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Adult dacryocystocele

Abstracts

The dacryocystocele represents a dilated lacrimal sac and is often considered as having a congenital etiology. However, dacryocystocele is a rare disease in adults. The clinical feature is characterized by a painless bulge in the medial region of the orbit, below the medial canthal ligament. The lacrimal excretion test, endoscopy, computerized tomography and magnetic resonance image are used to make the diagnosis of dacryocystocele. Similar to pediatric patients, endoscopic marsupialization of the cyst with nasal stenting seems to be the appropriate therapy. In some cases the treatment consists of external dacryocystorhinostomy.

Orbit; Nasolacrimal duct obstruction; Dacryocystocele; Dacryocystorhinostomy


A dacriocistocele representa um saco lacrimal dilatado e é frequentemente considerada como tendo uma etiologia congênita. No entanto, dacriocistocele é uma doença rara em adultos. A característica clínica se caracteriza por um abaulamento indolor na região medial da órbita, inferior ao ligamento cantal medial. O teste de excreção lacrimal, endoscopia, tomografia computadorizada e ressonância magnética são utilizados para fazer o diagnóstico de dacriocistocele. Semelhante a pacientes pediátricos, marsupialização endoscópica do cisto nasal e a colocação de stent parece ser a terapêutica adequada. Em alguns casos o tratamento consiste na dacriocistorrinostomia externa


INTRODUCTION

Dacryocystocele is characterised by a dilated lacrimal sac and is often regarded as a congenital condition. However, it is a rare disease in adults. It presents as a painless bulge in the medial region of the orbit below the medial ligament. Its mechanism involves an acquired obstruction at the level of Krause’s valve and obstruction proximal to the common canaliculus, at the level of Rosenmuller’s valve.(1Lai PC, Wang JK, Liao SL. A case of dacryocystocele in an adult. Jpn J Ophthalmol. 2004;48(4):419-21.

Xiao MY, Tang LS, Zhu H, Li YJ, Li HL, Wu XR. Adult nasolacrimal sac mucocele. Ophthalmologica. 2008;222(1):21-6.
-3Ansari SA, Pak J, Shields M. Pathology and imaging of the lacrimal drainage system. Neuroimaging Clin N Am. 2005;15(1):221-37.)

In a patient with nasal obstruction, the differential diagnosis includes masses such as haemangioma, glioma, encephalocele, and dermoid cyst, as well as congenital bone anomalies such as choanal atresia.(4Lelli GJ, Levy RL. Epidermoid cyst masquerading as dacryocystocele: case report and review. Orbit. 2011;30(2):114-5.,5Debnam JM, Esmaeli B, Ginsberg LE. Imaging characteristics of dacryocystocele diagnosed after surgery for sinonasal cancer. AJNR Am J Neuroradiol. 2007;28(10):1872-5.)

The tear secretion test, endoscopy, computed tomography, and magnetic resonance imaging can be used to diagnose the condition.

Dacryocystocele in adults should be considered as an obstruction of the nasolacrimal duct, and intranasal examination should be performed before external dacryocystorhinostomy.(6Weber AL, Rodriguez-DeVelasquez A, Lucarelli MJ, Cheng HM. Normal anatomy and lesions of the lacrimal sac and duct: evaluated by dacryocystography, computed tomography, and MR imaging. Neuroimaging Clin N Am. 1996;6(1):199-217.

Berlucchi M, Tomenzoli D, Trimarchi M, Lombardi D, Battaglia G, Nicolai P. [Dacryocystocele in the adult: etiology, diagnosis and treatment]. ActaOtorhinolaryngol Ital. 2001;21(2):100-4.
-8Day S, Hwang TN, Pletcher SD, Bhatki A, McCulley TJ. Interactive image- guided endoscopic dacryocystorhinostomy. Ophthal Plast Reconstr Surg. 2008;24(4):338-40.) Similar to paediatric patients, endoscopic marsupialization of the nasal cyst with stent placement seems to be the appropriate therapeutic approach. Some cases can be treated with external dacryocystorhinostomy.(9Plaza G, Nogueira A, González R, Ferrando J, Toledano N. Surgical treatment of familial dacryocystocele and lacrimal puncta agenesis. Ophthal Plast Reconstr Surg. 2009;25(1):52-3.

10 Song HY, Lee DH, Ahn H, Kim JH, Kang SG, Yoon HK, et al. Lacrimal system obstruction treated with lacrimal polyurethane stents: outcome of removal of occluded stents. Radiology. 1998;208(3):689-94.
-1111 Yazici B, Yazici Z, Parlak M. Treatment of nasolacrimal duct obstruction in adults with polyurethane stent. Am J Ophthalmol. 2001;131(1): 37-43.)

