Intracranial solitary fibrous tumor in a 15-year-old girl: illustrative case

Kuan Lu Medical Imaging Center, The Second People’s Hospital of Yibin, Neuroimaging Big Data Research Center, Clinical Research and Translational Center, The Second People’s Hospital of Yibin, Yibin, China
Department of Radiology, The Affiliated Hospital of Southwest Medical University, Luzhou, China; and

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Xiaoqing Qu Medical Imaging Center, The Second People’s Hospital of Yibin, Neuroimaging Big Data Research Center, Clinical Research and Translational Center, The Second People’s Hospital of Yibin, Yibin, China
Department of Radiology, The Affiliated Hospital of Southwest Medical University, Luzhou, China; and

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Jingcheng Jiang Department of Neurosurgery, The Second People’s Hospital of Yibin, Yibin, China

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Quanjun Zheng Medical Imaging Center, The Second People’s Hospital of Yibin, Neuroimaging Big Data Research Center, Clinical Research and Translational Center, The Second People’s Hospital of Yibin, Yibin, China

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Yongsheng Ao Medical Imaging Center, The Second People’s Hospital of Yibin, Neuroimaging Big Data Research Center, Clinical Research and Translational Center, The Second People’s Hospital of Yibin, Yibin, China
Department of Radiology, The Affiliated Hospital of Southwest Medical University, Luzhou, China; and

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Lihua Qiu Medical Imaging Center, The Second People’s Hospital of Yibin, Neuroimaging Big Data Research Center, Clinical Research and Translational Center, The Second People’s Hospital of Yibin, Yibin, China
Department of Radiology, The Affiliated Hospital of Southwest Medical University, Luzhou, China; and

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BACKGROUND

Solitary fibrous tumor (SFT) is an infrequent spindle cell tumor derived from mesenchymal tissue, which can manifest in diverse anatomical locations, primarily in the pleural cavity and infrequently in the central nervous system. SFT is predominantly observed in individuals aged between 40 and 50 years old, with a slightly higher occurrence in males than in females.

OBSERVATIONS

This case report describes a female, age 15, who had migraines for 2 months prior to the diagnosis of an intracranial tumor. Computed tomography and magnetic resonance imaging showed a mass located in the right parietooccipital region with surrounding edema and a compressed right lateral ventricle. Neurosurgery was utilized to successfully remove the mass, and single intracranial fibrous tumor (grade I) was identified by postoperative pathological analysis. During an 8-month follow-up period, the patient did not experience any recurrences.

LESSONS

SFT is often misdiagnosed as meningioma because of their similar imaging characteristics. However, identifying the distinctive features of SFT on magnetic resonance imaging can distinguish it from meningioma and help to select appropriate treatment. The complete preoperative imaging data for this case indicated a highly vascularized tumor. Preoperative vascular embolization treatment reduced any difficulties during the subsequent tumor resection and minimized intraoperative bleeding.

ABBREVIATIONS

ASL = arterial spin labeling; CBF = cerebral blood flow; CNS = central nervous system; CT = computed tomography; DTI = diffusion tensor imaging; HPC = hemangiopericytoma; MRI = magnetic resonance imaging; MRS = magnetic resonance spectroscopy; NAA = N-acetylaspartate; SFT = solitary fibrous tumor; WHO = World Health Organization

BACKGROUND

Solitary fibrous tumor (SFT) is an infrequent spindle cell tumor derived from mesenchymal tissue, which can manifest in diverse anatomical locations, primarily in the pleural cavity and infrequently in the central nervous system. SFT is predominantly observed in individuals aged between 40 and 50 years old, with a slightly higher occurrence in males than in females.

OBSERVATIONS

This case report describes a female, age 15, who had migraines for 2 months prior to the diagnosis of an intracranial tumor. Computed tomography and magnetic resonance imaging showed a mass located in the right parietooccipital region with surrounding edema and a compressed right lateral ventricle. Neurosurgery was utilized to successfully remove the mass, and single intracranial fibrous tumor (grade I) was identified by postoperative pathological analysis. During an 8-month follow-up period, the patient did not experience any recurrences.

