Computed tomography and magnetic resonance imaging findings of acquired dacryocystoceles

    Computed Tomography and Magnetic Resonance Imaging Findings of Acquired Dacryocystoceles Erkan G¨okc¸e, Murat Beyhan, Berat Acu, Helin Deniz Demir PII: DOI: Reference:

S0899-7071(13)00340-9 doi: 10.1016/j.clinimag.2013.12.016 JCT 7542

To appear in:

Journal of Clinical Imaging

Received date: Revised date: Accepted date:

6 September 2013 10 December 2013 19 December 2013

Please cite this article as: G¨ok¸ce Erkan, Beyhan Murat, Acu Berat, Deniz Demir Helin, Computed Tomography and Magnetic Resonance Imaging Findings of Acquired Dacryocystoceles, Journal of Clinical Imaging (2014), doi: 10.1016/j.clinimag.2013.12.016

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ACCEPTED MANUSCRIPT Computed Tomography and Magnetic Resonance Imaging Findings of Acquired

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Dacryocystoceles

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Short title: CT and MRI Findings of Acquired Dacryocystoceles

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1. Erkan Gökçe, Asist Prof . 1

3. Berat Acu, Asist Prof. 3 4. Helin Deniz Demir, Asist Prof . 4

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2. Murat Beyhan, M.D.2

1. Gaziosmanpaşa University, School of Medicine, Department of Radiology, Tokat.

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2. Zile State Hospital, Department of Radiology, Tokat 3. Osmangazi University, School of Medicine, Department of Radiology, Tokat.

Corresponding:

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4. Gaziosmanpaşa University School of Medicine, Department of Ophthalmology, Tokat.

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Erkan Gökçe, Asist. Prof.

Gaziosmanpaşa University, School of Medicine, Department of Radiology, 60200 Tokat, Turkey

Phone: +903562133294 Mobile phone : +90 542 3798986 Fax: +903562133179 e-mail: [email protected] second e-mail : [email protected]

ACCEPTED MANUSCRIPT Computed Tomography and Magnetic Resonance Imaging Findings of Acquired Dacryocystoceles

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Introduction

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Dacryocystocele, also known as lacrimal sac mucocele, amniocele, amniotocele, or dacryocele, is a pathologic condition frequently observed in newborns as a result of congenital lacrimal system stenosis [1]. Acquired form which can develop due to various

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etiologies, on the other hand, is rarer [2-4]. Macroscopically, dacryocystocele is diffused, cystic and often bluish enlargement of lacrimal sac. Histopathologically, on the other hand,

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dacryocystoceles are filled with secretions from their epithelial linings containing goblet cells and submucosal accessory seromucinous glands, as well as some entrapped tears [3]. In the present study, radiological imaging findings of dacryocystoceles detected in patients who presented themselves with complaints such as epiphora, orbital cellulitis and lump in medial canthal area or stroke and to whom one or more of orbita, maxillofacial and brain CT or MRI

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Materials and Methods

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techniques were applied.

In this study, after obtaining the approval of local ethics, we retrospectively analyzed

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the CT and MRI examinations in the PACS system (GE Healthcare, Milwaukee, WI, USA) of dacryocystoceles in patients. A Total of 11 patients including 8 female and 3 males with dacryocystoceles on their radiologic examinations between May 2010 and July 2013 were

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included in the study. Age of the patients in the study ranged from 9 to 86 (mean 55.4±25.4). CT and/or MRI examinations performed because of medial canthal swelling after maxillofacial trauma in one patient, headache in one patient, unilateral epiphora in one patient, cerebrovascular disease in two patients, unilateral swelling in canthal area in two patients, and swelling, erythema and purulent discharge in eyelid and/or right medial canthal area in four patients. Nine patients underwent ophthalmic examination, and their lacrimal systems were irrigated and probed. Ophthalmic examination was not performed in two patients who were incidentally found to have dacryocystocele in their brain CT and MRI examinations. CT examinations were carried out on 2- and 8-detector CT machines (Siemens Somatom sprit, Erlangen-Germany and GE LightSpeed Ultra, Milwaukee-USA). The MRI examinations were performed on a 1.5 T (Signa excite HD; GE Healthcare, Milwaukee, WI,

ACCEPTED MANUSCRIPT USA) machine, using an 8-channel neuro-vascular coil. In CT and MRI examinations, dimensions, localizations, (unilateral or bilateral and sides), presence of nasolacrimal duct and lacrimal fossa dilatation, infection findings in dacryocystoceles and orbital structures, and

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other accompanying anomalies such as dacryoliths were studied. Six patients had clinical

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indications of seven infected dacryocystoceles while the remaining five patients had six noninfected dacryocystoceles. In order to evaluate possible density differences between infected and non-infected dacryocystoceles, densities of dacryocystoceles were measured in non-

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contrast CT examinations using three different cross-sections, and the average density was calculated.

