Is Routine Excision of Dysplastic Testicular Remnants/Nubbins Associated with Nonpalpable Testis Necessary? Is Routine Fixation of Contralateral Solitary Testis Indicated? A Survey on the Prevalent Practice among Indian Pediatric Surgeons

Abstract

Background and Aims:

Dysplastic nubbin also referred to as testicular regression syndrome (TRS) is found in 5% of cases of the Non palpable testis (NPT). There is no consensus on the excision of the above and fixation of the contralateral solitary testis. We aimed to survey the prevalent practice of the same among members of the Indian Association of Pediatric Surgeons (IAPS).

Methods:

A structured questionnaire was sent through group e-mail and social media platforms to IAPS members to identify their practices in management.

Results:

A total of 132 surgeons responded to the questionnaire. Excision of intra-abdominal and inguinoscrotal TRS remnants was practiced by 84% (95% confidence interval [CI] 77%–89%) and 82% (95% CI 74%–87%). Fixation of contralateral solitary testis was practiced by 62% (95% CI 53%–70%) in the above scenario. Among the respondents, 30% reported encountering torsion of solitary testis during their career and this experience was a significant factor (P = 0.01) in deciding contralateral orchidopexy. Scrotal infection/necrosis was not encountered by a majority (72%) and it was not a deterrent factor in preventing contralateral orchidopexy (P = 0.68).

Conclusions:

The majority of pediatric surgeons favored the removal of intra-abdominal/inguinoscrotal TRS remnants identified during laparoscopy for NPT. A majority favored sutureless fixation of the contralateral solitary testis.

INTRODUCTION

Testicular regression syndrome (TRS), or “vanishing testis syndrome (VTS),” is found during laparoscopy in around 5% of cases of nonpalpable undescended testis (NPT).[1] It is characterized by the presence of hypoplastic spermatic vessels and vas deferens ending up blind or in a macroscopically abnormal testicular tissue often referred to as “nubbin.” Alternatively, during laparoscopy, vas and vessels may be found exiting a closed internal ring and TRS remnants may be located in the inguinoscrotal location.[2] Although the exact etiology is unclear, recent studies support a vascular accident or antenatal torsion theory.[3]

Histopathological features of TRS/VTS remnants include fibrosis, dystrophic calcification, and hemosiderin deposition in association with identifiable testicular/paratesticular structures.[4] The optimal management of testicular remnants associated with TRS/VTS/NPT is debatable with some studies supporting its routine excision, whereas others questioning its necessity.[1,5,6,7,8,9,10,11]

In addition, management of the solitary testis on the contralateral side is further debatable with several authors in favor of routine fixation, whereas some are not so keen.[12,13,14] Contralateral torsion after unilateral orchidopexy is reported[15] and several studies have shown the presence of abnormal fixation[16] of contralateral testis in those with TRS. Bell-clapper deformity, a known predisposition for testicular torsion is noted in as high as 12% of autopsy specimens[17] and several authors have stressed the importance of protecting solitary testis[13] from catastrophic loss. However, there is no consensus in the literature regarding the fixation of the remaining testis.[12]

In a condition as rare as TRS/NPT, only a long-term strict follow-up of 30–40 years can reveal the true incidence of malignancy. However, a survey of existing practices may throw some light on the current thinking of pediatric surgeons and their concerns regarding malignancy risk. So far, there are only two surveys published from UK and France[12,14] in this regard and there is no such survey from India. Practices which are contrary to information available in standard textbooks are fraught with the danger of litigation. The chapter on undescended testis in the recent Indian Association of Pediatric Surgeons (IAPS) textbook of pediatric surgery, favors fixation of the contralateral side in VTS.[18] With increasing litigations for excision of testicular nubbin and loss of testis in solitary testis, it is better if the widely prevalent practice in India by a large group of surgeons is available in Indian literature so that people can safeguard themselves in future litigation. In this survey, we sought the opinion of members of IAPS to find out their current practice in the management of testicular nubbin/TRS remnants and contralateral solitary testis. In addition, we conducted a literature review of the above situations to compare the practice worldwide.

