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Braz J Otorhinolaryngol. 2014;xxx(xx):xxx---xxx 1

Brazilian Journal of

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CASE REPORT 3

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Mucocele de saco lacrimal

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Lacrimal sac mucocele夽

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Silvia Bona do Nascimento a , Marcio Ricardo Barros Pio b , Ana Baleska Rodrigues a,∗ , Talline Priscila Magalhães Jurity a , Jéssica Coelho de Sá a , Anísio Neto de Oliveira Castelo Branco a a b

University Center (UNINOVAFAPI), Teresina, PI, Brazil Hospital Flávio Santos, Teresina, PI, Brazil

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Received 15 August 2012; accepted 15 December 2012

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Introduction

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A dacryocystocele is characterized by nasolacrimal duct (NLD) obstruction and a dilated lacrimal sac (LS). The lacrimal sac mucocele (LSM) supervenes when the LS becomes filled with mucopurulent material.1 Importantly, in addition to the character of the entrapped discharge, over a period of years the mucocele represents a chronic condition that causes bone erosion and remodeling with lacrimal sac expansion, and the formation of a cavity.1 This article aimed to describe this condition, which is rare in adults, by discussing the mechanisms of its formation, and its differential diagnosis and treatment.

Case report A 54-year-old female patient presented with a 12-year history of excessive tearing of the right eye. After four years, she noticed a progressive obstruction of the right nasal cavity which over the last two years, also involved the left



Please cite this article as: do Nascimento SB, Barros Pio MR, Rodrigues AB, Magalhães Jurity TP, de Sá JC, de Oliveira Castelo Branco AN. Lacrimal sac mucocele. 2014. Braz J Otorhinolaryngol. http://dx.doi.org/10.1016/j.bjorl.2014.05.001 ∗ Corresponding author. E-mail: [email protected] (A.B. Rodrigues).

nasal cavity. In the last year, a tender bulging near the canthus of the right eye could be noted. She had sought medical care many times for this condition; oral antibiotics were prescribed and a transient improvement was achieved. The patient had no comorbidities. On physical examination, she presented with right eye epiphora and a 1.2 cm diameter cystic lesion, next to the medial canthus of the right eyeball; the overlying skin was erythematous (Fig. 1A). No drainage could be made from the lesion or through the lacrimal canaliculi. Anterior rhinoscopy revealed total obstruction of the right nasal cavity with vestibule stenosis, and a reduced left nasal cavity lumen from a septal deviation. Additionally movement of the right medial rectus muscle was limited. The remainder of the ENT examination showed no abnormalities. Paranasal sinus tomography showed a soft-tissue density lesion occupying the right ethmoid and maxillary sinuses and the nasal cavity, with erosion of the right lamina papyracea with ipsilateral compression of the medial rectus muscle and eyeball displacement. Additionally, there was nasal septal erosion and displacement into to the left nasal cavity, partially obliterating its lumen consistent with a mucocele (Fig. 1B). Surgical treatment was elected. During surgery, a cystic lesion filled with mucopurulent material arising from the ipsilateral lacrimal sac was found, as demonstrated by right lacrimal duct probing. Since the mucocele occupied the entire right nasal cavity, rupture was inevitable, and marsupialization into the nasal cavity was performed. At a six-month postoperative follow-up, the patient was symptom-free.

1808-8694/© 2014 Associac ¸ão Brasileira de Otorrinolaringologia e Cirurgia Cérvico-Facial. Published by Elsevier Editora Ltda. All rights reserved.

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Figure 1 (A) Macroscopic appearance of the lesion in the medial region to the eyeball. (B) Computed tomography (CT) of paranasal sinuses, coronal section.

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Discussion Although the true incidence of LSM is not known, it is a clinical condition rarely affecting adult individuals.2 It is characterized by a collection of lacrimal discharge at the medial canthus of the eye due to nasolacrimal duct obstruction, initially producing a dacryocystocele, but with subsequent infectious and inflammatory processes changing the character of the entrapped material, to mucoid or mucopurulent.3,4 Even at the mucocele stage, the clinical features are associated with pain only in infectious flareups.3 On physical examination, LSM is characterized by a solid mass at the medial canthus, whether or not associated with cellulitis of the overlying skin.1,3 In adulthood, LSM usually results from chronic acquired obstruction of NLD1,2 and secondary blockage of its canaliculi, thus preventing tear secretion reflux via several mechanisms.3,5 The first mechanism involves mucosal swelling resulting from chronic inflammation around the valve of Rossenmuller, which might prevent reflux drainage from the sac. In addition, LS lateral strain compresses canaliculi and might also contribute to compress and seal the lower lacrimal punctum.3 It is now classified as a dacryocystocele. When its content is infected, resulting fibrosis often completely occludes backward tear drainage from the LS. Thus, a cystic purulent collection is formed, a mucocele or mucopyocele.2 The acquired NLD obstruction, in the genesis of the process, might be due to chronic infections, (mainly caused by bacteria and fungi of low pathogenicity),1,6 dacryoliths,7 anatomical changes, such as abnormal ethmoid cells, facial fractures, or complications of nasal surgery.1 A few drugs, such as the chemotherapeutic agents docetaxel and fluorouracil or I-131 have also been implicated in NLD stenosis.1,8 Rarer causes include Stevens-Johnson syndrome, Wegener’s granulomatosis, sarcoidosis,1,6 and inflammatory bowel disease,9 in addition to herpetic10 and chlamydial infections.11 Nasolacrimal sac or duct neoplasms, or those found in adjacent structures are exceedingly rare causes of NLD obstruction although they are more serious. Their rarity questions the usefulness of routine biopsy during

