Abstract
Dedifferentiated liposarcoma (DDLS) has a relatively poor prognosis, however this neoplasm rarely occurs in the head and neck. To date, no definite protocol has been established for the diagnosis and treatment of head and neck DDLS. The present study reports the case of a 69-year-old male patient with DDLS of the oral floor. To the best of our knowledge, this is the first documented case of oral floor DDLS. In addition, this is the first reported case with the development of a second primary malignancy following the treatment of head and neck DDLS. A literature review of 50 cases of head and neck DDLS revealed that preoperative biopsy is not reliable for the diagnosis of these tumors and an accurate pathological diagnosis with total resection is preferred.
Keywords: dedifferentiated liposarcoma, biopsy, second primary malignancy, oral cavity, oral floor
Introduction
Liposarcoma (LS) is the most common tumor among sarcomas of the soft tissue (−20% of the tumors in adults) (1). This neoplasm was first described by Virchow (2) in 1857 and has been well documented thereafter (3,4). LS is categorized into four subgroups: atypical lipomatous tumor (ALT)/well-differentiated liposarcoma (WDLS), myxoid liposarcoma, pleomorphic liposarcoma, and dedifferentiated liposarcoma (DDLS) (5). Among these, DDLS is defined as a subtype of ALT/WDLS with non-lipogenic lesions (heterogenous lesions in one tumor) (5). DDLS has a high degree of malignancy; hence, its recurrence and metastasis rates are higher than those of other types of LS (6,7). DDLS can develop anywhere in the body; however, the head and neck (H&N) is a relatively rare site of occurrence of this lesion (7,8). The pathological features of DDLS are well defined (5,9). Here we report the case of a 69-year-old male patient with DDLS of the oral floor. It was difficult to determine the diagnosis clinically. Furthermore, to date, no definite protocol has been established for the diagnosis and treatment of H&N DDLS.
Case study
Written informed consent was obtained from the patient for the publication of this case report and accompanying images. The report was submitted for ethical review to the Ethics Committee of the University of the Ryukyus (Okinawa, Japan), which waived the requirement for review per institutional protocol because the study does not contain content that requires ethical approval. The Ethics Committee approved the submission and publication of the manuscript.
A 69-year-old man presented to the Department of Oral and Maxillofacial Surgery at Ryukyu University Hospital. He had noticed a slow-growing mass in his mouth and experienced difficulty in talking for approximately 1 year. Physical examination revealed a painless, smooth, and non-tender (firm) mass at the floor of the mouth (Fig. 1). The mass was covered by an intact mucosa. The Wharton duct was not involved by the mass, and clear saliva could be expressed from the sublingual gland duct. The patient's facial appearance was symmetrical, and there was no cervical lymphadenopathy. He had a history of alcohol consumption and was a current smoker, with no history of malignancy. The patient was being treated for diabetes mellitus. His brother had a history of colorectal cancer. Contrast-enhanced computed tomography (CT) demonstrated a large heterogenous mass under the tongue that seemed to push the hyoglossus muscles, but no invasive lesion was present. The margins of the lesion were well defined. The adipose-like section of the mass was partially suspected. No other lesions were detected in the H&N, bones, and lungs. Contrast-enhanced magnetic resonance imaging (MRI) demonstrated a 50×39×43 mm lesion that pushed the hyoglossus muscle into the sublingual space and seemed to contain heterogeneous components (Fig. 2). Most of the mass revealed low-signals in T1-weighted image and high-signals in T2-image. On the other hand, at the bottom of the mass, fat signals were partially detected. No other lesion was present. Based on the findings, the oral floor lesion was considered a tumor or cyst; however, an apparent clinical diagnosis could not be made. Moreover, performing biopsy for an oral floor is difficult (10). Therefore, we planned for surgical resection and accurate pathological examination.
Figure 1.
Intraoral photograph taken at the initial examination. Physical findings indicated a painless, smooth, and non-tender (firm) mass located at the floor of the mouth. The mass was covered with an intact mucosa.
Figure 2.
MRI images. (A and B) Coronal sections. (C and D) Axial sections. An MRI scan revealed a 50×39×43 mm lesion that pushed the hyoglossus muscle into the sublingual space and seemed to contain heterogeneous components. (A) A contrast-enhanced fat-suppression T1-weighted image. (B) A fat-suppression T2-weighted image. (C) A T1-weighted image showed high-signals (yellow arrow) indicating a fatty lesion. (D) In contrast, a contrast-enhanced fat-suppression T1-weighted image showed the lesion had low-signals (yellow arrow). Most of the mass revealed low-signals in T1-weighted image and high-signals in T2-image. Most of the mass revealed low-signals in T1-weighted image and high-signals in T2-image. On the other hand, the bottom of the mass revealed fatty lesion. MRI, magnetic resonance imaging.
