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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 55  |  Issue : 4  |  Page : 310-312

Management of isolated subconjunctival hemangioma masquerading as ocular surface squamous neoplasia


Department of Cornea and Refractive Surgery, The Eye Foundation, Coimbatore, Tamil Nadu, India

Date of Web Publication25-Apr-2018

Correspondence Address:
Dr. Shreesha Kumar Kodavoor
The Eye Foundation, Coimbatore, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/tjosr.tjosr_40_17

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  Abstract 


The purpose of this article is to report a case of isolated subconjunctival cavernous hemangioma, masquerading as ocular surface squamous neoplasia (OSSN) in a young male. A 40-year-old male presented with complaints of elevated vascular nodular lesions with feeder vessels in both eyes (right eye > left eye), in temporal limbus for the right eye, and in temporal bulbar conjunctiva for the left eye. Provisional diagnosis of both eyes' OSSN was made. Wide excisional biopsy with cryotherapy was performed for the right eye, and bare sclera was covered with conjunctival autograft. Histopathology report of the lesion showed subconjunctival cavernous hemangioma with no malignancy. The patient did not show any recurrence of lesion in the right eye at 1-year follow-up. Isolated conjunctival hemangioma is rare, but it can be considered as a differential diagnosis of vascular limbal nodules.

Keywords: Conjunctival autografting, conjunctival cavernous hemangioma, excision biopsy, ocular surface squamous neoplasia, ultraviolet rays


How to cite this article:
Kodavoor SK, Sarvate N. Management of isolated subconjunctival hemangioma masquerading as ocular surface squamous neoplasia. TNOA J Ophthalmic Sci Res 2017;55:310-2

How to cite this URL:
Kodavoor SK, Sarvate N. Management of isolated subconjunctival hemangioma masquerading as ocular surface squamous neoplasia. TNOA J Ophthalmic Sci Res [serial online] 2017 [cited 2019 Dec 15];55:310-2. Available from: http://www.tnoajosr.com/text.asp?2017/55/4/310/231129




  Introduction Top


Vascular tumors of the conjunctiva are not very common. They are divided into two groups – vascular tumors and vascular abnormalities – which include low flow (venous), high flow (arterial), and lymphatic lesions.[1] One example of such lesions is hemangioma which is a developmental vascular malformation, and another example is hamartoma which is an overgrowth of mature tissue that exists normally in the conjunctiva. Conjunctival hemangioma is nothing but the proliferation of blood vessels within the substantia propria. Ocular surface squamous neoplasia (OSSN) is a disease of the elderly [2] having predilection for the interpalpebral area – mostly the corneoscleral limbus [2] in majority of the cases. We report a rare case of a chronic, bilateral subconjunctival hemangioma in a young male which masqueraded as OSSN and which was successfully managed by wide excision biopsy with conjunctival autografting.


  Case Report Top


A 40-year-old male presented with complaints of redness associated with foreign body sensation in both eyes (right eye > left eye) for the past 5 years which was intermittent, recurrent, and not associated with any pain, watering, or discharge. He had no prior history of any eye surgery or trauma. He was a marketing executive by profession requiring prolonged traveling on a two-wheeler daily. There was a history of multiple treatment regimens, including artificial tears, topical nonsteroidal anti-inflammatory drugs, and topical corticosteroids which had been tried with limited success, and there was recurrence of redness after stopping the medications. He had no systemic illness, and there was no lymphadenopathy. On examination, his best-corrected visual acuity was 20/20 both eye and intraocular pressure was 11 and 10 mm of Hg in the right and left eye, respectively, measured by Goldmann applanation tonometry. On slit-lamp examination of the right eye, anterior segment showed a 7 mm × 4 mm × 3 mm elevated vascular nodular lesion within the temporal limbus with two large, dilated, tortuous feeder vessels at 7 and 11 o' clock position [Figure 1]a. Left eye anterior segment showed a smaller nodular lesion 1 mm away from limbus in the temporal bulbar conjunctiva measuring 3 mm × 4 mm × 1 mm with three large feeder vessels at 1, 4, and 5 o' clock, respectively [Figure 1]b. His anterior chamber [AC] was quiet and fundus examination was normal in both eyes. An ultrasound biomicroscopy was done in both eyes which showed no extension of the lesion to deep tissues such as sclera and ciliary body. The ELISA test for human immunodeficiency virus, done after taking patient's informed consent, was negative. On basis of history and clinical examination, provisional diagnosis of both eyes' OSSN was made and wide excision biopsy with conjunctival autografting was planned for the right eye and written informed consent was obtained for the same.
Figure 1: (a) Vascular raised lesion of temporal limbus in Right Eye (b) Raised conjunctival lesion in temporal bulbar conjunctive of Left Eye with feeder vessels

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He underwent uneventful surgery in the right eye by wide excision biopsy of the lesion with 2 mm of clear margins on the conjunctival side and 1 mm of clear margin on the corneal side by no-touch technique. After the excision, double-freeze cryotherapy was performed to the resected margins and scleral bed. Then, a tenon-free conjunctival autograft (CAG) was fashioned from superior bulbar conjunctiva and secured over the bare sclera using fibrin glue.