METHODS

A structured questionnaire was sent to members of IAPS through group e-mail, social media platforms (WhatsApp and Facebook groups), etc., to identify their practices in the management of intra-abdominal/inguinoscrotal TRS remnants/nubbins and further management of the contralateral solitary testis. The questionnaire was anonymous and the experience of the surgeon was not factored in as NPT is a common condition often managed widely by those in all stages of their pediatric surgical career. The participants were also asked if they had witnessed necrosis of a solitary testis following orchidopexy, or torsion of a solitary testis. The type of fixation (suture/sutureless) and their advice regarding testicular prosthesis were also sought. The questionnaire was prepared using SurveyMonkey mobile application (surveymonkey.com). The results were expressed as percentage (95% confidence interval [CI]) and a comparison of subgroup percentage was performed using Chi-square test.

RESULTS

A total of 132 surgeons responded to the questionnaire. All questions were answered by all of them. We did not have the actual number of surgeons who saw/received the message, to assess the participation rate. Table 1 summarizes the basic questions and the responses. Excision of intra-abdominal and inguinoscrotal TRS remnants was practiced by 84% (95% CI 77%–89%) and 82% (95% CI 74%–87%) of pediatric surgeons. Fixation of contralateral solitary testis was practiced by 62% (95% CI 53%–70%) in the above scenario. A subgroup analysis of their individual experiences in their decision for/against contralateral orchidopexy is shown in Table 2. Among those who fix contralateral testis, their personal experience with torsion in the solitary testis was a significant factor (P = 0.01). Among the respondents, 30% reported encountering torsion of solitary testis during their career! Scrotal infection/necrosis was not encountered by a majority (72%) and it was not a deterrent factor in preventing contralateral orchidopexy (P = 0.68). Figure 1 compares the practice of respondents to fix contralateral testis (responses to questions 8, 9, 3, and 10) based on their torsion risk perception. While 98% and 78% were worried about torsion in intravaginal torsion (IVT) and extravaginal torsion (EVT), respectively, only 52% had this concern for solitary testis associated with NPT/TRS. This concern was reduced further to 20% in solitary testis following orchidectomy for testicular tumor. Testicular prosthesis insertion was advised by 76%.

Table 1.

Questionnaire on the management of nubbins/nonpalpable testis testicular regression syndrome remnants and contralateral solitary testis

Questions	Majority response	n (%)	95% CI
1. When a nubbin/TRS remnant is found intra-abdominal during laparoscopy for NPT will you excise it?	Yes	111/132 (84)	77-89
2. When a closed ring with vas and vessels exiting is found at laparoscopy for NPT will you explore groin/scrotum and excise the nubbin/TRS remnant?	Yes	108/132 (82)	74-87
3. When testis is absent/excised will you fix the contralateral solitary testis?	Yes	82/132 (62)	53-70
4. What is your preference for fixing contralateral testis	Sutureless	69/132 (52)	44-60
5. Do you recommend a testicular prosthesis in whom the testis is absent/has been removed?	Yes	101/132 (76)	68-83
6. Have you encountered torsion in a solitary testis?	No	92/132 (70)	62-77
7. Have encountered scrotal infection/necrosis?	No	95/132 (72)	64-79
8. Do you fix the contralateral testis in older children with intravaginal torsion?	Yes	129/132 (98)	93-100
9. Do you fix the contralateral testis in neonatal extravaginal torsion?	Yes	103/132 (78)	70-84
10. Do you fix the contralateral testis after excision for testicular tumor?	No	106/132 (80)	73-86

NPT: Nonpalpable testis, TRS: Testicular regression syndrome, CI: Confidence interval

Table 2.

Subgroup analysis on questions 6 and 7

	Fix contra lateral testis	Don't fix contralateral testis	χ 2
Have encountered torsion of solitary testis
Yes	31	9	P=0.01
No	51	41
Have encountered scrotal infection/necrosis
Yes	24	13	P=0.68
No	58	37

Figure 1.

Black portion of the bars represents the percentage of respondents practicing contralateral testicular fixation based on their torsion risk perception: IVT: Intravaginal torsion, EVT: Extravaginal torsion, NPT: Nonpalpable testis, TRS: Testicular regression syndrome

