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 Table of Contents    
CASE REPORT
Year : 2020  |  Volume : 13  |  Issue : 1  |  Page : 43-45  

Intralesional bleomycin: A treatment modality for conjunctival lymphangioma


Department of Oculoplasty, Dr. Shroff's Charity Eye Hospital, New Delhi, India

Date of Submission11-Feb-2019
Date of Decision15-Sep-2019
Date of Acceptance16-Dec-2019
Date of Web Publication17-Feb-2020

Correspondence Address:
Dr. Smriti Bansal
Department of Oculoplasty, Dr. Shroff's Charity Eye Hospital, Daryaganj, New Delhi
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ojo.OJO_44_2019

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   Abstract 


Lymphangioma is a benign hamartomatous lesion of vascular system which frequently involves orbit and ocular adnexa. A 35-year-old male patient presented with a subconjunctival mass extending from limbus to fornices. There was a history of recurrent swellings following episodes of upper respiratory tract infection. Bleomycin is an antineoplastic agent which acts on endothelial cells of the vascular channels, helps in collapsing the cystic cavity, and prevents its recurrence. We report a case where only bleomycin injection has been used intralesionally at multiple spots and patients have shown good cosmetic outcome without any recurrence noted after 1 year of follow-up.

Keywords: Bleomycin, conjunctival, intralesional, lymphangioma


How to cite this article:
Bansal S, Das S. Intralesional bleomycin: A treatment modality for conjunctival lymphangioma. Oman J Ophthalmol 2020;13:43-5

How to cite this URL:
Bansal S, Das S. Intralesional bleomycin: A treatment modality for conjunctival lymphangioma. Oman J Ophthalmol [serial online] 2020 [cited 2020 May 30];13:43-5. Available from: http://www.ojoonline.org/text.asp?2020/13/1/43/278556




   Introduction Top


Lymphangioma is a vascular malformation involving veno-lymphatic system. These are rare entities and benign in nature. Orbital lymphangioma represents 0.3%–4% of all orbital tumors.[1] These can be classified as deep orbital lymphangioma or superficial lymphangioma or can be combined depending on the location of the mass. Deep orbital lymphangioma presents itself with restriction of extraocular motility due to spontaneous bleeding in them, whereas surface lymphangioma can present as subconjunctival mass.[2]

Conjunctival lymphangioma represents the superficial component of lymphangioma and can be present rarely in isolated form. It mostly presents as unilateral case but can be seen bilaterally in association with syndromes such as Turner's syndrome or Nonne–Milroy–Meige disease.[3] Patients commonly present after an episode of hemorrhage within the lesion following the formation of chocolate cyst after trauma or due to an episode of upper respiratory tract infection.[2]

Bleomycin is an antineoplastic agent, now being used as a sclerotherapeutic agent in such arteriovenous malformations.[3] The reported case is of conjunctival lymphangioma which was treated conservatively with intralesional bleomycin only, without surgical excision. Only three cases of conjunctival lymphangioma have been reported so far, where excision of the mass was adopted as a primary treatment modality and it is the first case where only intralesional sclerotherapy has been used.[4],[5],[6]


   Case Report Top


A 35-year-old male with complaints of a red-colored mass in the right eye since childhood reported to our institution. He gave a history of recurrent swelling in the right eye following episodes of upper respiratory tract infections. He did not have any other systemic illness. On ocular examination, his best-corrected visual acuity was 20/20 in both eyes for distance and N6 for near. Anterior- and posterior-segment examinations were within normal limits in both the eyes. On external examination, a red-colored subconjunctival mass was present extending from limbus to fornices, involving three-fourth quadrants from 11'o clock to 7'o clock [Figure 1]a [Figure 1]b [Figure 1]c [Figure 1]d. It was associated with mild upper eyelid ptosis due to mechanical effect of the mass and fullness below the lower lid medially obscuring the tear trough. Ocular motility was full and free in all gazes. Magnetic resonance imaging was done to confirm the diagnosis and to assess the posterior extent of the lesion. It showed a multiloculated cystic lesion involving anterior part of the orbit which was isointense on T1 and hyperintense on T2 [Figure 1]e and [Figure 1]f. Based on clinical and imaging findings, the diagnosis of an orbital lymphangioma was confirmed.
Figure 1: (a-d) The right eye of the patient showing a subconjunctival cystic red-colored mass extending from 11'o clock to 7'o clock in primary, inferior, superior, and lateral gaze with mild mechanical ptosis and lower lid fullness. (e and f) Magnetic resonance imaging of the same patient showing multiloculated occupying anterior orbit and isointense on T1 and hyperintense on T2, respectively

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It was decided to take a conservative approach, and therefore, intralesional bleomycin was injected taking all aseptic precautions. Bleomycin comes in a lyophilized powder form in a concentration of 15 units in a single vial. This was diluted with 3 ml of sterile water which making it as 5 units/ml (equivalent to 5 mg/ml). One milliliter of this reconstituted bleomycin was then further diluted with lignocaine and water (4:1) making it as 1 unit (mg)/ml.[7] The recommended dose is 0.5 mg/kg, and the total cumulative dose should not exceed 5 ml at any particular time.[8] The patient received injection bleomycin three times, at an interval of 1 month each. On the first visit, he was injected four units in the inferior quadrant at multiple sites with a 30G hypodermic needle, and the response was assessed. Following this, subsequently, four units in the superior quadrant, and finally, two units near the caruncular area were injected on the second and third visits. A total cumulated dose of ten units was injected over a period of 3 months [Figure 2].
Figure 2: (a-d) The right eye of the patient showing complete fibrosis of the lesion in primary, inferior, superior, and lateral gaze, respectively, after 6 months of giving injection bleomycin

