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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 1  |  Issue : 4  |  Page : 736-738

Orbital venous malformation masquerading as lacrimal mucocele


Department of Oculoplasty, Indira Gandhi Eye Hospital and Research Centre, Lucknow, Uttar Pradesh, India

Date of Submission16-Jan-2021
Date of Acceptance09-Apr-2021
Date of Web Publication09-Oct-2021

Correspondence Address:
Dr. Nidhi Pandey
Department of Oculoplasty, Indira Gandhi Eye Hospital, 1 B. N. Road, Qaiserbagh, Lucknow - 226010, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_122_21

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  Abstract 

While mucocele of the lacrimal sac typical presents as a medial canthal lesion located below the medial canthal tendon, there may be an occasional simulating clinical condition. We present a 17-year-old patient with epiphora who had an orbital venous malformation masquerading as a lacrimal mucocele.

Keywords: Orbit, masquerade, lacrimal mucocele, venous malformation, epiphora


How to cite this article:
Pandey N. Orbital venous malformation masquerading as lacrimal mucocele. Indian J Ophthalmol Case Rep 2021;1:736-8

How to cite this URL:
Pandey N. Orbital venous malformation masquerading as lacrimal mucocele. Indian J Ophthalmol Case Rep [serial online] 2021 [cited 2021 Oct 18];1:736-8. Available from: https://www.ijoreports.in/text.asp?2021/1/4/736/327623



Lacrimal sac area lesions may simulate a mucocele.[1] Swelling below the medial canthal tendon is typically related to pathologies of the lacrimal sac origin. Benign or malignant lesions not related to lacrimal sac rarely present with swelling below the medial canthal tendon.[2] We report a case of venous malformation simulating a mucocele.


  Case Report Top


A 17-year-old male presented with a gradually progressive, painless swelling near the inner corner of the right eye and watering for the past 2 years. On examination, a diffuse, soft, partially compressible soft mass, measuring approximately 12 mm × 10 mm and indistinct posterior border was noted in the area of lacrimal sac [Figure 1]a. The uncorrected visual acuity of both eyes was 6/6, and the rest of the ocular and systemic examination was unremarkable. There was no regurgitation on pressing the lacrimal sac, however, the patient felt some fluid in the nose occasionally when the mass was pressed a few times. Syringing was patent. With differential diagnoses of atonic lacrimal sac, lacrimal sac diverticula, sinus mucocele, epidermal inclusion cyst, dacryolith, vascular mass, or a neoplasm in mind, a computed tomography (CT) dacryocystography was planned. It revealed dye only in the proximal part of the sac, and no lacrimal fossa mass was discernible [Figure 2]a and [Figure 2]b. A dacryocystorhinostomy (DCR) with bicanalicular intubation was planned to relieve functional epiphora and decompress the sac.
Figure 1: (a) Diffuse mass simulating a lacrimal sac mucocele; (b) postoperative 3 months follow-up

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Figure 2: CT dacryocystography showing dye in the proximal portion of lacrimal sac in a) axial and b) coronal section

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At the time of surgery, on separating the orbicularis oculi muscle fibers, a bluish-colored mass was noted, and it did not communicate with the lacrimal system on passing a metal probe through the puncta. The mass was non pulsatile on palpation and nondistensible when the patient was instructed to perform Valsalva maneuver. The surgical plan was changed to excision of the mass and biopsy. DCR was abandoned. Careful blunt separation from adjoining tissues was performed. The mass was found connected to angular vein and one minor vessel. The connection was ligated using 6-0 vicryl sutures as an additional step to prevent bleeding because the flow dynamics were not ascertained preoperatively. Cyanoacrylate glue was injected distal to the ligation point [Figure 3]a, [Figure 3]b, [Figure 3]c. The mass was then easily excised. It measured 20 mm in its maximum dimension, and histopathology (HP) revealed several dilated vascular spaces filled with organized blood consistent with a venous vascular malformation [Figure 4]a and [Figure 4]b. At 1 week follow-up, the skin around the incision site showed discoloration, but it faded away after 1 month. The epiphora was relieved [Figure 1]b. The lacrimal system remained patent, and the patient is doing well at 3 months postoperation follow-up [Figure 1]b.
Figure 3: (a) Bluish-colored mass noted after separating the orbicularis muscle (b) mass delineated from adjacent tissue by blunt dissection; (c) glue injection in the mass after ligation of vessel connections

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Figure 4: (a) Gross morphology showing an irregular mass measuring 20 mm in maximum dimension. (b) Histopathology: H & E–stained slide (40 × magnification) shows several irregularly dilated vascular spaces filled with organizing blood

