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Table of Contents
CASE REPORT
Year : 2013  |  Volume : 5  |  Issue : 4  |  Page : 302-304  

Adrenal rests associated with ectopic testis in an adult: Clinical significance


1 Department of Surgery, Armed Forces Hospital, Southern Region, Khamis Mushait, Saudi Arabia
2 Department of Pathology, Armed Forces Hospital, Southern Region, Khamis Mushait, Saudi Arabia
3 Department of Pathology, College of Medicine, King Khalid University, Abha, Saudi Arabia

Date of Submission19-Nov-2012
Date of Acceptance24-Mar-2012
Date of Web Publication24-Oct-2013

Correspondence Address:
M Ezzedien Rabie
Department of Surgery, Armed Forces Hospital, Southern Region P.O. Box 101, Khamis Mushait
Saudi Arabia
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DOI: 10.4103/0974-7796.120307

PMID: 24311918

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   Abstract 

Ectopic adrenal tissue may be present along the path of the testis and discovered during groin surgery. The condition has been mainly reported in the pediatric population and very rarely in adults. Here, we report on the case of an adult male with undescended testis who received orchidectomy, and ectopic adrenal tissue was discovered in the removed specimen. The clinical significance of such a condition is discussed.

Keywords: Adrenal ectopia, orchidectomy, undescended testis


How to cite this article:
Rabie M E, Shah MT, Jamil S, Al-Shraim MM. Adrenal rests associated with ectopic testis in an adult: Clinical significance. Urol Ann 2013;5:302-4

How to cite this URL:
Rabie M E, Shah MT, Jamil S, Al-Shraim MM. Adrenal rests associated with ectopic testis in an adult: Clinical significance. Urol Ann [serial online] 2013 [cited 2020 Jan 25];5:302-4. Available from: http://www.urologyannals.com/text.asp?2013/5/4/302/120307


   Introduction Top


Adrenal glandular tissue can be aberrantly located in many locations, including the inguinal region. [1] Ectopic adrenal tissue is rarely reported in adult population, and may pose a diagnostic challenge. The pathogenesis of such a rare entity is still not fully understood. [2] The ectopic adrenal tissue can potentially undergo hyperplasia and neoplasia; [3],[4] therefore, surgical resection is advisable whenever incidentally found.


   Case Report Top


A 26-year-old man, married with two children, presented with a swelling in the right groin, which was present since early childhood. His past history was irrelevant. On examination, he was generally well and his vital signs were normal as well as his chest and abdomen. Local examination revealed a reducible inguinoscrotal hernia on the right side, and an undescended testis on the left, with underdeveloped hemiscrotum.

His routine laboratory investigations were unremarkable and computerized axial tomography (CT)/ultrasound (US) scans of the inguinal region showed normally located right testis with an inguinoscrotal hernia, while on the left side, the testis was lying at the level of the deep inguinal ring. At operation: A right indirect inguinal hernia was found for which mesh repair was performed. On the left side, the testis, expectedly, was found at the deep ring and orchidectomy was done.

The patient recovered well and was discharged in good condition for follow-up in the outpatient clinic.

Histopathological examination of the excised testis showed atrophic changes, with adjacent adrenal rests in which the three cortical layers were represented [Figure 1], [Figure 2], [Figure 3] and [Figure 4].
Figure 1: Photomicrograph (original magnification ×20; H and E stain) shows the testis (yellow arrow) with adjacent adrenal rest (red arrow)

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Figure 2: Photomicrograph (original magnification ×10; H and E stain) showing adrenal rest with its three layers preserved

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Figure 3: Magnified view of Figure 2 (original magnification ×40; H and E stain)

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Figure 4: Photomicrograph (original magnification ×40; H and E stain) shows atrophic changes in the undescended testis

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   Discussion Top


Ectopic adrenal tissue in a juxta renal position was first reported by Morgagni in 1740. [1] Subsequently, other sites, including the inguinoscrotal path of the testis, have been reported. The estimated incidence of adrenal ectopia is 1.2-2% of groin operations. These nodules may be found along the spermatic cord, in the hernia sac, or between the testis and epididymis [2],[3],[4],[5] and association with undescended testis have been suggested. [6] The condition has been primarily reported in the pediatric population and, very rarely, in adults. [2],[7] In their search in the English literature, Mendez et al. [2] found 117 reported cases in infants and children and only 25 cases in adults. Adrenal ectopia at this site is encountered mainly in males [2],[3],[4],[5],[8] and very rarely in females where the ectopic tissue lies in a paraovarian location. [9]

Grossly, ectopic adrenal tissues appear oval in shape, 1-5 mm in size and have a bright yellow or orange color, [5] and resemblance to metastatic clear cell carcinoma has been observed. [7] Histologically, the three adrenal cortical layers are present, surrounded by a fibrous capsule. [5],[4]

