Testicular fibrous hamartoma : A case report

Fibrous hamartoma (FH) of childhood is uncommon benign tumor. They are generally seen in the head and neck region, gastrointestinal system and lung. As in the case we report, they may also occur in other unusual sites such as groin and testis. The clinical presentation is almost always a mass or swelling. We hereby report a case with FH in atrophic testis which is unusual location and clinical presentation.


INTRODUCTION
Prepubertal testicular tumors are different from those that appear during adulthood [1].
Historically, most of these tumors have been considered malignant, but more recent studies indicate that there are more cases of benign testicular tumors than malignant tumors during infancy and childhood.
Fibrous hamartoma (FH) is a benign, soft tissue growth, which was described by Reye in 1956 [2].This uncommon benign tumor is a focal malformation resembling a neoplasm, composed of an overgrowth of mature cells and tissues that normally occur in the affected area.Nevertheless, these mature cells and tissues are disorganized [3].
Although they are generally reported in the head and neck region, gastrointestinal system and lung, it seldom may involve genitourinary anatomical sites.The most common clinical presentation of the cases with FH is almost always a mass or swelling.
We hereby report a pediatric case of FH which located in atrophic left testis.The histological findings were consistent with testicular fibrous hamartoma (TFH).

DISCUSSION
Hamartomas may be seen anywhere in the body and are often seen in infancy and childhood.Hence, it has been believed that lesions are developmental aberrations [4].This clinical finding doesn't help to distinguish tumor from other situations, even our case was admitted to our hospital because of testicular atrophy.
Benign tumors of testis are as common as malignant ones in pediatric age group.
Bujons et al in 24 years period, reported 15 testicular tumors in children<13 in which 12 (80%) were benign lesions and the remaining 3 were malignant.The most common benign lesion was the epidermoid cyst, a far greater percentage than the 3% described in literature.Other benign lesions included teratom, hemangioma, fibrous hamartoma, splenogonadal fusion [1]. In

A 7 -
year-old boy presented to department of pediatric surgery at Abant Izzet Baysal University Hospital.The major symptom of the patient was left testicular atrophy.There was no history of previous scrotal swelling, trauma, fever or loss of weight.In the ultrasound examination, size of left testis was smaller compared to the right one and contained hipoechoic, oval shaped areas.Testicular tumor's markers and results of hematological and biochemical analysis were within normal limits.Left orchiectomy was performed.In macroscopic examination, testis dimension was 1.5x1.3x0.4 cm.Its cut surface was smooth and dirty-white.The histological section prepared from the material showed, disorganized matur tissue that composed of fibrocollagen stroma, vessels, muscular and adipose tissue (Fig. 1A, B, C and D).Neither evidence of a malignant lesion nor intratubular germ cell neoplasia was detected on histological assessment.
They are commonly seen in boys and rarely occur under two years of age[5].Our case was seven years old.The tumor has been reported to occur in different sites of the body, but it is generally found in the upper torso.Genitourinary sites were unusual locations.An extensive review of the literature found only 8 reports of FH in the genital region[6].The paratesticular area is a complex anatomical area and histogenetically this area is composed of a variety of epithelial, mesothelial and mesenchymal components.Neoplasms arising from this region therefore form a heterogeneous group of tumors with different behavioral patterns and have a extensive differential diagnosis.The clinical presentation is often a mass or swelling[7].