LESSONS

SFT is often misdiagnosed as meningioma because of their similar imaging characteristics. However, identifying the distinctive features of SFT on magnetic resonance imaging can distinguish it from meningioma and help to select appropriate treatment. The complete preoperative imaging data for this case indicated a highly vascularized tumor. Preoperative vascular embolization treatment reduced any difficulties during the subsequent tumor resection and minimized intraoperative bleeding.

ABBREVIATIONS

ASL = arterial spin labeling; CBF = cerebral blood flow; CNS = central nervous system; CT = computed tomography; DTI = diffusion tensor imaging; HPC = hemangiopericytoma; MRI = magnetic resonance imaging; MRS = magnetic resonance spectroscopy; NAA = N-acetylaspartate; SFT = solitary fibrous tumor; WHO = World Health Organization

Intracranial solitary fibrous tumor (SFT), also referred to as “hemangiopericytoma” (HPC) in the past, is a rare type of tumor composing less than 1% of all intracranial tumors.1 The 2016 classification of tumors in the central nervous system (CNS) by the World Health Organization (WHO) has merged SFT and HPC into a unified entity referred to as “SFT/HPC.”2 In the 2021 WHO classification, SFT is categorized as a nonmeningeal mesenchymal epithelial tumor and graded into three levels, namely 1, 2, and 3.3 SFT typically develops in middle-aged and older individuals, demonstrating a higher incidence during the fourth and fifth decades of life, and has a slight male predominance.4 In this case, we report on a histologically confirmed diagnosis of a WHO grade 1 intracranial SFT in an adolescent patient. To the best of our knowledge, this case report provides the most comprehensive documented instance of intracranial SFT in a teenager, along with accompanying imaging data. The imaging data for this case were thorough, encompassing pre- and postenhancement computed tomography (CT) and magnetic resonance imaging (MRI), diffusion tensor imaging (DTI), magnetic resonance spectroscopy (MRS), and arterial spin labeling (ASL).

Illustrative Case

A 15-year-old female patient reported experiencing headaches for 2 months and sought medical attention after discovering the mass outside of the hospital 3 days prior. Neurological examination showed no abnormalities, and laboratory evaluations were within normal ranges. The first CT scan of the head revealed a mass with an increased density, surrounding edema, mass effect, compression of the right lateral ventricle, and resorption of bone in the adjacent area. Additionally, a right-to-left midline shift was also observed. The mass displayed significant enhancement on the contrast-enhanced scan, exhibiting numerous neovascularizations (Fig. 1).

FIG. 1
FIG. 1

Preoperative nonenhanced CT (A) shows a hyperdense mass adjacent to the right parieto-occipital region, with no calcification. Postcontrast CT (B and C) reveals a markedly enhanced mass with abundant neovascularization. Axial CT (D) shows bone resorption in the adjacent bones.

MRI revealed a mass with mixed-intensity signals, was isointense on T1-weighted imaging, and demonstrated both hyper- and hypointense signals on T2-weighted imaging. The mass measured approximately 71 × 62 × 60 mm. The tumor demonstrated varied enhancement on the contrast-enhanced scan, and noticeable edema of the brain tissue was evident in the surrounding area. The adjacent meninges were thickened and enhanced, and a dural tail sign was present. DTI indicated compression and displacement of the white matter fiber tracts in the affected region. MRS revealed a heightened choline peak and the absence of an N-acetylaspartate (NAA) peak, indicating an extracerebral origin. ASL revealed marginal hyperperfusion and central hypoperfusion (Fig. 2).

