A Suspected Metastatic Liposarcoma of the Inferior Alveolar Nerve Canal: A Case Report and Literature Review

AUTHORS

Sara Pourshahidi 1 , Marzieh Yousefian 1 , * , Soheila Manifar 1 , Farzaneh Mosavat 1 , Mohsen Esfandbod 2 , Behnaz Jahanbin 3 , Samaneh Salarvand 3

1 Department of Oral and Maxillofacial Medicine, Faculty of Dentistry, Tehran University of Medical Sciences, Tehran, Iran

2 Department of Internal Medicine, School of Medicine, Tehran University of Medical Sciences, Tehran, Iran

3 Pathology Department, Cancer Research Institute, Tehran University of Medical Sciences, Tehran, Iran

How to Cite: Pourshahidi S, Yousefian M, Manifar S, Mosavat F , Esfandbod M , et al. A Suspected Metastatic Liposarcoma of the Inferior Alveolar Nerve Canal: A Case Report and Literature Review, Ann Mil Health Sci Res. Online ahead of Print ; 18(4):e108733. doi: 10.5812/amh.108733.

ARTICLE INFORMATION

Annals of Military and Health Sciences Research: 18 (4); e108733
Published Online: December 29, 2020
Article Type: Case Report
Received: August 22, 2020
Revised: September 27, 2020
Accepted: September 29, 2020
Crossmark
Crossmark
CHECKING
READ FULL TEXT

Abstract

Introduction: Liposarcoma is the most common soft tissue sarcoma, but it is relatively scarce in the head and neck. The metastatic liposarcoma is more infrequent than the primary one.

Case Presentation: Here is presented a suspected case of metastatic liposarcoma in the inferior alveolar nerve canal in a 40-year-old female patient with a chief complaint of the chin and lower lip numbness and medical history of liposarcoma in the thigh. The present study was a case report for metastatic liposarcoma in the head and neck regions, according to the English language literature found in PubMed and Google Scholar. The search was performed using the keywords "Metastatic Liposarcoma" and "Head" and "Neck".

Conclusions: According to the findings, the reported case may be the first case of the metastasis of liposarcoma in the inferior alveolar nerve canal. Ominous signs, such as numbness, should be considered in patients with a history of cancer, due to their warning for metastasis or recurrence of the tumor.

1. Introduction

Soft tissue sarcomas are rare among adult malignant tumors (0.7%) (1). Liposarcoma, which was first introduced by Virchow in 1857 (2, 3), is a tumor due to malignant transformation of adipose tissue (4). It is the most common soft tissue sarcoma (5); 30% of liposarcomas have a myxoid pathologic feature (6). This tumor often occurs in the retroperitoneum and extremities (7) and adults aged 40-60 years, with slightly more prevalent in males (8). It is relatively rare in the head and neck regions (5%-9%) (9, 10). The metastatic cases reported in the head and neck region are even rarer in comparison to primary tumors. Here is reported a case of suspected metastatic liposarcoma tumor in the inferior alveolar nerve canal.

2. Case Presentation

A 40-year-old female patient was referred to the Oral and Maxillofacial Medicine Department of Tehran University of Medical Sciences. Her chief complaint was numbness of the chin and the lower lip, started one month ago from the right side and gradually extended to the left side. However, the main dysesthesia still focused on the right side. On history, she had painful swelling in the right thigh, surgically removed two years ago. The lesion was myxoid liposarcoma without lymphatic or vascular invasion.

After surgery, she underwent radiotherapy. After one year, a tumor was excised from her thorax, which was myxoid/round cell liposarcoma, based on histopathology findings.

Upon examination, bilateral dysesthesia of the skin of the lower lip and chin was observed. The mandibular incisors of both sides were vital. Based on these data, panoramic radiography was ordered, in which an irregularity was observed in the alveolar nerve canal on the right side.

For further examination, a cone beam computerized tomography (CBCT) was performed on her mandible, showing two radiolucent areas (6.4 × 9.6 and 17.1 × 4 mm) with ill-defined borders on the right side. The lesions were close to the inferior alveolar canal, and the cortical border of the canal was not detectable in the proximity of the lesion. No cortical expansion or thinning was observed in buccal and lingual plates (Figure 1).

Figure 1. (a) Two radiolucent areas (6.4 × 9.6 and 17.1 × 4 mm) with no cortical expansion and perforation in the reconstructed panoramic-like view of cone-beam computed tomography; (b) Cross-section view of the anterior lesion; (c) Cross-section view of the posterior lesion

The patient was referred to her oncologist to investigate possible metastasis of liposarcoma to the mandible.

