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Perinatal testicular torsion: preoperative radiological findings and the argument for urgent surgical exploration
Journal of Pediatric Surgery (2008) 43, 1563–1565
www.elsevier.com/locate/jpedsurg
Perinatal testicular torsion: preoperative radiological findings and the argument for urgent surgical exploration Sally J. Ahmed a , George W. Kaplan a,b , Marvalyn E. DeCambre a,b,⁎ a
University of California, San Diego, USA Rady Children's Hospital San Diego, USA
b
Received 1 October 2007; revised 8 March 2008; accepted 10 March 2008
Key words: Bilateral; Neonatal; Testicular; Torsion; Emergency; Scrotum
Abstract Perinatal testicular torsion is an infrequent event, the management of which has been controversial. Occurrence is rare, estimated at 1 in 7500 newborns (Kaplan, G. W., Silber, I.: Neonatal torsion—to pex or not? In: Urologic surgery in neonates and young infants. Edited by King, L.R. Philadelphia: W.B. Saunders Co., 1988; Chapter 20, pp. 386-395). The frequency of bilateral perinatal torsion is up to 22% (J Urol. 2005;174:1579). Here, we describe two cases of bilateral asynchronous perinatal torsion, in which the only presenting abnormality on exam after birth was a unilateral scrotal mass. These cases illustrate that contralateral perinatal torsion may be present even when physical exam findings suggest unilateral involvement. © 2008 Elsevier Inc. All rights reserved.
1. Case 1 A 4450-g term male infant, born to a 28-year-old primigravida, was noted to have a left hydrocele on prenatal ultrasound 2 weeks before delivery. The prenatal course and delivery were otherwise unremarkable. Right sided scrotal swelling was noted on the first neonatal examination and on arrival to the tertiary-care facility at 24 hours of age. The contralateral scrotal sac (left) had a normal-appearing smaller hemiscrotum. Although the Doppler ultrasound demonstrated no vascular flow in the affected testis and heterogeneous central echogenicity, the contralateral testis appeared too small for conclusive Doppler evaluation. After informed consent, the decision was made for immediate surgical exploration. Intraoperative findings were twisting of the tunica vaginalis, with
⁎ Corresponding author. Tel.: +1 858 279 8527; fax: +1 858 279 8876. 0022-3468/$ – see front matter © 2008 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2008.03.056
purple discoloration of the testis and epididymis on the right. The left testis was pale-pink and atrophic (Fig. 1A). At surgical exploration, bilateral asynchronous testicular torsion (extravaginal) was diagnosed, and orchiectomies were performed. Pathology findings confirmed bilateral torsion, and testicles were nearly entirely replaced with necrotic debris.
2. Case 2 A 3235-g term male born to a 23-year-old primigravida was noted to have an enlarged left testis and smaller normal appearing hemiscrotum on initial examination after birth at a referring community hospital. On arrival at our tertiary-care facility, he was noted to have an enlarged, firm left scrotal mass with minimal overlying skin discoloration. The contralateral testis (right) was equally firm on palpation but half the size of its counterpart with
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Fig. 1 Surgical findings. A, Case 1. Tunica vaginalis is opened on right and on left. Right testis and tunica is blue/black from torsion. Left testis is atrophic and the tunica is pink (representing revascularization). B, Case 2. On the right, tunica vaginalis is opened and the testis blue/ black. On the left, the twisting of the tunica may be seen at the junction of the scrotal skin (blue appearance below and pink above the twist).
normal overlying skin. Ultrasound findings were absent blood flow of left testis and central blood flow of the contralateral smaller right testis with heterogeneous parenchyma. After informed consent, immediate surgical exploration ensued. Intraoperative findings included twisted tunica vaginalis (720°, left) and blue/purple discoloration of cord structures, testes, and bilateral epidydimii (Fig. 1B). Bilateral orchiectomies were performed. Pathology findings confirmed bilateral torsion and testicles with necrotic debris and associated calcifications.