FIG. 2
FIG. 2

Preoperative axial T1-weighted (A), T2-weighted (B), and fluid-attenuated inversion recovery (FLAIR; C) images show an isointense mass adjacent to the right parietooccipital region. The lesion appears mixed hyper- and hypointense on T2 FLAIR imaging compared to cortical gray matter. Diffusion-weighted imaging (D) and apparent diffusion coefficient (ADC; E) suggest slight restriction of diffusion. Contrast-enhanced T1-weighted images (F–H) show heterogeneous and vivid enhancement of the tumor with the dural tail sign. DTI (I) reveals slight damage and compression of the white matter fiber tracts in the lesion area. ASL (J) shows marginal hyperperfusion and central hypoperfusion. MRS (K) shows increased choline peak and no obvious NAA peak in the lesion area, consistent with an extracerebral origin.

After extensive consultation with the patient and her surrogate decision-maker, the surgical team opted for intervention to excise the tumor. The patient underwent preoperative interventional embolization of the supplying arteries. Angiography revealed that the blood supply was from the right external carotid arterial system, which includes the occipital, posterior meningeal, and middle meningeal arteries, as well as the right internal carotid arterial system, including the meningeal pituitary trunk and distal branches of the middle cerebral artery. The responsible vessel was embolized using a slow injection of glue. On review, the occupied vessel was reduced by approximately 70%, and contrast development was significantly delayed. The remaining angiographic results were normal.

During the operation, a tumor was located at the foundation of the transverse sinus and tentorium cerebelli, and it was sufficiently perfused with blood. Initially, the tumor was separated from the base of the transverse sinus and tentorium before it was carefully dissected along the tumor capsule. The excision was performed under microscopic observation, utilizing microscissors and an ultrasonic scalpel (cavitron ultrasonic surgical aspirator) together with a bipolar electrocoagulation tool. Ultimately, the tumor was removed in stages. Due to the abundance of thick and numerous tumor-perforating arteries, along with turbulent hemorrhaging, the process of excising the mass in stages proved to be quite challenging. Hence, a section of the swollen brain tissue, specifically the right parietal and occipital lobes, was excised, and an incision was made along the arachnoid membrane space of the tumor. The brain tumor was gradually removed, and ultimately the tumor tissue that was attached to the transverse sinus was cauterized. Arteries and veins located on the surface of the brain tissue were isolated and protected. The tumor was completely removed under microscopic examination.

Pathological analysis confirmed the presence of a solitary fibrous tumor (grade 1 according to the 2021 WHO classification criteria), characterized by histomorphology and immunophenotype, including CD34 (+), SATA6 (+), PR (−), EMA (−), GFAP (−), S100 (−), P53 (−), and Ki-67 (+, about 1%; Fig. 3). The patient experienced no postoperative functional deficits. Postoperative imaging revealed the absence of any definite tumor tissue, implying complete removal of the tumor. The patient remained asymptomatic and did not show signs of recurrence during the 8-month follow-up period (Fig. 4).

FIG. 3
FIG. 3

Histopathological section illustrating anaplastic SFT. Hematoxylin and eosin, original magnification ×400.

FIG. 4
FIG. 4

Postoperative axial T1-weighted (A), T2-weighted (B), and T2 FLAIR (C) images showed that the original right parietooccipital mass had been removed. Currently, lamellar encephalomalacia was formed in the surgical area, the posterior horn of the adjacent right lateral ventricle was obviously dilated, the parietooccipital region of the skull was partially absent, the adjacent part of meningeal brain tissue was bulged, and the midline structure was not significantly displaced. DWI (D) shows no abnormal high signal in the surgical area. There was no obvious abnormal enhancement in contrast-enhanced scanning (E–H).

Patient Informed Consent

The necessary patient informed consent was obtained in this study.

