Solitary Fibrous Tumor in Pelvis Extended to Transverse Mesocolon and Peritoneum

Mostafa Hoseini¹, Alireza Negahi², Farnaz Vosough³, Borna Farazmand⁴, Fereshteh Vosough⁵, Behnood Farazmand⁶, Masoud Haghighi Kian⁷*

¹Professor of Surgery, Head and Neck Surgery, Iran University of Medical Sciences, Tehran, Iran.

²Assistant Professor, Department of Surgery, Iran University of Medical Sciences, Tehran, Iran.

³Medical Doctor, Tehran University of Medical Sciences, Tehran, Iran.

⁴Department of Radiation Oncology, Tehran University of Medical Sciences, Tehran, Iran.

⁵Medical Doctor, Iran University of Medical Sciences, Tehran, Iran.

⁶Resident of Surgery, Iran University of Medical Sciences, Tehran, Iran.

⁷Assistant Professor, Department of Surgery, Iran University of Medical Sciences, Tehran Iran.

*Corresponding Author E-mail: mhaghighikian@iran.ir

ABSTRACT:

Solitary fibrous tumor (SFT) is a rare, spindle-cell neoplasm, which is classified as mesenchymal neoplasm emanating from ubiquitous dendritic interstitial cells. SFTs are benign tumor with a low rate of malignancy developed in intrathoracic areas and its growth is rarely observed in extrapleural locations. Based on to the rarity of malignant SFT, there is no certain treatment to the inoperable malignant. This case study is a report of a man with SFT of the pelvis, transverse mesocolon, and peritoneum. So far, more than 58 patients with SFT of the pelvic have been reported in the literature, out of which 26% of them were malignant either clinically or histologically. The female/male ratio was equal within the age range of 27-83 years. The size of reported tumors were ranged 1-33 cm. Malignancy were reported in 26% of cases.

KEYWORDS: Mesenchymal neoplasm, Solitary fibrous tumor, Pelvis, Extrapleural locations.

INTRODUCTION:

Solitary fibrous tumor (SFT) is a rare, non-functional, spindle-cell neoplasm, categorized as mesenchymal neoplasm originating from ubiquitous dendritic interstitial cells^1,2,3. The incidence of SFT is estimated 1.4 per million population⁴. Based on a study conducted by Okike et al., the estimation of SFT incidence is 2.8 per 100000⁵.

Mainly, SFTs is not usually observed in extrapleural sites of body⁶. Accordingly, only about 30% of SFTs occur in extrathoracic locations⁷. Commonly, SFTs is known as benign tumor and the rate of its malignancy is 12–22%².

Considering the rarity of malignant SFT, there is no certain treatment to the inoperable malignant. The recurrence of certain SFTs is different with regard to its malignancy, meaning that it may occur more than 10 years after the resection of the original tumor¹. The rate of mortality in patients with malignant SFTs is estimated 10–20%⁸.

