Intermittent Torsion of the Spermatic Cord Portends an Increased Risk of Acute Testicular Infarction

Intermittent Torsion of the Spermatic Cord Portends an Increased Risk of Acute Testicular Infarction

Intermittent Torsion of the Spermatic Cord Portends an Increased Risk of Acute Testicular Infarction Matthew H. Hayn,* Daniel B. Herz, Mark F. Belling...

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Intermittent Torsion of the Spermatic Cord Portends an Increased Risk of Acute Testicular Infarction Matthew H. Hayn,* Daniel B. Herz, Mark F. Bellinger and Francis X. Schneck From the Department of Urology, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania, and Division of Pediatric Urology, Dartmouth-Hitchcock Medical Center, Children’s Hospital at Dartmouth (DBH), Lebanon, New Hampshire

Purpose: Intermittent torsion of the spermatic cord is an episode of acute torsion of the spermatic cord that resolves spontaneously. We compared the outcomes in boys with intermittent spermatic cord torsion treated electively with testicular fixation with those in boys with a history of recurrent scrotal pain who required emergent operation for acute spermatic cord torsion without spontaneous resolution. Materials and Methods: A retrospective review revealed 17 boys who required emergency operation for acute spermatic cord torsion and 30 who underwent elective surgery for intermittent spermatic cord torsion. The clinical presentation, number of recurrent painful episodes, lead time to operation, prior alternate diagnoses, intraoperative findings and clinical outcomes were recorded. Results: There was a mean of 2 recurrent painful episodes in the elective group and 3 in the emergency group (p ⬍0.005). In the elective group all boys were cured of pain after bilateral testicular fixation with 100% testicular preservation at a mean of 4 months of followup. In the emergency group at a mean of 10 months of followup the testicular preservation rate was 47% (p ⬍0.01). Intraoperatively an ipsilateral bell clapper malformation was found in 100% of boys in each group. A contralateral bell clapper malformation was noted in 90% and 88% of boys in the elective and emergency groups, respectively. Conclusions: When diagnosed accurately, intermittent spermatic cord torsion can be treated with elective testicular fixation with an excellent outcome. Misdiagnosis may create a cohort of boys with intermittent spermatic cord torsion who are at risk for acute unresolved torsion and potential testicular loss. Urologists should be proactive in recommending elective scrotal exploration when intermittent spermatic cord torsion is a likely diagnosis. Key Words: testis, spermatic cord torsion, pain, scrotum

lthough ITSC is a well established clinical entity,1,2 it remains a diagnosis of exclusion. ITSC is characterized by recurrent episodes of severe hemiscrotal pain of sudden onset and short duration, punctuated by pain-free intervals that may last for hours, days, weeks or months. ITSC has been variously reported to occur in approximately 6% of boys with recurrent hemiscrotal pain1– 4 and in approximately 50% to 62% of boys with ATSC.1–3,5– 8 Often lacking the striking and persistent symptoms of acute torsion, ITSC may be misdiagnosed or deemphasized in favor of a more common alternative source of recurrent hemiscrotal pain such as epididymitis, or torsion of a testicular or epididymal appendage. Indeed, during an episode of recurrent pain signs and symptoms may be identical to those of acute torsion. The testicle may be high riding, tender and erythematous with an absent cremasteric reflex, and it may have a horizontal lie.9,10 The urine is typically sterile and without pyuria, and the boy is afebrile. More importantly the signs and symptoms during an acute episode of ITSC may mimic epididymitis or torsion of a rudimentary appendage, although ITSC resolves relatively rapidly, while the other conditions have lingering symptoms. Depending on

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Study received institutional review board approval. * Correspondence: Department of Urology, University of Pittsburgh Medical Center, Kaufmann Medical Building, Suite 700, 3471 Fifth Ave., Pittsburgh, Pennsylvania 15213 (telephone: 412-6924100; FAX: 412-692-4101; e-mail: [email protected]).