CASE REPORT

A white, 42-year-old woman presented with epiphora in the right eye and progressive ipsilateral nasal congestion starting 4 months earlier. She had no history of epistaxis, facial fracture, or sinus and nasal disorders. Ophthalmic examination found a soft mass in the medial region of the right orbit (Figure 1). Biomicroscopy showed no hyperaemia in the bulbar conjunctiva and no anterior chamber reaction. The Milder test was intensely positive. Catheterisation found a soft stop after introducing the Bowman probe 9 mm into the canaliculus. The Jones 1 test was negative. Examination of the left eye was normal, including eyelid position. Right anterior rhinoscopy found no mass below the inferior turbinate.The patient was prescribed prophylactic antibiotic therapy and cold packs, and the dacryocystocele resolved after 2 weeks, progressing to chronic dacryocystitis. The patient will undergo external dacryocystorhinostomy.

Figure 1
Frontal and lateral images of right dacryocystocele

DISCUSSION

Dacryocystocele in the medial orbital region is a relatively rare condition, but congenital dacryocystocele has been recognised as an individual disease since 1933(1212 Duke-Elder SS, editor. System of ophthalmology: normal and abnormal development: congenital deformities. St Louis: Mosby; 1964. Vol. 3, Pt 2. p. 934-41.). Congenital dacryocystocele is found almost exclusively in paediatric patients(1313 Rand PK, Ball WS Jr, Kulwin DR. Congenital nasolacrimal mucoceles: CT evaluation. Radiology. 1989;173(3):691-4.), and it has a specific natural history, clinical features, disease mechanism, and treatment. Lacrimal sac dilation occurs because of distal nasolacrimal duct obstruction and functional obstruction of the common canaliculus. In cases of nasolacrimal duct obstruction, secretions can accumulate in the lacrimal sac, culminating in its dilation and obstructing the common canaliculus. Another mechanism proposed for dacryocystocele involves folds in the common canaliculus due to a dilated lacrimal sac and Rosenmuller’s valve malfunction secondary to oedema and inflammation.(1313 Rand PK, Ball WS Jr, Kulwin DR. Congenital nasolacrimal mucoceles: CT evaluation. Radiology. 1989;173(3):691-4.) Children with dacryocystocele are treated with antibiotics, massage, and cold compresses, and if medical treatment is ineffective, probing and irrigation of the lacrimal pathway associated with intranasal endoscopic marsupialisation of the cyst are then indicated.(1414 Shashy RG, Durairaj V, Holmes JM, Hohberger GG, Thompson DM, Kasperbauer JL. Congenital dacryocystocele associated with intranasal cysts: diagnosis and management. Laryngoscope. 2003;113(1):37-40. Erratum in Laryngoscope. 2005;115(4):759. Durairaj, Vikram [corrected to Durairaj, Vikram D].)

There are a few reports of dacryocystocele in adults(1515 Yip CC, McCulley TJ, Kersten RC, Bowen AT, Alam S, Kulwin DR. Adult nasolacrimal duct mucocele. Arch Ophthalmol. 2003;121(7):1065-6.). Its clinical picture includes painless bulging of the medial region of the orbit below the medial ligament. The lacrimal secretion test, endoscopy, computed tomography, and magnetic resonance imaging can be used to diagnose the condition.(1616 PerryLJ, Jakobiec FA, Zakka FR, Rubin PA. Giant dacryocystomucopyoce in na adult a review of lacrimal sac enlargements with clinical and histopathologic differencial diagnoses. Sury Ophthalmol. 2012;57(5):474-85.) The disease mechanism of dacryocystocele is similar in adults and children, but the nasolacrimal duct obstruction in adults is acquired. A complication of dacryocystocele is chronic dacryocystitis.(1717 Meyer JR, Quint DJ, Holmes JM, Wiatrak BJ. Infected congenital mucocele of the nasolacrimal duct. AJNR Am J Neuroradiol. 1993;14(4):1008-10.)

Treatment involves placement of a nasolacrimal stent or dacryocystorhinostomy.(1818 Perena MF, Castillo J, Medrano J, De Gregorio MA, Loras E, Cristobal JA. Nasolacrimal polyurethane stent placement: preliminary results. Eur J Ophthalmol. 2001;11(1):25-30.,1919 Eloy P, Martinez A, Leruth E, Levecq L, Bertrand B. Endonasal endoscopic dacryocystorhinostomy for a primary dacryocystocele in an adult. B-ENT. 2009;5(3):179-82.)

It may be necessary to differentiate dacryocystocele from a tumour of the lacrimal sac.(2020 Fliss DM, Freeman JL, Hurwitz JJ, Heathcote JG. Mucoepidermoid carcinoma of the lacrimal sac: a report of two cases, with observations on the histogenesis. Can J Ophthalmol. 1993;28(5):228-35.,2121 Sabet SJ, Tarbet KJ, Lemke BN, Smith ME, Albert DM. Granular cell tumor of the lacrimal sac and nasolacrimal duct: no invasive behavior with incomplete resection. Ophthalmology. 2000;107(11):1992-4.)Adult dacryocystocele can be adequately diagnosed through studies such as ultrasound imaging, computed tomography, and magnetic resonance imaging. Computed tomography has become the most important method for locating and characterising an orbital mass. CT can determine the correct anatomical diagnosis by showing the dacryocystocele in the lacrimal sac and is also helpful in differentiating dacryocystocele from other masses that can affect the medial canthus in adults. An orbital tumour in the medial orbital region such as haemangioma, neurofibroma, lymphangioma, or rhabdomyosarcoma can be seen on CT as a solid mass.

