Review Article Sep 2012; Vol 22 (No 3), Pp: 281-289

Iran J Pediatr Review Article Sep 2012; Vol 22 (No 3), Pp: 281-289

Neonatal Testicular Torsion; a Review Article

Muhammad Riaz-Ul-haq, MBBS, FCPS, FEBPS; Diaa Eldin Abdelhamid Mahdi, and Elbagir Uthman Elhassan

King Faisal Hospital, Taif, Saudi Arabia Nov 02, 2011 Mar 18, 2012 Apr 01, 2012

Received: ; Final Revision: ; Accepted: Abstract

Neonatal testicular torsion, also known as perinatal testicular torsion is a subject of debate among surgeons. Neonatal testicular torsion either intrauterine or postnatal results into extravaginal torsion which is a different entity than intravaginal type but has the same devastating consequences if not diagnosed and managed well in time. Testicular torsion results into acute ischemia with its resultant sequelae such as abnormality of testicular function and fertility. Urgent surgical exploration and fixation of the other testis are the key points in the management. General anesthesia is not a contraindication for exploration as thought Iranianbefore.Journal Diagnosisof Pediatrics and ,controversiesVolume 22 (Number on 3management), Sep 2012, Pages of :testicular281-289 torsion are discussed in this review.

Key Words:

Testis; Testicle; Testicular Torsion; Neonatal; Perinatal Introduction

Testicular torsion was first described in 1840 by postnatal torsion is an acute manifestation with Delasiauve considerable tenderness[5] and swelling of a [1] previously normal testicle .

. The condition was first [2]time reported A review of the urological literature in the newborn by Taylor in 1897 . But it has demonstrates no consistent pattern regarding the taken a long time for it to be recognized as a potential etiologies of PTT. Possible theories vascular emergency that needs prompt diagnosis include difficult labor, breech presentation, high and urgent exploration. It was not until 1907 birth weight, an[5,6] over reactive cremasteric reflex when Rigby and Howard wrote their classic paper and multiparity . Despite the type or mechanism on torsion that this[3] entity gained widespread of torsion, PTT is an unexpected finding for clinical acceptance . Neonatal testicular torsion neonatologists and parents. Antenatal ultrasound also known as perinatal testicular torsion (PTT) is is not sensitive in detecting this abnormality; thus, a rare event with controversies regarding its it is commonly diagnosed during the routine etiology, presentation, surgical management and postnatal physical examination. The pediatric sequelae. Initially, PTT was thought of one distinct urologist or pediatric surgeon is usually consulted entity. Recently it has been subcategorized as in the first few hours of life and is faced with either occurring prenatally[4] in utero or postnatally formulating a management plan. There is much in the first month of life . controversy regarding the optimal management. Prenatal torsion is marked by minimal to no In this article, we will try to elaborate different discomfort and few localized findings. Conversely, options of management and review of literature. * Corresponding Author; Address: Specialist Pediatric Surgeon, King Faisal Hospital, Taif- Saudi Arabia E-mail: [email protected]

Embryology and Anatomy

6 the lobules are formed by septations arising from SRY mediastinuminner layers testis of the tunica albuginea. The Shortly after weeks' gestation, the testis- seminiferous tubules converge toward the determining gene on chromosome Y directly rete testes and unite to form larger affects the differentiation of the indifferent gonad tubules, which in turn form a network of tubules, into a testis. Around 6-7 weeks' gestation, Sertoli the efferent. The tubules rete testes unite to form cells develop and secrete Müllerian inhibitory efferent tubuleshead at the of thesuperior epididymis pole of the testes. substance (MIS),9 which leads to the regression of These (10-15 in number) the female genital organs. constitute the uctus epididymis . The efferent Around the weeks' gestation, Leydig cells start tubules then unite to form a single lumen producing testosterone, which promotes structure called d (Fig. 1). development of the Wolffian duct into portions of The ductus epididymis forms the body and the the male genital tract. Because of the differential tail of the epididymis, which is located on the growth of the fetus, the testicles move into the posterolateral aspectvas of deferens the testis. The tail of the pelvis, close to the internal ring. But initially it epididymis undertakes an acute turn and

