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| CASE REPORT |
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| Year : 2023 | Volume
: 3
| Issue : 2 | Page : 493-495 |
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Giant lacrimal mucocele in an adult – A case report
Haimanti Choudhury, Himadri Choudhury, Hirendra K Choudhury
Department of Cataract and Oculoplasty Services, Choudhury Eye Hospital and Research Centre, Shyamaprasad Road (Shillongpatty), Silchar, Assam, India
| Date of Submission | 24-Dec-2022 |
| Date of Acceptance | 21-Feb-2023 |
| Date of Web Publication | 28-Apr-2023 |
Correspondence Address:
Haimanti Choudhury
Choudhury Eye Hospital and Research Centre, Shyamaprasad Road (Shillongpatty), Silchar - 788 001, Assam
India
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/IJO.IJO_3346_22
Encysted lacrimal mucocele refers to diffuse, centrifugal enlargement of the lacrimal sac resulting from combined proximal and distal obstructions in lacrimal drainage system. Its presence in adults is an extremely rare event. The authors describe a case of giant lacrimal mucocele in an adult female presenting with only mild epiphora. The patient underwent aspiration of pus followed by external dacryocystorhinostomy (DCR). Patient's symptoms of epiphora resolved after surgery.
Keywords: Dacryocystorhinostomy, epiphora, medial canthal swelling, mucocele
How to cite this article:
Choudhury H, Choudhury H, Choudhury HK. Giant lacrimal mucocele in an adult – A case report. Indian J Ophthalmol Case Rep 2023;3:493-5 |
Lacrimal mucocele refers to diffuse, centrifugal enlargement of the lacrimal sac resulting from combined proximal and distal obstructions in a lacrimal drainage system.[1] In adults, it is secondary to inflammation, trauma, tumour, nasal surgery or dacryocystitis sequelae.[2] Clinical examination, lacrimal irrigation, computerized tomography and dacryocystography are used to diagnose the condition.[2] Lacrimal mucocele is treated with endoscopic or external dacryocystorhinostomy (DCR) with the placement of nasolacrimal stent. The authors report a case of giant lacrimal mucocele in an adult, idiopathic in nature, presenting with mild epiphora. Written informed consent was obtained from the patient for the publication of case details and photographs.
| Case Report | |  |
A 56-year-old female presented with mild epiphora and swelling in the inner corner of the left eye for 3 years. The swelling was small to begin with, and gradually increased in size. There was no history of dacryocystitis, trauma, surgery or discharge from swelling. On examination, nontender, nonmobile, subcutaneous mass at the medial canthus of about 4 × 3.5 × 2 cm was detected [Figure 1]. The regurgitation test was negative. Syringing showed blockage of a nasolacrimal duct with regurgitation of clear fluid from the opposite punctum. The best-corrected visual acuity in both eyes was 6/6. Rest ophthalmic examination was within normal. CT scan orbit revealed nonenhancing, low-density, well-defined, thin walled cystic lesion at the medial margin of the orbit. There were no signs of bony erosion. Features were consistent with idiopathic acquired lacrimal mucocele. | Figure 1: 56 year old female with medial canthal swelling causing narrowing of palpebral aperture of left eye
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After necessary preoperative investigations, the case was taken up for DCR. ENT consultation ruled out any nasal pathology. The surgery was performed under local anaesthesia. Prior to making the incision, mucopurulent fluid was aspirated with 10 cc syringe fitted with 18 G (gauge) needle. Around 15 ml aspirate was obtained. Standard external DCR was performed. A large part of the lacrimal sac flap was excised since the size was very big [Figure 2]. Intraoperative probing was done to confirm the patency of both canaliculi. Silicone intubation was not done as there was no canalicular block. She was started on systemic and topical antibiotics, and systemic anti-inflammatory in the postoperative period for one week. | Figure 2: Pieces of redundant lacrimal sac, excised prior to making of anatomosis
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Postoperatively the patient's swelling subsided [Figure 3]. Syringing was patent on the first postoperative day and was repeated after one week and one month. It was patent on all occasions. The sac aspirate was sent for microbiological evaluation. The culture was sterile after 72 h, and microscopic examination did not reveal any organism. | Figure 3: 56 year old female showing subsidence of medial canthal swelling one week after DCR
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| Discussion | | |
Lacrimal mucocele are non-neoplastic profuse enlargement of the lacrimal sac by mucopurulent material and results from combined proximal and distal obstructions at the level of the common canalicular opening and the nasolacrimal duct, respectively. These cases may or may not have epiphora on presentation and generally do not have any history of acute inflammation in the past. Imaging of mucocele shows fluid-filled structure with thin rim enhancement, without any solid internal structures.[3] It is necessary to differentiate the mucocele from the lacrimal sac tumour. Diagnosis is based on clinical examination, dacryocystography and computed tomography. An orbital tumour in the medial orbit such as haemangioma, lymphangioma, neurofibroma, or rhabdomysarcoma can be seen on CT as a solid mass.[4]
As mucocele is initially sterile, conservative management with topical antibiotic, digital massage and probing of nasolacrimal duct relieves occlusion in congenital cases, but dacyocystorhinostomy is the treatment of choice in adults.[5] In adults, nasolacrimal duct obstruction leads to chronic dacryocystitis, which in turn leads to epiphora, mucocele, recurrent acute on chronic dacryocystitis, pyocele and lacrimal fistula if left untreated.[6] The present case is unusual as the patient developed mucocele over a 3-year period, but never developed any attack of dacryocystitis.