The oncologist preferred to order a total body FDG-PET/CT (fluorodeoxyglucose-positron emission tomography/computed tomography) scan. Its report indicated soft tissue densities in the posterior trunk, especially on the left side. Then, a core needle biopsy of the bone marrow was performed. Microscopic examination of bone marrow revealed sarcoma with a myxoid background containing fat cells and rare lipoblasts. The stroma had arborizing, thin-walled vessels replacing normal hematopoietic elements. The pathology report was compatible with metastatic liposarcoma (Figure 2).

Figure 2. Involvement of marrow spaces with sarcoma composed of atypical adipocytes and few lipoblasts in the vascularized myxoid background (4x10 magnification)

According to the extent of the disease, the oncologist decided to use the palliative chemotherapy regime of AIM (Adriamycin, Ifosfamide, Mesna).

Surgery is the standard treatment of liposarcoma. If surgical treatment is not sufficient or the tumor progresses, chemotherapy and radiography are employed, which were applied to the reported patient. Unfortunately, the patient was lost to follow-up.

3. Discussion

Liposarcoma is the malignant tumor of lipoid cells. The reported cases of head and neck liposarcoma are few, mostly occur in the neck, face, larynx, dura, orbit, oral cavity, ears, mandible, and parotid, respectively (11). The metastatic cases to the head and neck region are extremely rare. Studies on metastatic liposarcoma in the head and neck region, using the keywords "Metastatic Liposarcoma" and "Head" and "Neck", were searched in the English language database of PubMed and the search engine of Google Scholar, and finally, a total of 25 cases were found (Table 1).

Table 1. Metastatic Liposarcoma in the Head and Neck Region
AuthorPatient's AgePatient's GenderPrimary AreaMetastatic Area
Trabelsi (12)42MThighParotid
Daly (13)46MLower extremitiesLarynx
Zagzoog (14)43FLower extremitiesSkull base
Haft (14)52MThighBrain
Paraf (14)27MHeartBrain
Arepally (14)56MThighBrain
Fitzpatrick (14)74FThighBrain
Kumar (14)73FThighBrain
Joshua (15)68MLeft shoulderRight gingival mucosa
Tysome (15)51MThighThyroid
Fabi (15)53MRetroperitoneumBilateral orbits
Ohashi (15)38MLeft tibiaSubmandibular region
Ohashi (15)41MLeft kneeAnterior neck
Brandwein-Gensler (15)86FThighThyroid
Azar (15)30FThighThyroid
Bashir (15)55FButtockThyroid
Bailey (15)54FSublingualParafalcine dura mater
Utsunomiya (15)44MThighDura mater and skull base
Fezza and Sinard (15)75FAbdomenOrbit
Cheng (15)Not specifiedNot specifiedButtockDura mater
Tijl (15)52FNot specifiedOrbit
Nasr (15)57FRetroperitoneumOrbit
Peison (15)50MRetroperitoneumScalp
Abdalla (15)43MThighOrbit
Tatsis (16)78MScrotalTongue

According to the current review report, there was no metastatic case of liposarcoma to the mandibular nerve canal, and the reported case might be the first one with mental numbness as the first sign of metastasis. Although clinical signs, panoramic radiographic view, and CBCT confirmed the presence of the lesion in the mandibular canal, PET-FDG CT scan was negative for any lesion in the site. The false-negative result of the PET-FDG CT scan can be related to the low activity of the tumor, while the method is based on the metabolic activity of the lesion (17). The small tumor size (18, 19) and the spatial resolution limitations related to PET-FDG CT scan (17) were the other reasons for the false-negative results.

Although the mandibular lesion was not detected by PET-FDG CT scan, it led to finding other metastatic lesions.

3.1. Conclusion

Signs and symptoms of patients with a history of cancer should be considered more cautiously since they could be the first trace of cancer metastases or recurrence.

Footnotes

References

  • 1.

    Chang HR, Hajdu SI, Collin C, Brennan MF. The prognostic value of histologic subtypes in primary extremity liposarcoma. Cancer. 1989;64(7):1514-20. doi: 10.1002/1097-0142(19891001)64:7<1514::aid-cncr2820640726>3.0.co;2-2.

  • 2.

    Kim YB, Leem DH, Baek JA, Ko SO. Atypical lipomatous tumor/well-differentiated liposarcoma of the gingiva: a case report and review of literature. J Oral Maxillofac Surg. 2014;72(2):431-9. doi: 10.1016/j.joms.2013.06.222. [PubMed: 23992781].

  • 3.

    Virchow R. A case of malignant occurring in part in the form of fat neuroma tumors. Virchows Arch Pathol Anat. 1857;11(3):281-8.

  • 4.

