Solitary fibrous tumour of the lacrimal sac presenting with recurrent dacryocystitis

Solitary fibrous tumour of the lacrimal sac presenting with recurrent dacryocystitis

Correspondence recession-resection surgery on the horizontal rectus muscles. The cycles disappeared completely after surgery. Supported by: This work...

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Correspondence recession-resection surgery on the horizontal rectus muscles. The cycles disappeared completely after surgery.

Supported by: This work was supported by the Natural Science Foundation of Shandong Province ZR2009CM134.

Luxin Ma, Dong Kong, Zhaoshan Fan, Jie Zhao Provincial Hospital Affiliated to Shandong University, Jinan, P. R. China Correspondence to: Luxin Ma, MD: [email protected] REFERENCES

4. Pillai P, Dhand UK. Cyclic esotropia with central nervous system disease: report of two cases. J Pediatr Ophthalmol Strabismus. 1987;24:237-41. 5. Frenkel RE, Brodsky MC, Spoor TC. Adult-onset cyclic esotropia and optic atrophy. J Clin Neuroophthalmol. 1986;6:27-30. 6. Troost BT, Abel L, Noreika J, Genovese FM. Acquired cyclic esotropia in an adult. Am J Ophthalmol. 1981;91:8-13. 7. Uemura Y, Tomita M, Tanaka Y. Consecutive cyclic esotropia. J Pediatr Ophthalmol. 1977;14:278-80. 8. Muchnick RS, Sanfilippo S, Dunlap EA. Cyclic esotropia developing after strabismus surgery. Arch Ophthalmol. 1976;94:459-60. 9. Riordan-Eva P, Vickers SF, McCarry B, Lee JP. Cyclic strabismus without binocular function. J Pediatr Ophthalmol Strabismus. 1993;30:106-8. 10. Garg SJ, Archer SM. Consecutive cyclic exotropia after surgery for adult-onset cyclic esotropia. J AAPOS. 2007;11:412-3. 11. Di Meo A, Costagliola C, Della Corte M, Romano A, Foria C, Di Costanzo A. Adult-onset cyclic esotropia: a case report. Optom Vis Sci. 2013;90:e95-8.

1. Costenbader FD, Mousel DK. Cyclic Esotropia. Arch Ophthalmol. 1964;71:180-1. 2. Hutcheson KA, Lambert SR. Cyclic esotropia after a traumatic sixth nerve palsy in a child. J AAPOS. 1998;2:376-7. 3. Cole MD, Hay A, Eagling EM. Cyclic esotropia in a patient with unilateral traumatic aphakia: case report. Br. J. Ophthalmol. 1988;72:305-8.

Solitary fibrous tumour of the lacrimal sac presenting with recurrent dacryocystitis Solitary fibrous tumour is a rare spindle cell neoplasm that frequently arises in the pleura and mediastinum.1 This tumour has, however, been reported in numerous other locations and may be misdiagnosed because of variable clinical presentations and histologic appearances. We present a case of solitary fibrous tumour of the lacrimal sac. A 44-year-old female presented with excessive tearing and a slow-growing soft mass in the lacrimal sac fossa, just below the right medial canthal tendon, clinically consistent with a mucocele. She was treated with oral antibiotics in the past for a right dacryocystitis. She presented again in January 2014 with recurrent dacryocystitis. Examination confirmed the presence of a 1.5-cm smooth, firm mass. The left side and the remaining ophthalmic examination were unremarkable. Ultrasound revealed a hypoechoic, solid mass (1.5  1.0  0.9 cm). Increased vascular flow was noted via Doppler ultrasound (Fig. 1). CT scan revealed a discrete lesion in the lacrimal sac fossa consistent with a mucocele of the lacrimal sac. A tumour was not suspected based on clinical or imaging studies. There was no bone erosion. A right dacryocystorhinostomy, insertion of Crawford tube, and partial debulking of middle turbinate were performed. The lacrimal sac wall appeared thickened, consistent with chronic infection. Invasion into surrounding structures was not evident. Histopathologic examination revealed a moderately cellular, spindle cell neoplasm with prominent vascularity. The cells, admixed with thick collagen bands, were arranged in fascicle. The tumour cells exhibited minimal pleomorphism. Necrosis and mitotic figures were not identified (Fig. 1C).