The patient underwent surgical resection of the mass under general anesthesia. The mass had no adhesions to the surrounding tissue. The excised specimen was a 60×45×45 mm capsulated mass. The resected mass showed two areas: A pale yellow (fatty) area and milky-white (non-fatty) area; however, no cystic lesion was found (Fig. 3). Histopathological examination also revealed two distinct areas, but the findings were contrasting (Fig. 4A): i) The milky-white area contained a dedifferentiated area which was composed of spindle cell and pleomorphic cell with patchy necrosis. Spindle cell showed a fascicular architecture with hyperchromatic plump nuclei and eosinophilic cytoplasm. Bizarre multinucleate giant cells were occasionally seen (Fig. 4B); ii) the yellow area was a well-differentiated area, which demonstrated adipocytic proliferation with hyperchromatic stromal cells (Fig. 4C). The two areas mostly transitioned abruptly, and partly transitioned gradually. Immunohistochemical examination revealed positive results for S-100 in the adipocytic cells, whereas it revealed partial positive results for SMA, desmin and CDK4, but negative for caldesmon or MDM2 in the dedifferentiated component. Based on the findings, DDLS (FNCLCC system grade 2) was diagnosed. The tumor was clinically resected; however, histological surgical margin was positive. Therefore, postoperative radiotherapy (RT) (total 60 Gy) was performed to treat the residual tumor and to prevent the recurrence or metastasis of the disease (3,11). At 5 years 8 months postoperatively, no sign of local recurrence or distant metastasis of DDLS had been found, until the time of writing this report. However, pleomorphic LS of the chest wall was detected after 5 years 2 months postoperatively. The patient was treated and followed up at another hospital (Fukuoka University Hospital, Fukuoka, Japan) to this writing. Histologically, atypical spindle-shaped cells, bizarre giant cells, and lipoblast-like cells were revealed in the chest wall tumor. These cells were negative for MDM2 or CDK4. Further, no DDLS component was observed. Therefore, the chest wall tumor was considered a second primary tumor rather than a metastasis of DDLS.
Figure 3.
The resected mass comprised a pale yellow (fatty) area, and a milky-white solid (non-fatty) area; however, no cystic lesion was observed.
Figure 4.
Histological examination of the specimen. (A) Hematoxylin and eosin staining revealed that the specimen comprised two distinct areas, *indicates the well-differentiated area and **indicates the dedifferentiated area. Magnification, ×100. (B) In the dedifferentiated area, bizarre multinucleate giant cells were occasionally observed. Magnification, ×200. (C) In the well-differentiated area, adipocytic proliferation with hyperchromatic stromal cells was observed. Magnification, ×200.
Discussion
To the best of our knowledge, this is the first documented case of oral floor DDLS. Furthermore, our case is the first to exhibit the development of a second primary malignancy (SPM) after the treatment of H&N DDLS. We searched the English literature for H&N DDLS cases that occurred between 1979 and 2017, using Pubmed and Google Scholar. The exclusion criteria were i) cases from non-English literature, ii) cases in which DDLS metastasis from non-H&N regions was apparent (12,13) and iii) a case in which it could not be determined whether the lesion was a DDLS or WDLS (14). We identified 50 cases [excluding the cases in Tirumani et al (12) study, where the number of cases was stated as ‘not applicable’ (NA)], which are listed in Table I (7,11,12,15–42). However, no patient had oral floor DDLS. This list includes 2 cases of tongue DDLS (19,22), but clinical information regarding these cases was sparse. Therefore, we could not confirm whether the DDLSs involved the oral floor in these cases. As described in Table I, DDLS has been reported to develop at various sites in the H&N region (7,11,12,15–42). Among these, the most common site was the larynx (6 patients), followed by the cheek (5 patients), neck (3 patients), orbit (3 patients), pyriform sinus (3 patients), buccal area (2 patients), tongue (2 patients), parotid gland, pharyngeal space, posterior neck, paralaryngeal area, nose, maxillary gingiva, and oral floor (current case), i.e., anywhere in the H&N region (Table I). The mean age was 58.78±17.27 years (range, 20–86 years), with a male/female ratio of 1.8:1. Most of the patients (except for the NA case) underwent