Clinical and Experimental Ophthalmology 2014; 42: 785–804 doi: 10.1111/ceo.12305

Letters to the Editor

Idiopathic dacryoadenitis mimicking a primary intraocular tumour in a young girl A 17-year-old Caucasian female was evaluated for an intraocular mass oculus sinister (OS) of 1-month duration with headache, periorbital pain and mild ptosis OS. Computed tomography (CT) of the brain was obtained and was normal. Another consultation prompted the diagnosis of an allergy treated with artificial tears and topical ketotifen drops with some improvement. One week before presenting to Massachusetts Eye and Ear Infirmary, she began seeing a dark shadow nasally OS. Funduscopic exam revealed an intraocular mass (Fig. 1a). Her past ocular history included a tennis ball trauma OS 1 year ago without any ocular sequelae. Her general history was unremarkable. On ophthalmic examination, her visual acuity was 6/6 oculus dexter and 6.0/7.5 OS; the intraocular pressures were 11 mmHg bilaterally. There were 2 mm of upper eyelid ptosis OS without proptosis. Slit-lamp biomicroscopy revealed a posterior polar cataract OS. Dilated funduscopic examination again displayed an elevated superotemporal mass OS (Fig. 1a). Fluorescein angiography showed early choroidal hypoperfusion in the area of the lesion (Fig. 1b) without any leakage. B-scan ultrasonography established that the lesion was 3.01 mm in height (Fig. 1c). Dynamic ultrasonography demonstrated that the lesion changed location in different fields of gaze. The suspicion for an extraocular mass therefore was high. A magnetic resonance imaging with gadolinium failed to separate the left orbital mass from the lacrimal gland, which, due to its firm character, was felt to have indented the globe (Fig. 1d). There was no bone destruction. The patient was then referred to the Oculoplastics Service. The lacrimal gland lesion was excised via lid crease orbitotomy with preservation of the orbital rim. The mass measured 2.2 × 1.8 × 0.9 cm and had a tan appearance and firm consistency (Fig. 1e,f). The surviving epithelial units of the lacrimal gland were entirely ductular encircled by collagen (Fig. 2a) and interstitial inflammatory cells. The lacrimal nerves and vessels were also enveloped by thick bundles of collagen (Fig. 2b). The periodic acid Schiff stain revealed stasis of secretions in some of the lumens (Fig. 2c). Cytokeratin immunohistochemical staining highlighted the surviving epithelial units (Fig. 2d). The inflammatory infiltrate was polymorphous, containing

small lymphocytes, plasma cells, pale staining histiocytes, and both polymorphonuclear and eosinophilic leukocytes (Fig. 2e). CD68/163 immunostain for histiocytes established their florid presence (Fig. 2f). The pathologic diagnosis was idiopathic dacryoadenitis. Unlike the current case, dacryoadenitis usually presents hyperacutely with conjunctival injection, chemosis, proptosis and frequently periorbital swelling and erythema.1–4 It represents an idiopathic, non-specific orbital inflammation. Lacrimal infection occurs due to viruses (EBV, Mumps, CMV, HSV and VZV) and more rarely, with bacteria such as Staphylococcus aureus and Streptococcus pneumonia.1,2 As with inflammations, CT usually shows an oblong diffuse enlargement of the lacrimal gland. Idiopathic sclerosing orbital inflammation represents 7.8% of all inflammatory lesions and requires immunomodulatory treatment in addition to corticosteroids.3 Its paediatric variant is usually responsive to corticosteroids.4,5 As in the current case, paediatric patients often have extensive eosinophilic leukocytic infiltration in their involved tissues and eosinophilic leukocytosis in the peripheral blood.5 The current patient had a very atypical presentation. She did not have any of the classic symptoms, but instead displayed a subacute evolution with mild ptosis and a mass in the left fundus with a correlating visual field defect. This constellation of symptoms raised clinical suspicion of a malignant epithelial lesion, notably of an adenoid cystic carcinoma. This tumour has a bimodal age distribution, with a major peak diagnosed around 40, and a smaller one found in the teenage years.6 It is important to carefully execute the clinical examination with multiple diagnostic modalities. Prompt surgical intervention should be undertaken to clarify the nature of a lesion with an atypical presentation.

Competing/conflicts of interest: No stated conflict of interest. Funding sources: No stated funding sources. © 2014 Royal Australian and New Zealand College of Ophthalmologists

Thanos D Papakostas MD,1 Frederick A Jakobiec MD DSc,2 Jason Mantagos MD,3 Alia Rashid MD,2 Aaron Fay MD4 and Demetrios G Vavvas MD PhD1 1 4 Retina Service, Oculoplastics Service, Department of Ophthalmology, 2David G. Cogan Laboratory of Ophthalmic Pathology, Massachusetts Eye and Ear Infirmary, and 3Department of Ophthalmology, Children’s Hospital, Harvard Medical School, Boston, Massachusetts, USA Received 21 January 2014; accepted 1 February 2014.