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  Discussion Top


Vascular lesions of the orbit comprise 7% of all orbital pathologies.[3] Khan et al.[1] reported three cases of varix of angular vein presenting as lacrimal sac mucocele. No intervention was done as the lesions were small and asymptomatic. Nasr and Huaman[4] and Mudgil et al.[5] have described isolated varix in the area of lacrimal sac, increasing in size on Valsalva maneuver, treated with ligation and excision. ALSwaina and ALSuhaibani[6] and Imperial et al.[7] have reported low flow venous malformation presenting as lacrimal sac mucocele identified on imaging as a well-defined mass separate from lacrimal sac. The mass was excised and confirmed by histopathology (HP). In all previous case reports, the lesion was identified preoperatively by their flow characteristics and/or on imaging, and decision to observe or excise was based on the amount of symptoms the patients had [Table 1]. Assessing the flow dynamic of a vascular lesion is a critical first step in unraveling the etiology. Useful clinical exam techniques involve orbital palpation, ocular auscultation, and clinical observation of the lesion with and without Valsalva maneuver. Specific imaging modalities such as contrast-enhanced dynamic magnetic resonance imaging (MRI) along with a magnetic resonance angiography (MRA) help in assessing the flow dynamics of vascular lesion.[8] A simple CT scan if not done both in prone and supine posture will miss a low flow small nondistensible lesion, like in our case. The nature of the mass in our case did not conform to the clinical and imaging characteristics typical of a vascular lesion, and the complaint of epiphora along with the patient's response to fluid in the nose sometimes on pressing the mass repeatedly led to a misdiagnosis of the atonic sac. Peroperatively, the lesion was nondistensible and sans any signs of high flow; therefore, a careful approach to ligate vessel connection followed by glue embolization was done, and the mass could be excised completely. Venous malformations respond to sclerosant therapy and surgical excision with intraluminal glue application when indicated.[9],[10],[11] This case highlights the importance of meticulous clinical examination and specific imaging instructions to assess the etiology of lesions around the lacrimal sac area to prevent catastrophic complications of mismanagement of vascular malformations.
Table 1: Table comparing previously reported cases of vascular masses in lacrimal sac area with our case

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  Conclusion Top


Low-flow vascular mass in the lacrimal fossa area may simulate a mucocele. A thorough clinical examination and imaging to assess flow dynamics cannot be emphasized enough to diagnose such lesions and prevent peroperative surprises. On encountering such lesions, it is imperative to carefully assess vascular connections and follow a step-by-step approach to delineate it by ligation and glue injections before excision.

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.
Khan SR, Burton BJL, Beaconsfield M, Rose GE. The varix of angular vein. Eye 2004;18:645-7.  Back to cited text no. 1
    
2.
Stefanyszyn MA, Hidayat AA, Pe'er JJ, Flanagan JC. Lacrimal sac tumors. Ophthal Plast Reconstr Surg 1994;10:169-84.  Back to cited text no. 2
    
3.
Sundar G. Vascular lesions of the orbit: Conceptual approach and recent advances. Indian J Ophthalmol Case Rep2018;66:3-6.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Nasr AM, Huaman AM. Anterior orbital varix presenting as a lacrimal sac mucocoele. Ophthalmic Plast Reconstr Surg 1998;14:193-7.  Back to cited text no. 4
    
5.
Mudgil AV, Meyer DR, Dipillo MA. Varix of the angular vein manifesting as a medial canthal mass. Am J Ophthalmol 1993;116:245-6.  Back to cited text no. 5
    
6.
ALSwaina NF, ALSuhaibani AH. Low flow venous malformation lesion presented with medial canthal swelling simulating swelling of the lacrimal sac origin: A case report. Int J Health Sci (Qassim). 2015;9:185-8.  Back to cited text no. 6
    
7.
Imperial MT, Cheah E, Fong KS. An unusual clinical presentation of angular vein varix. Orbit 2006;25:141-3.  Back to cited text no. 7
    
8.
Stacey AW, Gemmete JJ, Kahana A. Management of orbital and periocular vascular anomalies. Ophthalmic Plast Reconstr Surg 2015;31:427-36.  Back to cited text no. 8
    
9.
Garcia DD, Heran MKS, Amadi AJ, Rootman J. Low outflow distensible venous malformations of the anterior orbit: Presentation, hemodynamic factors, and management. Ophthalmic Plast Reconstr Surg 2011;27:38-43.  Back to cited text no. 9
    
10.
Lin T, Zhu L, He Y. Clinical outcome observation of the embolization of orbital vascular malformation with medical glue under direct intra-operative view. BMC Ophthalmol 2018;18:330.  Back to cited text no. 10
    
11.
Bosch J, Garcia-Pagan JC. Prevention of variceal rebleeding. Lancet 2003;361:952-4.  Back to cited text no. 11
    


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