Extrathoracic SFTs are larger than those in other sites and show more aggressive characteristics¹. The diagnosis of SFTs is performed by computed tomography (CT) and magnetic resonance imaging (MRI). Large amounts of operational data concerning several years’ operation are now becoming available, mainly in middle-large sized organizations⁹. Now, with the recent explosive growth of the Internet, all these computers are becoming interconnected in a global communications network¹⁰. But in the complex modern world, we are surrounded by ethical issues in all facets of our lives. Consequently, there has been a heightened interest in the field of ethics, in an attempt to gain a better understanding of how these issues influence us¹¹. However, it is difficult to diagnose SFT due to the difficulties in the determination of the precise location of the pelvic SFT¹. So far, more than 50 patients with SFT in the pelvic have been reported, 30% of whom were malignant either clinically or histologically¹. In the present study, we reported a case of a male patient with SFT in the pelvis expanded to both transverse mesocolon and peritoneum. Soni and Burje (2011) studied Tumor Detection Algorithm Using Boundary Based Approach. The method makes use of an adaptive color metric from the red, green, and blue (RGB) planes that contain information to discriminate the tumor from the background. Using this suitable coordinate transformation, the image is segmented. The tumor portion is then extracted from the segmented image and borders are drawn¹². Shekar, Krithega Devi and Sathish Kumar (2013) studied Analysis of Lymphoma Neoplasm and their Phenotypes. Histopathologic diagnosis was B-cell lymphoma in 4 (50%), T-cell Lymphoma in 3 (37.5%). 2 patients (20%) were diagnosed with Hodgkin’s Lymphoma. Diseases sites were para-aortic nodes (52%), cervical (8%), peripheral nodes (33%), others 6%¹³. Jain (2014) studied Vibration Analysis of Fibrous Composite Plates with Central Circular Hole. The analytical treatment of such type of problem is very difficult; hence, finite element method is adopted for whole analysis. The finite element formulation is carried out in ANSYS package. The work is most appreciated in aerospace and marine industry where the study will provide the guidelines technique to designer for laminate configuration as per their requirements¹⁴. Devand and Sarma (2014) studied Solitary Wave Solution of Higher order Kadomstev-Petviashvili Equation for Complex Plasma. The analytical solution of K-P equation and other higher order nonlinear wave equation such as mK-P equation are derived by well-known tanh-method and dust acoustic solitary waves and shock waves propagation for different situation are reported, which could be relevant in case of different space and astrophysical plasmas including Saturn’s F-ring and also in laboratory observation for both positive and negative dust charge fluctuation¹⁵. Khande and Udapi(2014) studied a comparative study to assess the level of depression among the elderly residing in old age home and with the family. The major findings indicated that the level of Depression among the elderly residing in old age home were severe to moderate and elderly residing with the family had mild to moderate level of depression¹⁶.

CASE REPORT:

The case in this study was a 60-year-old man referred to the clinic for the abdominal pain after motor vehicle collisions (MVC). He had the history of mild abdominal pain and distention for a year. He was a taxi driver and bump into another car.

Vital signs of patient were unstable and the focused assessment with sonography in trauma was positive at the time of his arrival at the emergency department. Therefore, the patient was transferred to an operation room, where two intrapelvic masses were observed in the abdominal exploration. The masses were bleeding; therefore, they were grossly removed to control bleeding. Moreover, two other tumors, one located in the transverse mesocolon region and the other one in the peritoneum were diagnosed. These tumors were not resected because the patient was unstable and no bleeding was observed.

The resected mass were transferred to the laboratory for the pathological examination. According to the obtained results of T2-weighted magnetic resonance imaging (MRI), several masses with a funicular or linear pattern and an extensive low-signal intensity area were observed. Moreover, high-signal-intensity areas suggesting the foci of hemorrhage with or without necrosis was detected in T1-weighted MRI.

Based on the CT scan images, there were remaining masses (….cm in longest diameter) in the cavity of the pelvis, a mass in the peritoneum, and multiple masses in the colon. These masses were well- defined and smooth, which ranged in size from few millimeters to Centimeters. Postop was uneventful. The patient became candidate for the the resection of two remained tumors.

Uniform spindle cells with dense keloid-like ropy collagen were observed in the Biopsy of the mass lesion. Thick collagen bundle was observed in biopsy without any changes in myxomatous. The equally-sized tumor cells shaped round to oval nuclei in the biopsy. A number of factors, including mild enhances of chromatin, nuclear shape asymmetry, and thickness of nuclear membrane in some areas affected the limitation of the cellular atypia of each tumor cell. A variety of cellularity in different areas was observed in biopsy. There were less conspicuous cytological atypia and lower percentage of MIB-1-positive tumor cells in the regions of lower cellularity. However, slightly more nuclear atypia was observed in the higher cellularity areas. Aforementioned histological criteria were in favor of benign SFT.