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   Discussion Top


Lymphangiomas are benign hamartomatous malformations involving the lymphatic system which remains undetected till bleeding occurs in the cystic spaces, leading to either proptosis or ptosis with extraocular motility restriction. Most of the cases present after an episode of upper respiratory tract infection or minor trauma.[2]

Isolated conjunctival lymphangioma is a rare case which presents as either a subconjunctival mass or a nodule with an average age of presentation at 25 years of age.[9] They represent superficial component of orbital lymphangiomas. Most of the patients present after an episode of hemorrhage in these malformations, leading to restriction in extraocular motility. Rootman et al. reported a single case of conjunctival lymphangioma in their series of orbital lymphangioma.[6] Seca et al. and Raj et al. have reported cases of conjunctival lymphangioma of a 55-year-old and a 60-year-old patients, in whom complete excision was adopted as a treatment modality.[4],[5] In both cases, the lesion was restricted to inferior quadrant and was amenable for excision.[4],[5] In this case, the lesion was occupying three-fourth of the quadrant which was difficult to excise.

Bleomycin is a chemotherapeutic agent derived from Streptomyces verticillus and was first used in cases of pleural effusion and other vascular anomalies.[10] It also has antimicrobial and antiangiogenic properties, but the basic mechanism of action in vascular anomalies is due to its sclerosing effect on the endothelium of the abnormal vasculature when injected intralesionally. Intralesional bleomycin has been effectively used as a treatment modality for orbital lymphangiomas.[10] Gooding andMeyer also showed its effectiveness in refractory deep orbital lymphangiomas where all patients showed favorable clinical outcomes.[11] Bleomycin injections have also been used as an effective treatment of basal cell carcinoma and Kaposi sarcoma.[3] There are reports of using bleomycin in cases of refractory periocular capillary hemangiomas too.[12]

This case presented with a large subconjunctival mass since childhood extending from limbus to fornices, with a recurrent history of swelling and chemosis, following episodes of the upper respiratory tract infections. Surgical excision was not possible due to the extent of the lesion. Bleomycin was injected at multiple sites under topical anesthesia over a period of 3 months with an interval of 4 weeks. The effect of bleomycin was assessed before giving the next injection. After 1 year of follow-up, the subconjunctival mass was completely fibrosed with a total cumulated dose of 10 units, and there was no recurrence reported. The patient was satisfied with a good cosmetic outcome.


   Conclusion Top


Intralesional bleomycin is a good alternative to surgical excision in cases of surface lymphangiomas where it is not possible to remove the entire mass, and the patient is also concerned about cosmesis. It can also be used as an adjunct therapy to surgical debulking, where continuous negative pressure prevents chances of 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.
Russin JJ, Rangel-Castilla L, Kalani YS, Spetzler RF. Surgical management, outcomes and recurrence rate of orbital lymphangiomas. JSM Neurosurg Spine 2014;2:1030.  Back to cited text no. 1
    
2.
Tunç M, Sadri E, Char DH. Orbital lymphangioma: An analysis of 26 patients. Br J Ophthalmol 1999;83:76-80.  Back to cited text no. 2
    
3.
Meyer D, Gooding C. Intralesional bleomycin as an adjunct therapeutic modality in eyelid and extraocular malignancies and tumors. Middle East Afr J Ophthalmol 2015;22:410-4.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Seca M, Borges P, Reimão P, Gomes M, Meireles A. Conjunctival lymphangioma: A case report and brief review of the literature. Case Rep Ophthalmol Med 2012;2012:836573.  Back to cited text no. 4
    
5.
Raj A, Nagpal RC, Harsh M, Bahadur H. Conjunctival lymphangioma: A unique case report and review of literature. JCOR 2017;5:142-44.  Back to cited text no. 5
    
6.
Rootman J, Hay E, Graeb D, Miller R. Orbital-adnexal lymphangiomas. A spectrum of hemodynamically isolated vascular hamartomas. Ophthalmology 1986;93:1558-70.  Back to cited text no. 6
    
7.
Raichura ND, Alam MS, Noronha VO, Mukherjee B. A prospective study of the role of intralesional bleomycin in orbital lymphangioma. J AAPOS 2017;21:146-51.  Back to cited text no. 7
    
8.
Kumar V, Kumar P, Pandey A, Gupta DK, Shukla RC, Sharma SP, et al. Intralesional bleomycin in lymphangioma: An effective and safe non-operative modality of treatment. J Cutan Aesthet Surg 2012;5:133-6.  Back to cited text no. 8
[PUBMED]  [Full text]  
9.
Jones IS. Lymphangiomas of the ocular adnexa. An analysis of sixty-two cases. Am J Ophthalmol 1961;51:481-509.  Back to cited text no. 9
    
10.
Lee KH, Han SH, Yoon JS. Successful treatment of orbital lymphangioma with intralesional bleomycin and application of continuous negative pressure. Korean J Ophthalmol 2015;29:70-2.  Back to cited text no. 10
    
11.
Gooding C, Meyer D. Intralesional bleomycin: A potential treatment for refractory orbital lymphangiomas. Ophthalmic Plast Reconstr Surg 2014;30:e65-7.  Back to cited text no. 11
    
12.
Smit DP, Meyer D. Intralesional bleomycin for the treatment of periocular capillary hemangiomas. Indian J Ophthalmol 2012;60:326-8.  Back to cited text no. 12
  [Full text]  


    Figures

  [Figure 1], [Figure 2]



 

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