Solitary neurofibroma of the floor of the mouth: rare localization at lingual nerve with intraoral excision

Abstract

Background Neurofibromas (NF) are benign tumors of the peripheral nerves that are composed of Schwann cells, perineural-like cells and fibroblasts. The differential diagnosis for a solitary intraneural variant of neurofibroma arising in the floor of the mouth is broad and includes a submandibular gland neoplasm and adenopathy, among others. The intraoral approach is the best choice for a medium-sized lesion. Case presentation We report a rare case of a solitary neurofibroma of the floor of the mouth in a 31-year-old male. The patient consulted the dental emergency department for acute pain of the left mandible. Systematic clinical examination revealed the presence of a mass in the left mouth floor. The panoramic x-ray was not conclusive and the magnetic resonance imaging (MRI) revealed a well-defined soft tissue lesion with homogenous isosignal intensity on the T1-weighted image, high intensity signal on the T2-weighted image and heterogeneous enhancement following contrast-enhancement on the T1-weighted Fast Sat image. The surgical excision of the soft-tissue neoplasm was accomplished by an intraoral approach. The specimen was sent for histopathologic analysis and Immunohistochemical studies which confirmed the diagnosis of a myxoid predominant intraneural solitary neurofibroma. Conclusion The diagnosis of neurofibroma was confirmed by histopathological evaluation and immunohistochemical studies which also excluded other entities in the histopathologic differential diagnosis including schwannoma and a malignant peripheral nerve sheath tumor among other. Localized (solitary) neurofibromas most often occur as sporadic lesions, however; diagnosis of a solitary neurofibroma prompts clinical evaluation to exclude the remote possibility of neurofibromatosis. The purpose of this case report is to raise awareness of the uncommon presentation of neurofibroma and to document the successful management of such a lesion using an intraoral approach.

Large solitary neurofibroma of the cheek mucosa: a case report

“…Their etiological factor is unclear, some authors believe that solitary neurofibromas seem to contain hyper material of a hamartomatous and non-neoplastic nature. Maxillofacial lesions usually originate from branches of the trigeminal and Research, Society and Development, v. 10, n. 2, e56810212905, 2021 (CC BY 4. (Chrysomali et al, 1997;Broly et al, 2019). The neural origin of the lesion presented in this report is thought to arise from the buccal nerve, as after its surgical excision the main patient complaint was paresthesia in the right inner cheek and posterior vestibular mucosa.…”

“…Solitary oral neurofibromas consist of slow-growing, rarely painful submucosal masses, and neurological disturbances are often uncommon (Broly et al, 2019). They mainly appear in the third decade of life although occurrence between 10 months and 70 years old has been described in the literature, with no gender predilection (Depprich et al, 2009;Marocchio et al, 2007;Salla et al, 2011).…”

“…The neural origin of the lesion presented in this report is thought to arise from the buccal nerve, as after its surgical excision the main patient complaint was paresthesia in the right inner cheek and posterior vestibular mucosa. Broly et al, (2019) reviewed the literature and found 26 cases of neurofibromas in the oral cavity reported in English literature with just one case located at the cheek. All of which were found to developing from trigeminal branches.…”

“…Neurofibroma sometimes is found as a firm oval or rounded-shape mass located in subcutaneous/submucosal tissues, thus making a differential diagnosis with lipoma, fibroma and sharing the same neural origin with other tumors (eg. schwannoma, traumatic neuroma, nerve sheath myxoma) (Chrysomali et al, 1997;Broly et al, 2019). Therefore, MRI and histological examination are mandatory to confirm the diagnostic.…”

Solitary Neurofibroma of the Hard Palate: A Case Report and Literature Review

“…Neurofibromas are common in the tongue and buccal mucosa in the oral cavity, but approximately 20–60% of oral neurofibromas are associated with neurofibromatosis [ 14 ]. In 2019, Broly et al [ 15 ] reviewed 26 cases of isolated neurofibromas occurring in the oral cavity [ 15 ], and intraoral occurrences were very rare. However, because the oral cavity is composed of various tissues such as the tongue, buccal mucosa, gingiva, floor, and palate, the neurofibromas that develop there have different nerves of origin and different surrounding tissues that are removed during surgery.…”