According to the immunohistochemical findings, CD34, CD99, bcl-2, and vimentin of the tumors were positive; while smooth muscle actin (a-SMA), S-100, estrogen receptor (ER), progesterone receptor (PgR), CD10, desmin, caldesmon, c-kit, pan-cytokeratin, myogenin, and myoD1 were negative respectively. Histological complete remission (CR) was confirmed by biopsy, which was in line with microscopical and immunohistochemical findings.

SEARCHING PROCESS:

In this literature review, articles reported the cases with the pelvis SFT were evaluated through searching databases, such as PubMed, Science Direct, and Google Scholar. The search process was accomplished using the following keywords in combination with Solitary Fibrous Tumor: “Pelvis”, “Colon”, or “Peritoneum”. We searched through all case reports or case series published in English up to 2018. The studies with insufficient data were excluded from the previously-made table, although we reported all articles.

In general, we found 58 articles reporting FST in the pelvis, out of which 10 cases with the malignant pelvic SFT were reported. Table 1 shows the characteristics of the reported cases with SFT pelvic. Except cases reported in Table 1, four, four, three, and four cases were also reported by Tian et al.¹⁷, Baldi et al.¹⁸, Park et al.¹⁹, and Hasegawa et al.²⁰, respectively. Moreover, some cases with pelvic FST were reported by Goodlad et al.²¹, Trastour et al.²², Guo et al.²³, and Tanaka et al.²⁴, which due to no complete data were not inserted in Table 1. Based on the obtained results, abdominal pain was the most frequent complaints of cases referring to the hospitals before the diagnosis of SFT. The female/male ratio was 1:1 and the age range was 27-83 years. The size of reported tumors were within the range of 1-33 cm. Malignancy was reported in 26% of cases. The duration of follow-up was 4-126 months.

