Austin J Clin Case Rep. 2015;2(1): 1067.
Subramanya Sharma S1*, Ramakrishnan K2, Vijayalakshmi D2 and Saravanan C3
1Department of Oral & Maxillofacial Surgery, Adhiparasakthi Dental College & Hospital, India
2Department of Oral Pathology & Microbiology, Adhiparasakthi Dental College & Hospital, India
3Department of Oral & Maxillofacial Surgery, SRM Dental College & Hospital, India
*Corresponding author: Subramanya Sharma S, Department of Oral & Maxillofacial Surgery, Adhiparasakthi Dental College & Hospital, Melmaruvathur, Tamilnadu, India
Received: December 11, 2014; Accepted: March 23, 2015; Published: April 16, 2015
Leiomyoma is a slow-growing, benign tumor of smooth muscle origin presenting clinically as a soft tissue tumor. It occurs more commonly in the gastrointestinal tract, skin, uterus and the subcutaneous tissues. Oral leiomyomas are quite rare. Reported tumors have commonly presented in the lips, tongue, buccal mucosa, palate and mandible. Most of the reported cases are in the 4th and 5th decades of life with very few cases reported in paediatric age group. Most of the lesions are small, asymptomatic and present as a smooth elevated area with pigmentation. We present a case report of an 8-yearold Indian (Asian) boy with a large, exophytic mass in the lingual aspect of the mandible. The size, the site and the age at presentation makes this a rare case worth to be recorded.
Keywords: Smooth muscle; Leiomyoma; Immunohistochemistry
Leiomyoma is a benign neoplasm of smooth muscle origin. General absence of smooth muscles in the oral region makes it a rare tumor. Several histological variants described are; Solid Leiomyoma, vascular leiomyoma and Epithelioid variety [1,2]. Vascular leiomyoma, otherwise known as angiomyoma is the most common variant reported in the oral cavity [2,3]. Leiomyoma shows a male predilection of 1.4: 1. It usually presents itself around the 4th to 5th decades of life. The common oral sites are lips, tongue and buccal mucosa. Various other sites including the palate, gingiva and the mandible have been reported [4-6]. Incidence of malignant transformation has not been reported [1,2,7].
We report the case of a male child, who presented with an Oral Leiomyoma.
An 8-year-old Indian (Asian) boy reported to the out-patient clinic of the Department of Oral & Maxillofacial Surgery with the complaint of a large mass on the lingual aspect of his right mandible in relation to the molar region. The lesion has been present since 6 months and has been growing gradually. It got traumatized frequently during mastication, associated with minor bleeding episodes.
Clinical examination revealed a painless, quite large, solitary, pedunculated, exophytic mass with an irregular, surface in the traumatized areas and other areas were pale pink in color (Figure 1). It measured about 3 x 4 cm in size extending anteriorly from the deciduous first molar to the retromolar area posteriorly on the right lingual aspect of alveolar mucosa and gingiva. Superiorly, the lesion extended to the line of occlusion and inferiorly upto the floor of mouth. Medially the lesion was adherent to the lingual aspect of mandible. The lesion was firm in consistency. No regional lymphadenopathy was evident. Differential diagnoses for the peripheral, oral exophytic lesion are Inflammatory Hyperplasias and peripheral benign mesenchymal tumors. An excisional biopsy was done and the specimen was sent to the Oral Pathology Department (Figure 2).
Figure 1: Intraoral lesion.
Figure 2: Excised specimen.
The routine H & E section of the excised lesion showed a parakeratinized stratified squamous surface epithelium associated with the underlying tumor parenchyma which was composed of interlacing fascicles of spindle cells with eosinophilic cytoplasm and elongated, pale staining, vesicular nuclei (Figure 3). The nuclei were blunt ended, typical of smooth muscle nuclei (Figure 4). The presence of few compressed slit-like vessels was evident. No mitotic activity was seen. The lesion was classified as a benign spindle cell neoplasm and histological differential diagnosis include Leiomyoma, Myofibroblastic tumor, Fibrous histiocytoma and Neurilemoma. The absence of histiocytes, multinucleated giant cells and absence of Antoni type A, type B, verocay bodies differentiated the lesion from Fibrous Histiocytoma and Neurilemoma respectively.
Figure 3: Photomicrograph showing interlacing fascicles of spindle cells with blunt ended nuclei.
Figure 4: Photomicrograph showing spindle cells with blunt ended nuclei.
Immunohistochemistry (IHC) studies were performed for further differentiating the spindle cell neoplasms with the following markers namely S-100, SMA (Smooth Muscle Actin), Muscle specific Actin (HHF-35), Desmin, Caldesmon, Vimentin, Calponin, and Ki67. Strong positive expression was noted for SMA (Figure 5), Desmin (Figure 6) and Caldesmon (Figure 7). S-100, Calponin and HHF-35 were negatively expressed. Ki -67 labelling index was insignificant. Thus the tumor was diagnosed as Leiomyoma.
Figure 5: Photomicrograph showing diffuse positivity for SMA.
Figure 6: Photomicrograph showing positivity for Desmin.
Figure 7: Photomicrograph showing positivity for Caldesmon.
Oral leiomyoma accounts for less than 1% of all leiomyomas. Oral leiomyoma is hypothesized to originate from vascular smooth muscles and excretory ducts of salivary glands. In the oral cavity, vascular or angiomyoma is the most common variant followed by the solid variant. Epithelioid variant (Leiomyoblastoma) is said to be extremely rare accounting for less than 1% . In the oral cavity it rarely attains a huge size with our case possibly the largest reported [2,3,9].
Histopathologically leiomyoma may be confused with other spindle cell neoplasms. We suggest that an oral leiomyoma, in special occasions such as involving young individuals needs Immunohistochemical stains with a panel of antibodies like Caldesmon, Desmin, HHF-35, Calponin, and Ki-67 to confirm the histopathological diagnosis. The typical cigar-shaped nuclei arranged in whirls and interlacing bundles and the brighter eosinophilic cytoplasm in leiomyoma differentiates it from a Myofibroblastic tumor [1,2,7,10]. Neurilemomas are S-100 positive . Both leiomyomas and myofibromas express SMA [12,13]. Leiomyomas express Desmin and Caldesmon whereas Myofibroblastic tumors are negative [14,15]. Vimentin, HHF-35, Calponin and Ki67 are positive for Myofibroblastic tumors and negative for leiomyomas [16,17]. Therefore, Immunohistochemistry plays a vital role in distinguishing the spindle cell neoplasms.
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Citation: Subramanya Sharma S, Ramakrishnan K, Vijayalakshmi D and Saravanan C. Leiomyoma of Oral Cavity in a Young Child. Austin J Clin Case Rep. 2015;2(1): 1067. ISSN : 2381-912X