contrast-enhanced CT or MRI for initial staging; however, no patient underwent positron emission tomography (PET) for initial staging (date not shown in Table I). Some patients underwent PET as an additional detection test after the first surgery (31) or as follow-up of radical surgery (37,42). For H&N DDLS, the outcomes are reportedly good with wide surgical excision (11). No patients underwent preoperative therapy, but 12 patients (including our case) underwent postoperative RT. No patient underwent postoperative chemotherapy, but one patient underwent therapy for the recurrence of the tumor (15). Given the sparse clinical details, the present literature review was unable to report any conclusions regarding treatment suggestions. Of 24 patients (except for the NA case), 3 (12.5%) reported recurrence and 1 (our case) developed SPM (4.2%); no patient with regional recurrence or distant metastasis was identified. However, case reports with long-term follow-up are limited. Of the 20 patients whose follow-up duration was reported, only 6 (30%) and 8 (40%) patients were followed up for >5 and 2 years, respectively. Meanwhile, cases of recurrence after 23 years of follow-up (16) and six recurrences over 26 years of follow-up (22) have been reported. Our case exhibited no recurrence or metastasis during 5 years of follow-up; however, SPM (pleomorphic LS of the chest wall) developed at 5 years after the H&N DDLS resection. We could not determine why the current patient developed SPM because there have been no reports of SPM in H&N DDLS cases to date. Lupo et al (43), reported on the statistical analysis of 8,785 sarcoma (at all regions of the body, including H&N) survivors diagnosed between 1992 and 2012 from the Surveillance, Epidemiology, and End Results database, using standardized incidence ratios. Among these, LS survivors (257 patients) had a relatively high SPM risk; however, there were no details regarding the DDLS survivors (30 patients) (43). To date, reports of SPMs in DDLS (at all regions of the body) cases are sparse (44). Therefore, our case indicates the possibility of SPM developing not only in the H&N region but also at all DDLS sites. According to the size of oral region LSs, lesions of >5.0 or >3.6 cm were reported as prognostic factors for recurrence, metastasis, or death (22,45). We researched the relationship between the size of H&N DDLS lesions and recurrence; however, no definitive data were found because of the sparsity of clinical information.
Table I.
DDLSs in the head and neck region.
| First author | Year | Age | Gender | Site | Size (cm) | Type of on biopsy | Histological diagnosis based on biopsy findings | Grade and histological type of DDLS | Postoperative RT | Follow-up data | (Refs.) |
|---|---|---|---|---|---|---|---|---|---|---|---|
| Tobey | 1979 | 61 | M | Larynx | NA | (+) | LS | (−) | (−) | Approximately 6 months; . recurrence and mortality | (15) |
| McCormick | 1994 | 62 | M | Larynx | NA | NA | NA | NA | NA | 23 years; recurrence | (16) |
| Henricks | 1997 | NA | NA | H&N | NA | NA | NA | NA | NA | NA | (17) |
| Henricks | 1997 | NA | NA | Larynx | NA | NA | NA | NA | NA | NA | (17) |
| Henricks | 1997 | NA | NA | Buccal | NA | NA | NA | NA | NA | NA | (17) |
| Cai | 2001 | 54 | F | Orbit | >2 | NA | NA | (−) | NA | NA | (18) |
| Nascimento | 2002 | 83 | F | Tongue | 2.5 | NA | NA | NA | NA | NA | (19) |
| Diamond | 2002 | 57 | M | Cheek | NA | (+) | Suggestive of neurofibroma | (−) | (+): 66 Gy | 12 months; NED | (20) |
| Gonzalez-Lois | 2002 | 69 | M | Pyriform sinus | >3 | (+) | Lipoma | (−) | (−) | 6 months; NED | (21) |
| Fanburg-Smith | 2002 | 39 | M | Tongue | 6 | NA | NA | Low-grade | NA | 6 years; NED | (22) |
| Fanburg-Smith | 2002 | 56 | M | Buccal (mucosa) | 5 | NA | NA | High-grade, focal myxoid features | NA | 26 years; 6 recurrences, but alive | (22) |
| Fanburg-Smith | 2002 | 67 | F | Parotid grand | 5.5 | NA | NA | High-grade | NA | 17 years; NED | (22) |
| Roza | 2004 | 61 | M | Cheek | 7 | (−) | (−) | (−) | (+) | Lost to follow-up | (23) |
| Cunha | 2005 | 42 | F | Cheek | 6 | (−) | (−) | (−) | (+) | 1 year; NED | (24) |
| Angiero | 2006 | 62 | M | Cheek | 3 | Incisional | LS | NA | (−) | 7 years; NED | (25) |
| Giordano | 2006 | 50 | M | Pyriform sinus | 5 | (−) | (−) | Low-grade | (−) | 6 months;, NED | (26) |
| Powitzky | 2007 | 63 | M | Larynx | 4.5 | (+) | Myxoid LS | High-grade, with myxomatous degeneration and clement rhabdomyosarcoma | (+): 70.2 Gy | 16 months; NED | (11) |