Table 1: the characteristic of reported cases with pelvic SFT

Authors References	Age	Sex	Site	Tumor size (cm)	Chief Complaints	Imaging studies	Malignancy	Treatment	Follow up (months)
Young et al. ²⁵	59	N/A	Adnexal mass on pelvic examination	9	N/A	CT	N/A	Resection	No
Fukunaga et al.²⁶	33	Female	Right renal peripelvis	3.0×2.5×2.5	Abdominal pain	CT, IVU, angiography	No	Right nephrectomy	90
Fukunaga et al.²⁶	36	Female	Left renal peripelvis	2.0×1.5× 1.5	Abdominal pain	CT, MRI	No	· Left nephrectomy	12
Yoshida et al.²⁷	62	Male	Pelvis cavity	11× 9×7	Abdominoperineal and excision of the rectum	CT, MRI	No	Laparotomy+ perineal approach	10
Lasota et al.²⁸	52	Female	Pelvic	5×4	Abdominal discomfort	CT	No	resection	36
Yazaki et al.²⁹	70	Male	Right renal pelvis	6.0×4.5×4.0	Abdominal mass	CT, IVU, MRI, angiography	No	· Radical nephrectomy	60
Wiessner et al.³⁰	50	Male	Pelvic cavity	N/A	During routine screening	CT	No	Resection	24
Madhuvrata et al.³¹	55	Female	Pelvis to the left of midline	10×9×6	Lower abdominal pain, frequency of Micturition, urgency and urge incontinence	CT	No	Laparotomy	No
Ishikawa et al.³²	64	Male	Pelvic mass adjacent to the prostate	12.5×9.5×8.3	Complaining of difficult voiding and constipation	CT	No	Resection	No
Nagase et al.³³	60	Male	Left side of the pelvic cavity	14×9	Hypoglycemic crisis	CT, MRI	Yes	Laparotomy Resection	No
Vossough et al.³⁴	61	Male	Pelvic peritoneum	9×6×11	Increased urinary frequency, left flank pain radiating to the scrotum, and deep pelvic pain	CT, MRI	Yes	Laparotomy	No
Yi et al.³⁵	65	Male	Pelvic	17×13	Urinary retention and intermittent diarrhea alternating with constipation	CT	No	RT, Chemotherapy Resection	12
Chu et al.³⁶	78	Female	Pelvic mass attached to the uterus	N/A	Low abdominal pain with a palpable abdominal mass Lesion	CT	No	Exploratory laparotomy	12
Kawamura et al.³⁷	74	Female	Pelvic cavity	3.8	Constipation	CT, MRI	Yes	RT Chemo-embolization	12
Santo et al.³⁸	49	Male	Mass posterolateral to the bladder	10	N/A	MRI	No	Resection	6
Wat et al.³⁹	63	Female	Pelvis Rectum and sigmoid Colon to the right	14×11×14 cm	Abdominal pain of two months	CT	No	Resection and RT Laparotomy	No
Joe et al.⁴⁰	34	Male	Prostate	12×7.6×9	Abdominal discomfort, diarrhea, and urinary frequency	CT, MRI,	Yes	Resection	No
Xue et al.⁴¹	47	female	Pelvis	4×3×2.5	Massive bleeding	CT, MRI	No	Laparotomy	12
Wagner et al.⁴²	28	Male	Small pelvis	18×15×11	Left flank pain	CT, MRI, PET	Yes	Resection and RT	No
Katsuno et al.⁴³	56	Female	Right posterior aspect of the rectum	9×7.5×5	N/A	CT	No	Trans-sacral approach	No
Hata et al.⁴⁴	83	Male	Pelvis	10	Hypoglycemic attacks	CT	Yes	Resection	No
Kurisaki-Arakawa et al.⁴⁵	70	Female	Pelvis	17×17×13	Exertional dyspnea	CT, MRI	No	Resection	Died after 4 months
Hosaka et al.⁴⁶	62	Female	Right pelvic cavity and had expanded to the right buttock	14	Hypoglycemic Attack and noticed right sciatic pain	CT, MRI	Yes	Resection	No
Schutt et al.⁴⁷	64	female	Pelvic	14.5×9×9	Urinary incontinence	CT, MRI	Yes	Laparotomy with resection of the retroperitoneal mass	22
Wang et al.⁴⁸	56	male	Pelvic	9.5	Constipation	CT, MRI	Yes	Resection	No
	75	Female	Pelvic	5.5	Abdominal pain	CT, MRI	No	Resection	76
	68	male	Pelvic	2.5	Abdominal pain	CT, MRI	No	Resection + adjuvant chemotherapy	126
Kim et al.⁴⁹	52	Female	Pelvic cavity	12×9	N/A	CT	No	Surgical resection	36
Tsashimi et al.⁵⁰	64	Male	Pelvic cavity	10×8×7	Pollakiuria	CT	No	Resection
Yokoyama et al.⁵¹	63	Male	Pelvic cavity	30×25×19	Frequent urination	CT, MRI	No	Resection	24
Soda et al.⁵²	27	Male	Pelvic	16	Severe urinary retention and constipation	CT	No	Resection	12
Dozier et al.⁵³	41	Male	Upper pelvis bladder serosa	28×21×18	Weight loss and progressive abdominal bloating	CT	Yes	Resection	8
Lee et al.⁵⁴	71	Female	Peripelvis	N/A	Abdominal pain	CT	No	Resection	No
Zerón-Medina et al.⁵⁵	54	Male	Pelvic peritoneum	N/A	Acute urinary retention and weight loss	CT	No	Resection	No
Shoji et al.⁵⁶	41	Male	Pelvis	12.5×9.5×12.9	Urinary retention	US, CT	Yes	Resection	12
Boe et al.⁵⁷	52	male	Pelvic cavity	12×9×20	N/A	CT, MRI	No	Complete surgical resection	No
Jarlot et al.⁵⁸	71	Male	Pelvic cavity	N/A	Acute urinary retention	CT, MRI	No	Resection	15
Jarlot et al.⁵⁸	51	Male	Behind the bladder and displacing the rectum laterally	14	Widespread dorsolumbar pain	CT, MRI	No	Resection	No
Ando et al.⁷	71	Male	Pelvic cavity	10×7×10	Intermittent urine stream and post micturition dribbling	CT, MRI	Yes	Surgical resection	24
Pata et al.⁵⁹	76	Male	Pelvic cavity	17×10×9	Low abdominal pain and acute urinary retention and constipation	CT, MRI	No	Surgical resection	60
Gao, et al.⁶⁰	48	male	Pelvic cavity	8.0×5.3	Abdominal pain	CT, MRI	Yes	Surgical resection	48
Ishihara et al.⁶	72	Male	Pelvic	7	Intermittent loss of consciousness and hypoglycemic attacks Recurrent disease	CT, MRI	Yes	Surgical resection	9
Sueblinvong et al.⁶¹	54	Female	Left pelvic peritoneum	10×16×9.5	Recurrent disease, Pelvic discomfort thought to be caused by uterine leiomyomata	CT, MRI	Yes	Surgical resection	12
	73	female	Pelvic	N/A	Urinary retention	CT, MRI	No	Laparotomy	21
	48	Female	Pelvic	N/A	N/A	CT, MRI	No	Exploratory laparotomy	7