0022-5347/08/1804-1729/0 THE JOURNAL OF UROLOGY® Copyright © 2008 by AMERICAN UROLOGICAL ASSOCIATION

timing diagnostic ultrasound or nuclear scan may be normal or show absent, decreased or increased flow (reperfusion hyperemia).11 ITSC may lead to testicular atrophy marked by peritubular fibrosis and azoospermia.12 Alternatively ITSC, which is frank acute torsion, may fail to resolve spontaneously. In these cases despite prompt intervention the operative salvage rate is only 42% to 67%.3,13–16 Therefore, it is paramount that ITSC be recognized by its recurrent nature and characteristic pain-free intervals. The history in most cases may be an indication for prompt surgery since the risk of testicular damage outweighs the morbidity of elective bilateral orchiopexy.3,5,17 The premise and driving force for this report are our belief that boys with recurrent hemiscrotal pain characteristic of ITSC are at high risk for testicular infarction or testicular atrophy if spontaneous detorsion fails to occur. Our study objectives were 3-fold. 1) We compared the history, clinical presentation, operative findings and testicular preservation rates between 2 cohorts of boys, that is those treated with elective testicular fixation after resolved ITSC and those who required emergency surgical detorsion or orchiectomy with contralateral testicular fixation in the face of acute torsion without resolution. 2) We tested our diagnostic accuracy for detecting ITSC by comparing our operative findings with clinical impressions. 3) We established whether boys with ITSC and boys with ATSC with a

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Vol. 180, 1729-1732, October 2008 Printed in U.S.A. DOI:10.1016/j.juro.2008.03.101

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history of recurrent scrotal pain represent the same group that is surgically intercepted at different points on the same timeline. MATERIALS AND METHODS During the 18-year period from 1988 to 2006 a retrospective chart review revealed 76 boys who required emergency operation for ATSC. This retrospective chart review was approved by the institutional review board at our institution. Of these boys 51 with no history of scrotal pain were excluded from review. The remaining 23 boys with ATSC had a history of recurrent scrotal pain consistent with ITSC. Of these boys 17 were clinically evaluable and they were designated the emergency group. During the same 18-year period another cohort of 32 boys with recurrent scrotal pain were treated electively with bilateral testicular fixation for ITSC. Two boys were excluded from review because of prior ipsilateral inguinal surgery for varicocele or inguinal hernia. The remaining 30 boys were designated the elective group. Charts were reviewed to determine the number of prior RPEs and previous diagnoses of epididymitis or orchitis, torsion of a testicular or epididymal appendage, urinary tract infection, unexplained fever, hydrocele, severe scrotal trauma, cryptorchidism, varicocele or inguinal hernia. The interval between the first episode of scrotal pain and surgery was designated the lead time. Operative reports were reviewed with attention to intrascrotal anatomy and BCD. Surgical outcomes as well as any episodes of recurrent torsion or scrotal pain were likewise recorded. All operations were performed at Children’s Hospital of Pittsburgh by one of us (FXS or MFB). At followup the scrotal contents were examined to estimate any discrepancy in testicular size or consistency. In patients with the 2 testes in situ the contralateral testicle served as a control organ for the ipsilateral testis. Statistical analysis was performed to determine the significance of reported variables between the 2 groups using the 1-tailed t test for 2 unpaired groups, assuming equal variance. RESULTS Of the 47 boys with ITSC in this study 17 presented to the emergency room at Children’s Hospital of Pittsburgh with ATSC and required emergency operative detorsion (emergency group) (table 1). There was a clear history of recurrent scrotal pain consistent with prior ITSC in all 17 emergency group boys. The remaining 30 boys were referred to our office and were electively operated on to treat ITSC (elective group). These 30 boys were asymptomatic at the time of office evaluation. Mean age in the emergency group was 14

No. pts: No. overall testicular loss (%): Infarction at exploration Postop atrophy Mean followup (mos)

Emergency

Elective

17 9 (53) 6 3 8

30 0, p ⬍0.01 0 0 4

No patient had perioperative complications, postoperative torsion or recurrent testicular pain.