  • Work conducted at the Lacrimal Pathways Unit, Federal University of São Paulo (UNIFESP), São Paulo/SP, Brazil.

REFERÊNCIAS

  • 1
    Lai PC, Wang JK, Liao SL. A case of dacryocystocele in an adult. Jpn J Ophthalmol. 2004;48(4):419-21.
  • 2
    Xiao MY, Tang LS, Zhu H, Li YJ, Li HL, Wu XR. Adult nasolacrimal sac mucocele. Ophthalmologica. 2008;222(1):21-6.
  • 3
    Ansari SA, Pak J, Shields M. Pathology and imaging of the lacrimal drainage system. Neuroimaging Clin N Am. 2005;15(1):221-37.
  • 4
    Lelli GJ, Levy RL. Epidermoid cyst masquerading as dacryocystocele: case report and review. Orbit. 2011;30(2):114-5.
  • 5
    Debnam JM, Esmaeli B, Ginsberg LE. Imaging characteristics of dacryocystocele diagnosed after surgery for sinonasal cancer. AJNR Am J Neuroradiol. 2007;28(10):1872-5.
  • 6
    Weber AL, Rodriguez-DeVelasquez A, Lucarelli MJ, Cheng HM. Normal anatomy and lesions of the lacrimal sac and duct: evaluated by dacryocystography, computed tomography, and MR imaging. Neuroimaging Clin N Am. 1996;6(1):199-217.
  • 7
    Berlucchi M, Tomenzoli D, Trimarchi M, Lombardi D, Battaglia G, Nicolai P. [Dacryocystocele in the adult: etiology, diagnosis and treatment]. ActaOtorhinolaryngol Ital. 2001;21(2):100-4.
  • 8
    Day S, Hwang TN, Pletcher SD, Bhatki A, McCulley TJ. Interactive image- guided endoscopic dacryocystorhinostomy. Ophthal Plast Reconstr Surg. 2008;24(4):338-40.
  • 9
    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(1):52-3.
  • 10
    Song HY, Lee DH, Ahn H, Kim JH, Kang SG, Yoon HK, et al. Lacrimal system obstruction treated with lacrimal polyurethane stents: outcome of removal of occluded stents. Radiology. 1998;208(3):689-94.
  • 11
    Yazici B, Yazici Z, Parlak M. Treatment of nasolacrimal duct obstruction in adults with polyurethane stent. Am J Ophthalmol. 2001;131(1): 37-43.
  • 12
    Duke-Elder SS, editor. System of ophthalmology: normal and abnormal development: congenital deformities. St Louis: Mosby; 1964. Vol. 3, Pt 2. p. 934-41.
  • 13
    Rand PK, Ball WS Jr, Kulwin DR. Congenital nasolacrimal mucoceles: CT evaluation. Radiology. 1989;173(3):691-4.
  • 14
    Shashy RG, Durairaj V, Holmes JM, Hohberger GG, Thompson DM, Kasperbauer JL. Congenital dacryocystocele associated with intranasal cysts: diagnosis and management. Laryngoscope. 2003;113(1):37-40. Erratum in Laryngoscope. 2005;115(4):759. Durairaj, Vikram [corrected to Durairaj, Vikram D].
  • 15
    Yip CC, McCulley TJ, Kersten RC, Bowen AT, Alam S, Kulwin DR. Adult nasolacrimal duct mucocele. Arch Ophthalmol. 2003;121(7):1065-6.
  • 16
    PerryLJ, Jakobiec FA, Zakka FR, Rubin PA. Giant dacryocystomucopyoce in na adult a review of lacrimal sac enlargements with clinical and histopathologic differencial diagnoses. Sury Ophthalmol. 2012;57(5):474-85.
  • 17
    Meyer JR, Quint DJ, Holmes JM, Wiatrak BJ. Infected congenital mucocele of the nasolacrimal duct. AJNR Am J Neuroradiol. 1993;14(4):1008-10.
  • 18
    Perena MF, Castillo J, Medrano J, De Gregorio MA, Loras E, Cristobal JA. Nasolacrimal polyurethane stent placement: preliminary results. Eur J Ophthalmol. 2001;11(1):25-30.
  • 19
    Eloy P, Martinez A, Leruth E, Levecq L, Bertrand B. Endonasal endoscopic dacryocystorhinostomy for a primary dacryocystocele in an adult. B-ENT. 2009;5(3):179-82.
  • 20
    Fliss DM, Freeman JL, Hurwitz JJ, Heathcote JG. Mucoepidermoid carcinoma of the lacrimal sac: a report of two cases, with observations on the histogenesis. Can J Ophthalmol. 1993;28(5):228-35.
  • 21
    Sabet SJ, Tarbet KJ, Lemke BN, Smith ME, Albert DM. Granular cell tumor of the lacrimal sac and nasolacrimal duct: no invasive behavior with incomplete resection. Ophthalmology. 2000;107(11):1992-4.

Publication Dates

  • Publication in this collection
    Sep-Oct 2014

History

  • Received
    11 Nov 2011
  • Accepted
    03 Sept 2012
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