*Positron Emission Tomography (PET); No Data (N/A); Ultrasound Sonography (US)

RT: radiation therapy

CT: computed tomography

MRI: magnetic resonance imaging

US: ultrasound

PET: positron emission tomography

IVU: intravenous urogram

DISCUSSION:

Here, we found 58 cases of extrapleural SFT in the pelvic cavity in 9 studies (no detailed information was available for 19 cases). This is the first time that SFT has been diagnosed as a pleural lesion despite the fact that studies on the extrapleural SFT have been conducted since the 1990s. Many studies have reported SFT in the pleura, which showed that SFT is typically a benign lesion and occurs equally in both genders and in all age groups. SFTs have been measured to be 1–33cm in size. These characteristics are the same as those of extrapleural ones.

Extrapleural SFT is equally distributed between the sexes and shares the same statistics as pelvic SFT. The age range of people with pelvic SFT is 22-83 years (mean: 50 years). There is a difference between the mean age of males and females with pelvic SFT (females: 40 years, males: 58 years)⁸. The size of the extrapleural SFT lesions ranged was within the range of 1.8-33cm. The mean size of pelvic SFT was found to be 5–22cm (mean: 13cm).

Among the reported cases of pelvic SFT, 16 out of 58 cases were designated as malignant and benign, respectively (four cases were deemed to have an uncertain status). Based on this result, it is possible that the rate of malignancy is higher in pelvis SFT as compares to other type of SFT. Accordingly, this higher rate of malignancy in pelvis SFT may be related to its size, particularly since FST is larger when located in the abdomen or pelvic cavity. It has also been shown that there is a correlation between SFT bigger than 10 cm and the probability of metastasis⁶². Therefore, the size of FST should be considered as the main indicators of malignancy^3,8,63. Based on a study conducted by Kawamura et al., there is a very high possibility of SFT growing rapidly in the abdominal or pelvic cavity¹. Further, the researcher reported an elderly woman with inoperable malignant SFT, which is the first report explaining the use of therapeutic response for inoperable malignant SFT¹.

Histologically, malignant SFT relapsed or metastasized in 68–80% of cases^64,65; therefore, even in the cases with small and benign tumors, careful follow up is necessary. Excisional surgery and radiation therapy are two traditional treatments of soft-tissue sarcomas. Resection is the primary choice of therapy for malignant SFT. Although, no standard forms of therapy have been established for malignant SFT due to its rarity, according to its localized features, complete resection is commonly performed in cases with malignancy⁶³. On the other hand, radiation therapy can be used to shrink the tumor, as was proven effective in a study by Kawamura et al. In their study, Kawamura et al. reported a case, in which there was a remarkable response to radiation therapy, although the mass had not regressed¹. Based on one study, the rate of mortality associated with adjuvant radiation therapy was not higher than surgery⁶⁶.

