|LETTERS TO EDITOR
|Year : 2021 | Volume
| Issue : 1 | Page : 147-149
Leiomyoma of the tunica albuginea: A rare cause of intrascrotal mass
Department of Pathology, Indira Gandhi Medical College, Shimla, Himachal Pradesh, India
|Date of Submission||18-Oct-2020|
|Date of Decision||12-Jan-2021|
|Date of Acceptance||07-Feb-2021|
|Date of Web Publication||26-Mar-2021|
Set No 14, Type VI Quarters, IAS Colony, Meheli, Shimla, Himachal Pradesh
Source of Support: None, Conflict of Interest: None
|How to cite this article:|
Mardi K. Leiomyoma of the tunica albuginea: A rare cause of intrascrotal mass. Cancer Res Stat Treat 2021;4:147-9
Leiomyomas are benign tumors that originate from the smooth muscles and are commonly seen in the uterus. However, they have also been reported to occur in the renal pelvis, bladder, spermatic cord, epididymis, prostate gland, scrotum, and the glans penis.,,,, Rare cases of primary ovarian leiomyoma, leiomyoma of the testis, and leiomyoma of the kidney have also been reported. Leiomyomas of the tunica albuginea are extremely rare, and to the best of our knowledge, only 10 cases have been reported so far.,,,,,,,,, We present a case report to highlight the importance of awareness about tunica albuginea leiomyomas, which can be clinically mistaken for malignant testicular tumors.
A 37-year-old gentleman presented to our hospital with enlargement of the right testis for 1 year. On examination, the right testis was found to be enlarged and nontender. A clinical diagnosis of chronic epididymo-orchitis was made. An ultrasonography (USG) revealed a heterogeneous mass measuring 6 cm × 2.5 cm in the right scrotum separate from the testis. A possibility of paratesticular leiomyoma was suggested. A contrast-enhanced computed tomography scan revealed a right paratesticular mass with separate planes from the testis in relation to the spermatic cord. A possibility of rhabdomyosarcoma was suggested. Serum levels of lactate dehydrogenase, alpha-fetoprotein, and human chorionic gonadotropin were within normal limits. A right radical high inguinal orchiectomy was performed. Intraoperatively, a hard mass measuring 8 cm × 5 cm arising from the tunica albuginea of the right testis was seen. On gross examination, a well-encapsulated, well-circumscribed mass measuring 6.5 cm × 6 cm × 3 cm arising from the tunica at the lower pole of the testis was observed. The cut surface of the mass was firm, homogeneously grayish-yellow in color with a whorled appearance [Figure 1].
|Figure 1: Large, well-circumscribed tumor arising from the tunica at the lower pole of testis and has yellowish grey cut surface with whorled appearance|
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Microscopic examination revealed interlacing bundles of spindle cells arranged in a fascicular pattern. These cells had elongated nuclei with blunt ends and dispersed fine chromatin and small to inconspicuous nucleoli [Figure 2]. Focal areas showed these tumor cells arranged in a palisading pattern. The tumor cells were separated by well-vascularized connective tissue. Areas of hyalinization were seen. Mitosis, hemorrhage, or necrosis was not seen. The mitotic activity was low (0–1/10 high power field). Immunohistochemistry was performed using desmin, smooth muscle actin (SMA), vimentin, and S-100 antibodies. The smooth muscle origin of the tumor cells was confirmed by the strong desmin positivity [Figure 3], along with the positivity for SMA and vimentin. Therefore, a diagnosis of leiomyoma was made.
|Figure 2: Well-circumscribed tumor consisting of spindle cells arranged in a whorled pattern and fascicles (H and E, ×10)|
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|Figure 3: Smooth muscle nature of tumor cells was confirmed by desmin positivity (IHC, ×10)|
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A variety of both benign and malignant tumors can arise from the tunica albuginea. They constitute about 15% of all intrascrotal tumors in the older age groups. Albert and Mininberg reported the first case of leiomyoma arising from the tunica albuginea in 1972.
Although rare in the genitourinary tract, leiomyomas can originate from any structure containing smooth muscles, most commonly the renal capsule. The histogenesis of leiomyomas of the testis is not clear. They may arise due to smooth muscle differentiation from myocytes in the wall of the seminiferous tubules, myoid cells, or progenitors present in the vascular smooth muscles. Apart from smooth muscle of the blood vessels, they may also arise from totipotent teratomas.
The most common age of presentation for leiomyoma is the fifth decade of life. These tumors do not show any predilection to the side of occurrence in the testis and rarely present as bilateral swellings. After an extensive review of literature, it appears that the majority of patients present with a painless swelling, ranging from 0.5 to 10 cm in diameter.
A possible differential diagnosis is an inflammatory myofibroblastic tumor (IMT). These are neoplasms of proliferating myofibroblasts, with a variable inflammatory component. Immunohistochemically, unlike IMTs, leiomyomas of the tunica albuginea are positive for desmin, as was seen in this case. Apart from IMTs, fibroma and solitary fibrous tumors also resemble leiomyomas under a light microscope. Leiomyomas stain positive for SMA, desmin, and caldesmon, whereas solitary fibrous tumors are negative for desmin. Thus, desmin is useful for distinguishing these tumors. Other tumors to be excluded are neurofibromas, schwannomas, and adenomatoid tumors. Neurofibromas and schwannomas are positive for S-100. Adenomatoid tumors show multiple irregular spaces (vacuolated cytoplasm) lined by a layer of flat or cuboidal epithelial cells, surrounded by collagenous stroma and muscle fibers, and are positive for tumor markers HMBE1 and calretinin.