DISCUSSION

Around 20% of undescended testes are nonpalpable and in around 5%, the vas and vessels end blind or in an atrophic dysplastic nubbin also referred to as TRS, or “VTS.”[1] These dysplastic nubbins can be found intra-abdominally or along the path of descent (laparoscopically vas and vessels exiting a closed internal ring) and there is no consensus on the excision of these structures. The main reason for the debate over its management is the variable incidence of viable germ cells (GCs) and seminiferous tubules (SNTs) reported in different studies.[2,3,5] Gao et al.[2] reported that GCs were present only in 2.1% cases and SNTs in 6.3% and therefore proposed that surgical removal of VTS remnants in an inguinal or scrotal position may not be necessary. Nataraja et al.,[8] on the other hand, found GC in 10% and SNT in 24% of TRS and suggested the removal of TRS remnants due to a future malignancy risk. In a recent systematic review, Nataraja et al.[9] reported that 1 in 20 resected testicular remnants had viable GCs and 1 in 10 had SNTs. Wood and Elder[19] reported a relative risk of 2.75–8 for testicular cancer in cryptorchidism. They reported that 5%–15% of scrotal testicular remnants contained germinal tissue, but malignancy was rarely reported in them. Although the exact potential for malignant transformation is unclear, given the potential for malignant degeneration, Storm et al., Bader et al., and Nataraja et al.[1,6,8] felt that testicular nubbins should be removed since these patients are already under anesthetic and its excision hardly takes a few minutes more. In this review, 85% of respondents felt that intra-abdominal nubbins should be removed and 84% felt that inguinoscrotal TRS remnants should be removed.

Another vexatious question is whether to fix the contralateral solitary testis to prevent a theoretical torsion risk. A survey by Mishriki et al.[14] in 1992 reported that 47% never fix a solitary testis. Since some respondents reported a devastating loss of solitary testes, Mishriki et al.[14] felt that contralateral testicular fixation after an orchiectomy was mandatory. Bellinger found abnormal testicular fixation in 5/6 contralateral testes explored following the diagnosis of monorchia.[16] Harris et al. found 13 cases of bell-clapper deformity in 15 patients,[13] and hence concluded that the remaining testis should be fixed. Al-Zahem and Shun[20] examined contralateral testis associated with VTS and found that 71% had testicular abnormalities, whereas 16% had an abnormality that could directly predispose to metachronous torsion. In a survey by Harper et al.,[12] fixation of contralateral solitary testis was practiced by respondents in 100% of IVT, 57% of neonatal EVT, 46% of NPT, and 28% of those who underwent orchidectomy for tumor. Similarly, in the current survey also, the respondents favored fixation of contralateral solitary testis in 98% of childhood IVT, 78% of neonatal EVT, 62% of NPT, and 30% after orchidectomy for tumor. Both these findings show that the fear of contralateral torsion is less among surgeons when dealing with solitary testis after excision of intra-abdominal or scrotal nubbin/TRS remnant. Surveys like this and the one by Harper et al.[12] can only tell regarding the widely prevalent practice among pediatric surgeons. Although scientific evidence does not show clearly whether fixation is necessary, the catastrophic loss of solitary testis, leading to potential anorchia is a serious concern making the majority of respondents in this survey (62%) to go for fixation of the contralateral solitary testis. In addition, a staggering 30% of respondents reported having encountered torsion in a solitary testis. In fact, the concern regarding torsion of the only testis was a significant factor among those who chose fixation of contralateral testis in this survey.

Further controversy is whether to go for suture fixation[21] or sutureless fixation of the remaining contralateral testis. In this survey, 52% favored sutureless fixation of the solitary testis (either subdartos pouch or some eversion techniques) in TRS/NPT. Among the respondents, 76% recommended testicular prosthesis to be inserted later and 48% reported that these patients do not turn up for prosthesis insertion despite explaining the cosmetic benefits.[22]

Figure 2 summarizes the majority practice followed by surveyed IAPS members in the management of testicular nubbins/TRS remnants. One of the main limitations of this study is the nature of the study (survey based on surgeon's practice rather than evidence). Furthermore, the number of respondents is relatively small compared to the total number of practicing pediatric surgeons. In addition, we have not segregated responses based on years of experience. Despite these limitations, we feel that the surveyed cohort may be a representative sample and their majority opinion may provide an overview of the current practice in the management of TRS remnants and solitary testis.

Figure 2.

Prevalent practice among IAPS members on the management of dysplastic testicular remnants and solitary testis associated with NPT. Solid lines represent the results based on this survey while dotted lines the well-known management of intra-abdominal testis. NPT: Nonpalpable testis, IAPS: Indian Association of Pediatric Surgeons

CONCLUSIONS

The majority of pediatric surgeons favored the removal of intra-abdominal/inguinoscrotal TRS remnants identified during laparoscopy for NPT. A majority favored sutureless fixation of the contralateral solitary testis.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.