develops[8] retroperitoneally adjacent to the continues as the , which joins the kidney . At about the third month of intra uterine spermatictunica cord. vaginalis The testis and epididymis are life, the gubernaculum testis develops and extends vested by an extension of the peritoneum called from the genital tubercle to the inferior pole of the the ; this covers all but the testis via the inguinal canal. The peritoneum later posterior side (Fig. 1). encircles the testis completely forming a The testis is suspended from the spermatic cord mesentery, the mesorchium. The inferior main like an object at the end of a rope. The contents of part of the gubernaculum attaches to the scrotal the spermatic cord include the vas deferens and its

skin pouch[8] and the minor, superior part artery, the testicular artery, the pampiniform disappears . plexux of veins, lymphatic vessels, and the The testes are paired structures suspended in sympathetic nerves. All of the aforementioned the scrotum by the spermatic cord. They measure structures are enclosed in facial layers derived approximately 2.0 × 3.0 × 4.0 cm in the adult and from the oblique muscles of the il abdomen.ioinguinal Loops nerve weigh 15-20 g, but the measures are variable in of cremasteric muscle encircle the spermatic cord

the neonate.tunica albuginea and scrotum, innervated by the [8] Each testis is surrounded by a fibrous capsule testicularand are responsible artery for the cremasteric reflex . called the . The testis contains The blood supply to the testis isdeferential mainly from artery the seminiferous tubules, which are tightly coiled and , a branch of the abdominal aorta. arranged in wedge shaped lobules. The margins of Contribution is also made by the ,

Fig. 1. Anatomy of testis

1. Head of epididymis 2. Rete testis 3. Body of epididymis 4. Tail of epididymis semineferous tubules 5. Efferent ductules 6. Tunica vaginalis 7. Tunica albuginea 8. Convoluted 9. Testicular septa semineferous tubules 10. Straight 11. Testicular lobules Iran J Pediatr; Vol 22 (No 3), Sep 2012 283

a branch of thecremasteric hypogastric branch or superior vesical type of testicular torsion, the testis, epididymis,[9] artery; it primarily supplies the vas deferens. In and tunica vaginalis twist on the spermatic cord addition, the of the inferior (Fig. 3a,4). In result of twisting ischemicaway from changes the epigastric artery forms a network over the tunica midlinesuch as swelling, degeneration, necrosis and and forms an anastomosis at the testicular infarction occur. Torsion is usually mediastinum. The testicular artery enterscapsular the due to the orientation of cremasteric arteriestestis through the mediastinum and branches muscle° fibers. The degree° of torsion varies from under the tunica albugineacentripetal to form arteries 180 to more than 720 . The severity of torsion . The capsular arteries send radial depends upon the degree of twist. In result of branches,U known as the , into torsion, both venous and arterial blood supplies the substance of the testis. The centripetal arteries are hindered and ultimately results into gangrene form loops near their ends, increasing the of the testis. PTT may be unilateral or bilateral, effective area of supply. The scrotal wall is and bilateral torsion can be synchronous or supplied by the pudendal artery, which is not a asynchronous. Baglaj MIntravaginal et al mentioned torsion incidence contenttesticular of the veins spermatic cord and thus not of bilateral synchronous[10] torsion as 67% and involved in testicular torsion (Figpampiniform. 2). plexus asynchronous 33% . occurs The exit from the mediastinum more commonly in the peripubertal period than at and form a plexus called the . other times. This type of testicular torsion is The plexus then combines into a dominant vein, associated with a bell-clapper deformity (Fig 3b). which follows the testicular artery into the Perinatal History spermatic cord. The left testicular vein drains into the left renal vein, whereas the right testicular prenatal vein enters directly into the inferior vena cava. history In case of perinatal testicular torsion, Pathology and Pathogenesis is very important. It should include pre eclampsia, gestationalBirth history diabetes, twin gestation, Extravaginal large size for gestational age, presence of prenatal torsionTwo types of testicular torsion are recognized, and hydronephrosis. should include mode each has slightly different etiologies. of delivery, as well as presence or absence of occurs in fetuses and in neonates. With this nuchal cord, meconium aspiration and prolonged

Fig. 2a: Arterial supply of testis and epididymis

1) Testicular artery 2) Deferential artery 3)2b Cremasteric: Transverse artery section of the testis

4)Internal spermatic fascia 5)Parietal and visceral lamina of tunica vaginalis 6)Testicular artery 7)Capsular arteries 8)Centripetal arteries 9)Vas deference 284 Neonatal Testicular Torsion; M Riaz-Ul-haq, et al