A review of literature has revealed similar cases, but the management done in the present case is different from other reported cases. Perry LJ et al.[7] reported a giant dacryocystomucopyocele 4 × 5 cm in an adult woman, developed over 25 years and resulting in globe displacement. Kamath SJ et al.[8] reported an encysted lacrimal mucopyocele 2.7 × 2.5 × 1.2 cm in an adult male, secondary to chronic dacryocystitis. Krisnamurthy G et al.[9] reported an encysted mucocele of 3 × 3 cm with orbital extension. Pooja R Jain et al. reported the largest dacryocystomucopyocele (5.0 × 4.8 × 4.0 cm) developed over 15 months in an adult male.[6] All these cases were managed by complete cyst excision or dacryocystectomy (DCT). Anwar MJ et al.[2] reported an adult male with idiopathic acquired encysted dacryocystocele 2 × 2 cm, managed by external DCR with silicone tube intubation. Woo KI et al.[10] reported four cases of dacryocystocele (maximum size being 1.5 × 2.5 cm) which was managed with DCR combined with internal punctoplasty (2 cases), canaliculorhinostomy (1 case) and conjunctivodacryocystorhinostomy (1 case).
Surgical management of lacrimal mucocele includes external or endoscopic DCR with or without silicon tube intubation or complete surgical excision of the cystic sac (DCT).[6] But there is no consensus as to the best surgical approach as adult cases are relatively infrequent.[7] The authors feel that a well-performed DCR with meticulous anastomosis is sufficient to alleviate both epiphora and swelling. The excessively long sac flap needs to be trimmed to anastomose well with the appropriately fashioned nasal flap. Placement of silicon tube is only warranted in the event of documented canalicular obstruction. Surgical excision of the sac will inflict the subject to lifelong epiphora.
| Conclusion | | |
Medial canthal swelling with or without epiphora in adults may present a diagnostic dilemma. Lacrimal mucocele can be diagnosed with thorough clinical examination and syringing of nasolacrimal passage. CT scan plays a decisive role in differentiating such swelling from other masses or tumour. Dacryocystorhinostomy with or without silicon tube intubation offers the best surgical option.
Ethical approval
The study was approved by the institutional Ethics Committee.
Patient's consent form
Obtained.
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 | | |
| 1. | Berlucchi M, Tomenzoli D, Trimarchi M, Lombardi D, Battaglia G, Nicolai P. [Dacryocystocele in the adult: Etiology, diagnosis and treatment]. Acta Otorhinolaryngol Ital 2001;21:100-4.  |
| 2. | Anwar MJ, Choudhry SA, Aadil M, Asif A, Ameer A. Idiopathic acquired dacryocystocele presenting only with epiphora: A very rare case report. Cureus 2017;9:e1653. |
| 3. | Bothra N, Wagh RD, Ali MJ. Masquerades of acquired dacryocystocele. Clin Ophthalmol 2020;14:1855-8. |
| 4. | do Nascimento SB, Rodrigues AB, Jurity TP, de Sá JC, Castelo Branco AN. Lacrimal sac mucocele. Braz J Otorhinolaryngol 2014;80:540-1. |
| 5. | Plaza G, Nogueira A, González R, Ferrando J, Toledano N. Surgical treatment of familial dacryocystocele and lacrimal puncta agenesis. Ophthal Plast Reconstr Surg 2009;25:52-3. |
| 6. | Jain PR, Tiwari RP, Barot RK, Mane A, Bhatt VD. A rare case of giant dacryocystomucopyocele. J Clin Diagn Res 2021;15:ND01-2. |
| 7. | Perry LJ, Jakobiec FA, Zakka FR, Rubin PA. Giant dacryocystomucopyocele in an adult: A review of lacrimal sac enlargements with clinical and histopathologic differential diagnoses. Surv Ophthalmol 2012;57:474-85. |
| 8. | Kamath SJ, Saha D, Krishna U, Kolavala B. A large encysted lacrimal sac mucocele- A typical presentation. Int J Curr Res 2017;9:59920-2. |
| 9. | Krisnamurthy G, Padmavar BU, Desai Y. An encysted lacrimal mucocele with orbital extension. Indian J Opthalmol 1977;25:40-1. |
| 10. | Woo KI, Kim YD. Four cases of dacryocystocele. Korean J Ophthalmol 1997;11:65-9. |
[Figure 1], [Figure 2], [Figure 3]
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