Letters to the Editor

Figure 1. Clinical, diagnostic and gross features of lacrimal gland mass.(a) Optos wide-angle colour fundus photograph showing the superotemporal elevated lesion (arrows). (b) Optos wide-angle fluorescein angiogram demonstrates early choroidal hypoperfusion in the area of the lesion (arrows) from compression and indentation caused by firm, enlarged lacrimal gland. (c) B-scan ultrasonogram found that the lesion measures 3.01 mm in height. (d) Axial (upper panel) and coronal (lower panel) magnetic resonance imaging (MRI), T1 sequence post-contrast showing an oblong enlargement of the lacrimal gland (crossed arrows) indenting the globe (uncrossed arrow). There is no adjacent bone destruction. (e) Gross appearance of the external aspect of the excised lesion displaying roughened, irregular surface. (f) On cut surface, a solid, pink-white lesion is well circumscribed by a pseudocapsule. The whiteness indicates the presence of collagen. LG, contralateral non-enlarged lacrimal gland. © 2014 Royal Australian and New Zealand College of Ophthalmologists

Letters to the Editor


Figure 2. Histopathologic and immunohistochemical features of lacrimal gland mass. (a) Surviving ductules of lacrimal gland parenchyma are surrounded by collagen sheaths (arrows). (b) Thickly textured collagen bands envelop peripheral nerve units (arrows) accounting for the patient’s periorbital pain and headache. (c) Many of the ductular units have periodic acid Schiff-positive luminal secretions (arrows), also highlighted in the inset. (d) Pancytokeratin immunostaining discloses irregular shapes of epithelial ductular remnants of the lacrimal gland (arrows). The crossed arrow indicates a collection of non-staining lymphocytes. (e) Polymorphous, non-granulomatous inflammatory infiltrate of the lacrimal gland is composed of lymphocytes, plasma cells (arrows), and pale staining histiocytes (crossed arrow) admixed with polymorphonuclear and eosinophilic leukocytes which occasionally formed microabscesses. The inset depicts a collection of eosinophils. (f) Myriad CD68-positive histiocytes were present among the chronic inflammatory cells. (a and b, Masson trichrome, ×200; c, Periodic acid Schiff, ×200, inset ×600; d, Immunoperoxidase reaction, ×40; e, Hematoxylin and eosin, main panel and inset ×400; f, Immunoperoxidase reaction, ×400). © 2014 Royal Australian and New Zealand College of Ophthalmologists



Letter To The Editor

REFERENCES 1. Jakobiec FA, Bilyk JR, Font RL. The orbit. In: Spencer WH, ed. Ophthalmic Pathology; An atlas and Textbook, Vol. 4. Philadelphia: Saunders WB, Saunder Co, 1996; 2810–58. 2. Harris GJ, Snyder RW. Lacrimal gland abscess. Am J Ophthalmol 1987; 104: 193–4. 3. Pemberton JD, Fay A. Idiopathic sclerosing orbital inflammation: a review of demographics, clinical presentation, imaging, pathology, treatment, and outcome. Ophthal Plast Reconstr Surg 2012 Jan-Feb; 28: 79–83. 4. Mottow LS, Jakobiec FA. Idiopathic inflammatory orbital pseudotumor in childhood. I. Clinical characteristics. Arch Ophthalmol 1978 Aug; 96: 1410–7. 5. Mottow-Lippa L, Jakobiec FA, Smith M. Idiopathic inflammatory orbital pseudotumor in childhood. II. Results of diagnostic tests and biopsies. Ophthalmology 1981 Jun; 88: 565–74. 6. Tellado MV, McLean IW, Specht CS, Varga J. Adenoid cystic carcinomas of the lacrimal gland in childhood and adolescence. Ophthalmology 1997 Oct; 104: 1622–5.

Surgical blues: vitreous staining with trypan blue during cataract surgery Trypan blue can be used to stain the anterior capsule of the eye during cataract surgery.1 In patients with a history of

zonular dehiscence, there have also been incidences of trypan blue inadvertently staining the vitreous.2,3 Herein, we describe a case of trypan blue staining the vitreous during cataract surgery in an eye with no signs of zonular dehiscence. A healthy 72-year-old woman with a history of bilateral laser peripheral iridotomies presented with a Snellen visual acuity of right 6/12 and left 1/36. She denied any history of ocular trauma. Clinical examination revealed a hypermature nuclear sclerotic cataract in the left eye, with no signs of pseudoexfoliation syndrome, phacodonesis or iridodonesis. Peripheral iridotomies were patent bilaterally. Fundal examination was unremarkable. The patient underwent left cataract surgery under peribulbar anaesthesia. A clear corneal incision was followed by gentle administration of trypan blue (1 mg/mL) into the anterior chamber, which was filled with an air bubble. After 10 s, the anterior chamber was irrigated with balanced saline solution, and sodium hyaluronate 1% (Provisc, Alcon Laboratories, Fort Worth, TX, USA) viscoelastic material was injected into the anterior chamber. Continuous curvilinear capsulorrhexis and hydrodissection was performed, and ‘divide and conquer’ phacoemulsification was used to remove the nucleus. During segment removal, it became apparent that no red reflex was visible. There was no evidence of choroidal haemorrhage or zonular dehiscence. No view of the fundus was possible with indirect ophthalmoscopy and the patient was left aphakic due to uncertainty of the capsule integrity. Day 1 postoperatively, the intraocular pressure was 20 mmHg. There was no relative afferent pupillary defect and B-mode ultrasound scan demonstrated an attached retina with no choroidal abnormality or signs of

Figure 1. Day 1 postoperatively shows a normal red reflex appearance in the right eye (right) and a blue fundal appearance due to trypan blue vitreous staining in the left eye (left).

Competing/conflicts of interest: No stated conflict of interest. Funding sources: No stated funding sources. © 2014 Royal Australian and New Zealand College of Ophthalmologists

Copyright of Clinical & Experimental Ophthalmology is the property of Wiley-Blackwell and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.

Idiopathic dacryoadenitis mimicking a primary intraocular tumour in a young girl.

Idiopathic dacryoadenitis mimicking a primary intraocular tumour in a young girl. - PDF Download Free
1MB Sizes 0 Downloads 0 Views