3. Discussion Perinatal testicular torsion accounts for about 12% of all childhood torsions [1], occurring prenatally in 72% of cases [2]. Physical examination findings of torsion is an enlarged, firm, nontender testis with scrotal discoloration in neonates N3 kg [1]. There is no predilection of laterality as exemplified by our 2 cases. Classic operative findings include twisting or rotation of the tunica vaginalis and its
contents on a vertical axis, also considered extravaginal torsion. Extra-vaginal type of testicular torsion is seen primarily in neonates owing to the loose attachments of the tunica to the scrotal wall [3]. In infants, secure attachment of the tunica vaginalis to the scrotal wall is thought to occur within several weeks of birth [1,4]. The intravaginal type of testicular torsion is seen, typically, in older children and adults, where twists or rotations of the testicular cord structures occur within the tunica vaginalis [3]. Experimental animal models of testicular ischemia have shown loss of spermatogenesis at 4 to 6 hours and that of hormonal function at 10 to 12 hours after diminished testicular blood flow. Empirical salvage rates of neonatal torsion are low, approximately 5% in combined perinatal and neonatal torsion groups [5]. Urgent scrotal exploration in cases of neonatal torsion remains controversial [4], the rationale being that some experts view poor salvage rates of the affected gonad(s) and concerns for increased anesthetic risks in the neonatal period [6] as deterrents to urgent exploration. However, fixation of the tunica vaginalis on the contralateral side is uncertain for several weeks after birth, as is the risk of contralateral torsion
Fig. 2 Ultrasound architecture for both testes and both cases. Case 1: A1, the echotexture of the clinically swollen red right testis is heterogeneous; A2, the contralateral left testis has smaller dimensions. Case 2. B1, the echotexture of the right testis is heterogeneous; B2, the clinically swollen left testis has demonstrable heterogeneous echotexture and larger dimensions.
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Fig. 3 Contralateral testicular flow. A, Case 1. The Doppler flow demonstrates blood flow to the contralateral smaller left testis. B, Case 2. The Doppler flow demonstrates blood flow to the contralateral smaller right testis with demonstrable heterogeneous echotexture.
[4]. In 1 retrospective review, 22% of neonates with examination findings consistent with unilateral neonatal testicular torsion had contralateral torsion [7] at surgical exploration despite the normal appearance before surgery. This underscores the importance of bilateral exploration at the time of orchiectomy for unilateral neonatal torsion. In our patients, radiology findings were consistent for the heterogeneous appearance of the contralateral testes (Fig. 2), whereas central testicular blood flow to the contralateral testis was either equivocal or present (Fig. 3). Cases presented here and by Yerkes et al [7] found central vascular flow to the contralateral testes was misleading. The heterogeneous appearance of the testis seemed to correlate better with the intraoperative findings. In both of our cases, the contralateral testes appeared smaller pre- and postoperatively, and vascular flow was present, indicated by the appearance of the tunica vaginalis (see Fig. 1). This may reflect revascularization of the previously twisted or rotated tunica vaginalis after torsiondetorsion events. These cases may, in aggregate, represent the pathogenesis for testicular atrophy when a testis that was palpated at birth appears absent years later (ie, “vanishing testis” syndrome). These cases argue for urgent surgical exploration upon diagnosis of a unilateral perinatal scrotal mass with or without the use of the scrotal ultrasound in order to save or uncover bilateral pathology. Both the salvage and detection of bilateral perinatal testicular torsion have tremendous implications on the hormonal milieu in which the male child will develop. Knowing the status of the contralateral testes prepares families for hormonal therapy and possible testicular implants. Some experts also argue against surgical exploration because of the belief that Leydig cell function may be preserved by leaving the torsed testes in situ. Based
on the pathologic findings, neither of these 2 cases supports that belief. Finally, we argue that ultrasound imaging may be falsely reassuring during the evaluation of perinatal torsion. We believe immediate exploration is necessary not only to address the acute process of the affected scrotum but also to evaluate the contralateral side and offer fixation if the testis is viable. This approach may offer the best outcome and perhaps minimize the need for hormonal supplementation later. We hope these case reports serve pediatricians and surgeons in their practice to seek subspecialty evaluation or perform scrotal exploration for fixation immediately, in their respective clinical roles. We also propose perinatal torsion may play role in the pathogenesis of the vanishing testis syndrome.
References [1] Driver CP, Losty PD. Neonatal testicular torsion. Br J Urol 1998;82:855. [2] Das S, Singer A. Controversies of perinatal torsion of the spermatic cord: a review, survey and recommendations. J Urol 1990;143:231. [3] Traubici J, Daneman A, Navarro O, et al. Original report. Testicular torsion in neonates and infants: sonographic features in 30 patients. AJR Am J Roentgenol 2003;180:1143. [4] Kaplan GW, Retik A, Snyder HM III, et al. Neonatal torsion: immediate surgical exploration versus conservative management. Presented at the American Urologic Association, San Francisco, CA; 2004. [5] Kaplan GW, Silber I. Neonatal torsion—to pex or not? In: King LR, editor. Urologic surgery in neonates and young infants. Philadelphia: W. B. Saunders Co; 1988. p. 386-95. [Chapter 20]. [6] Warner LO, Teitelbaum DH, Caniano DA, et al. Inguinal herniorrhaphy in young infants: perianesthetic complications and associated risk factors. J Clin Anesth 1992;4:455. [7] Yerkes EB, Robertson FM, Gitlin J, et al. Management of perinatal torsion: today, tomorrow or never? J Urol 2005;174:1579.