Discussion

Observations

Intracranial SFT constitutes only 0.4% of all intracranial tumors and represents a rare tumor that originates from meningeal mesenchyme.5–7 SFT is often located in the intracranial region outside of the brain, commonly presenting as lesions near the skull base, sagittal sinus, falx cerebri, and tentorium cerebelli.5,8 The location of SFT within the brain is comparable to that of meningioma. Our case corresponds with previous reports of SFT in the areas of the transverse sinus and tentorium cerebelli. Most SFTs are indeed solitary, but instances of multiple lesions can occur. In terms of malignancy, intracranial SFTs are regarded as borderline tumors, with the majority of cases exhibiting benign characteristics and 10% to 20% displaying malignant or potentially malignant features. Meningioma, on the other hand, is one of the most common types of primary central nervous system tumors, accounting for 20% of all intracranial tumors.9 It is frequently noted in regions with high levels of arachnoid granulations.10 However, intracranial meningiomas are uncommon in children, accounting for only 0.4% to 4.1% of pediatric tumors and merely 1.5% to 1.8% of all intracranial meningiomas.11 They constitute just 1.1% to 2.9% of all CNS tumors and display a slight male predominance.12 Pediatric meningiomas tend to be larger and have a higher incidence of cystic components and a high grade than meningiomas found in the adult population.11 They are more likely to occur in unusual locations, such as the ventricular system, craniocervical junction, and skull base. Additionally, they have a higher chance of recurring12,13 and are less frequently observed to have a dural attachment. Although most meningiomas have a positive prognosis after complete surgical removal,14 distinguishing between SFT and meningioma can present challenges in routine clinical practice given their identical manifestations on conventional diagnostic imaging.15,16 However, significant differences exist in their biological behavior, treatment options, surgical planning, and prognosis.17 SFT displays a heightened level of biological aggressiveness, resulting in a recurrence rate of 60% postresection and a tendency to metastasize to extracranial organs at a rate of 20%.6,18 Therefore, resection followed by radiotherapy or chemotherapy is the primary treatment option for SFT.17 After our surgical procedure, the pathological findings revealed a WHO grade 1 SFT, indicating no need for postoperative chemotherapy or radiotherapy. However, the resection of SFT can result in profuse intraoperative bleeding because of the abundant blood supply of these tumors. Preoperative embolization is recommended for reducing the risk of intraoperative bleeding and improving resectability.19 Preoperative embolization was conducted in our case. Therefore, it is essential to distinguish SFT from meningioma preoperatively.17

The primary danger of undergoing surgery is the tumor’s high vascularity, with the possibility of invading the venous sinus, which can cause significant bleeding throughout the procedure. Predicting the preoperative blood supply and efficiently managing the supplying arteries can reduce surgical blood loss and improve the likelihood of complete tumor resection. Another surgical risk involves complications that may arise from preoperative tumor embolization. Previous research suggests that particle embolization in certain hypervascular tumors can increase the chances of postoperative tumor swelling and bleeding.20 In this specific case, the preoperative imaging data demonstrated that the tumor possessed an abundant blood supply, as well as significant blood vessels. As a result, interventional embolization of blood vessels supplying the area was performed initially to reduce bleeding during surgery. Fortunately, there were no complications resulting from the procedure.

Key points for distinguishing between intracranial SFT and meningioma comprise the detection of a meningeal tail sign, which is present in both tumor types. However, meningioma is more frequently diagnosed in middle-aged women and often displays calcification and a broader base than SFT. The bone changes neighboring the area can aid in discerning intracranial SFT from meningioma. Adjacent bone thickening is often observed in meningiomas, but erosion is commonly seen in intracranial SFT.21,22 In addition, MRS is helpful for the differential diagnosis of intracranial SFT and meningioma. It has been reported in the literature21,23 that MRS of meningioma can show elevated glutamate and alanine peaks, but no glutamate and alanine peaks were found in intracranial SFT. In addition to the decreased levels of NAA and creatine and the increased levels of choline and myo-inositol in SFT, some lesions can show lactate peak and lipid peak, which have not been described in meningiomas. This may be one of the distinguishing features between intracranial SFT and meningioma. In our case, the tumor showed an elevated choline peak and no apparent NAA peak on MRS analysis, consistent with previous reports.21,23 At present, only one study has reported that magnetic resonance perfusion imaging of intracranial SFT results in a significant increase in cerebral blood volume (7- to 7.5-fold relative cerebral blood volume compared to normal white matter).21 In this case, we used ASL to investigate the cerebral blood flow (CBF) value of tumor parenchyma and found that the hyperperfusion area at the edge of the mass was 269.6 (mL/100 mg/min) and that the regional CBF value was 4.3, which is generally consistent with the literature.21 In contrast, most of the meningiomas showed hyperperfusion, which was more uniform or gradually decreased from the center to the periphery. The marginal hyperperfusion of SFT may be used to differentiate it from meningioma, and further confirmation is needed in more cases in the future.