The excised lesion was sent for histopathological examination. Histopathology report showed a subconjunctival cavernous hemangioma with multiple vascular spaces, intense plasma cell infiltration around vessels, and keratinization of overlying conjunctiva, with no dysplasia or malignancy [Figure 2]. Postoperatively, the patient was given a course of topical antibiotic-steroid combination in tapering dose over 6 weeks, topical lubricants for 6 weeks. At 6-week follow-up of right eye, the patient was doing well with the CAG in situ [Figure 3]a. At 6-month follow-up, slit lamp examination showed a well-apposed graft [Figure 3]b. At 1-year follow-up, the right eye was absolutely quiet with no evidence of any recurrence.
Figure 2: Histopathology of Right Eye showing a subconjunctival hemangioma showing multiple vascular spaces without any dysplasia or malignancy

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Figure 3: (a) Right Eye postoperatively 6 months of follow–up showing graft in situ (b) 1–year follow–up showing a quiet eye with no recurrence of lesion

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  Discussion Top


The most common vascular lesions of the conjunctiva are capillary hemangioma, lymphangioma, and pyogenic granuloma.[3] On the other hand, isolated subconjunctival cavernous hemangioma is very rare, and very few cases had been previously reported. Capillary hemangioma is more common in pediatric age group and presents in early life with features of proptosis. Cavernous hemangioma on the other hand is seen commonly in adults (4th–5th decades) with a higher female preponderance and also presents commonly as proptosis involving the extraconal orbital space. To the best of our knowledge, no case of bilateral conjunctival cavernous hemangioma with purely palpebral involvement has been reported in the literature. In a series of 1643 conjunctival tumors, Shields et al. found only four (0.2%) cases with cavernous hemangioma.[3] Elsas and Green reported two (0.2%) case of cavernous hemangioma of conjunctiva among 302 epibulbar tumors in children collected over a 50-year period.[4] Aghaei and Shahraki [5] and Malik et al.[6] have reported a case of unilateral isolated conjunctival hemangioma in a young male and a elderly female, respectively. A case of cavernous conjunctival hemangioma managed by surgery also has been reported.[7] Majority of the patients presenting with such lesions are usually asymptomatic apart from few which complain of poor cosmesis or symptoms associated with tear film abnormality. However, Kiratli et al. have reported three patients with conjunctival cavernous hemangioma in association with multiple recurrent episodes of subconjunctival hemorrhage.[8]

With the history of chronicity of lesion, strong history of ultraviolet (UV) light exposure associated with patient's occupation, and clinical appearance, we made a provisional diagnosis of OSSN in both eyes, but the histopathology ruled it out. Chronic UV rays exposure associated with his occupation was probably the important risk factor for the development of these lesions in our case. During excision biopsy, utmost care was taken to remove the lesion in toto with clear margins, and there was no evidence of any recurrence seen at 1-year follow-up. Since the lesion in the left eye was smaller and the patient was relatively asymptomatic, it was managed conservatively and the patient was asked to follow-up periodically. What makes this case interesting is a rare bilateral presentation of subconjunctival cavernous hemangioma with clinical findings mimicking it as OSSN although the patient was young. The subconjunctival hemangiomas are rare vascular conjunctival lesions, and bilateral presentation is even rarer. Subconjunctival cavernous hemangioma can be considered as an important differential diagnosis of a vascular limbal nodule and has to be confirmed by histopathological examination. Since these neoplasms are benign, careful surgical excision is associated with no recurrence.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Shields CL, Shields JA. Tumors of the conjunctiva and cornea. Surv Ophthalmol 2004;49:3-24.  Back to cited text no. 1
[PUBMED]    
2.
Chen C, Louis D, Dodd T, Muecke J. Mitomycin C as an adjunct in the treatment of localised ocular surface squamous neoplasia. Br J Ophthalmol 2004;88:17-8.  Back to cited text no. 2
[PUBMED]    
3.
Shields JA, Mashayekhi A, Kligman BE, Kunz WB, Criss J, Eagle RC Jr., et al. Vascular tumors of the conjunctiva in 140 cases. Ophthalmology 2011;118:1747-53.  Back to cited text no. 3
    
4.
Elsas FJ, Green WR. Epibulbar tumors in childhood. Am J Ophthalmol 1975;79:1001-7.  Back to cited text no. 4
[PUBMED]    
5.
Aghaei H, Shahraki K. Isolated cavernous hemangioma of the conjunctiva: Case report and review of literature. Iran J Ophthalmol 2013;25:317-9.  Back to cited text no. 5
    
6.
Malik A, Bhala S, Arya SK, Narang S, Punia RP, Sood S, et al. Isolated cavernous hemangioma of conjunctiva. Ophthal Plast Reconstr Surg 2010;26:385-6.  Back to cited text no. 6
    
7.
Rao MR, Patankar VL, Reddy V. Cavernous haemangioma of conjunctiva (a case report). Indian J Ophthalmol 1989;37:37-8.  Back to cited text no. 7
[PUBMED]  [Full text]  
8.
Kiratli H, Uzun S, Tarlan B, Tanas Ö. Recurrent subconjunctival hemorrhage due to cavernous hemangioma of the conjunctiva. Can J Ophthalmol 2012;47:318-20.  Back to cited text no. 8
    


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  [Figure 1], [Figure 2], [Figure 3]



 

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