13 4 83

_ _ - -

a S t a r t of Hormone

50

1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 I7 18 19

A G E ( Y e a r s )

Fig. 1. Height centile chart showing patient’s height prior to hormone therapy (well below 25th centile) and the effect of hormone treatment to induce puber- ty (around 40th centile at age 18).

Scand J Urol Nephrol25

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320 S. Liu et al.

tion (8). It is said to present acutely rather than developing slowly in utero ( 1 1) and may have a predilection for preterm infants (9). In the few cases (1, 2, 3, 7, 10) where immediate explora- tion had been performed, the torsion was found to be mostly extravaginal (torsion of the sper- matic cord and very rarely intravaginal (the type more commonly seen in older children and adults). Recently, there had been increasing rec- ognition of bilateral testicular torsion as a cause of secondary testicular atrophy and bilateral an- orchidism (6).

In the case of the above patient, immediate surgery was not performed but the presence of bilateral scrotal swelling at birth and the ab- sence of testes with intact spermatic cords at subsequent exploration indicate that some acute destructive pathological process must have occurred at or around birth, giving rise to the delayed complication of testicular atrophy and failure of development of secondary sexual characteristics. It is difficult to envisage any condition other than bilateral torsion which could have caused such comprehensive destruc- tion of testicular tissue. Other differential diag- noses of scrotal swelling in the neonate include strangulated hernia, testicular tumour, haema- tocele, torsion of the appendix testes, bilateral testicular infarction ( 5 ) and bilateral scrotal mass secondary to neonatal adrenal haemor- rhage (4), but whatever the provisional diagno- sis immediate exploration is indicated. Radio- logical investigation, particularly ultrasound, may be of help to confirm the diagnosis. Typi- cally, this shows an area of hypoechoecity with a bright peripheral rim ( 1 1, 12). Other investi- gations like urine microscopy and culture may help to exclude other differential diagnoses.

In view of the morbidity and serious conse- quences of this pathology whose diagnosis re- quired a high index of suspicion, we conclude that neonatal bilateral testicular torsion, al- though uncommon, must be suspected in all cases of bilateral scrotal swelling in the new- born.

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REFERENCES Atallah MW, Ippolito JJ, Rubin BW. Intrauter- ine bilateral torsion of the spermatic cord. J Urol

Bachor R, Frohneberg D, Heymer B et al. Bilater- al intrauterine testicular torsion. Urologe-A

Fenton EJ, OSullivan DC. A further case report of bilateral torsion of testes in a newborn. Irish Med J 1983; 76: 242. Giacoia GP, Cravens JD. Neonatal adrenal hem- orrhage presenting as scrotal hematoma. J Urol

Hara S, Izumi T , Ohmae H et al. A case of bi- lateral testicular infarction without spermatitor- sion. Hinyokika-Kiyo 1984; 30: 947-952. Herzog B, Hadziselimovic F, Strebel C. Primary and secondary testicular atrophy. Euro J Pediatr

Kay R, Strong DW, Tank ES. Bilateral spermatic cord torsion in the neonate. J Urol 1980; 123: 293. Pierson M, Vidailhet M, Wuilbercq L et al. Total testicular regression syndrome or anorchidism. Arch Fr Pediatr 1983; 40: 767-773. Ryken TC, Turner JW, Haynes T. Bilateral tes- ticular torsion in a pre-term neonate. J Urol

Weingarten JL, Garofalo FA, Cromie WJ. Bi- lateral synchronous neonatal torsion of the sper- matic cord. Urology 1990; 35: 135-136. Zafaranloo S, Gerald PS, Wise G. Bilateral neo- natal testicular torsion: ultrasonic evaluation. J Urol 1986; 21: 151-153. Zerin JM, DiPietro MA, Grignon A et al. Testic- ular infarction in the newborn: ultrasonic find- ings. Pediatr Radio1 1990; 20: 329-330.

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