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Orbital Chondromyxoid Fibroma Osteal Space, Pushing the Inferior and Medial Periosteal Border Into the Orbital Soft Tissue

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Orbital Chondromyxoid Fibroma Osteal Space, Pushing the Inferior and Medial Periosteal Border Into the Orbital Soft Tissue cal Center, No. 325, Section 2, Cheng-Kung Road, Neihu the posterior pole (Figure 1A). Computed tomogra- 114, Taipei, Taiwan ([email protected]). phy disclosed a superotemporal, noninfiltrative orbital Financial Disclosure: None reported. mass with erosion of the adjacent frontal bone (Figure 1B and C). Differential diagnosis included lacrimal gland tu- 1. Janatpour KA, Choo PH, Lloyd WC III. Primary orbital peripheral T-cell lym- phoma: histologic, immunophenotypic, and genotypic features. Arch mors, atypical dermoid cysts, and benign fibro-osseous Ophthalmol. 2007;125(9):1289-1292. lesions such as osteoma, fibrous dysplasia, or ossifying 2. Woog JJ, Kim Y-D, Yeatts RP, et al. Natural killer/T-cell lymphoma with ocu- lar and adnexal involvement. Ophthalmology. 2006;113(1):140-147. fibromyxoid tumor of soft parts. 3. Coupland SE, Foss HD, Assaf C, et al. T-cell and T/natural killer-cell lym- The patient underwent transcutaneous extraperios- phomas involving ocular and ocular adnexal tissues: a clinicopathologic, im- munohistochemical, and molecular study of seven cases. Ophthalmology. 1999; teal orbitotomy. Intraoperatively, the periosteum was sepa- 106(11):2109-2120. rated by blunt dissection from the bone in the periph- eral portions of the mass both superiorly and inferiorly. The mass proved to be located mainly in the extraperi- Orbital Chondromyxoid Fibroma osteal space, pushing the inferior and medial periosteal border into the orbital soft tissue. The tumor centrally rimary tumors of orbital bone are rare, constitut- ing up to 2% of all orbital masses.1 Chondromyx- showed a close connection to the bony wall. En bloc re- oid fibroma (CMF) is one of the least common section including the tumor, surrounding periosteum, and P adjacent bony wall was performed. tumors of bone, composing less than 1% of bone tu- ϫ ϫ mors and less than 2% of benign bone tumors.2,3 Apart Macroscopically, the mass measured 20 15 7 from brief case reports,4-6 orbital CMF has not been clearly mm. Histopathological examination revealed a CMF documented in the ophthalmic literature. To our knowl- (Figure 2A-C). The soft-tissue mass was surrounded edge, we report for the first time the clinicopathological inferomedially by periosteum. The periosteum showed features and management options of an orbital CMF aris- calcification superiorly and laterally corresponding to the ing from the frontal bone. radio-dense margins on the computed tomographic scan. The lateral borders of the mass consisted of bone. The cel- Report of a Case. A 37-year-old woman had slowly pro- lular elements displaying a low proliferation rate (MIB- gressive swelling of the left upper eyelid temporally as- 1Ͻ1%) were positive for S-100B protein in the central chon- sociated with occasional headache and shooting pain for droid area and positive for vimentin and smooth muscle 3 years. On examination, visual acuity was 20/20 OU. The actin in the peripheral fibroblastic area but negative for des- left eye showed 4 mm of proptosis with downward dis- min, CD68, CD34, and CD31. Ultrastructural findings placement, mild blepharoptosis, and choroidal folds at (Figure 2D and E) supported the diagnosis of CMF. A B C D Figure 1. Clinical features of an orbital chondromyxoid fibroma arising from the frontal bone in a 37-year-old woman. A, Slowly progressive proptosis, downward displacement, and mild blepharoptosis of the patient’s left eye, of 3 years’ duration. B, Axial computed tomographic scan showing a circumscribed, round to ovoid, superotemporal orbital mass with radio-dense margins and a lucent central component isodense to muscle tissue indenting the globe inferiorly. C, Axial computed tomographic scan with bone window settings clearly delineating the incompletely formed sclerotic rim surrounding the round to ovoid mass with focal erosion (arrow) and thinning of the adjacent frontal bone. D, Marked improvement of proptosis, downward displacement, and blepharoptosis without evidence of recurrence or metastasis 2 years after transcutaneous extraperiosteal orbitotomy with en bloc resection including the tumor, surrounding periosteum, and adjacent bony wall. (REPRINTED) ARCH OPHTHALMOL / VOL 127 (NO. 8), AUG 2009 WWW.ARCHOPHTHALMOL.COM 1072 ©2009 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/29/2021 A B,D Figure 2. Histopathological and ultrastructural findings of an orbital chondromyxoid fibroma arising from the frontal bone. A, Histopathological section showing an overview of the orbital mass surrounded in part by periosteum (arrows) and bony elements (arrowheads) (Masson