Clinical Findings

delivery. Jonathan D Kaye described 10 patients in whom prenatal history was known, 5 (50%) had at least one significant prenatal finding. Two After obtaining a detailed history, examination of mothers had preeclampsia, two mothers had the spine, back, perineum, groin and the scrotum gestational diabetes, 1 neonate was large for should be performed. Examination of the abdomen gestational age and 1 neonate was diagnosed is also important to exclude other problems giving prenatally with hydronephrosis. Two patients rise to such symptomatology. On physical were product of twin gestation. 5 of 10 patients examination, the affected hemiscrotum is swollen

were delivered[11] with some sort of delivery and frequently erythematous. The normal complication . separation of the testis from the epididymis may while postnatal torsion has not been linked to not be palpable. An elevated, horizontal lie of the any specific risk factors, such is not the case for affected testis and skin pitting at the scrotal base prenatal torsion. This condition has been may provide evidenceblue-dot in signsupport of the diagnosis. associated with breach presentation and With transillumination, the ischemic testicle may traumatic delivery. Most prenatal torsions occur in be visualized ( ). Typically, no pain

full term neonates with[12] the mean birth weight in relief occurs with scrotal elevation, as observed one series being 3.6 kg . All of these factors can with acute epididymitis. The spermatic cord is potentially increase intra uterine pressure as well typically thickened and tender. Tenderness alone

as pressure[13] in the birth canal during may be indicative of acute epididymitis. In a parturition . It is plausible that such pressure practical sense, PTT includes[15] neonates with 5 may, in turn, stimulate a brisk cremasteric definite clinical pictures .

response in[14] the setting of loose tunic scrotal 1) If torsion occurs in prenatal period vanishing far from attachment . So a detailed prenatal history and testisbirth, the newborn will be born with an absent mode of delivery are mandatory and certainly in or a nubbin testis. In the first case ( postnatal torsion history of initial well-being, ), the patient should be treated as irritability, vomiting, and sudden scrotal swelling cryptorchid at an older age. In the second case should also be considered. he should be approached as a sequel of a long standing intrauterine testicular torsion.

Fig. 3: Types of testis torsion

a) Extra-vaginal b) Intra-vaginal (Bell-Clapper Deformity) Iran J Pediatr; Vol 22 (No 3), Sep 2012 285

WORK UP

2) If torsion occurs in prenatal period vanishing far from testisbirth, the newborn will be born with an absent In addition to careful clinical examination and or a nubbin testis. In the first case ( evaluation, Doppler sonography is increasingly

), the patient should be treated as used in the management[16 -of18] patients with suspicion cryptorchid at an older age. In the second case for testicular torsion . This can mainly be he should be approached as a sequel of a long attributed to the quick technical progress with

standing intrauterineseveral weeks testicular torsion. excellent anatomical imaging[19] and simultaneous 3) If torsion occurs in the prenatal period far portrayal of blood flow . High frequency from birth ( ), typically the child transducers, power Doppler and tissue harmonic will present since birth a regular, firm, functions are increasingly used in daily routine painless scrotal mass, often in the upper part care. However to prove testicular torsion, surgical of the hemiscrotum, smaller than the exploration of the scrotum is still fairly often contralateral normal testis, very attached to performed. Taken into consideration that less mostthan the scrotal wall, without acute inflammatory infants30% of may the infants be managed with[17] anconservatively acute scrotum suffer signs, andseveral which days does not transmit light. from testicular torsion , it is obvious that 4) If torsion occurs in the prenatal period near . Despite birth ( ), the newborn will present reports on strongly varying results concerning the

since birth a firm and painless scrotal mass, reliability for detecting[20] testicular torsion by bigger or similar in size than the contralateral Doppler sonography , recent studies[21,22] show an normal testis, without acute inflammatory increasing sensitivity of this technique . signs and whichfew daysdoes not or transmit several light. hours In neonates color Doppler sonography (CDU) 5) If torsion occurs in prenatal period very near shows an enlarged, heterogenous testis, thickened to birth ( ), the tunica albuginea with rim like hyperechoic newborn will be born with acute reflections (calcifications) at the transitional zone inflammatory scrotal signs: a painful, between testis and tunica albuginea. Hypoechoic

enlarged, bluish or reddish hemiscrotum with central area[23] may also be evident which shows an enlarged and sometimes elevated testis necrosis . In neonatal torsion CDU may be used