years (range 10 to 18). In the elective group the mean age was 12 years (range 7 to 17). None of the boys in either group had a history of sexual activity, unexplained fever, documented urinary tract infection, cryptorchidism or significant scrotal trauma. Of the 17 boys in the emergency group 12 (71%) had prior diagnoses made by other physicians of epididymitis, or torsion of a testicular or epididymal appendage. Four boys (24%) in this group had prior diagnoses of hernia or hydrocele. In the elective group only 7 boys (23%) had a history of alternate diagnoses. Mean lead time in the emergency and elective groups was 10 and 3 months, respectively. The average number of RPEs was 2.0 (range 1 to 3) in the elective group and 2.9 (range 1 to 6) in the emergency group (p ⬍0.005). To avoid selection bias the final episode that required emergency detorsion was not included in this portion of the analysis. Prior painful episodes were described as recurrent and self-limited but severe enough to seek medical attention, lasting between 1 and 4 hours in the emergency group, and ½ to 6 hours in the elective group. Pain resolution was followed by pain-free intervals that lasted in some cases for days and in others for weeks or months. Eight of 17 boys (47%) in the emergency group and 10 of 30 (33%) in the elective group experienced nausea, vomiting or abdominal pain. No patient had dysuria, hematuria, pyuria, bacteriuria or fever. No hernia or hydroceles were detected in either group. None of the emergency group boys had an ipsilateral cremasteric reflex and only 2 (12%) had a contralateral cremasteric reflex. None of the prepubertal boys had a discernable horizontal lie of either testis. Of postpubertal boys a horizontal lie of the testis was detected in 6 of 13 (46%) and 6 of 12 (50%) in the elective and emergency groups, respectively. At emergency operation 6 testes were removed because of infarction and contralateral testicular fixation was performed. Of the remaining 11 testes that had surgical detorsion and orchiopexy 8 were found to be viable and 3 were atrophic at followup. BCD was noted in all detorsed testes and contralateral BCD was found in 15 of 17 (88%). In the elective group all ipsilateral testes and 27 of 30 contralateral testes (90%) had BCD. BCD is classically described as a loss

TABLE 3. Overall testicular preservation in emergency group

TABLE 1. Historical and operative data

No. pts Mean age (yrs) Mean No. RPEs Mean lead time (mos) Mean pain duration (hrs) No. prior alternate diagnosis (%) % BCD (ipsilat/contralat)

TABLE 2. Postoperative data

Emergency

Elective

17 14 3 10 2 16 (94) 100/88

30 12 2, p ⬍0.005 3 3 7 (23) 100/90

No. RPEs (No. testes)

No. Testes Preserved (%)

1 (2) 2 (4) 3 (8) 4 (1) 5 (1) 6 (1)

2 (100) 2 (50) 4 (50)* 0 0 0

* Two testes were atrophied on 4 to 6-week postoperative examination.

SPERMATIC CORD TORSION AND INFARCTION RISK of the posterior attachments of the testis and spermatic cord structures to the tunica vaginalis, which allows the testis to rotate on the long axis of the spermatic cord. Followup revealed no further episodes of testicular pain or torsion in either group. In the elective group there was no change in symptomatic testis size or consistency. Patients in the emergency group were followed a mean of 8 months, while those in the elective group were followed a mean of 4 months. The overall rate of ipsilateral testicular preservation in the emergency group was 47% with no episodes of symptomatic contralateral torsion after fixation (table 2). In the elective group bilateral testicular fixation cured all boys of recurrent pain and all testes retained normal size and consistency. DISCUSSION Our data show similarities and differences between the 2 study groups. Mean age, the duration and character of recurrent pain, absent alternate pathology and the ubiquitous BCD were similarities that prompted us to look further into the reasons for the disparate clinical outcome. In the emergency group 6 of the 17 acutely torsed testes (35%) were grossly infarcted and surgically removed. Three of the other 11 testes (27%) that were preserved and treated with orchiopexy were atrophic at followup for an overall testicular preservation rate of 47% in the emergency group. This is in contrast to 100% ipsilateral testicular preservation in the elective group without subsequent episodes of acute torsion in either testis. Our data show that the emergency group had a greater number of RPEs, a longer lead time (delay in diagnosis) and a higher incidence of alternate diagnoses (presumed missed diagnoses) before presentation. The data also support our postulate that boys with a greater number of recurrent painful episodes are at greater risk for testicular infarction. In other words, each subsequent episode risks the chance that spontaneous detorsion may not occur. Table 3 shows that testes found to be grossly infarcted at operation or atrophic postoperatively were in boys with a greater number of RPEs. Our ability to predict which boys with a history of recurrent scrotal pain would be at highest risk for ATSC is often limited to a qualitative assessment of the frequency and intensity of pain. Therefore, we believe that primary care physicians should consider all boys with recurrent testicular pain in the absence of urinary infection, trauma, epididymitis, hernia or hydrocele to have ITSC until proven otherwise. These boys should be referred promptly for urological evaluation. It is our opinion that the urologist by training and experience should be most capable of determining by examination and history which boys should undergo elective scrotal exploration. It should be said that on occasion one might perform exploration in a patient who would be found not to have BCD. This represents a missed diagnosis. If intermittent torsion is suspected, operative intervention should ideally be considered before an acute event occurs. Creagh et al reported that acute torsion developed in 10% of patients with intermittent torsion while they waited for surgery.1 Parents and patients should be advised that this may occur and require acute intervention. Intraoperative findings of BCD have been highly associated with ITSC.9 If ipsilateral BCD is found at operation, contralateral