| Saeed | 2007 | 56 | F | Orbit | NA | (+) | DDLS grade 2 | Grade 2 | (+): 60 Gy | NED | (27) |
| Rogers | 2010 | 83 | M | Pharyngeal space | 8.6 | FNA | No evidence of malignancy | NA | (+): 64 Gy | 19 months; NED | (28) |
| Gritli | 2010 | NA | NA | Neck | NA | NA | NA | NA | (+) | NED | (29) |
| Endo | 2010 | 48 | M | Neck | 5 | (−) | (−) | Low-grade | (−) | 1 year; NED | (30) |
| Makeieff | 2010 | 62 | F | Larynx | 8 | (+) | A possible gastrointestinal stromal tumor (malignant) | NA | (+) | NED | (31) |
| Stomeo | 2012 | 76 | M | Cheek | 12+10 | Incisional | Lipomatous lesion | NA | (Refused by the patient) | 2 years; death with NED | (32) |
| Zhang | 2011 | 23 | F | Orbit | NA | (−) | (−) | NA | (+) | 16 months; NED | (33) |
| Blumberg | 2012 | 65 | M | Paratracheal | 4.7 | FNA | Failure | Low-grade, with meningothelial-like whorling | (−) | NED | (34) |
| Wang | 2012 | 20 | F | Neck | 5 | (−) | (−) | With an osteosarcomatous component | (−) | 5 months; NED | (35) |
| Zreik | 2015 | 86 | M | Posterior neck | 9.3 | US guided FNA | Suggestive of DDLS | NA | (+) | 4 months; NED | (36) |
| Gerry | 2014 | NA | NA | H&N (number of cases, 16) | NA | NA | NA | NA | NA | NA | (7) |
| Petersson | 2014 | 61 | F | Paralaryngeal | 6 | CT guided | Deceptively mild histopathological features (benign) | Suggestive of a partially benign dedifferentiated component | (+) | The case was reported during postoperative RT | (37) |
| Jour | 2015 | NA | NA | Larynx | NA | NA | NA | NA | NA | NA | (38) |
| Tirumani | 2015 | NA | NA | H&N (number of cases, NA) | NA | NA | NA | NA | NA | NA | (12) |
| Saâda-Bouzid | 2015 | 63 | M | Nose | NA | NA | NA | NA | NA | NA | (39) |
| Ishii | 2016 | NA | NA | H&N (number of cases, 2) | NA | NA | NA | NA | NA | NA | (40) |
| Riva | 2016 | 81 | M | Pyriform sinus | 21 | (−) | (−) | Grade 2 according to FNCLCC | (−) | 1 year; NED | (41) |
| Enomoto | 2017 | 28 | F | Maxillary gingiva | NA | (+) | DDLS | Grade 3 according to FNCLCC | (−) | 30 months; NED | (42) |
| Current case | / | 70 | M | Oral floor | 6 | (−) | (−) | Grade 2 according to FNCLCC | (+): 60 Gy | 5 years; second primary cancer of the chest wall (pleomorphic LS) |
NA, not applicable; M, male; F, female; LS, liposarcoma; DDLS, dedifferentiated liposarcoma; H&N, head and neck; FNA, fine needle aspiration; US, ultrasonography; CT, computed tomography; NED, no evidence of disease.
So far, no accurate protocol for DDLS (in all regions of the body, including H&N) management has been established (5,9). For both LS of the whole body and H&N, surgical resection is the standard treatment (7). However, the effects of pre- and postoperative therapy have been inaccurately reported so far (38). DDLS is a rare condition, and experimental DDLS models are lacking, leading to a delay in the development of suitable therapeutic strategies (46). Furthermore, DDLS may have site-specific characteristics. Henricks et al (17), studied 155 DDLS cases and concluded that retroperitoneal DDLS has a significantly worse prognosis than does DDLS at other sites. However, reports of H&N DDLS cases remain sparse because this is a relatively rare site for this tumor (7,37). Therefore, the accumulation of H&N DDLS cases with detailed clinical information and long-term follow-up is needed to establish a novel therapeutic protocol. We speculate that hidden H&N DDLS cases of recurrence, metastasis, or SPM exist.
Another important issue highlighted in this study is that biopsy (either incisional biopsy or fine needle aspiration) is not reliable for the diagnosis of DDLS. Table I shows that biopsy results have reported in 13 cases; however, DDLS was diagnosed in only 3 cases (23.1%). Even worse, 6 cases (46.2%) were misdiagnosed as benign lesions (5 cases) or ‘failures’ (1 case). DDLS generally involves heterogeneous lesions and occasionally presents as kinds of lesions (11,34,35,37). Petersson and Murugasu (37), reported a case of a unique DDLS lesion with a partly deceptively benign-appearing dedifferentiated component, leading to the misdiagnosis of DDLS on biopsy. Some studies have confirmed that WDLS and DDLS belong to the same group (14,47,48) because DDLS is well defined as a disease caused by progression from WDLS to a high- or low-grade lesion (34,38). Importantly, DDLS has a poorer 5-year disease-specific and overall survival rates compared with WDLS (7). Therefore, accurate pathological diagnosis with total resection is preferred to clearly distinguish DDLS from other LSs.
In conclusion, the current patient was the first documented case of oral floor DDLS. Furthermore, our case was the first reported case of SPM development after the treatment of H&N DDLS. After the first DDLS description in 1979 (49), the present study detected 50 cases of H&N DDLS. Our literature review indicated that preoperative biopsy is not reliable for the diagnosis of H&N DDLS, and accurate pathological diagnosis with total resection is preferred. Statistical analyses could not be performed, due to the small number of patients and sparse clinical information. Therefore, additional cases with long-term follow-up and well-described clinical information are needed to develop new protocols for H&N DDLS patients.
Acknowledgements
The authors would like to thank Professor Masaki Fujita, Dr Akira Nakao, Dr Hisako Kushima (Department of Respiratory Medicine, Fukuoka University Hospital, Fukuoka, Japan) and Dr Asahi Nagata (Departments of General Thoracic Breast and Pediatric Surgery, Fukuoka University Hospital, Fukuoka, Japan) who contributed to clinical patient diagnosis and treatment of pleomorphic LS of the chest wall.
Glossary
Abbreviations
- DDLS
dedifferentiated liposarcoma
- LS
liposarcoma
- ALT
atypical lipomatous tumor
- WDLS
well-differentiated liposarcoma
- H&N
head and neck
- CT
computed tomography
- MRI
magnetic resonance imaging
- RT
radiotherapy
- SPM
second primary malignancy
- NA
not applicable
- PET
positron emission tomography
Funding
No funding was received.
Availability of data and materials
The datasets used and/or analyzed during the current study are available from the corresponding author on reasonable request.
Authors' contributions
FN and TM acquired the data, performed the literature review and edited the manuscript. AA made substantial contributions to the concept and design of the study. TN, AM, NM and KN acquired the data and gave clinical advice. HM, NM and AA revised the manuscript. HM and NY evaluated the specimens and gave histopathological advice. TM was a major contributor in writing the manuscript.
Ethics approval and consent to participate
The report was submitted for ethical review to the Ethics Committee of the University of the Ryukyus (Okinawa, Japan), which waived the requirement for a review, since the study does not contain any protocols requiring ethical approval. The Ethics Committee approved the submission and publication of the manuscript.
Consent for publication
Written informed consent was obtained from the patient for the publication of this case report, including their clinical data and accompanying images.
Competing interests
The authors declare that they have no competing interests.
References
- 1.Crago AM, Socci ND, DeCarolis P, O'Connor R, Taylor BS, Qin LX, Antonescu CR, Singer S. Copy number losses define subgroups of dedifferentiated liposarcoma with poor prognosis and genomic instability. Clin Cancer Res. 2012;18:1334–1340. doi: 10.1158/1078-0432.CCR-11-2820. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 2.Virchow R. A case of malignant occurring in part in the form of fat neuroma tumors. Virchows Arch Pathol Anat. 1857;11:281–288. doi: 10.1007/BF01995372. (In Deutsch) [DOI] [Google Scholar]
- 3.Zagars GK, Goswitz MS, Pollack A. Liposarcoma: Outcome and prognostic factors following conservation surgery and radiation therapy. Int J Radiat Oncol Biol Phys. 1996;36:311–319. doi: 10.1016/S0360-3016(96)00265-9. [DOI] [PubMed] [Google Scholar]
- 4.Brennan MF, Antonescu CR, Alektiar KM, Maki RG. Liposarcoma. Management of soft tissue sarcoma. Springer Int Publ. 2016:PP105–124. [Google Scholar]
- 5.Dei Tos AP, Marino-Enriquez A, Pedeutour F, Rossi S. Dedifferentiated Liposarcoma. WHO Classification of Tumours of Soft Tissue and Bone. In: Fletcher C, Bridge J, Hogendoorn P, Mertens F, editors. 4th. Lyon, IARC Press; 2013. pp. PP37–38. [Google Scholar]
- 6.Darouichi M, Garibotto V, Christen B, Roud AF, Pazera A, Renggli JC, Willi JP, Ratib O, Qanadli S. FDG PET-CT in detection of diaphragmatic metastasis of dedifferentiated liposarcoma: A case report. Eur J Radiol Extra. 2011;77:e35–e38. doi: 10.1016/j.ejrex.2010.12.008. [DOI] [Google Scholar]
- 7.Gerry D, Fox NF, Spruill LS, Lentsch EJ. Liposarcoma of the head and neck: Analysis of 318 cases with comparison to non-head and neck sites. Head Neck. 2014;36:393–400. doi: 10.1002/hed.23311. [DOI] [PubMed] [Google Scholar]
- 8.Mariño-Enríquez A, Hornick JL, Dal Cin P, Cibas ES, Qian X. Dedifferentiated liposarcoma and pleomorphic liposarcoma: A comparative study of cytomorphology and MDM2/CDK4 expression on fine-needle aspiration. Cancer Cytopathol. 2014;122:128–137. doi: 10.1002/cncy.21362. [DOI] [PubMed] [Google Scholar]
- 9.Goldblum J, Weiss S, Folpe AL, editors. 6th. Elsevier Saunders; Philadelphia, PA: 2013. Liposarcoma. In: Enzinger and Weiss's soft tissue tumors; pp. 484–523. [Google Scholar]
- 10.Ariji Y, Gotoh M, Naitoh M, Izumi M, Shimozato K, Kurita K, Maeda H, Ariji E. Magnetic resonance imaging assessment of tumorous lesions in the floor of the mouth: Case reports and review of the literature. Oral Radiol. 2006;22:18. doi: 10.1007/s11282-006-0040-0. [DOI] [Google Scholar]
- 11.Powitzky R, Powitzky ES, Garcia R. Liposarcoma of the larynx. Ann Otol Rhinol Laryngol. 2007;116:418–424. doi: 10.1177/000348940711600605. [DOI] [PubMed] [Google Scholar]
- 12.Tirumani SH, Tirumani H, Jagannathan JP, Shinagare AB, Hornick JL, Ramaiya NH, Wagner AJ. Metastasis in dedifferentiated liposarcoma: Predictors and outcome in 148 patients. Eur J Surg Oncol. 2015;41:899–904. doi: 10.1016/j.ejso.2015.01.012. [DOI] [PubMed] [Google Scholar]
- 13.McElderry J, McKenney JK, Stack BC. High-grade liposarcoma metastatic to the gingival mucosa: Case report and literature review. Am J Otolaryngol. 2008;29:130–134. doi: 10.1016/j.amjoto.2007.04.001. [DOI] [PubMed] [Google Scholar]
- 14.Sioletic S, Dal Cin P, Fletcher CD, Hornick JL. Well-differentiated and dedifferentiated liposarcomas with prominent myxoid stroma: Analysis of 56 cases. Histopathology. 2013;62:287–293. doi: 10.1111/j.1365-2559.2012.04348.x. [DOI] [PubMed] [Google Scholar]
- 15.Tobey DN, Wheelis RF, Yarington CT., Jr Electron microscopy in the diagnosis of liposarcoma and fibrosarcoma of the larynx. Ann Otol Rhinol Laryngol. 1979;88:867–871. doi: 10.1177/000348947908800623. [DOI] [PubMed] [Google Scholar]
- 16.McCormick D, Mentzel T, Beham A, Fletcher CD. Dedifferentiated liposarcoma. Clinicopathologic analysis of 32 cases suggesting a better prognostic subgroup among pleomorphic sarcomas. Am J Surg Pathol. 1994;18:1213–1223. doi: 10.1097/00000478-199412000-00004. [DOI] [PubMed] [Google Scholar]
- 17.Henricks WH, Chu YC, Goldblum JR, Weiss SW. Dedifferentiated liposarcoma: A clinicopathological analysis of 155 cases with a proposal for an expanded definition of dedifferentiation. Am J Surg Pathol. 1997;21:271–281. doi: 10.1097/00000478-199703000-00002. [DOI] [PubMed] [Google Scholar]
- 18.Cai YC, McMenamin ME, Rose G, Sandy CJ, Cree IA, Fletcher CD. Primary liposarcoma of the orbit: A clinicopathologic study of seven cases. Ann Diagn Pathol. 2001;5:255–266. doi: 10.1053/adpa.2001.27911. [DOI] [PubMed] [Google Scholar]
- 19.Nascimento AF, McMenamin ME, Fletcher CD. Liposarcomas/atypical lipomatous tumors of the oral cavity: A clinicopathologic study of 23 cases. Ann Diagn Pathol. 2002;6:83–93. doi: 10.1053/adpa.2002.32375. [DOI] [PubMed] [Google Scholar]
- 20.Diamond C, Prince ME, Covert AA, Morris SF. Dedifferentiated liposarcoma of the cheek: Case report and literature review. J Otolaryngol. 2002;31:125–128. doi: 10.2310/7070.2002.19022. [DOI] [PubMed] [Google Scholar]
- 21.González-Lois C, Ibarrola C, Ballestín C, Martánez-Tello FJ. Dedifferentiated liposarcoma of the pyriform sinus: Report of a case and review of the literature. Int J Surg Pathol. 2002;10:75–79. doi: 10.1177/106689690201000113. [DOI] [PubMed] [Google Scholar]
