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.f�?�?x1�������VVVVVVVj�����fj�*2��y*{*{*{*{*{*{*$,,h�.��*V����*VV�*�����VVy*��y*��V�&@VV!'���D}܊��J��&U*$�*0�*�&0n/��n/!'!'�n/V�'t��|�:d���*�*��*����jjjndjjjnjjjVVVVVV����ORBITAL SOLITARY FIBROUS TUMOR: REPORT OF A THREE CASES SERIES.

F. Vilardell, V. Huerva, A. Ab�, MC. S�nchez, X. Matias-Guiu.


INTRODUCTION

The solitary fibrous tumor (SFT) is a mesenchymal tumour initially reported affecting pleura (�solitary pleural fibrous tumor�), but increasingly diagnosed arising elsewhere in the body, including eyelid, orbit or lacrimal area. The SFT in the orbit presents as painless and slowly growing masses. Histologically the tumor is composed by cells resembling fibroblasts and showing among them prominent branching vascular channels giving rise to an �hemangiopericytoid� appearance. Usually, the tumor cells show strong and diffuse immunoreactivity to CD34. Usually are benign but may be aggressive. Although the differential diagnosis has been done with orbital hemangiopericytoma, schwannoma and fibrous histiocytoma, recent reports are proposing the terminology of SFT to designate all these fibrous tumours arising in the orbit. We describe three cases diagnosed in our institution in a short lapse of time.


Case 1

A 29-year-old black African man with unknown pathologic antecedents comes to our centre consulting for two years evolution protrusion of the left eyeball, which has increased during the last year. He refers occasional frontal-temporal headache but neither ocular pain nor blurred vision or diplopia. No visual loss was noticeable. With a diagnostic orientation of cavernous haemangioma versus lacrimal gland tumor, a cranial computed tomography (CT) with and without contrast showed a 30 x 18 mm lobulated mass located in the outer-external region of the left orbit. This lesion showed a strong contrast enhancement and was conditioning a displacement of the eyeball in a forward and inner direction, causing proptosis. No orbital bone infiltration was observed. Provided its location, this tumor might arise in the left lacrimal gland. The tumor was removed though eyelid creases under general anesthesia. A localized pseudoencapsulated mass was appreciable and removed. The surgical specimen was submitted to the pathology department. 

Grossly, the lesion appeared as a pinkish nodular mass measuring 29 x 17.6 mm. Histological examination by means haematoxylin & eosin staining (Figure 1) showed a well-circumscribed, partially encapsulated tumor composed of spindle shaped cells arranged in a patternless pattern with many branching thin walled vessels, giving in some areas a vaguely hemangiopericytoid architecture. The tumor cells showed oval to spindle bland nucleus, with little to moderate nuclear pleomorphism, inconspicuous to prominent nucleoli, and moderate amount of cytoplasm with indistinct cell boundaries. Few mitotic figures could be observed. An immunohistochemical panel was performed, showing positivity of the tumor cells for vimentin and CD34.

Case 2

A 46-year-old Caucasian man presents with a nodular-shaped lesion in the right lower eyelid, attached to deep planes and measuring 2 cm in maximum diameter, which had appeared two years previously. The patient refers the same size during this time. A surgical excision was proposed. In this case a direct horizontal approach may be possible and no adherence to the skin was remarkable. During the intervention an encapsulated mass was observed with adherence only to the posterior fat tissue. The histology and immunolabeling with CD34 and CD99 was compatible with solitary fibrous tumor (Figure 2). No relapse was noticeable during a year of follow-up.

Case 3 

A 38-year-old Caucasian female consults for edema and swelling of her superior right eyelid. A mild proptosis was noticeable. No visual loss was referred us. In the biomicroscopic examination, congestion of deep vessels in the nasal conjunctiva was visible. The orbital computed CT showed a nodular expansive mass of 17 mm in maximum diameter attached to the eyeball and the upper edge of the musculus rectus internus. The eyeball, optic nerve and remaining orbital muscles were normal. An excision biopsy for histologic study was proposed. Through a medial conjunctival approach, the pseudoencapsulated lesion was visible. Multiple adherences to the medial rectus and orbital fat were appreciable. The histologic study (Figure 3) showed a proliferation of spindle shaped cells arranged in a vaguely hemangiopericytoid architecture. Once again the tumor cells showed oval to spindle bland nucleus, with little to moderate nuclear pleomorphism, and showing positivity of the tumor cells for vimentin, CD34 and CD99. No relapse has been observed during a follow-up of 6 months. 

DISCUSSION

The solitary fibrous tumor (SFT) tipically presents in middle age adults, being the average 50 years. Although it was classically depicted affecting pleura (�solitary pleural fibrous tumor�), its actual anatomic distribution is very wide, and SFT have been described arising in bladder, central nervous system, ear, eye, kidney, liver, nasal and oral cavities, prostate, thyroid gland, and soft tissues from any location. So, the mesothelial origin of these tumors has been questioned, being a fibroblastic differentiation more probable.  Infrequently may cause hypoglycemia as paraneoplastic syndrome, due to production of IGF (insulin-like growth factor).
The first orbital SFTs were described by Dorfman et al (1) and Westra et al (2) in 1994. The SFT present as painless and slowly and self-limited growing masses. The symptoms are only due to contiguity growing. Grossly are solid, well circumscribed, solitary and non-encapsulated or partially encapsulated masses, with a whitish and firm cut surface. Histologically, the tumor is composed by cells mimicking fibroblasts, arranged with a patternless pattern but showing among them prominent branching vascular channels giving rise to a peculiar architecture called �hemangiopericytoid�. Most tumors formerly identified as soft tissue hemangiopericytomas, are currently recognized as solitary fibrous tumors. Collagen deposition can be observed, ranging from very little to keloid-like fibrosis. Some cases of SFT with cystic areas have been reported (3). Usually, the tumor cells show strong and diffuse immunoreactivity to vimentin, CD34 and bcl-2. More variable reactivity to CD99 may be also found. Muscular markers, cytokeratins, S-100 and factor VIII are negative. Usually are benign but may be aggressive. The SFT with malignant histological features may show an infiltrative growing pattern, and can originate metastases in up to 15-20% of cases (4). Presence of necrosis and infiltrative margins are associated to locally aggressive or malignant tumors. Exceptionally, tumours with a benign histological appearance can metastasize. Features indicative of worse prognosis are size larger than 10 cm in diameter, a mitotic index higher than four figures of mitosis in ten high-power fields, market nuclear atypia and affected surgical margins. A cell rich variant of SFT is the tumor traditionally known as giant cell angiofibroma of orbit. This tumor arises in eyelid, near to the lacrimal gland, and like the usual SFT are well delimitated but nonencapsulated. The giant cell angiofibroma histologically shows a collagenized or myxoid background, and floret type multinucleated giant cells lining the vascular channels, but in fact the presence of giant cells has been reported in extrapleural SFTs (5). Traditionally, the main differential diagnosis has been done with orbital hemangiopericytoma, schwannoma, fibrous histiocytoma and optic nerve meningioma (6). In opposition to many other anatomic regions where hemangiopericytoma are a controversial entity sine long time ago, the orbital region is a location where hemangiopericytomas are still currently diagnosed. These tumors resembling SFT seem to be better encapsulated and show more prominent vessels and a dense pericellular staining of reticulin, but in opposition to SFT usually show a clear positivity to CD99 but a focal or weak immunostaining to CD34. Their behaviour seems to be more aggressive compared to SFT (7). Fibrous histiocytomas are the most common mesenchymal tumor in the orbital region and may be benign, locally aggressive or malignant. Microscopically are fibrous tumors with a storiform pattern. Furusato E et al. (8), after review the morphology and CD34, CD99, Bcl-2, Ki-67, and p53 immunostaining pattern of 41 orbital fibrous tumours from their files, currently claim that hemangiopericytomas, SFT, giant cell angiofibromas and fibrous histiocytomas are actually the same tumour, despite some variation in degree of cellularity, stromal collagen, presence of giant cells or immunoreactivity pattern, and should be designated as solitary fibrous tumour. In conclusion, orbital solitary fibrous tumor (SFT) is being proposed as the encompassing terminology for hemangiopericytoma, giant cell angiofibroma (GCAF), and fibrous histiocytoma of the orbit (9).


