Multiple Spindle Cell Lipomas of Tongue in 7-Year-Old Child: A Rare Presentation

Lipomas are among the most common tumors of the human body, with relative rarity in the oral cavity. The first case of lingual lipoma was reported by Barling in 1858 and cited by Guillou et al. ¹ Spindle cell lipoma (SCL), a histologically distinct variant of lipoma, arises in the subcutis of the posterior neck, upper back, or shoulder. SCLs rarely occur within the oral cavity and, in particular, rarely involve the tongue.

Case Report

Here, we present a rare case of multiple SCLs of the tongue in a 7-year-old boy, with approval from the Institutional Review Board of Kanti Children’s Hospital. The patient presented in the ear, nose, and throat outpatient department of a tertiary center, with multiple painless elastic nodules in the right lateral margin and tip of tongue. The patient noticed multiple small swellings in tongue about a year back, which were progressively increasing in size and causing difficulty in speech and mastication. All tumors were polypoid, covered by healthy mucosa, and ranged in size from 5 to 30 mm ( Figure 1 ). On extraoral examination, no masses were identified on the head and neck. Submandibular or cervical lymphadenopathy was unremarkable. A clinical diagnosis of multiple tongue lipomas was made. The tumors were resected surgically under intravenous anesthesia, and histologic examination was done. Histopathology showed intersecting fascicles of spindle-shaped cells admixed with sheets of mature adipocytes. The spindle-shaped cells showed mild pleomorphism with occasional mitotic figures, focal areas of myxoid changes, and scattered mast cells with entrapment of striated muscle ( Figure 2 ). Immunohistochemistry was not performed in this case. The patient was followed up at 6 months with no recurrence.

Figure 1.

Clinical image pretreatment.

Figure 2.

Histopathologic findings.

Discussion

SCL is an asymptomatic benign neoplasm and was first described in 1975 by Enzinger and Harvey ² as an uncommon variant of lipoma. Patients with SCL of tongue are older adults (mean, 58.2 years) with a male predilection (male:female, 1.9:1). ³ We presume that this is the first case report of multiple lingual SCLs in a pediatric population. SCLs present clinically as a painless mass or swelling, often with a history of slow growth over long duration. SCLs are typically solitary, although bilateral SCLs of tongue have been reported. On gross examination, SLCs are well-circumscribed neoplasms with firm consistency. The cut surface varies from yellow to gray-white, the latter representing the spindle cell component of the tumor. SCLs are composed of bland spindle cells with minimal pleomorphism and mitoses, ⁴ as in our case. When a diagnosis of lingual SCL is considered, the possibility of an atypical lipomatous tumor or well-differentiated liposarcoma should be kept in mind. A recent review of oral cavity atypical lipomatous tumors and well-differentiated liposarcomas showed involvement of the tongue in 55% of cases. Similar to SCLs, liposarcomas of the tongue present clinically as painless, slow-growing masses. On histologic examination, atypical lipomatous tumor and well-differentiated liposarcoma can exhibit a spindle cell component that can lead to confusion with SCL. ⁵ The spindle cells in SCLs are usually positive for CD34 in immunohistochemistry. CD34 expression is common in various other lipomatous and nonlipomatous soft tissue tumors; thus, this marker has a limited role in the diagnosis of SCL, and diagnosis is established primarily on histologic findings.

Conclusion

SCL is an asymptomatic benign soft tissue neoplasm that is an uncommon variant of lipoma. SCL arising in the tongue is very rare. The lesion is typically of slow-growing nature, encapsulated, and usually symptom free. SCL may be easily mistaken for other lipomatous and nonlipomatous neoplasms, benign and malignant. Surgical excision is the elective treatment of choice and involves easy removal from surrounding tissues. Lingual SCL follows a benign clinical course with no risk of recurrence, but long-term follow-up is mandatory.

Author Contributions

Sonika Dhari Shrestha, drafting and compiling of article, getting consent from patient’s father and approval from Institutional Review Board at Kanti Children’s Hospital; Uma Bhatta, pathology, help with HPE image production and findings; Anuj Kayastha, general surgery, drafting of article; Tridip Bahadur Pantha, supervision and correction of drafted article.

Disclosures

Competing interests: None.

Sponsorships: None.

Funding source: None.