trichrome, C,E original magnification ϫ12.5). The letters indicate the locations of figure parts B, C, D, and E. B, Centrally located hypocellular area with spindle-shaped or stellate cells in a myxoid matrix (hematoxylin-eosin, original magnification ϫ200). C, Peripherally located hypercellular area with many rather uniform spindle-shaped cells and focal calcification (hematoxylin-eosin, original ϫ B C magnification 100). D, Transmission electron microscopy of the central hypocellular area showing stellate chondroblastlike cells with irregularly shaped, euchromatin-rich nuclei with inconspicuous nucleoli, prominent dilated rough endoplasmic reticulum, bundles of intermediate filaments, and isolated lipid droplets (bar=5 µm). E, Transmission electron microscopy of the peripheral hypercellular area showing rather spindle-shaped fibroblastic cells with oval nuclei and moderate amounts of rough endoplasmic reticulum (bar=4 µm). D E Postoperatively, recovery was fast and unremark- teum, and adjacent bony wall was performed. Complete able. Two years after surgery, the patient showed marked en bloc resection is important regarding both histo- improvement of proptosis, downward displacement, and pathological diagnosis and the prevention of tumor re- blepharoptosis without evidence of recurrence or me- currence. Simple curettage may favor underdiagnosis tastasis (Figure 1D). and explain in part why the entity has been rarely documented. Comment. Chondromyxoid fibroma manifests most In conclusion, CMF should be considered as a rare be- frequently in the second and third decades of life, more nign lesion in the differential diagnosis of primary orbital often in males than in females.2,3 The long bones are the bone tumors. most common site, followed by the flat bones and the bones of the hands and feet.3 Craniofacial involvement Ludwig M. Heindl, MD is relatively rare.3 Histopathological differential diag- Kerstin U. Amann, MD nosis includes chondrosarcoma, enchondroma, or Arndt Hartmann, MD chordoma.2 Friedrich E. Kruse, MD Chondromyxoid fibroma with orbital involvement has Leonard M. Holbach, MD been reported only in brief case reports.4-6 Hashimoto et al4 and Cruz et al5 described a CMF of the ethmoid sinus Correspondence: Dr Heindl, Department of Ophthal- destroying the medial orbital wall. Wolf et al6 reported mology and University Eye Hospital, University Erlangen- an intracranial CMF of the frontal-sphenoid junction with Nürnberg, Schwabachanlage 6, 91054 Erlangen, Ger- secondary orbital involvement. In our patient, there many ([email protected]). was—to our knowledge for the first time—marked down- Financial Disclosure: None reported. ward displacement and indentation of the globe with cho- Previous Presentation: This study was presented at the roidal folds caused by progressive orbital tumor growth. 47th Annual Meeting of the European Ophthalmic Pa- Because the adjacent periosteum was intact, we elected thology Society; June 2008; Besanc¸on, France. to take an extraperiosteal approach, separating tumor and Additional Contributions: Mathias Werner, MD, pro- periosteum from bone as much as possible. In the area of vided expert evaluation. close adhesion to the adjacent frontal bone, an en bloc 1. Selva D, White VA, O’Connell JX, Rootman J. Primary bone tumors of the resection including the tumor, surrounding perios- orbit. Surv Ophthalmol. 2004;49(3):328-342. (REPRINTED) ARCH OPHTHALMOL / VOL 127 (NO. 8), AUG 2009 WWW.ARCHOPHTHALMOL.COM 1073 ©2009 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/29/2021 2. Zillmer DA, Dorfman HD. Chondromyxoid fibroma of bone: thirty-six cases with positive excision margins 2 years before the refer- with clinicopathologic correlation. Hum Pathol. 1989;20(10):952-964. 3. Wu CT, Inwards CY, O’Laughlin S, Rock MG, Beabout JW, Unni KK. Chon- ral. He developed right facial paralysis and lower eyelid dromyxoid fibroma of bone: a clinicopathologic review of 278 cases. Hum Pathol. ectropion a few weeks after excision. He underwent sur- 1998;29(5):438-446. gical repair of paralytic ectropion presumed to be due to 4. Hashimoto M, Izumi J, Sakuma I, Iwama T, Watarai J. Chondromyxoid fi- broma of the ethmoid sinus. Neuroradiology. 1998;40(9):577-579. idiopathic Bell’s palsy. The ectropion recurred after 8 5. Cruz AA, Mesquita IM, Becker AN, Chahud F. Orbital invasion by chondro- months. Magnetic resonance imaging revealed an infra- myxoid fibroma of the ethmoid sinus. Ophthal Plast Reconstr Surg. 2007;23(5): 427-428. orbital right cheek mass that extended into the mastica- 6. Wolf DA, Chaljub G, Maggio W, Gelman BB. Intracranial chondromyxoid tor space. The patient was referred to the M. D. Ander- fibroma: report of a case and review of the literature. Arch Pathol Lab Med. son Cancer Center for further management. The histologic 1997;121(6):626-630. sections of the original lesion
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