    McCulloch TM, Makielski KH, McNutt MA. Head and neck liposarcoma. A histopathologic reevaluation of reported cases. Arch Otolaryngol Head Neck Surg. 1992;118(10):1045-9. doi: 10.1001/archotol.1992.01880100035010. [PubMed: 1389054].

  • 5.

    Dei Tos AP. Liposarcoma: new entities and evolving concepts. Ann Diagn Pathol. 2000;4(4):252-66. doi: 10.1053/adpa.2000.8133. [PubMed: 10982304].

  • 6.

    Orvieto E, Furlanetto A, Laurino L, Dei Tos AP. Myxoid and round cell liposarcoma: a spectrum of myxoid adipocytic neoplasia. Semin Diagn Pathol. 2001;18(4):267-73. [PubMed: 11757867].

  • 7.

    Mack TM. Sarcomas and other malignancies of soft tissue, retroperitoneum, peritoneum, pleura, heart, mediastinum, and spleen. Cancer. 1995;75(S1):211-44.

  • 8.

    Enzinger FM, Weiss SW. Soft Tissue Tumors,. 3rd edn ed. Inc., St Louis: Mosby Year Book; 1995.

  • 9.

    Golledge J, Fisher C, Rhys-Evans PH. Head and neck liposarcoma. Cancer. 1995;76(6):1051-8. doi: 10.1002/1097-0142(19950915)76:6<1051::aid-cncr2820760620>3.0.co;2-4.

  • 10.

    Stewart MG, Schwartz MR, Alford BR. Atypical and malignant lipomatous lesions of the head and neck. Arch Otolaryngol Head Neck Surg. 1994;120(10):1151-5. doi: 10.1001/archotol.1994.01880340091016. [PubMed: 7917199].

  • 11.

    Weiss SW, Goldblum JR, Folpe AL. Enzinger and Weiss's soft tissue tumors. Elsevier Health Sciences; 2007.

  • 12.

    Trabelsi A, Ben Abdelkrim S, Jemni H, Stita W, Ouni C, Dhouibi A, et al. Metastatic liposarcoma to the parotid. J Oncol. 2008;2008:715153. doi: 10.1155/2008/715153. [PubMed: 19259332]. [PubMed Central: PMC2648632].

  • 13.

    Daly SF, Sciubba J, Tufano RP. Lower-extremity liposarcoma metastatic to the larynx: case report. Ear Nose Throat J. 2006;85(3):185-6. 189. [PubMed: 16615602].

  • 14.

    Zagzoog N, Ra G, Koziarz A, Provias J, Sommer D, Almenawer SA, et al. Metastatic Liposarcoma of the Skull Base: A Case Report and Review of Literature. Neurosurgery. 2017;80(4):219-23. doi: 10.1093/neuros/nyw157. [PubMed: 28362929].

  • 15.

    McElderry J, McKenney JK, Stack BC. High-grade liposarcoma metastatic to the gingival mucosa: case report and literature review. Am J Otolaryngol. 2008;29(2):130-4. doi: 10.1016/j.amjoto.2007.04.001. [PubMed: 18314026].

  • 16.

    Tatsis D, Deligiannidis D, Papaemmanouil S, Vahtsevanos K. Metastatic myxoid liposarcoma to the tongue: a unique intraoral metastasis. Contemp Oncol (Pozn). 2020;24(3):203-5. doi: 10.5114/wo.2020.97483. [PubMed: 33235548]. [PubMed Central: PMC7670184].

  • 17.

    Chang JM, Lee HJ, Goo JM, Lee HY, Lee JJ, Chung JK, et al. False positive and false negative FDG-PET scans in various thoracic diseases. Korean J Radiol. 2006;7(1):57-69. doi: 10.3348/kjr.2006.7.1.57. [PubMed: 16549957]. [PubMed Central: PMC2667579].

  • 18.

    Goo JM, Im JG, Do KH, Yeo JS, Seo JB, Kim HY, et al. Pulmonary tuberculoma evaluated by means of FDG PET: findings in 10 cases. Radiology. 2000;216(1):117-21. doi: 10.1148/radiology.216.1.r00jl19117. [PubMed: 10887236].

  • 19.

    Kostakoglu L, Agress HJ, Goldsmith SJ. Clinical role of FDG PET in evaluation of cancer patients. Radiographics. 2003;23(2):315-40. quiz 533. doi: 10.1148/rg.232025705. [PubMed: 12640150].

  • Copyright © 2020, Annals of Military and Health Sciences Research. This is an open-access article distributed under the terms of the Creative Commons Attribution-NonCommercial 4.0 International License (http://creativecommons.org/licenses/by-nc/4.0/) which permits copy and redistribute the material just in noncommercial usages, provided the original work is properly cited.
    COMMENTS

    LEAVE A COMMENT HERE: