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ORIGINAL ARTICLE
Year : 2017  |  Volume : 13  |  Issue : 4  |  Page : 660-663

Treatment outcomes of malignant fibrous histiocytoma of the maxillary sinus


National Cancer Centre, Chinese Academy of Medical Sciences, Cancer Hospital of Peking Union Medical College, Beijing 100021, China

Date of Web Publication13-Sep-2017

Correspondence Address:
Dezhi Li
National Cancer Centre, Chinese Academy of Medical Sciences, Cancer Hospital of Peking Union Medical College, Beijing 100021
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jcrt.JCRT_29_17

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 > Abstract 

Background: Malignant fibrous histiocytoma (MFH) is an uncommon neoplasm of maxillary sinus. This study was designed to investigate the clinical courses and summarize the experience of the treatment outcomes.
Materials and Methods: This is a retrospective analysis which included patients with MFH of the maxillary sinus from January 1980 to December 2008 treated in our institute. Survival data were analyzed by means of the Kaplan–Meier method using univariate analysis and Cox regression model using multivariate analysis.
Results: The study included 31 patients. Of which, one patient received radical radiotherapy, and thirty patients received surgical resection. Of these, thirty patients received preoperative radiotherapy, and 11 patients received postoperative radiotherapy. Radical resection with clear surgical margins was obtained in twelve patients (80%) after preoperative radiotherapy, and in eight patients (53.3%) with postoperative radiotherapy (P = 0.221). The 3-year overall survival rate (OS) and recurrence-free survival rate (RFS) were 59.0% and 43.5%, respectively. Patients treated with a radical resection had significantly better survival than patients with a nonradical resection. Three-year OS was 79.8% and 28.1%, 3-year RFS was 61.9% and 18.5%, respectively (P = 0.001 and 0.029, respectively). Local recurrence rate (LRR) was lower in patients resected with clear surgical margins than patients with unclear or uncertain margins, and the 3 years LRR was 33.3% and 72.2%, respectively (P = 0.052).
Conclusions: Surgical resection is the mainstay of treatment for MFH of the maxillary sinus. A radical resection with clear margins is essential for excellent local control and long-term survival.

Keywords: Malignant fibrous histiocytoma, maxillary sinus, prognosism, radiotherapy, surgical resection


How to cite this article:
Huang H, Li D, Wang X, Wu Y, Liu S, Xu Z. Treatment outcomes of malignant fibrous histiocytoma of the maxillary sinus. J Can Res Ther 2017;13:660-3

How to cite this URL:
Huang H, Li D, Wang X, Wu Y, Liu S, Xu Z. Treatment outcomes of malignant fibrous histiocytoma of the maxillary sinus. J Can Res Ther [serial online] 2017 [cited 2018 Nov 16];13:660-3. Available from: http://www.cancerjournal.net/text.asp?2017/13/4/660/214469


 > Introduction Top


In 1964, O'Brien and Stout first reported a kind of adult soft-tissue sarcoma with complicated structures which is now called as malignant fibrous histiocytoma (MFH).[1] MFH mostly occurs in the extremities and retroperitoneum with a very low rate (<10%) in the head and neck region [2] and rarely occurs in the maxillary sinus.[3] The mainstay of MFH treatment in the head and neck region is surgical resection with wide margins,[4],[5] and neck dissection is unnecessary in the vast majority of cases due to its uncommon regional metastases.[6],[7] Adjuvant radiotherapy is recommended as the tumor is unresectable or clear surgical margins are not achieved.[5],[6],[7] However, analysis about MFH of the maxillary sinus is rare, and till date, there were no randomized clinical trials due to its low incidence rate. Currently, the most appropriate treatment strategy is still unclear.[8],[9],[10] Hence, this study retrospectively analyzed a series of MFH of the maxillary sinus treated in our institution to indicate an appropriate treatment strategy.


 > Materials and Methods Top


Design

This study was a retrospective clinical analysis of MFH of the maxillary sinus. All information and data were extracted from the electronic medical records of the National Cancer Center/Cancer Hospital, Chinese Academy of Medical Sciences. Data from January 1980 to December 2008 were collected, including patient demographics, T stage, N status, treatment methods, and outcomes. Tumor was restaged according to the American Joint Committee on Cancer TNM staging Classification for the Paranasal Sinuses (7th ed., 2010).[11] This study was approved by the Institutional Ethics Review Board.

Case selection

Thirty-one cases were included in this study. Eligibility criteria included primary MFH of maxillary sinuses, which was pathologically diagnosed by endoscopic biopsy or maxillary sinusotomy, no distant metastases identified by chest X-ray and abdominal B-mode ultrasound examination, and completed medical records. Exclusion criteria included any previous treatment including surgical resection, chemotherapy or radiotherapy, any synchronous systemic disease or other conditions contraindicated for general anesthesia operation, and incomplete follow-up records.

Treatment

The treatment strategy was decided by a multidisciplinary team who are experienced in surgical oncology, radiation oncology, radiology, and tumor pathology. Most patients received combined treatment modalities including radiotherapy and surgery. No patient received chemotherapy as initial or adjuvant therapy. As lymph node metastasis is rare for MFH, only patients with metastatic or suspicious enlarged lymph node underwent neck dissection.

Sixteen patients received preoperative radiotherapy. They were treated using the standard three-field technique or a three-dimensional conformal by computerized tomography-planning. The average cumulative dose was 48.25 Gy (32 Gy–70 Gy). One patient received radical radiation with a cumulative dose of 70 Gy (2 Gy per fraction) and refused salvage surgery without tumor remission. Fifteen patients received reevaluation of the tumor and ceased radiation when cumulative dose was up to 40–60 Gy. After a rest of three to 4 weeks, all the 15 patients received surgical resection. Three patients underwent extended resection of the lateral nasal wall including the anterior skull base and the orbit as being invaded. Six patients underwent resection of the entire maxilla; six patients underwent extended resection of the maxilla, including muscles around the sinus or the orbit, as being invaded. All the 15 patients used split skin graft to cover the wound. Only one patient underwent unilateral neck dissection (Level I to V).

Fifteen patients received surgical resection first. Two patients underwent subtotal resection of the maxilla. Thirteen patients underwent extended resection of maxilla including the orbit or skull base as being invaded. No patient underwent neck dissection. All patients used split skin graft to cover the wound. Eleven patients received postoperative radiotherapy after a rest of 4 to 6 weeks as planned, while other four refused.

Follow-up and statistical analysis

The patients were followed up every 3 months during the 1st year, every 6 months in the 2nd year and every year thereafter. The survival data were obtained from the electronic medical records or by mails/telephone calls. The primary end-points were the overall survival (OS) which is defined as the time from starting the treatment till death and recurrence-free survival rate (RFS) which is defined as the time from starting the treatment till tumor recurrence or death. The secondary end-points were local recurrence rate (LRR), which was diagnosed by the histopathological examination after biopsy or surgical resection. The statistical analysis was conducted using SPSS 18.0 (SPSS Inc., Chicago, IL, USA). Survival data were analyzed by means of the Kaplan–Meier method and survival curves were compared using the log-rank test in univariate analysis. All statistical tests were two-sided and P < 0.05 was considered as statistically significant.


 > Results Top


Clinicopathological characteristics

Twenty patients were male and eleven were female with a mean age of 43.48 ± 10.91 years. Three cases were staged as T2, 11 cases were staged as T3, and 17 as T4. Only one case was diagnosed with ipsilateral lymph node metastasis by pathological examination after neck dissection and was staged as N2b [Table 1]. We tried to judge if the tumor was resected radically with clear surgical margins according to the intraoperative findings and intraoperative frozen pathological examination. Patients were divided into two groups accordingly. If the tumor was resected with clear margins on the surrounding normal tissues, and the suspicious tissue was identified with no tumor by frozen pathological examination, the patient was grouped as radical resection with clear margins (completed resection group). If the tumor was resected just around the tumor capsule or the margin was confirmed with tumor residual by frozen pathological examination, or margins containing residual tumor because of some key structures being invaded such as skull base, carotid artery, or sinus cavernous and with no possibility for reresection, the patient was grouped as nonradical resection with unclear or uncertain margins (uncompleted resection group). Accordingly, completed resection were obtained in 12 patients (80%) after preoperative radiation, and in eight patients (53.3%) without preoperative radiation (log-rank value was 2.400, P = 0.221) [Table 2].
Table 1: Clinicopathological characteristics

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Table 2: Surgical margin status according to treatment strategy

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Outcomes

The median time of death for 11 patients who died before the follow-up deadline was 16 months (4–114 months). All the patients died within 36 months except one at 114 months. The median survival time of the remaining twenty patients was 55 months (7–273 months). Local recurrence was occurred in 12 patients, seven of which were located in the orbital region (of which 1 invaded skull base, 1 invaded infratemporal fossa, 1 invaded ethmoid sinus), four cases in the hard palate region, and one case in the pterygopalatine fossa. Regional lymph node recurrence occurred in two patients and distant recurrence in three patients. The 3-year cumulative LRR was 46.7%, the 3-year cumulative cervical lymph node recurrence rate was 7.8%, and the 3-year cumulative distant metastasis rate was 14.2%. The 3-year OS rate was 59.0%, and the 3-year RFS rate was 43.5% [Figure 1]. Eleven patients died, 1 of local failure, 3 of local recurrence, 1 of cervical lymph node recurrence, 3 of distant recurrence, and 3 of unknown causes. Local recurrence occurred in 3 of the 4 patients who were treated with surgical resection alone. After a reresection, all of the three patients survived for more than 5 years.
Figure 1: Kaplan-Meier curves for overall and recurrence-free survival. (a) Overall survival. The 3-year overall survival was 59.0%. (b) Recurrence-free survival. The 3-year recurrence-free survival was 43.5%

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Survival analysis

Univariate analysis showed that there were no significant differences in the survival rate which are stratified by factors such as age and sex. Survival of the patients with completed resection was significantly better than patients with uncompleted resection. The 3-year OS was 79.8% and 28.1%, the 3-year RFS was 61.9% and 18.5%, respectively. Local recurrence was lower in patients in completed resection group than patients in uncompleted resection group though there was no significant difference. Three-year LRR was 33.3% and 72.2%, respectively [Table 3]. The vast majority of patients in this series were in stage T3/T4 and only one patient with lymph node metastasis, we did not analysis the impact of T stage and N status on prognosis.
Table 3: Survival analysis in univariate analysis with log-rank test

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 > Discussion Top


MFH occurs rarely in the maxillary sinus, and lack specific symptoms and signs in its early stage. Most tumors are locally advanced when they are presented to the clinic. Locally advanced disease is a great challenge for radical resection. Surgery is the mainstay of treatment for MFH of the head and neck,[4],[5] and aggressive surgical resection with clear margins is vital for tumor elimination and survival.[12],[13] For a radical resection, the minimum width of the margin is recommended as 3 cm beyond the edge of the tumor.[14] However, the maxillary sinus is so close to its surrounding structures such as the orbit and the skull base that it is impossible to get a wide margin beyond 3 cm. In our series, the surgical margin was carried out on the surrounding normal fascia plane if possible. For the majority of our cases, a wide range of surgical resection was achieved, including the orbital cavity, infratemporal fossa, or the skull base if invaded. Twenty of the thirty cases had clear surgical margins (66.7%), and ten cases had unclear or uncertain surgical margins (33.3%). Local recurrence of patients, when treated with a radical resection (with clear surgical margins), was much lower than patients with a nonradical resection (with unclear or uncertain surgical margins) (33.3% vs. 72.2%) though showed no significant differences. Patients treated with a radical resection had significantly improved OS and RFS than with a nonradical resection. Multivariable analysis showed that radical resection with clear surgical margins was an independent factor. All of the three patients with surgical resection alone survived for more than 5 years after salvage resection for local recurrence. Hence, MFH may be cured as long as the radical resection with clear surgical margins was obtained. Radical resection acts as the foundation for the treatment of MFH of the maxillary sinus.

The role of radiotherapy for MFH of the maxillary sinus is still in controversy. Some authors believed that MFH of the maxillary sinus is not sensitive to radiotherapy, radiation cannot effectively control the tumor progression, and radical resection of the maxilla should be performed as early as possible.[15] Others concluded that radiotherapy can effectively reduce the tumor volume,[16] and it also play an important role when the surgery is unfeasible or in an adjunctive setting, either pre- or post-operative.[5] Our data show that patients obtained radical resection with clear surgical margins after preoperative radiation although the difference did not reach significance. Radiation tends to make the surgical resection more radical. We found that local recurrence was much lower in patients treated with a radical resection and radical resection was an independent factor for RFS. Thus, we think that preoperative radiotherapy was effective for patients with MFH in terms of local control.

Owing to the low incidence rate, the sample size of the MFH of the maxillary sinus is small. Treatment ideas, surgical and radiotherapy technology improves during the long period of study (1980–2008), and this study was retrospective, bias may exist. Because of the small sample size, we failed to do multivariate analysis to determine the independent factor for recurrence and survival.

However, we confirmed that surgical resection was the mainstay of treatment, and a radical resection with clear surgical margins was essential for excellent local control and long-term survival. We also suggested reresection whenever possible in cases of local recurrence. More clinical research was needed to further evaluate the treatment methods, particularly in the role of radiotherapy.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
 > References Top

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O'brien JE, Stout AP. Malignant fibrous xanthomas. Cancer 1964;17:1445-55.  Back to cited text no. 1
    
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Salcedo-Hernández RA, Lino-Silva LS, Luna-Ortiz K. Maxillary sinus sarcomas: Epidemiological and clinicopathological experience of 25 years in a national reference cancer center. Indian J Otolaryngol Head Neck Surg 2014;66:359-64.  Back to cited text no. 4
    
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Peng KA, Grogan T, Wang MB. Head and neck sarcomas: Analysis of the SEER database. Otolaryngol Head Neck Surg 2014;151:627-33.  Back to cited text no. 5
    
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Blitzer A, Lawson W, Zak FG, Biller HF, Som ML. Clinical-pathological determinants in prognosis of fibrous histiocytomas of head and neck. Laryngoscope 1981;91:2053-70.  Back to cited text no. 6
    
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Barnes L, Kanbour A. Malignant fibrous histiocytoma of the head and neck. A report of 12 cases. Arch Otolaryngol Head Neck Surg 1988;114:1149-56.  Back to cited text no. 7
    
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Vuity D, Bogdan S, Csurgay K, Sapi Z, Nemeth Z. Malignant fibrous histiocytoma/undifferentiated high-grade pleomorphic sarcoma of the maxillary sinus: Report of a case and review of the literature. Pathol Oncol Res 2013;19:605-9.  Back to cited text no. 8
    
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Yanagi Y, Murakami J, Hisatomi M, Katase N, Nagatsuka H, Asaumi J. A case of malignant fibrous histiocytoma of the maxillary sinus. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2010;109:e99-104.  Back to cited text no. 9
    
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Sadri D, Yazdi I. Postradiation malignant fibrous histiocytoma of the maxillary sinus. Arch Iran Med 2007;10:393-6.  Back to cited text no. 10
    
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Edge S, Byrd D, Compton C, Fritz A. AJCC Cancer Staging Handbook: From the AJCC Cancer Staging Manual. 7th ed. New York: Springer; 2009.  Back to cited text no. 11
    
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Hardison SA, Davis PL rd, Browne JD. Malignant fibrous histiocytoma of the head and neck: A case series. Am J Otolaryngol 2013;34:10-5.  Back to cited text no. 12
    
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Clark DW, Moore BA, Patel SR, Guadagnolo BA, Roberts DB, Sturgis EM. Malignant fibrous histiocytoma of the head and neck region. Head Neck 2011;33:303-8.  Back to cited text no. 13
    
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Wu X, Qi Y, Tang P. A comparison of malignant fibrous histiocytoma of head, neck and extremities. Chin Med J (Engl) 2000;113:532-5.  Back to cited text no. 14
    
15.
Sawyer R, Webb DL, Wittich DJ. Head and neck malignant fibrous histiocytomas. Ear Nose Throat J 1993;72:299-302.  Back to cited text no. 15
    
16.
Sasaki R, Sakai S, Itoh M, Murata M, Honda M, Aozasa K. Malignant fibrous histiocytoma in the maxillary sinus. Xanthoma-like change of the tumor after radiotherapy. Laryngoscope 1983;93:202-4.  Back to cited text no. 16
    


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    Tables

  [Table 1], [Table 2], [Table 3]



 

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