Address correspondence to: Felip Vilardell Villellas, Servei d�Anatomia Patol�gica
Hospital Universitari Arnau de Vilanova, Alcalde Rovira Roure 80, 25198, Lleida
Tel 973 248100; fvilardell.lleida.ics HYPERLINK "mailto:fvilardell@arnau.scs.es" @gencat.cat

Competing interests: None to declare
Patient consent: obtained
Ethics approval: This study was conducted with the approval of the Hospital Arnau de Vilanova Ethics Committee
Provenance and peer review: Not commissioned; externally peer reviewed


REFERENCES

Dorman D.M, To K, Dickersin GR, Rosenberg AE, Pilch BZ. 1994. Am J Surg Pathol. 1994 Mar; 18(3):281-7.
Westra WH, Gerald WL, Rosai J. Solitary fibrous tumor. Consistent CD34 immunoreactivity and occurrence in the orbit. Am J Surg Pathol. 1994. 18(10):992-8.
Feuerman J.M, Flint A, Elner V.M. Cystic solitary fibrous tumor of the orbit. 2010. Arch Ophthalmol 128(3):385-387.
Hanau CA, Miettinen M. Solitary fibrous tumor: Histological and immunohistochemical spectrum of benign and malignant variants presenting at different sites. 1995 Hum Pathol; 26:440-9.
Hasegawa T, Matsuno Y, Shimoda T, Hasegawa F, Sano T, Hirohashi S. Extrathoracic solitary fibrous tumors: their histological variability and potentially aggressive behavior. Hum Pathol. 1999; 30:1464�1473.
 HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=Bernardini%20FP%5BAuthor%5D&cauthor=true&cauthor_uid=12867407" Bernardini F.P,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=de%20Conciliis%20C%5BAuthor%5D&cauthor=true&cauthor_uid=12867407" de Conciliis C,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=Schneider%20S%5BAuthor%5D&cauthor=true&cauthor_uid=12867407" Schneider S,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=Kersten%20RC%5BAuthor%5D&cauthor=true&cauthor_uid=12867407" Kersten R.C,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=Kulwin%20DR%5BAuthor%5D&cauthor=true&cauthor_uid=12867407" Kulwin D.R. Solitary fibrous tumor of the orbit: is it rare? Report of a case series and review of the literature. 2003. Ophthalmology 110(7):1442-8.
Ali M.J., Honavar S.G., Naik M.N., Vemuganti G.K. Orbital solitary fibrous tumor: A clinicopathologic correlation and review of literature. 2011. Oman J Ophthalmol 4(3):147-9.
Furusato E, Valenzuela I.A, Fanburg-Smith J.C, Auerbach A, Furusato B, Cameron J.D, Rushing E.J.  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed/21056898" Orbital solitary fibrous tumor: encompassing terminology for hemangiopericytoma, giant cell angiofibroma, and fibrous histiocytoma of the orbit: reappraisal of 41 cases. 2011. Hum Pathol Jan;42(1):120-8.
Son da H, Yoo S.H, Sa H.S, Cjo K.J. A solitary fibrous tumor with giant cells in the lacrimal gland: a case study. Korean J Pathol. 2013. Apr;47(2):158-62.

FIGURE LEGEND
Figure 1. Case 1: a), b) and c) Hematoxilin & eosin stained slide at original magnifications 40x, 100x and 400x respectively, showing a vaguely hemangiopericytoid architecture with oval to spindle bland nucleus cells; d) Strong CD34 immunolabeling (original magnification 100x); e) Smooth-muscle actin immunolabeling, highlighting the tumor branching vessels.
Figure 2. Case 2: a) Macro-micro vision of a whole section showing a nodular-shaped and encapsulated lesion; b) and c) Hematoxilin & eosin stained slide at original magnification 40x and 400x respectively; d) & e) Strong CD34 and CD99  immunolabeling (original magnification 100x).
Figure 3. Case 3: a) and b) Hematoxilin & eosin stained slide at original magnification 40x and 400x respectively, showing vaguely hemangiopericytoid architecture and oval to spindle bland nucleus; c) & d) Positive CD99 and CD34 immunostaining (original magnification 100x); e) Low Ki67 proliferation index (original magnification 100x).
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