Case Report

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Case Report

A 37mm Spindle Cell Lipoma on the Floor of the Mouth

Eve Malthiery,

¹

Valérie Costes-Martineau,

²

Marie-Alix Fauroux,

¹

and Jacques-Henri Torres

¹

1Service d’Odontologie, Centre Hospitalier Universitaire, 34295 Montpellier Cedex 5, France

2Laboratoire d’Anatomie et de Cytologie Pathologiques, Centre Hospitalier Universitaire, 34295 Montpellier Cedex 5, France

Correspondence should be addressed to Jacques-Henri Torres; j-torres@chu-montpellier.fr Received 21 April 2019; Revised 26 May 2019; Accepted 19 June 2019; Published 4 August 2019 Academic Editor: Eugenio Maiorano

Copyright © 2019 Eve Malthiery et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Spindle cell lipoma (SCL) is a rare variety of lipoma that mostly arises in male patients and rarely affects the oral cavity. The floor of the mouth is an uncommon site for SCL, and very few cases have been reported in this location. A case of SCL is reported in a 70-year-old woman who had noticed a swelling of the floor of the mouth without any functional consequence. Both ultrasonography and RMI suggested a diagnosis of ranula, whereas clinical palpation showed a nonfluctuant mass. The lesion was excised under local anesthesia. A 37 × 32 mm encapsulated yellow mass was removed. Histological features (mature adipocytes and CD34+ spindle cells) led to a diagnosis of SCL. Medical imaging assessment of this lesion could have been influenced by the high frequency of the ranulas in the floor of the mouth. This case appears to be quite infrequent because of its location (floor of the mouth), its size (over 3.5 cm), and the patient’s gender (female).

1. Introduction

Lipomas are not frequent in the oral cavity. They only repre- sent 0.1 to 5% of oral tumors [1]. They usually appear as soft yellowish nodular swellings covered by normal mucosa.

Lipomas seldom aﬀect the ﬂoor of the mouth (2 among the 46 lipomas reported by Fregnani et al., while 10 were in the buccal mucosa) [1].

Several histopathological types of lipomas have been described: ﬁbrolipoma, sialolipoma, myelolipoma, osteoli- poma, chondrolipoma, chondroid lipoma, angiolipoma, angiomyolipoma, myolipoma, intramuscular lipoma, pleomorphic lipoma, and spindle cell lipoma (SCL) [2]. Enzinger and Harveyﬁrst described SCL in 1975 [3]. These tumors typically arise in the subcutaneous tissue of the upper back, posterior neck, and shoulders of males aged 40–70 and scarcely involve the oral cavity [4].

In 2003, Fregnani et al. reported 46 cases of lipomas of the oral cavity [1]. Five of them were in the ﬂoor of the mouth, none of which were SCL. Although SCL was predom- inant in the lip in the Furlong et al. 2004 report of 95 lipomas of the oral region, only 1 SCL was detected among the 5 cases

reported in theﬂoor of the mouth [5]. In 2006, Billings et al.

found 16 published SCL cases of the oral cavity in the literature [4]. They reported 7 new cases of SCL, of which only 1 was located in the floor of the mouth. In their 2010 series of 41 cases of lipomas, Juliasse et al. reported 1 SCL among 3 cases of lipomas in the floor of the mouth [2]. In 2013, Manor et al. found 33 reported cases of SCL of the oral cavity and added 2 personal cases [6]. The average size of these lesions was 20.87 mm; male predilection was 2 : 1. Among these 35 cases, only 5 affected the floor of the mouth.

We are reporting a new case of SCL in theﬂoor of the mouth in a 70-year-old woman.

2. Observation

A 70-year-old Caucasian woman was referred to the hospital by her dentist for a swelling noticed 3 weeks before in the ﬂoor of the mouth. It had no functional consequence on her speech or mastication and neither on tongue mobility.

An echography was performed, which lead to the conclusion of a ranula. The patient’s medical conditions of osteo- porosis, hypothyroidism, and mild depression were treated

Volume 2019, Article ID 2138928, 4 pages https://doi.org/10.1155/2019/2138928

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by alendronic acid, levothyroxine, and ﬂuoxetine. Physical examination revealed a tender mass, in the right sublingual region, though its posterior limit could not be clearly deﬁned (Figure 1).