www.elsevier.com/locate/jpedsurg
Perinatal testicular torsion: preoperative radiological findings and the argument for urgent surgical exploration Sally J. Ahmed a , George W. Kaplan a,b , Marvalyn E. DeCambre a,b,⁎ a
University of California, San Diego, USA Rady Children's Hospital San Diego, USA
b
Received 1 October 2007; revised 8 March 2008; accepted 10 March 2008
Key words: Bilateral; Neonatal; Testicular; Torsion; Emergency; Scrotum
Abstract Perinatal testicular torsion is an infrequent event, the management of which has been controversial. Occurrence is rare, estimated at 1 in 7500 newborns (Kaplan, G. W., Silber, I.: Neonatal torsion—to pex or not? In: Urologic surgery in neonates and young infants. Edited by King, L.R. Philadelphia: W.B. Saunders Co., 1988; Chapter 20, pp. 386-395). The frequency of bilateral perinatal torsion is up to 22% (J Urol. 2005;174:1579). Here, we describe two cases of bilateral asynchronous perinatal torsion, in which the only presenting abnormality on exam after birth was a unilateral scrotal mass. These cases illustrate that contralateral perinatal torsion may be present even when physical exam findings suggest unilateral involvement. © 2008 Elsevier Inc. All rights reserved.
1. Case 1 A 4450-g term male infant, born to a 28-year-old primigravida, was noted to have a left hydrocele on prenatal ultrasound 2 weeks before delivery. The prenatal course and delivery were otherwise unremarkable. Right sided scrotal swelling was noted on the first neonatal examination and on arrival to the tertiary-care facility at 24 hours of age. The contralateral scrotal sac (left) had a normal-appearing smaller hemiscrotum. Although the Doppler ultrasound demonstrated no vascular flow in the affected testis and heterogeneous central echogenicity, the contralateral testis appeared too small for conclusive Doppler evaluation. After informed consent, the decision was made for immediate surgical exploration. Intraoperative findings were twisting of the tunica vaginalis, with
⁎ Corresponding author. Tel.: +1 858 279 8527; fax: +1 858 279 8876. 0022-3468/$ – see front matter © 2008 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2008.03.056
purple discoloration of the testis and epididymis on the right. The left testis was pale-pink and atrophic (Fig. 1A). At surgical exploration, bilateral asynchronous testicular torsion (extravaginal) was diagnosed, and orchiectomies were performed. Pathology findings confirmed bilateral torsion, and testicles were nearly entirely replaced with necrotic debris.
2. Case 2 A 3235-g term male born to a 23-year-old primigravida was noted to have an enlarged left testis and smaller normal appearing hemiscrotum on initial examination after birth at a referring community hospital. On arrival at our tertiary-care facility, he was noted to have an enlarged, firm left scrotal mass with minimal overlying skin discoloration. The contralateral testis (right) was equally firm on palpation but half the size of its counterpart with
1564
S.J. Ahmed et al.
Fig. 1 Surgical findings. A, Case 1. Tunica vaginalis is opened on right and on left. Right testis and tunica is blue/black from torsion. Left testis is atrophic and the tunica is pink (representing revascularization). B, Case 2. On the right, tunica vaginalis is opened and the testis blue/ black. On the left, the twisting of the tunica may be seen at the junction of the scrotal skin (blue appearance below and pink above the twist).
normal overlying skin. Ultrasound findings were absent blood flow of left testis and central blood flow of the contralateral smaller right testis with heterogeneous parenchyma. After informed consent, immediate surgical exploration ensued. Intraoperative findings included twisted tunica vaginalis (720°, left) and blue/purple discoloration of cord structures, testes, and bilateral epidydimii (Fig. 1B). Bilateral orchiectomies were performed. Pathology findings confirmed bilateral torsion and testicles with necrotic debris and associated calcifications.