- 22.Fanburg-Smith JC, Furlong MA, Childers EL. Liposarcoma of the oral and salivary gland region: A clinicopathologic study of 18 cases with emphasis on specific sites, morphologic subtypes, and clinical outcome. Mod Pathol. 2002;15:1020–1031. doi: 10.1097/01.MP.0000027625.79334.F5. [DOI] [PubMed] [Google Scholar]
- 23.de la Roza G, Baredes S, Aisner SC. Dedifferentiated liposarcoma of the cheek. Ann Diagn Pathol. 2004;8:352–357. doi: 10.1053/j.anndiagpath.2004.07.008. [DOI] [PubMed] [Google Scholar]
- 24.da Cunha IW, Kowalski LP, Soares FA. Dedifferentiated liposarcoma of the oral cavity with angiosarcomatous dedifferentiation. Virchows Arch. 2005;446:456–459. doi: 10.1007/s00428-005-1207-5. [DOI] [PubMed] [Google Scholar]
- 25.Angiero F, Sidoni A, Stefani M. Liposarcoma of the oral cavity-case reports of the pleomorphic and the dedifferentiated variants and a review of the literature. Anticancer Res. 2006;26:4857–4867. [PubMed] [Google Scholar]
- 26.Giordano G, Corcione L, Letizia G, Mercante G, Ferri T. Dedifferentiated liposarcoma of the pyriform sinus. Oral Oncol Extra. 2006;42:176–180. doi: 10.1016/j.ooe.2005.11.007. [DOI] [Google Scholar]
- 27.Saeed MU, Chang BY, Atherley C, Khandwala M, Merchant DW, Liddington M. A rare diagnosis of dedifferentiated liposarcoma of the orbit. Orbit. 2007;26:43–45. doi: 10.1080/01676830600666102. [DOI] [PubMed] [Google Scholar]
- 28.Rogers J, Patil Y, Strickland-Marmol L, Padhya T. Lipomatous tumors of the parapharyngeal space: Case series and literature review. Arch Otolaryngol Head Neck Surg. 2010;136:621–624. doi: 10.1001/archoto.2010.93. [DOI] [PubMed] [Google Scholar]
- 29.Gritli S, Khamassi K, Lachkhem A, Touati S, Chorfa A, Ben Makhlouf T, El May A, Gammoudi A. Head and neck liposarcomas: A 32 years experience. Auris Nasus Larynx. 2010;37:347–351. doi: 10.1016/j.anl.2009.08.003. [DOI] [PubMed] [Google Scholar]
- 30.Endo M, Oda Y, Harimaya K, Tamiya S, Yamamoto H, Kohashi K, Kurihara S, Setsu N, Matsuura S, Matono H, et al. Low-grade dedifferentiated liposarcoma of the neck: Magnetic resonance imaging and pathological correlation. J Orthop Sci. 2010;15:148–152. doi: 10.1007/s00776-009-1407-y. [DOI] [PubMed] [Google Scholar]
- 31.Makeieff M, Pelliccia P, Poizat F, Arnaud S, Rat F, Cupissol D, Guerrier B, Costes V. Laryngeal dedifferentiated liposarcoma. Eur Arch Otorhinolaryngol. 2010;267:991–994. doi: 10.1007/s00405-010-1234-y. [DOI] [PubMed] [Google Scholar]
- 32.Stomeo F, Bianchini C, Ciorba A, Padovani D, Pedriali M, Pelucchi S, Pastore A. Giant dedifferentiated liposarcoma of the right hemifacial area involving the oral cavity. Gerodontology. 2012;29:e1152–e1156. doi: 10.1111/j.1741-2358.2011.00455.x. [DOI] [PubMed] [Google Scholar]
- 33.Zhang JX, Ma JM, Wang NL. Dedifferentiated Orbital liposarcoma: A case report. Int J Ophthalmol. 2011;4:452–453. doi: 10.3980/j.issn.2222-3959.2011.04.26. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 34.Blumberg JM, Jedrych J, Costa J, Judson B. Cervical dedifferentiated liposarcoma with meningothelial-like whorling. Head Neck Pathol. 2012;6:476–480. doi: 10.1007/s12105-012-0354-7. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 35.Wang Y, Shi H. Dedifferentiated liposarcoma of the neck: CT findings. AJNR Am J Neuroradiol. 2012;33:E4–E6. doi: 10.3174/ajnr.A2431. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 36.Zreik R, Soyalp K, Ruiz S, Ward R, Dobin S, Chen X, Liu L, Rao A. Ultrasound-guided fine-needle aspiration of a posterior neck dedifferentiated liposarcoma with MDM2 fluorescence in situ hybridization performed on a Pap-stained smear. Diagn Cytopathol. 2015;43:320–324. doi: 10.1002/dc.23200. [DOI] [PubMed] [Google Scholar]
- 37.Petersson F, Murugasu E. Dedifferentiated liposarcoma of the deep (paralaryngeal) soft tissue: Lessons learnt from a case with a partly deceptively benign appearing dedifferentiated component. Head Neck Pathol. 2014;8:171–177. doi: 10.1007/s12105-013-0472-x. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 38.Jour G, Gullet A, Liu M, Hoch BL. Prognostic relevance of Fédération Nationale des centres de lutte contre le cancer grade and MDM2 amplification levels in dedifferentiated liposarcoma: A study of 50 cases. Mod Pathol. 2015;28:37–47. doi: 10.1038/modpathol.2014.88. [DOI] [PubMed] [Google Scholar]