Thus, on immunohistochemical examination, positive staining for SMA, caldesmon, and desmin is important to confirm the diagnosis of leiomyomas. S-100 negativity is necessary to exclude neurofibromas and schwannomas. Low mitotic activity (Ki-67 proliferation index was 4 in our case) is also suggestive of leiomyoma.
Despite the benign nature of this entity, the treatment of choice remains orchiectomy, as clinically it cannot be distinguished from malignancy. Thus, paratesticular masses must be properly evaluated and investigated to rule out the possibility of a malignancy. USG sometimes cannot differentiate paratesticular from intratesticular lesions. As the majority of these lesions are benign, a testis-sparing surgery can be performed. In our case, the mass was quite big, and therefore, an orchiectomy was done.
This case report highlights the fact that both the pathologist and the clinician should be aware of the occurrence of this rare entity called tunica albuginea leiomyoma. A testicular biopsy can aid in differentiating a benign tumor from a malignant one and help in planning the type of surgery to be undertaken. It is necessary to distinguish leiomyomas from malignant tumors of the testis to avoid an over-diagnosis, leading to radical surgeries, thereby helping in testis preservation.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal his identity, but anonymity cannot be guaranteed.
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Conflicts of interest
There are no conflicts of interest.
| References|| |
Belis JA, Post GJ, Rochman SC, Milam DF. Genitourinary leiomyomas. Urology 1979;13:424-9.
Borri A, Nesi G, Bencini L, Pernice LM. Bizarre leiomyoma of the epididymis. A case report. Minerva Urol Nefrol 2000;52:29-31.
Redman JF, Liang X, Ferguson MA, Savell VH. Leiomyoma of the glans penis in a child. J Urol 2000;164:791.
Rosen Y, Ambiavagar PC, Vuletin JC, Macchia RJ. Atypical leiomyoma of prostate. Urology 1980;15:183-5.
Park JW, Jeong BC, Seo SI, Jeon SS, Kwon GY, Lee HM. Leiomyoma of the urinary bladder: A series of nine cases and review of the literature. Urology 2010;76:1425-9.
Tomas D, Lenicek T, Tuckar N, Puljiz Z, Ledinsky M, Kruslin B. Primary ovarian leiomyoma associated with endometriotic cyst presenting with symptoms of acute appendicitis: A case report. Diagn Pathol 2009;4:25.
Kullolli VS, Kullolli S, Pawar S, Gautam D. Leiomyoma of testis-A case report. Indian J Surg 2011;73:233-5.
Brunocilla E, Pultrone CV, Schiavina R, Vagnoni V, Caprara G, Martorana G. Renal leiomyoma: Case report and literature review. Can Urol Assoc J 2012;6:E87-90.
Albert PS, Mininberg DT. Leiomyoma of the tunica albuginea. J Urol 1972;107:869-71.
Aus G, Boiesen PT. Bilateral leiomyoma of the tunica albuginea. Case report. Scand J Urol Nephrol 1991;25:79-80.
Chiaramonte RM. Leiomyoma of tunica albuginea of testis. Urology 1988;31:344-5.
Giyanani VL, Hennigan DB, Fowler M, Sanders TJ. Sonographic findings in leiomyoma of postorchiectomy scrotum. Urology 1985;25:204-6.
Lia-Beng T, Wei-Wuang H, Biing-Rorn C, Chia-Chun T. Bilateral synchronous leiomyomas of the testicular tunica albuginea. A case report and review of the literature. Int Urol Nephrol 1996;28:549-52.
Mak CW, Tzeng WS, Chou CK, Chen CY, Chang JM, Tzeng CC. Leiomyoma arising from the tunica albuginea of the testis: Sonographic findings. J Clin Ultrasound 2004;32:309-11.
Bremmer F, Kessel FJ, Behnes CL, Trojan L, Heinrich E. Leiomyoma of the tunica albuginea, a case report of a rare tumour of the testis and review of the literature. Diagn Pathol 2012;7:140.
Zabar K, Jalal AA, Alawi MM. Leiomyoma of tunica albuginea. Bahrain Med Bull 2014;36:114-6.
Wani R, Bhat N, Sheikh BA, Bhat S, Sheikh S, Mir ZN, et al
. Leiomyoma of tunica aluginea: A case report. Sch J Med Case Rep 2015;3:565-7.
Rakshith V. Vidyavathi K. Leiomyoma of tunica albuginea: A case report with review of literature. Clin Cancer Investig J 2016;5:37-9. [Full text]
McClellan DS, Roscher A. Intrascrotal tumors in the older male. Int Surg 1986;71:51-2.
Thomas J, Rifkin M, Nazeer T. Intratesticular leiomyoma of the body of the testis. J Ultrasound Med 1998;17:785-7.
[Figure 1], [Figure 2], [Figure 3]