3. Discussion Perinatal testicular torsion accounts for about 12% of all childhood torsions [1], occurring prenatally in 72% of cases [2]. Physical examination findings of torsion is an enlarged, firm, nontender testis with scrotal discoloration in neonates N3 kg [1]. There is no predilection of laterality as exemplified by our 2 cases. Classic operative findings include twisting or rotation of the tunica vaginalis and its
contents on a vertical axis, also considered extravaginal torsion. Extra-vaginal type of testicular torsion is seen primarily in neonates owing to the loose attachments of the tunica to the scrotal wall [3]. In infants, secure attachment of the tunica vaginalis to the scrotal wall is thought to occur within several weeks of birth [1,4]. The intravaginal type of testicular torsion is seen, typically, in older children and adults, where twists or rotations of the testicular cord structures occur within the tunica vaginalis [3]. Experimental animal models of testicular ischemia have shown loss of spermatogenesis at 4 to 6 hours and that of hormonal function at 10 to 12 hours after diminished testicular blood flow. Empirical salvage rates of neonatal torsion are low, approximately 5% in combined perinatal and neonatal torsion groups [5]. Urgent scrotal exploration in cases of neonatal torsion remains controversial [4], the rationale being that some experts view poor salvage rates of the affected gonad(s) and concerns for increased anesthetic risks in the neonatal period [6] as deterrents to urgent exploration. However, fixation of the tunica vaginalis on the contralateral side is uncertain for several weeks after birth, as is the risk of contralateral torsion
Fig. 2 Ultrasound architecture for both testes and both cases. Case 1: A1, the echotexture of the clinically swollen red right testis is heterogeneous; A2, the contralateral left testis has smaller dimensions. Case 2. B1, the echotexture of the right testis is heterogeneous; B2, the clinically swollen left testis has demonstrable heterogeneous echotexture and larger dimensions.
Perinatal testicular torsion
1565
Fig. 3 Contralateral testicular flow. A, Case 1. The Doppler flow demonstrates blood flow to the contralateral smaller left testis. B, Case 2. The Doppler flow demonstrates blood flow to the contralateral smaller right testis with demonstrable heterogeneous echotexture.
[4]. In 1 retrospective review, 22% of neonates with examination findings consistent with unilateral neonatal testicular torsion had contralateral torsion [7] at surgical exploration despite the normal appearance before surgery. This underscores the importance of bilateral exploration at the time of orchiectomy for unilateral neonatal torsion. In our patients, radiology findings were consistent for the heterogeneous appearance of the contralateral testes (Fig. 2), whereas central testicular blood flow to the contralateral testis was either equivocal or present (Fig. 3). Cases presented here and by Yerkes et al [7] found central vascular flow to the contralateral testes was misleading. The heterogeneous appearance of the testis seemed to correlate better with the intraoperative findings. In both of our cases, the contralateral testes appeared smaller pre- and postoperatively, and vascular flow was present, indicated by the appearance of the tunica vaginalis (see Fig. 1). This may reflect revascularization of the previously twisted or rotated tunica vaginalis after torsiondetorsion events. These cases may, in aggregate, represent the pathogenesis for testicular atrophy when a testis that was palpated at birth appears absent years later (ie, “vanishing testis” syndrome). These cases argue for urgent surgical exploration upon diagnosis of a unilateral perinatal scrotal mass with or without the use of the scrotal ultrasound in order to save or uncover bilateral pathology. Both the salvage and detection of bilateral perinatal testicular torsion have tremendous implications on the hormonal milieu in which the male child will develop. Knowing the status of the contralateral testes prepares families for hormonal therapy and possible testicular implants. Some experts also argue against surgical exploration because of the belief that Leydig cell function may be preserved by leaving the torsed testes in situ. Based
on the pathologic findings, neither of these 2 cases supports that belief. Finally, we argue that ultrasound imaging may be falsely reassuring during the evaluation of perinatal torsion. We believe immediate exploration is necessary not only to address the acute process of the affected scrotum but also to evaluate the contralateral side and offer fixation if the testis is viable. This approach may offer the best outcome and perhaps minimize the need for hormonal supplementation later. We hope these case reports serve pediatricians and surgeons in their practice to seek subspecialty evaluation or perform scrotal exploration for fixation immediately, in their respective clinical roles. We also propose perinatal torsion may play role in the pathogenesis of the vanishing testis syndrome.
References [1] Driver CP, Losty PD. Neonatal testicular torsion. Br J Urol 1998;82:855. [2] Das S, Singer A. Controversies of perinatal torsion of the spermatic cord: a review, survey and recommendations. J Urol 1990;143:231. [3] Traubici J, Daneman A, Navarro O, et al. Original report. Testicular torsion in neonates and infants: sonographic features in 30 patients. AJR Am J Roentgenol 2003;180:1143. [4] Kaplan GW, Retik A, Snyder HM III, et al. Neonatal torsion: immediate surgical exploration versus conservative management. Presented at the American Urologic Association, San Francisco, CA; 2004. [5] Kaplan GW, Silber I. Neonatal torsion—to pex or not? In: King LR, editor. Urologic surgery in neonates and young infants. Philadelphia: W. B. Saunders Co; 1988. p. 386-95. [Chapter 20]. [6] Warner LO, Teitelbaum DH, Caniano DA, et al. Inguinal herniorrhaphy in young infants: perianesthetic complications and associated risk factors. J Clin Anesth 1992;4:455. [7] Yerkes EB, Robertson FM, Gitlin J, et al. Management of perinatal torsion: today, tomorrow or never? J Urol 2005;174:1579.