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Sikiru Adekola Adebayo1, Ijeoma Nkemdilim Chibuzo2, Augustine Oghenewyin Takure3, Gabriel Olabiyi Ogun4, Michael Chigoziem Nweke5, Olayiwola Babatunde Shittu3, E Oluwabunmi Olapade-Olaopa3
1 Department of Surgery, University of Ibadan; Department of Surgery, University College Hospital, Ibadan, Nigeria
2 Department of Surgery, University College Hospital, Ibadan, Nigeria
3 Department of Surgery, University of Ibadan; Department of Surgery, University College Hospital, Ibadan, Nigeria
4 Department of College of Medicine, University of Ibadan; Department of Pathology, University College Hospital, Ibadan, Nigeria
5 Department of Pathology, University College Hospital, Ibadan, Nigeria
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Scrotal leiomyomata are rare benign tumours of the dartos muscle or subcutaneous tissues of the scrotum. We present a case of a 33-year-old male with a left hemiscrotal mass which was initially thought to be a sebaceous cyst, and later, a paratesticular tumour. Histology of the excised mass revealed a scrotal leiomyoma, an uncommon lesion.
Keywords: Dartos, leiomyoma, scrotum
| Introduction|| |
Scrotal leiomyomata (SL) are rare, benign, and usually solitary, sessile or pedunculated soft tissue tumours involving the dartos muscle or subcutaneous tissues of the scrotum.,, Soft tissue tumours may be superficial or deep. The superficial tumours originate from three sites, namely the erector pili, blood vessels and external genitalia. Genital tumours, which essentially involve the vulva, nipple and scrotum, account for less than 5% of soft tissue tumours.
Ninety-four cases of scrotal leiomyoma were found in a multinational literature review [Table 1]. The rarity of the lesion is further displayed by the report of Siegal and Gaffey who found 11 scrotal leiomyomata amongst 11,000 benign and malignant scrotal neoplasms – 0.01%; Fisher and Helwig found 4 SL amid 54 soft tissue tumours; Yokoyama et al found 4 SL among 19,000 benign soft tissue tumours; and Newman and Fletcher found 4 cases of SL when 32 genital tumours in patients of both genders were reviewed. It occurs more commonly in Caucasians. Few reports of SL in people of African descent were found in the literature reviewed.,,, Scrotal leiomyomata may mimic testicular or paratesticular tumours and are sometimes treated as such prior to histologic diagnosis.,,
|Table 1: Compilation of 94 cases of scrotal leiomyomata from various nations|
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| Case Report|| |
A 33-year-old Yoruba native of Southwestern Nigeria presented with 1-year history of a painless left hemiscrotal swelling that progressively increased in size. There was no pruritus or skin ulceration. He had no cough, fever, drenching night sweats or weight loss. There were no swellings elsewhere. Examination revealed a young male who had a hypopigmented, firm, irregular, non-tender 6 cm × 5 cm mass which was tethered to the skin of the left hemiscrotum. The left testis and cord structures, were palpable, separate from the mass and felt normal. There were distended superficial veins overlying the mass. The inguinal lymph nodes were not significantly enlarged. The right hemiscrotum, testis and cord structures were normal. A diagnosis of a sebaceous cyst of the scrotal skin was made.
A scrotal ultrasound scan showed a 54 mm × 40 mm mass of mixed echotexture arising from the scrotal skin and dartos tunica. Both testes were separate from the mass and of normal volume. Β-human chorionic gonadotrophin was 0.4 iu/l (normal values are <2.6 iu/l) whilst α-fetoprotein (AFP) was elevated at 10.3 ng/ml (normal values are <7.0 ng/ml). Due to the elevated AFP levels and a consequent suspicion of a paratesticular malignancy, he had an abdominopelvic ultrasound scan which showed a normal, smooth liver with a span of 13.6 cm and normal parenchymal echoes. No deposits were seen within it nor was there ductal dilatation. No para-aortic lymph nodes were seen.
He defaulted from care and represented with gross increase in the size of the mass, which now spanned almost the entire left hemiscrotum [Figure 1]. There was neither ulceration nor palpable inguinal lymphadenopathy still.
|Figure 1: Photograph of the scrotal mass at initial presentation (original)|
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A wide excision of the mass was done using local anaesthesia. An elliptical left hemiscrotal incision extending from the root of the penis to the fundus of the hemiscrotum was made. This spanned about 12 cm and encompassed the mass with a 2 cm normal skin margin [Figure 2]. The left testis and cord structures were separate from the scrotal mass. The mass was irregular, multinodular, arose from the scrotal wall, weighed 300 g and measured 11 cm × 8 cm × 8 cm. He was discharged home the same day on oral antibiotics and analgesics. His post-operative recovery was uneventful.