Testicular adrenal rests have been shown to enlarge considerably in patients with congenital adrenal hyperplasia due to hydroxylase deficiency. [10],[11] In such cases, they could not be differentiated from testicular tumors. [11] Additionally, excess hormone production by testicular adrenal rests may occasionally arise due to benign [10],[12] or, rarely, malignant [13] neoplastic transformation. These sequels stand behind the rationale for excising suspected nodules discovered during groin surgery. However, as their incidence is rare, a deliberate search for adrenal rests is not usually required. [3],[5] In the case presented here, the ectopic tissue was incidentally found upon examining the excised testis. The three layers of the adrenal cortex were found with no evidence of hyperplasia or neoplasia. This report adds another case to the very rarely reported testicular adrenal cortical ectopia in adults. Familiarity with the gross appearance of such tissue is required to in order to excise them once encountered.


   Conclusions Top


Adrenal rests may be incidentally found during groin surgery. As adrenal hyperplasia or neoplasia may rarely complicate their presence, familiarity with their gross appearance is important to enable excising them when encountered.


   Acknowledgments Top


The authors are grateful to the administrative staff at the Armed Forces Hospital-Southern Region and the College of Medicine, King Khalid University. We also convey our thanks to all histopathology technologists, including Mr. Ghazi Bargi, Mrs. Shehnaz Khan, and Mr. Saeed Al-Qhtani.

 
   References Top

1.Wiezel J. Ueber compensations-Hypertrophie der accessorischem nebennieren bei der Ratte. Zentral Blatt fuer Physiologie 1879;12:780. In: Schechter DC. Aberrant adrenal tissue. Ann Surg 1968;167:421-6.  Back to cited text no. 1
    
2.Mendez R, Tellado MG, Somoza I, Liras J, Sanchez-Abuin A, Pais E, et al. Ectopic adrenal tissue in the spermatic cord in pediatric patients: Surgical implications. Int Braz J Urol 2006;32:202-7.  Back to cited text no. 2
    
3.Ketata S, Ketata H, Sahnoun A, FakhFakh H, Bahloul A, Mhiri MN. Ectopic adrenal cortex tissue: An incidental finding during inguinoscrotal operations in pediatric patients. Urol Int 2008;81:316-9.  Back to cited text no. 3
    
4.Vaos G, Zavras N, Boukouvalea I. Ectopic adrenocortical tissue along the inguinoscrotal path of children. Int Surg 2006;91:125-8.  Back to cited text no. 4
    
5.Savaº C, Candir O, Bezir M, Cakmak M. Ectopic adrenocortical nodules along the spermatic cord of children. Int Urol Nephrol 2001;32:681-5.  Back to cited text no. 5
    
6.Ozel SK, Kazez A, Akpolat N. Presence of ectopic adrenocortical tissues in inguinoscrotal region suggests an association with undescended testis. Pediatr Surg Int 2007;23:171-5.  Back to cited text no. 6
    
7.El Demellawy D, Nasr A, Samkari A, Pastolero P, Alowami S. Aberrant adrenocortical tissue in hernia sac occurring in an adult: Case report and review of the literature. Hernia 2009;13:659-62.  Back to cited text no. 7
    
8.Sullivan JG, Gohel M, Kinder RB. Ectopic adrenocortical tissue found at groin exploration in children: Incidence in relation to diagnosis, age and sex. BJU Int 2005;95:407-10.  Back to cited text no. 8
    
9.Verdonk C, Guerin C, Lufkin E, Hodgson SF. Activation of virilizing adrenal rest tissues by excessive ACTH production. An unusual presentation of Nelson's syndrome. Am J Med 1982;73:455-9.  Back to cited text no. 9
    
10.Jin HY, Choi JH, Kim GH, Lee CS, Yoo HW. Testicular adrenal rest tumors in a patient with untreated congenital adrenal hyperplasia. Korean J Pediatr 2011;54:137-40.  Back to cited text no. 10
    
11.Karnak I, Senocak ME, Göðüþ S, Büyükpamukçu N, Hiçsönmez A. Testicular enlargement in patients with 11-hydroxylase deficiency. J Pediatr Surg 1997;32:756-8.  Back to cited text no. 11
    
12.Stikkelbroeck NM, Otten BJ, Pasic A, Jager GJ, Sweep CG, Noordam K, et al. High prevalence of testicular adrenal rest tumors, impaired spermatogenesis, and Leydig cell failure in adolescent and adult males with congenital adrenal hyperplasia. J Clin Endocrinol Metab 2001;86:5721-8.  Back to cited text no. 12
    
13.Jain SH, Sadow PM, Nosé V, Dluhy RG. A patient with ectopic cortisol production derived from malignant testicular masses. Nat Clin Pract Endocrinol Metab 2008;4:695-700.  Back to cited text no. 13
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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