Lessons

SFT, previously termed “hemangiopericytoma,” is a rare extraaxial intracranial tumor. Currently, there are only a few reports of intracranial SFT in children24,25 and infants,26,27 and our case report of SFT in an adolescent provides the first complete imaging data in this age group.

Differentiating SFT from meningioma is challenging but can be facilitated through careful analysis of characteristic imaging features, especially multimodal MRI. CT and MRI are valuable tools for diagnosis, grading, treatment planning, and follow-up monitoring of SFT. This case report highlights the occurrence of SFT in an adolescent patient, wherein comprehensive imaging examination enhances our understanding of this rare intracranial tumor.

Author Contributions

Conception and design: all authors. Acquisition of data: Lu, Qu, Jiang, Zheng, Ao. Analysis and interpretation of data: Lu, Qu, Jiang, Zheng, Ao. Drafting the article: Lu, Qu, Jiang, Zheng, Ao. Critically revising the article: Qiu, Lu. Reviewed submitted version of manuscript: Qiu, Lu, Jiang. Approved the final version of the manuscript on behalf of all authors: Qiu. Administrative/technical/material support: Qiu, Ao. Study supervision: Qiu, Lu, Qu, Jiang, Ao.

References

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  • FIG. 1

    Preoperative nonenhanced CT (A) shows a hyperdense mass adjacent to the right parieto-occipital region, with no calcification. Postcontrast CT (B and C) reveals a markedly enhanced mass with abundant neovascularization. Axial CT (D) shows bone resorption in the adjacent bones.

  • FIG. 2

    Preoperative axial T1-weighted (A), T2-weighted (B), and fluid-attenuated inversion recovery (FLAIR; C) images show an isointense mass adjacent to the right parietooccipital region. The lesion appears mixed hyper- and hypointense on T2 FLAIR imaging compared to cortical gray matter. Diffusion-weighted imaging (D) and apparent diffusion coefficient (ADC; E) suggest slight restriction of diffusion. Contrast-enhanced T1-weighted images (F–H) show heterogeneous and vivid enhancement of the tumor with the dural tail sign. DTI (I) reveals slight damage and compression of the white matter fiber tracts in the lesion area. ASL (J) shows marginal hyperperfusion and central hypoperfusion. MRS (K) shows increased choline peak and no obvious NAA peak in the lesion area, consistent with an extracerebral origin.

  • FIG. 3

    Histopathological section illustrating anaplastic SFT. Hematoxylin and eosin, original magnification ×400.

  • FIG. 4

    Postoperative axial T1-weighted (A), T2-weighted (B), and T2 FLAIR (C) images showed that the original right parietooccipital mass had been removed. Currently, lamellar encephalomalacia was formed in the surgical area, the posterior horn of the adjacent right lateral ventricle was obviously dilated, the parietooccipital region of the skull was partially absent, the adjacent part of meningeal brain tissue was bulged, and the midline structure was not significantly displaced. DWI (D) shows no abnormal high signal in the surgical area. There was no obvious abnormal enhancement in contrast-enhanced scanning (E–H).

  • 1

    Sardaro A, Mammucci P, Pisani AR, et al. Intracranial solitary fibrous tumor: a “new” challenge for PET radiopharmaceuticals. J Clin Med. 2022;11(16):4746.