The cut section of the excised mass had a whorled appearance. Photomicrographs [Figure 3]a and [Figure 3]b show sections of the scrotal mass biopsy with a benign mesenchymal neoplasm composed of proliferating smooth muscle cells disposed in long interlacing fascicles with interspersed areas of hyalinised stroma (at low magnification × 40). The component cells are of smooth muscle derivation and are characteristically spindle-shaped having fusiform ends and containing hyperchromatic cigar-shaped nuclei. These cells are benign, appearing with bland cytology. Occasionally, pleomorphic cells may be seen. This is still in keeping with a diagnosis of a scrotal leiomyoma. There were no areas of necrosis or atypical mitosis. The mitotic count was ≤1 mitosis/10 high-power fields. There were no tumour cells in the margin of the resected mass. Immunohistochemical staining showed a positive cytoplasmic expression of vimentin and desmin intermediate filaments [Figure 3]c and [Figure 3]d. Immunohistochemical stains carried out for cytokeratin expression (AE1/AE3), S100 and mitotic activity (Ki-67) were negative [Figure 3]e and [Figure 3]f. The final histological diagnosis was a leiomyoma of the scrotum.
|Figure 3: (a) Whorling fascicles of smooth muscle (H and E-stained × 40)-original. (b) Cigar-shaped bland nuclei with eosinophilic cytoplasm (H and E-stained × 100)-original. (c) Positive vimentin expression (vimentin immunohistochemical staining × 100)-original. (d) Positive desmin expression (desmin immunohistochemical staining × 100)-original. (e) Negative S100 expression by tumour (S100 immunohistochemical staining × 400)-original. (f) Negative S100 expression by tumour (S100 immunohistochemical staining × 400)-original|
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One year afterwards, there was neither local recurrence nor enlarged inguinal lymph node. Both testes were intrascrotal and of normal volume [Figure 4]. A repeat AFP showed persistence of the elevation. The liver function tests, however, were normal. He was to be monitored with serial hepatic ultrasound scans.
| Discussion|| |
A scrotal leiomyoma is a slow-growing tumour of the dartos muscle of the scrotum usually located within the corium, but which could extend across it. It typically occurs in Caucasians from the fourth to the ninth decades of life, with a peak in the fourth decade.,, The largest reported diameter was 14cm, with most being between 0.6 and 6 cm.[2–4,13] The largest reported weight was 8 kg. They may appear as solid hypoechoic masses on ultrasound scan. The cut sections of the lesion characteristically show a whorled appearance grossly and whorling fascicles of the tumour cells on histology.
Scrotal leiomyomata are classified into two groups, namely, typical and atypical leiomyomata. This is based on histological features namely: (i) size ≥ 5 cm in widest dimensions; (ii) presence of infiltrating margins; (iii) ≥ 5 mitotic figures per 10 high-power fields; and (iv) moderate cytological atypia. Those with one of the features are termed typical scrotal leiomyomata while possession of two features confers upon the lesion an atypical status.,, The atypical type of scrotal leiomyoma is even rarer. Possession of three or more features suggests a leiomyosarcoma. Based on these criteria, our index case was a typical SL.
Alpha fetoprotein (AFP) was elevated in this patient even after excision of the tumour. Typically, AFP serves as a tumour marker for malignant tumours like hepatoblastoma, hepatocellular carcinoma, non-seminomatous germ cell tumours (embryonal carcinoma, endodermal sinus tumours, yolk sac tumours) in men. Rarely, it may be elevated in gastric, pancreatic, biliary, breast and lung malignancies. Other situations in which AFP may be elevated in men hepatitis or cirrhosis. Normal values are less than 5.4 ng/ml and are usually less than 10ng/ml., The reference value at our centre is < 7.0 ng/ml. Despite close follow-up, no identifiable cause of elevated AFP was found in our patient. Hereditary persistence of AFP (HPAFP) is an autosomal dominant familial disorder, with a mutation on the 5' nucleotide of the gene coding for hepatic nuclear factor 1, resulting in increased AFP gene transcription and consequently, elevated AFP levels. Only 19 such families have been described.  None of his family members turned up to have their AFP levels determined to exclude HPAFP.
The predominant treatment offered for SL is surgical excision. Only one report of a recurrent lesion was found, with a subsequent histological diagnosis of a leiomyosarcoma and use of radium as an adjuvant.
| Conclusion|| |
Scrotal leiomyoma, a benign tumour of the dartos muscle of the scrotum, can mimic a sebaceous cyst or paratesticular tumour.
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 name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.
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Conflicts of interest
There are no conflicts of interest.
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Ijeoma Nkemdilim Chibuzo,
Department of Surgery, University College Hospital Ibadan, Ibadan, Oyo State
Source of Support: None, Conflict of Interest: None
[Figure 1], [Figure 2], [Figure 3], [Figure 4]
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