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scrotal exploration and fixation should be performed because there is an association of future contralateral torsion in 30% to 43% of cases despite a lack of symptoms and a high risk of contralateral BCD found at exploration.5 In our series testicular fixation cured all boys of symptoms in each group and there were no future episodes of torsion. This is in agreement with prior series showing that 94% of males achieved resolution of symptoms after bilateral scrotal orchiopexy.1,8,9,17 At exploration no patients in our series had the finding of normal ipsilateral testicular fixation. CONCLUSIONS In this retrospective study we identified 2 groups of boys with recurrent scrotal pain due to torsion of the spermatic cord. We believe that all of the boys in this study had the same natural history and pathophysiology, and they were simply intercepted surgically at different points on the timeline. The boys in the emergency group had a significant delay in diagnosis of ITSC, were more frequently given alternative diagnoses for recurrent scrotal pain and had a statistically significant higher number of RPEs than the elective group. However, testicular preservation remains the most noticeable difference between the subgroup of boys who required emergency operation for ATSC and those who were identified earlier and treated with elective testicular fixation. Furthermore, the fact that all boys in the elective group were cured of pain indicates that practitioners who are trained to recognize the classic history and absence of alternative pathological conditions can make the diagnosis of intermittent torsion accurately. Health care providers who are involved in the evaluation of boys with recurrent scrotal pain should regard the recurrent nature of the pain as nothing other than an impending sign of future recurrent torsion and they should prompt expedient referral to a pediatric urologist for elective scrotal exploration.

Abbreviations and Acronyms ATSC BCD ITSC RPE

⫽ ⫽ ⫽ ⫽

acute spermatic cord torsion bell clapper deformity intermittent spermatic cord torsion recurrent painful episode

REFERENCES 1.

Creagh TA, McDermott TE, McLean PA and Walsh A: Intermittent torsion of the testis. BMJ 1988; 297: 525. 2. Jones DJ: Recurrent subacute torsion: prospective study of effects on testicular morphology and function. J Urol 1991; 145: 297. 3. Cass AS: Elective orchiopexy for recurrent testicular torsion. J Urol 1982; 127: 253. 4. Knight PJ and Vassy LE: The diagnosis and treatment of the acute scrotum in children and adolescents. Ann Surg 1984; 200: 664. 5. Schulsinger D, Glassberg K and Strashun A: Intermittent torsion: association with horizontal lie of the testicle. J Urol 1991; 145: 1053. 6. Stillwell TJ and Kramer SA: Intermittent testicular torsion. Pediatrics 1986; 77: 908. 7. Vordermark JS 2nd: Intermittent testicular torsion: an underdiagnosed entity? J Urol 1988; 140: 386.

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Sellu DP and Lynn JA: Intermittent torsion of the testis. J R Coll Surg Edinb 1984; 29: 107. 9. Eaton SH, Cendron MA, Estrada CR, Bauer SB, Borer JG, Cilento BG et al: Intermittent testicular torsion: diagnostic features and management outcomes. J Urol 2005; 174: 1532. 10. Corriere JN Jr: Horizontal lie of the testicle: a diagnostic sign in torsion of the testis. J Urol 1972; 107: 616. 11. Paltiel HJ, Connolly LP, Atala A, Paltiel AD, Zurakowski D and Treves ST: Acute scrotal symptoms in boys with an indeterminate clinical presentation: comparison of color Doppler sonography and scintigraphy. Radiology 1998; 207: 223.

12. Krarup T: The testes after torsion. Br J Urol 1978; 50: 43. 13. Hermann D: The pediatric acute scrotum. Pediatr Ann 1989; 18: 198. 14. Williamson RC: Torsion of the testis and allied conditions. Br J Surg 1976; 63: 465. 15. Lindsey D and Stanisic TH: Diagnosis and management of testicular torsion: pitfalls and perils. Am J Emerg Med 1988; 6: 42. 16. Edelsberg JS and Surh YS: The acute scrotum. Emerg Med Clin North Am 1988; 6: 521. 17. Kamaledeen S and Surana R: Intermittent testicular pain: fix the testes. BJU Int 2003; 91: 406.