When the surgery on a patient is impossible or rejected, radiation therapy is used exclusively⁶⁷. The finding regarding the effectiveness of radiation therapy for high or low-grade tumors is controversial as in general, few soft-tissue sarcoma are sensitive to chemotherapeutic agents⁶⁸. Accordingly, the question remains unanswered as to whether primary systemic or intra-arterial chemotherapy, in combination with radiation therapy, can lead to tumor cells being killed.

Histological complete remission with dense hyalinized fibrosis was observed in the biopsy from FST after irradiation. Although the collagenous fibrotic component/matrix of SFT may remain unchanged, owing to radiation therapy, there could still be changes in neoplastic cells. Chemotherapeutic agents can lead to some biological modification occurring in the tumor cells, although the progress tumor continued with no changes highlighted in MRI scan¹.

It is difficult to make a clear diagnosis of SFT. Moreover, it can be diagnosed as a different type of malignancy or tumors, such as some sarcomas, malignant peripheral nerve sheath tumors (MPNST), melanoma, mesothelioma, poorly differentiated sex-cord stromal cell tumors, and sarcomatoid carcinoma. The primary differential diagnoses of SFT are leiomyosarcoma, synovial sarcoma and fibrosarcoma. When the basic morphology of SFT is not typical, the diagnosis becomes more difficult. Immunohistochemical staining for the diagnosis of SFT is essential due to broad differential diagnosis of this tumor. Commonly, CD34, bcl-2, and CD99 are positive, whereas, a-SMA, desmin, pan-cytokeratin, and S-100 protein are negative in SFT. The main marker for this tumor is positive CD34. However, this condition can be observed in some tumors, such as dermatofibrosarcoma protuberans, eruptive fibromas, and angiosarcoma. In such cases, the differential diagnosis should be considered according to staining against bcl-2, CD99, CD31, and factor VIII, in combination with HE morphology.

The other differential diagnosis is hemangiopericytoma, which is indistinguishable from SFT in most cases even when employing immunohistochemical observation. Since the world health organization determined the SFT criteria in 2002 by, it is possible that many tumors were called hemangiopericytomas were, in fact, SFT⁶⁹.

It is hard to predict the biological behavior of SFT⁷⁰. SFT is commonly benign, but malignant SFT also exists. The most prognostic way to identify malignant SFT is mitotic count (>4/10 HPF)⁷¹. Dedifferentiation within an SFT is a rare phenomenon that is more frequently observed in the malignant SFT^72,73,74. The diagnosis pattern of SFT includes a well-defined high-grade component with conventional SFT regions, along with increased mitotic activity and necrosis⁷³.

However, during the process of dedifferentiation, the expression of CD34 may be diminished⁴⁵. Due to an abrupt transition between the classical SFT region and the dedifferentiated part in dedifferentiated SFT, its accurate diagnosis is difficult. Conversely, the signal transducer and activator of transcription-6 (STAT6) is a highly sensitive and specific immunohistochemical marker for SFT, which can be applied to distinguish it from histologic mimics^75,76,77.

Furthermore, NGFI-A-binding protein 2 (NAB2) nuclear staining alone, as a diagnostic marker of SFT, is not sufficient⁷⁸. In dedifferentiated SFT, a strong and diffuse p53 overexpression is observable, while there are scattered p53-positive cells in the common SFT area. A relationship between the high p53 expression and conventional clinicopathologic prognostic characteristics was observed in previous studies, which indicated the overall survival of the patients with SFT^45,79. Therefore, aberrant p53 overexpression and p53 mutation play an important role in the dedifferentiated regions of different carcinomas^45,80,81.

Recent advances in diagnostics have shown that, the number of SFT, including the advanced malignant form, will be increased. However, there is not yet a sufficient amount of information on the diagnosis and treatment of SFT. This study suggested additional reports to determine a standard therapy for SFT patients, especially those with malignant lesion.

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Received on 09.03.2019 Modified on 26.05.2019

Research J. Pharm. and Tech. 2020; 13(4):1941-1948.

DOI: 10.5958/0974-360X.2020.00350.9