The swelling was not attached to the mandible bone or to other surrounding tissues. The mucosa covering the mass had a normal appearance and color. No inflammation, ero- sion, or ulceration were noted. Salivaryflow from the right submandibular gland was not affected by the swelling. Palpa- tion revealed a nonfluctuant mass without cervical lymphadenopathy.

An MRI scan was prescribed, which showed a 37 mm well-deﬁned lesion with both T1 and T2 hypersignals (Figure 2); the conclusion was that the sublingual mass could be a ranula. The patient was proposed an excision, and the procedure was performed under local anesthesia.

An encapsulated yellow mass was removed (Figure 3).

Histopathological examination established a diagnosis of spindle cell lipoma (Figure 4).

A proliferation of adipocytes in a myxoid background was seen, associated to small bland spindle cells with small chromatic nuclei without mitosis or atypia, low cellular den- sity, and without wavy collagenﬁbers. Adipocytes size was regular, without signiﬁcant anisocytosis unlike the recently described anisometric cell lipoma [7]. Immunohistochemical examination was HMGA2 and MDM2 negative. Spindle cells were immunoreactive for CD34 and negative for PS100 (Figures 5 and 6).

3. Discussion

Like in the case reported here, oral lipomas are usually asymptomatic. However, large lipomas have been reported to cause functional impairment: a lipoma measuring 5 cm disturbed lower denture stability [8]. An 8.5 cm sublingual lipoma has been reported to cause submandibular salivary gland swelling [9].

In the case series reported by Manor et al. [6], only one SCL in theﬂoor of the mouth was larger (45 mm) than the one reported here (37 mm). Despite the size of the lesion, local anesthesia was chosen for surgery in this case because of patient’s cooperation and preference to avoid general anesthesia.

In this case, ultrasonography could not eliminate a diagnosis of the ranula. Yet, a ranula typically appears as an anechoic or hypoechoic cystic component, whereas a Figure 1: Clinical aspect of the mass located in the right sublingual

region (tip of the tongue turned to the left side). The superior limit is above the occlusal surface of the molars.

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Figure 2: RMI features of the lesion: both T1- (a) and T2- (b) weighed images show hypersignal.

Figure 3: Resection specimen: 37 × 32 mm encapsulated yellow mass.

2 Case Reports in Dentistry

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lipoma has an isoechoic appearance with multiple internal fine echogenic lines [10]. Clinical palpation was more effi- cient, showing that the mass was not liquid. At this stage, the most likely clinical diagnosis was a sublingual gland pleomorphic adenoma. On RMI, imaging features of such lesions are typically T1 low signal and T2 marked hyperin- tensity [11]. The RMI exam, which has not been performed in the hospital, showed T1 and T2 hypersignals. However, the RMI report proposed a diagnosis of ranula. It may also be supposed that the location of the mass influenced the assessment; a ranula is indeed much more frequent than an adenoma or a lipoma of the floor of the mouth. Both lipoma and ranula are supposed to give homogenous internal structure, but lipoma T1-T2 images are expected to be high/high whereas ranula T1-T2 images are supposed to be low/markedly high [12]. In the present case, the RMI appearance was in favor of a lipoma.

From a histologic point of view, SCLs are made up of mature adipocytes and CD34+ spindle cells in a myxoid matrix with bands of birefringent collagen. Their histological diﬀerential diagnosis includes benign lesions such as classic

lipoma, schwannoma, neurofibroma, leiomyoma, solitary fibrous tumor (whose prognosis is regarded as uncertain) and truly malignant ones such as atypical lipomatous tumors (well-differentiated liposarcoma), and dermatofibrosarcoma protuberans [13]. In particular, secondary degenerative modifications and atrophy observed in SCL must not be mistaken with the malignant histologic features seen in lipo- sarcomas [14].

4. Conclusion

This case appears to be very infrequent because of its location (ﬂoor of the mouth), its size (over 3.5 cm), and the patient’s gender (female). It also reminds the clinicians to prudently consider data from imaging interpretation of tumors.

Conflicts of Interest

The authors declare that they have no conﬂicts of interest regarding the publication of this paper.

References

[1] E. R. Fregnani, F. R. Pires, R. Falzoni, M. A. Lopes, and P. A.