  • 2

    Kim BS, Kim Y, Kong DS, et al. Clinical outcomes of intracranial solitary fibrous tumor and hemangiopericytoma: analysis according to the 2016 WHO classification of central nervous system tumors. J Neurosurg. 2018;129(6):13841396.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 3

    Louis DN, Perry A, Wesseling P, et al. The 2021 WHO Classification of Tumors of the Central Nervous System: a summary. Neuro Oncol. 2021;23(8):12311251.

  • 4

    Barthelmeß S, Geddert H, Boltze C, et al. Solitary fibrous tumors/hemangiopericytomas with different variants of the NAB2-STAT6 gene fusion are characterized by specific histomorphology and distinct clinicopathological features. Am J Pathol. 2014;184(4):12091218.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 5

    Louis DN, Perry A, Reifenberger G, et al. The 2016 World Health Organization Classification of Tumors of the Central Nervous System: a summary. Acta Neuropathol. 2016;131(6):803820.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 6

    Ratneswaren T, Hogg FRA, Gallagher MJ, Ashkan K Surveillance for metastatic hemangiopericytoma-solitary fibrous tumors-systematic literature review on incidence, predictors and diagnosis of extra-cranial disease. J Neurooncol. 2018;138(3):447467.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 7

    Kinslow CJ, Bruce SS, Rae AI, et al. Solitary-fibrous tumor/hemangiopericytoma of the central nervous system: a population-based study. J Neurooncol. 2018;138(1):173182.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 8

    Moritani S, Ichihara S, Hasegawa M, et al. Dedifferentiation and progression of an intracranial solitary fibrous tumor: autopsy case of a Japanese woman with a history of radiation therapy of the head during infancy. Pathol Int. 2011;61(3):143149.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 9

    Longstreth WT Jr, Dennis LK, McGuire VM, Drangsholt MT, Koepsell TD Epidemiology of intracranial meningioma. Cancer. 1993;72(3):639648.

  • 10

    Goldbrunner R, Minniti G, Preusser M, et al. EANO guidelines for the diagnosis and treatment of meningiomas. Lancet Oncol. 2016;17(9):e383e391.

  • 11

    Lakhdar F, Arkha Y, El Ouahabi A, et al. Intracranial meningioma in children: different from adult forms? A series of 21 cases. Neurochirurgie. 2010;56(4):309314.

  • 12

    Pinto PS, Huisman TA, Ahn E, et al. Magnetic resonance imaging features of meningiomas in children and young adults: a retrospective analysis. J Neuroradiol. 2012;39(4):218226.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 13

    Phillips D, Auguste KI, Gupta N Meningiomas in children. Handb Clin Neurol. 2020;169:253259.

  • 14

    Wang N, Osswald M Meningiomas: overview and new directions in therapy. Semin Neurol. 2018;38(1):112120.

  • 15

    Meng Y, Chaohu W, Yi L, Jun P, Songtao Q Preoperative radiologic characters to predict hemangiopericytoma from angiomatous meningioma. Clin Neurol Neurosurg. 2015;138:7882.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 16

    Smith AB, Horkanyne-Szakaly I, Schroeder JW, Rushing EJ From the radiologic pathology archives: mass lesions of the dura: beyond meningioma-radiologic-pathologic correlation. Radiographics. 2014;34(2):295312.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 17

    Chen T, Jiang B, Zheng Y, et al. Differentiating intracranial solitary fibrous tumor/hemangiopericytoma from meningioma using diffusion-weighted imaging and susceptibility-weighted imaging. Neuroradiology. 2020;62(2):175184.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 18

    Sung KS, Moon JH, Kim EH, et al. Solitary fibrous tumor/hemangiopericytoma: treatment results based on the 2016 WHO classification. J Neurosurg. 2018;130(2):418425.

    • PubMed
    • Search Google Scholar
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  • 19

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