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Can J Ophthalmol 2014;49:e107–e108 0008-4182/14/$-see front matter & 2014 Canadian Ophthalmological Society. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jcjo.2014.05.012

Immunohistochemically, the tumour cells were positive for vimentin and CD34, and were negative for S-100, smooth muscle actin, and desmin (Fig. 1D). A reticulin stain confirmed the presence of reticular fibres between tumour cells. The patient had uneventful postoperative recovery and was symptom free at 1 month of follow-up. Approximately 200 cases of lacrimal sac tumours have been reported, the majority being (76%) epithelial tumours.2,3 Malignant epithelial tumours are more common. Squamous papilloma is the most common benign tumour,4 whereas carcinomas (squamous cell, transitional, mucoepidermoid, or adenocarcinoma) are the most common malignant neoplasms.3,4 Nonepithelial tumours of the lacrimal sac, including fibrous histiocytoma,1,5 schwannoma,6 hemangiopericytoma,3 lipoma,1,2 angiosarcoma,7 neurilemmoma,8 and neurofibroma,9 have been reported. Solitary fibrous tumour, melanoma, granulocytic sarcoma, and lymphoma are rare.1,3 Clinically, the tumours often present with epiphora, recurrent dacryocystitis, mass, and sometimes with blood regurgitation.4 These rare neoplasms are prone to misdiagnosis as dacryocystitis, thereby delaying appropriate treatment.6 Solitary fibrous tumour has been described in numerous locations, including the upper respiratory tract, sublingual gland, thyroid gland, breast, periosteum, brain, and the orbit. Although once considered to originate from mesothelial cells, a nonmesothelial origin is now largely accepted.1 By ultrasonography, they often have welldefined borders and a homogeneous echostructure. Histologically, a hemangiopericytoma-like appearance and diffuse sclerosing patterns are the most common patterns.10 They are generally described as proliferation of blandlooking spindled to oval epithelioid cells that form short

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Correspondence

Fig. 1 — (A) Ultrasound with (B) Doppler showing hypoechoic, solid and vascular mass (1.5  1.0  0.9 cm). Note considerable vascular flow within the mass. Photomicrographs showing (C) spindle cell proliferation in fibrocollagenous background and prominent vascularity, characteristic of solitary fibrous tumour. Hematoxylin and eosin (H&E) staining. Original magnification 25. (D) Immunocytochemical analysis showing strong expression for CD34 in the tumour cells. Original magnification 25.

fascicles or clusters, admixed with collagen bands, and a prominent branching vasculature. Mature adipocytes and giant multinucleated stromal cells may be present. Mitoses rarely exceed 3/10 high power field. The tumour is subclassified into fibroblastic and myofibroblastic categories, based on smooth muscles actin and desmin expression. Ten to 15% of solitary fibrous tumours are aggressive. Malignancy is evidenced by hypercellularity, cytologic atypia, necrosis, infiltrative margins, and high mitoses.6,11 Such features were not present in this case. The histologic appearance may overlap with other benign and malignant tumours, such as leiomyoma, nodular fasciitis, inflammatory myofibroblastic tumour, fibromatosis, and peripheral nerve sheath tumour. Immunocytochemically, most solitary fibrous tumours express CD34, which is a sensitive marker for these tumours.12 In contrast, nerve sheath tumours, fibromas, and fibrous histiocytomas generally lack CD34 expression.1 The lack of desmin and S-100

differentiates solitary fibrous tumours from tumours of muscle or nerve sheath origin. Table 1 lists the differential reactivity of the spindle cell tumours to immunohistochemical stains. In our case, based on the histologic and immunocytochemical features, solitary fibrous tumour was the final diagnosis. In the past, some such tumours may probably have been described as hemangiopericytoma. To the best of our knowledge, only 2 cases of solitary fibrous tumour have been published in the literature. Clinically, continuing chronic dacryocystitis, bloody tears, rapid growth of a mass, exophthalmos, and radiologic evidence of bony destruction are important clinical signs for tumour aggressiveness.6 Large tumour size, high cellularity, numerous mitoses, extensive pleomorphism, and the presence of necrosis are pathologic signs of tumour aggression.1 Overall, Ni et al.6 found that a significant number of malignant lacrimal sac tumours directly extended into the orbit and nasolacrimal duct, whereas 1 case extended into

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Correspondence Table 1—Immunohistochemical profile of (cytokeratin-negative and vimentin-positive) spindle cell neoplasm of the orbit Spindle cell tumour

CD34

S100

SMA

Melan A

Solitary fibrous tumour Fibrous histiocytoma Fibroma Leiomyoma Spindle cell lipoma Melanoma Neurofibroma Schwannoma Angiosarcoma

þþþ – þ/– – þ – þ þ þþ

– – – – – þ þ þ –

þ/– þ/– – þ – – – – –

– – – – – þ – – –

the skull. Lymphatic invasion and distant metastases have also been reported.6 Our case presented no clinical, radiologic, or histologic evidence of aggressiveness. Most authors recommend complete removal of the lacrimal sac tumour.13 Some malignant tumours require postoperative radiation or chemotherapy.14 However, several such tumours (e.g., adenocarcinomas) are chemoresistant.6 Because solitary fibrous tumours are chemoresistant, total excision is the preferred treatment. In cases of partial resection, local recurrence or malignant transformation may occur.1 Although in the case of benign lacrimal sac tumours, complete surgical excision leads to a cure, the outcome of malignant tumours depends on the tumour stage, appropriateness of treatment, and the pathologic characterization of the tumour.6 Although early-stage tumours confined to the lacrimal sac have good prognoses, in advanced cases with tumours extending beyond the lacrimal sac, the prognoses are less favourable. The prognosis of solitary fibrous tumour is difficult to predict. The tumour may recur locally after incomplete excision.1 Malignant transformation and a large size are also associated with poor prognosis.14 Hence longterm follow-up in all cases is advisable.