Vargas,“Lipomas of the oral cavity: clinical ﬁndings, histological classiﬁcation and proliferative activity of 46 cases,” Inter- national Journal of Oral and Maxillofacial Surgery, vol. 32, no. 1, pp. 49–53, 2003.

[2] L. E. R. Juliasse, C. F. W. Nonaka, L. P. Pinto, R. A. Freitas, and M. C. C. Miguel, “Lipomas of the oral cavity: clinical and histopathologic study of 41 cases in a Brazilian population,” European Archives of Oto-Rhino-Laryngology, vol. 267, no. 3, pp. 459–465, 2010.

[3] F. M. Enzinger and D. A. Harvey,“Spindle cell lipoma,” Can- cer, vol. 36, no. 5, pp. 1852–1859, 1975.

[4] S. D. Billings, J. D. Henley, D.-J. Summerlin, S. Vakili, and C. E.

Tomich,“Spindle cell lipoma of the oral cavity,” The American Journal of Dermatopathology, vol. 28, no. 1, pp. 28–31, 2006.

[5] M. A. Furlong, J. C. Fanburg-Smith, and E. L. B. Childers,

“Lipoma of the oral and maxillofacial region: site and subclas- siﬁcation of 125 cases,” Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontology, vol. 98, no. 4, pp. 441–450, 2004.

[6] E. Manor, N. Sion-Vardy, P. A. Brennan, and L. Bodner,

“Spindle cell lipoma of the oral cavity: a clinico-pathologic analysis of 35 reported cases,” Surgical Science, vol. 4, no. 03, pp. 196–201, 2013.

[7] H. L. Evans,“Anisometric cell lipoma: a predominantly subcutaneous fatty tumor with notable variation in fat cell size but not more than slight nuclear enlargement and atypia,” AJSP:

Reviews & Reports, vol. 21, pp. 195–199, 2019.

[8] Y. Anavi, M. Gross, and S. Calderon,“Disturbed lower denture stability due to lipoma in theﬂoor of the mouth,” Journal of Oral Rehabilitation, vol. 22, no. 1, pp. 83–85, 1995.

[9] A. R. Gray and G. R. Barker,“Sublingual lipoma: report of an unusually large lesion,” Journal of Oral and Maxillofacial Sur- gery, vol. 49, no. 7, pp. 747–750, 1991.

[10] L. Zhong, S. Zhao, G. Chen, and F. Ping,“Ultrasonographic appearance of lipoma in the oral and maxillofacial region,” Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontology, vol. 98, no. 6, pp. 738–740, 2004.

Figure 4: Histology showing mature adipocytes, uniform spindle cells, and short bundles of collagen in a myxoid matrix (HE staining, × 20).

Figure 5: Immunohistochemical anti-PS100 staining ( × 20):

adipocytes are PS100 positive.

Figure 6: Immunohistochemical anti-CD34 staining ( × 20):

adipocytes are CD34 negative and spindle cells are CD34 positive.

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[11] E. Matsusue, Y. Fujihara, E. Matsuda et al.,“Diﬀerentiating parotid tumors by quantitative signal intensity evaluation on MR imaging,” Clinical Imaging, vol. 46, pp. 37–43, 2017.

[12] T. Kurabayashi, M. Ida, M. Yasumoto et al.,“MRI of ranulas,”

Neuroradiology, vol. 42, no. 12, pp. 917–922, 2000.

[13] N. V. M. Milhan, A. S. R. Cavalcante, Y. M. F. S. Marques, Y. R. Carvalho, and A. L. Anbinder, “Spindle cell lipoma occurring in the buccal mucosa: an unusual location of this benign lipomatous neoplasm,” Case Reports in Pathology, vol. 2015, Article ID 805730, 5 pages, 2015.

[14] E. Manor, N. Sion-Vardy, B. Z. Joshua, and L. Bodner,“Oral lipoma: analysis of 58 new cases and review of the literature,” Annals of Diagnostic Pathology, vol. 15, no. 4, pp. 257–261, 2011.

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Case Report

Case Report

A 37mm Spindle Cell Lipoma on the Floor of the Mouth

Eve Malthiery,

Valérie Costes-Martineau,

Marie-Alix Fauroux,

and Jacques-Henri Torres

1. Introduction

2. Observation

3. Discussion

4. Conclusion

Conflicts of Interest

References

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