that does not transmit light, and a thickened to estimate the time elapsed[24] since the occurrence and painful cord. of intrauterine testis . A relatively short 6) If torsion occurs in postnatal period within the duration of torsion is characterized by mixed first month of life, the child will be born echogenecity. Prolonged intrauterine torsion

without any scrotal signs (occasionally a shows calcification and a hypervascular[24] ring of hydrocele) and the acute inflammatory signs tunica with a hypodense center . While will appear later. performing Doppler ultrasound, description of the

a b

Fig. 4: th Testis torsion. a: Erythema of the scrotum over torted testis (presented on 4 day of life with acute right hemiscrotum for 12 hrs), b: Intraoperative photo showing torted gangrenous testis 286 Neonatal Testicular Torsion; M Riaz-Ul-haq, et al

echogenecity and tissue structure of the testes and hematoma may be indistinguishable from a torted epididymis of both testes, scrotum volumetry testicle, demonstrating a hyperemic rim inside comparison and assessment of central and surrounding an area of decreased activity. Some

peripheral blood flow of the testicles at power and studies have claimed[27] it to be a better diagnostic CDU must be considered. In case of given modality than CDU . perfusion, the resistance index (RI) should be [25] Management included to recognize possible partial torsion . Special attention has to be paid to the fact that intrauterine and spontaneous reduction in intermittent torsion can postnatal in the first 30 days appear as reactive hyperperfusion of the testicular Perinatal testicular torsion ( parenchyma. Additionally, one should take into ) is an uncommon

consideration that in case of testicular torsion entity and represents about[28] 12% of all testicular with absentPulsed central Doppler blood sonography flow, peripheral torsions during infancy . It is thought that the perfusion can be maintained via collateral arterial majority (70%) of perinatal torsions are present at

supply. with delivery and 30%[29] develop postnatally in the first mechanical sector scanners is a better method[26] month of life . Review of literarture indicates than CDU for the diagnosis of testicular torsion . that there is controversial management of this But this technique studies only the testicular entity. We have tried to review the literature on arteries and omits the scrotal, paratesticular and optimum management of intrauterine and testicular surface vessels. postnatal testicular torsion and controversies Radionuclear scanning is another diagnostic whether contralateral testis should be fixed or not. modality in neonatal testicular torsion. We have also reviewed the literature on methods

Technetium-99m pertechnetate is the agent of of testicular fixation. [11] choice, with a pediatric dose of at least 5 mCi. Jonathan D. Kaye et al wrote their Typically, immediate radionuclide angiograms are recommendation in the light of their experience obtained, with subsequent static images as well. In on 15 cases of neonatal testicular torsion. the healthy patient, images show symmetric flow According to them in cases of bilateral testicular to the testes, and delayed images show uniformly torsion emergent bilateral exploration via inguinal symmetric activity. The appearanceusually <7 of testicularh approach should be performed and the surgeon torsion on scintigraphy depends upon the should err towards orchidopexy rather than chronicity. In acute torsionnubbin ( sign ), blood orchiectomy. If findings are present at birth and flow may range from normal to absent on the torsion is unilateral , they recommend ipsilateral involved side, and a may be visible. orchiectomy and contralateral orchidopexy (both The nubbin sign is a focal medial projection from via inguinal approach) after I month of age with the iliac artery representing reactive increased interim parental scrotal examination. If findings flow in the spermatic cord vessels terminating at are not present at birth (i.e. postnatal torsion), the site of torsion (This sign can also be seen in they advised emergent ipsilateral exploration and later stages). Static images demonstrate a contralateral dartos pouch orchidopexy (both via photopenic area in the involved testis. In the inguinal approach). subacute and late phases of torsion (missed Jose L Cuervo et al managed their patients by torsion), there is often increased flow to the considering whether the torsion was long standing affected hemiscrotum via the pudendal artery intrauterine, very near delivery or postnatal. For with a photopenicrim, doughnut, testis or andbull's a eyerim signof surrounding long standing intrauterine torsion they increased activity on static images. This has been recommended that as there is no urgency these called a . neonates should be operated on electively when Acute epididymitis generally appears as an area the child is in optimal clinical status to confirm the of focal or diffuse increased activity in the suspected diagnosis, to remove the affected testis, involved hemiscrotum. Testicular appendix and to explore the contralateral normal one. torsion has a variable appearance: it may have a According to them if torsion occurs in the prenatal normal scan or a focal area of increased or period very near to birth or in the postnatal period decreased activity. An abscess, tumor, or within the first month of life immediate Iran J Pediatr; Vol 22 (No 3), Sep 2012 287