- 39.Saâda-Bouzid E, Burel-Vandenbos F, Ranchère-Vince D, Birtwisle-Peyrottes I, Chetaille B, Bouvier C, Château MC, Peoc'h M, Battistella M, Bazin A, et al. Prognostic value of HMGA2, CDK4, and JUN amplification in well-differentiated and dedifferentiated liposarcomas. Mod Pathol. 2015;28:1404–1414. doi: 10.1038/modpathol.2015.96. [DOI] [PubMed] [Google Scholar]
- 40.Ishii T, Kohashi K, Iura K, Maekawa A, Bekki H, Yamada Y, Yamamoto H, Nabeshima K, Kawashima H, Iwamoto Y, Oda Y. Activation of the Akt-mTOR and MAPK pathways in dedifferentiated liposarcomas. Tumour Biol. 2016;37:4767–4776. doi: 10.1007/s13277-015-4232-2. [DOI] [PubMed] [Google Scholar]
- 41.Riva G, Sensini M, Corvino A, Vittone F, Garzaro M, Pecorari G. Rare giant pedunculated liposarcoma of the hypopharynx: Case report and review of literature. J Gastrointest Cancer. 2016;47:449–453. doi: 10.1007/s12029-015-9767-3. [DOI] [PubMed] [Google Scholar]
- 42.Enomoto A, Matsunaga K, Shimoide T, Mukai T, Uchihashi T, Hamada S. Dedifferentiated liposarcoma in the maxillary gingiva: A clinical report and review of the literature. J Oral Maxillofac Surg Med Pathol. 2017;29:542–545. doi: 10.1016/j.ajoms.2017.06.007. [DOI] [Google Scholar]
- 43.Lupo PJ, Brown AL, Hettmer S. Second malignancy risk among pediatric, adolescent, and young adult survivors of fusion-positive and fusion-negative sarcomas: Results from the SEER database, 1992 through 2012. Cancer. 2016 Aug 2; doi: 10.1002/cncr.30222. (Epub ahead of print) [DOI] [PubMed] [Google Scholar]
- 44.Özcan B, Çevener M, Yildiz A, Özdoğan M, Erdoğan O. Case report on the coincidence of retroperitoneal dedifferentiated giant liposarcoma and renal papillary cell carcinoma. Marmara Med J. 2017;30:50–53. doi: 10.5472/marumj.299404. [DOI] [Google Scholar]
- 45.Cheng J, Yu H, Wang L, Wang X, Shen G. Primary oral and maxillofacial liposarcoma: A clinicopathological and immunohistochemical study of eleven cases. Arch Med Sci. 2012;8:316–323. doi: 10.5114/aoms.2012.28560. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 46.Li H, Wozniak A, Sciot R, Cornillie J, Wellens J, Van Looy T, Vanleeuw U, Stas M, Hompes D, Debiec-Rychter M, Schöffski P. Pazopanib, a receptor tyrosine kinase inhibitor, suppresses tumor growth through angiogenesis in dedifferentiated liposarcoma xenograft models. Transl Oncol. 2014;7:665–671. doi: 10.1016/j.tranon.2014.09.007. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 47.Ray-Coquard I, Blay JY, Italiano A, Le Cesne A, Penel N, Zhi J, Heil F, Rueger R, Graves B, Ding M, et al. Effect of the MDM2 antagonist RG7112 on the P53 pathway in patients with MDM2-amplified, well-differentiated or dedifferentiated liposarcoma: An exploratory proof-of-mechanism study. Lancet Oncol. 2012;13:1133–1140. doi: 10.1016/S1470-2045(12)70474-6. [DOI] [PubMed] [Google Scholar]
- 48.Louis-Brennetot C, Coindre JM, Ferreira C, Pérot G, Terrier P, Aurias A. The CDKN2A/CDKN2B/CDK4/CCND1 pathway is pivotal in well-differentiated and dedifferentiated liposarcoma oncogenesis: An analysis of 104 tumors. Genes Chromosomes Cancer. 2011;50:896–907. doi: 10.1002/gcc.20909. [DOI] [PubMed] [Google Scholar]
- 49.Evans HL, Soule EH, Winkelmann RK. Atypical lipoma, atypical intramuscular lipoma, and well differentiated retroperitoneal liposarcoma: A reappraisal of 30 cases formerly classified as well differentiated liposarcoma. Cancer. 1979;43:574–584. doi: 10.1002/1097-0142(197902)43:2<574::AID-CNCR2820430226>3.0.CO;2-7. [DOI] [PubMed] [Google Scholar]
Associated Data
This section collects any data citations, data availability statements, or supplementary materials included in this article.
Data Availability Statement
The datasets used and/or analyzed during the current study are available from the corresponding author on reasonable request.