dacryocystorhinostomy.12 Since malignant neoplasms promote bone destruction, they are unlikely to produce an obstruction resulting in LSMs.1 Regardless of the causal factor, the acquired NLD obstruction is more common in adult females.7 As observed in the present case, the inflammatory and infectious process is prone to extend to and involve the overlying skin in LSMs; therefore spontaneous drainage is possibility, similar to the rupture of any abscess.1 Facial bone erosion is another possible long-term complication as demonstrated in the present case. Initially, bone destruction is due to inflammatory mediators, such as prostaglandin E2 and collagenases, produced by fibroblasts in the mucocele capsule.2 Subsequently, the pressure effect of the mucocele on surrounding walls also leads to bone destruction and remodeling.2 There is no consensus in the literature about the need of imaging studies for diagnosis.1---3 The macroscopic aspect of the lesion in the present case and other reported cases1 truly favors LSM as the clinical entity. However, imaging is essential if a different LS cause is suspected, in patients with significant nasal comorbidities, or when a thorough assessment with high resolution paranasal sinus, orbital, and nasal imaging is desired for preoperative planning.2 Computed tomography (CT) of paranasal sinuses is the first-line imaging modality, and any diagnostic questions should be resolved using magnetic resonance imaging (MRI). MRI is mainly valuable in the diagnosis of LS tumors, which are often malignant and may mimic a mucocele on CT.2 Differential diagnosis of LSM should consider dacryocystitis, lacrimal sac diverticulum, encephalocele, ethmoidal or maxillary mucocele, dermoid or epidermoid cysts, and benign or malignant neoplasms of LS or contiguous structures.1---12 Symptoms leading to a suspected neoplasm include a lesion found above the medial canthal ligament, severe pain, bloody discharge from lacrimal canaliculi, palpable mass around the LS, and bone destruction.1---12 As already highlighted, these lesions might present with repeat dacryocystitis, while the lacrimal system patency is still maintained.12 Up to 40% of patients with lacrimal sac tumors

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have a delay in diagnosis because the symptoms are similar to those resulting from lacrimal system obstruction by inflammation.12 This fact, on its own, warrants early imaging investigation of these cases. LSM treatment is more often performed by the ophthalmologist. However, the condition can extend into the nasal cavity, as in the present case, thus requiring an otorhinolaryngologist.13 LSM management can be surgical or conservative. In the adult, the first-line treatment is surgical, since the conservative treatment is often ineffective, and spontaneous regression is rare.5 Despite the reports of mucocele rupture into the nasal cavity and healing of the condition,2,4 most authors prefer the surgical approach since the spontaneous healing rate is likely under 2%.2 In addition to dacryocystorhinostomy, the use of a polyurethane stent in the nasolacrimal duct has been described, with satisfying long-term results.3 In the present case, dacryocystorhinostomy with mucocele marsupialization was chosen and achieved satisfactory long-term results.

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Final comments

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Despite its infrequent occurrence, adult LSM is more often a complication of chronic dacryocystitis caused by chronic NLD obstruction and secondary blockage of canaliculi. The first choice for evaluation is paranasal sinus computed tomography and the indicated treatment is surgical --- a dacryocystorhinostomy.

Conflicts of interest The authors declare no conflicts of interest.

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References

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1. Perry LJP, Jakobiec FA, Zakka FR, Rubin PAD. Giant dacryocystomucopyocele in an adult: a review of lacrimal sac enlargements with clinical and histopathologic differential diagnoses. Surv Ophthalmol. 2012;57:474---85. 2. Katarzyna EK, Harpreet A. Chronic dacryocystitis with spontaneous resolution of sac mucocele: fact or fiction. Ophthal Plast Reconstr Surg. 2011;27:e90---2. 3. Xiao MI, Tang LS, Zhu H, Li HJ, Li HL, Wu XR. Adult nasolacrimal sac mucocele. Ophthalmologica. 2008;222:21---6. 4. Yip CC, McCulley TJ, Kersten RC, Bowen AT, Alam S, Kulwin DR. Adult nasolacrimal duct mucocele. Arch Ophthalmol. 2003;121:1065---6. 5. Woo KI, Kim YD. Four cases of dacryocystocele. Kor J Ophthalmol. 1997;11:65---9. 6. Lindberg JV, McCormick SA. Primary acquired nasolacrimal duct obstruction. A clinicopathologic report and biopsy technique. Ophthalmology. 1986;93:1055---63. 7. Yazici B, Hammad AM, Meyer DR. Lacrimal sac dacryoliths predictive factors and clinical characteristics. Ophthalmology. 2001;108:1308---12. 8. Fard-Esfahani A, Farzanefar S, Fallahi B, Beiki D, Saghari M, Emami-Ardekani A, et al. Nasolacrimal duct obstruction as a complication of iodine-131 therapy in patients with thyroid cancer. Nucl Med Commun. 2012;33:1077---80. 9. Satchi K, McNab AA. Lacrimal obstruction in inflammatory bowel disease. Ophthal Plast Reconstr Surg. 2009;25:346---9. 10. Harley RD, Stefanyszyn MA, Apt L, Nelson LB. Herpetic canalicular obstruction. Ophthal Surg. 1987;18:367---70. 11. Tabbara KF, Bobb AA. Lacrimal system complications in trachoma. Ophthalmology. 1980;87:298---301. 12. Lee-Wing MW, Ashenhurst ME. Clinicopathologic analysis of 166 patients with primary acquired nasolacrimal duct Obstruction. Ophthalmology. 2001;108:2038---40. 13. Jin H-R, Shin S-O. Endoscopic marsupialization of bilateral lacrimal sac mucoceles with nasolacrimal duct cysts. Auris Nasus Larynx. 1999;26:441---5.

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