exploration should be carried out and one should[15] delay in operative intervention seems to be not spend valuable time in ultrasound studies . inappropriate from medical and medicolegal point Many investigators have recommended that of view. Taking into consideration a risk, however boys who present with suspected unilateral small of asynchronous torsion, bilateral emergent torsion within the first 30 days of age, the risks exploration is strongly[36 advised] in all neonates. associated with early surgery and anesthesia[4] Yerkes EB et al , are also in favor of outway the prospect of salvaging the testis . It is immediate surgical exploration. According to them now clear that the potential untoward anesthetic surgery is the best method available to confirm risks involving an otherwise healthy neonate testicular non viability and small but present risk quoted in prior studies are outdated and are not of asynchronous torsion in the perinatal period consistent with the[30] current standards of pediatric can be addressed early by pexing the contralateral anesthetic practice . Those opposed to emergent testis. They also mentioned that perinatal torsion surgical exploration of a suspected torted testis[31] is asymptomatic and in its early stages may show often cite the experience of Kaplan and Silber , no clinical signs of having occurred, thus early who reported a salvage rate of only 5% in their surgical exploration may detect asynchronous experience with babies who presented with torsion and allow its correction. testicular torsion. However, this percentage Another controversial issue is whether included all torted testes presenting in neonates. It contralateral orchidopexy is justified. Some inves- ignored the dramatic differences between an acute tigators suggested that since predisposing factors torsion presenting after birth and the more are lacking in extravaginal torsion, [37] there is no common entity of the long standing event of need for contralateral orchidopexy . On the antenatal testicular torsion. A[29] subsequent survey other hand the increasing number of reported conducted by Das and Singer suggested that in cases with bilateral intrauterine[38-40] torsion supports boys with neonatal torsion, at least 28% occurred a predisposing factor . although asynchronous postnatally. This suggests a potentially greater bilateral torsion is rare, it can, however occur at rate of testicular salvage than the disappointing any time and has been reported as[39] early as 48 results previously reported. Following a policy of hours after torsion on the other side . Kashif and managing postnatally diagnosed torsion in all Riazulhaq et al mentioned 11 cases of neonatal neonates in a fashion identical[32] to that practiced in testicular[41] torsion, opposite testis was fixed in all older boys, Pinto et al were able to salvage 2 cases . Mishriki et al are also of the[42] opinion to fix (20%) of 10 testes with emergent surgical a single testis whatever the cause is . So there is exploration.[33] [12] [34] consensus in exploring the contralateral side in Al-Salem , Guiney et al and Logino et al the same operation or soon after that, depending adopted the[35] policy of early surgical intervention. upon the[4,29,30,32,33,39,41,42] local conditions and clinical status of the John et al reviewed the literature on neonatal patient . testicular torsion. They described 77 boys treated But no consensus exists on how such fixation for neonatal torsion, no testes were salvaged. This should be achieved. There is wide variation in the dismal outcome underlines that immediate practice of pediatric surgeons, which may reflect surgical exploration, although commonly that all techniques are equally efficacious at pre- performed may not be saving[10]torted testes. empting recurrence, although some methods Baglaj M and Carachi R mentioned their purposely avoid breaching the integrity of the experience of 58 neonates with neonatal testicular tunica albuginea. Suture fixation[43] has been torsion. They strongly recommend an emergency described with bo[44]th absorbable and non operation in all males presenting with clinical absorbable sutures . But concerns regarding symptoms of unilateral or bilateral testicular testicular damage due to needle trauma have led torsion. They believe that prompt exploration some to adopt sutureless[45] fixation methods, such as serves its use as the most objective diagnostic Jaboulay[46] procedure or creation of a dartos method. They are of the opinion that clinical pouch . Simlpe suture fixation res[47]ults only in judgement by a pediatric surgeon or urologist is of fine adhesions at the suture points but when a utmost importance not the ultrasound. Although window in the tunica vaginalis was created, dense the prognosis for these patients is poor, an elective adhesions between the tunica vaginalis and the 288 Neonatal Testicular Torsion; M Riaz-Ul-haq, et al

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