Traumatic complete evulsion of the globe and optic nerve Evulsion means “extraction by force.” It is derived from the Latin verb vellere, meaning “to pull or pluck,” along with the prefix e, meaning “out.” Numerous cases of auto-enucleation have been previously reported in the psychiatric literature. However, evulsion of the globe as a result of trauma rarely has been described in the literature. We found only 14 previously documented patients in the English literature with evulsion of the eye as a result of injuries that were not selfinflicted. The documented mechanisms for evulsion include motor vehicle crash, eye pulled out by assailant, martial arts blow, train crash, falling onto corner of entertainment unit,

Presented as poster at the Canadian Ophthalmology Society Annual Meeting in Montreal, Que., June 14–17, 2013.

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Maher Kurdi, Larry Allen, Bret Wehrli, Subrata Chakrabarti Western University, London, Ont Correspondence to: Subrata Chakrabarti, MBBS: [email protected]

REFERENCES 1. Woo KL, Suh YL, Kim YD. Solitary fibrous tumor of the lacrimal sac. Ophthal Plast Reconstr Surg. 1999;15:450-3. 2. Flanagan JC, Stokes DB. Lacrimal sac tumours. Ophthalmology. 1978;85:1282-7. 3. Pe’er JJ, Stephanyszyn MA, Hidayat AA. Nonepithelial tumors of the lacrimal sac. Am J Ophthalmol. 1994;118:650-8. 4. Stephanyszyn MA, Hidayat AA, Pe’er JJ, Flanagan JC. Lacrimal sac tumors. Ophthalmic Plast Reconstr Surg. 1994;10:169-84. 5. Cole SH, Ferry AP. Fibrous histiocytoma (fibrous xanthoma) of the lacrimal sac. Arch Ophthalmol. 1978;96:1647-9. 6. Ni C, D’Amico DJ, Chi QF, Kuo PK. Tumors of the lacrimal sac: a clinicopathological analysis of 82 cases. Int Ophthalmol Clin. 1982;22:121-40. 7. Harry J, Ashton N. The pathology of tumors of the lacrimal sac. Trans Ophthalmol Soc UK. 1969;88:19-35. 8. Sen DK, Mohan H, Chatterjee PK. Neurilemmoma of the lacrimal sac. Eye Ear Nose Throat. 1971;50:56-7. 9. Filipowicz-Banachowa A, Sidorowicz E. Neurofibroma of the lacrimal sac. Klin Oczna. 1991;93:271-2. 10. Westra WH, Gerald WL, Rosai J. Solitary fibrous tumour. Am J Surg Pathol. 1994;18:281-7. 11. DeBacker CM, Bodker F, Putterman AM, Beckmann E. Solitary fibrous tumour of the orbit. Am J Opthalmol. 1996;121:447-9. 12. Lucas DR, Campbell RG, Fletcher CDM, et al. Solitary fibrous tumour of the orbit. Int J Surg Pathol. 1995;2:193-8. 13. Milder B. Neurofibroma of the lacrimal sac. Am J Ophthalmol. 1962;53:1016-8. 14. Hornblass A, Gabry JB. Diagnosis and treatment of lacrimal sac cysts. Ophthalmology. 1979;86:1655-61. Can J Ophthalmol 2014;49:e108–e110 0008-4182/14/$-see front matter & 2014 Canadian Ophthalmological Society. Published by Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jcjo.2014.05.011

and getting hit by a cow horn.1–10 This report details an additional case of traumatic evulsion of the globe. A 68-year-old male was referred from the emergency department to the on-call ophthalmology service. The patient reported falling out of bed and hitting his right orbit on the blunt stand of a floor lamp. He described no broken glass or damage to the lamp. On examination, right complete evulsion of the globe and optic nerve with sheath was noted. There was presence of a large orbital hematoma but no active bleeding (Fig. 1). We were unable to differentiate tissue or muscle clinically. His medical history included hypertension, pacemaker installation, and anticoagulant use. There was no history of psychiatric illness, psychiatric consultation, or medication use. Radiologic imaging showed no presence of an intraorbital foreign body (Figs. 2 and 3). The patient brought in a “clean” globe and nerve in a plastic freezer bag on ice (Fig. 4). The globe was then sent to pathology for a report. Pathologic record showed an

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