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A nine-year-old male patient, who had road accident based Lefort II fracture 18 months ago and who had swelling and purulent discharge in canthal area four months later, was subjected to non-contrast maxillofacial CT and contrast-enhanced orbita MRI. Contrastenhanced orbita MRI was applied to a 35-year-old women who had a growing swelling in right medial canthal area for the past three months. Non-contrast and contrast-enhanced orbita

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CT examinations were performed on a sixteen-year-old female patient who had swelling in right medial canthal area for three years and occasional epiphora and purulent discharge

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complaints and had swelling, erythema and purulent discharge in eyelid and right medial canthal area for 10 days, on a 50-year-old female patient who had dacryocystitis attacks in the

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last 10 years, on a 70-year-old female patient who had swelling in right medial canthal area for two years and had swelling, erythema and purulent discharge in eyelid and right medial

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canthal area for a week, on a 72-year-old female patient who had epiphora in right eye and swelling in medial canthal area for three years and had purulent discharge in last two weeks, on a 72-year-old female patient who had epiphora and swelling complaint in right medial canthal area for six months, and on a 54-year-old male patient who had epiphora in left eye for two years. Because of her headache, 71-year-old female patient had non-contrast brain MRI. Seventy-five-year old female patient who had cerebrovascular disease, growing swelling in bilateral medial canthal area for 10 years, recurring mucopurulent discharges and fever had non-contrast brain CT. Brain MRI examination records of this patient was existing in PACS system. Because of his cerebrovascular disease, non-contrast brain CT examination was performed on 86-year-old male patient. Results

ACCEPTED MANUSCRIPT A total of 13 dacryocystoceles cases were studied. Two patients had bilateral dacryocystoceles, while 4 patients had dacryocystocele on the left and 5 patients on the right. Volumes of dacryocystoceles varied between 0.25 to 3.42 ml (mean 1.60±0.97). In one of the

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patients, a post-trauma collapse in bone structures at the level of basal nose and

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dacryocystoceles in the form of bilateral rim containing a calcified wall were detected (Figure 1). Diameter increases of 6-12 mm were observed in a total of six lacrimal ducts in five patients. Unilateral expansion with only fluid density or intensity in lacrimal duct was

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detected in three patients (Figure 2). Dilatation of lacrimal duct was observed in one of the two patients with unilateral dacryocystoceles and metallic densities (3071 HU) that belonged

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to foreign objects in lacrimal duct on the same sides (Figure 3). Non-contrast CT scans of six patients with infected dacryocystoceles revealed a clear density increase in one patient and slight density increases in others, and contrast-enhanced CT or MRI examinations showed contrast enhancement in dacryocystocele walls. In two of these patients, contrast enhancement along with a density and volume increase were observed in soft tissues around the

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accompanying preseptal cellulitis (Figure 4). In addition, hyperdense dacryoliths of 3-4 mm size were observed in lacrimal sac and duct one of these patients who had infected

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dacryocystocele (Figure 4). In a case with infected bilateral dacryocystocele, homogeneous hyperdensity on CT and hypointensity in T2-weighted series of MRI were considered

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secondary to fungal sinusitis and dacryocystocele fungal content were determined (Figure 5). In eight of the dacryocystoceles (61.5%), there were enlargements in lacrimal fossa and remodeling in adjacent bone (Figure 2C ad 3). Densities of non-infected dacryocystoceles

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varied from 2 to 20 (mean 11.8±7.8) Haunsfield unit (HU), while those of the infected ones varied from 32 to 65 (median 35.0) HU. In none of dacryocystoceles and nasolacrimal ducts, contrast enhancement solid component or asymmetric wall thickening consistent with tumoral process was not observed. Age of the patients, and location, size, density, infection status and accompanying anomalies of dacryocystoceles are given in Table. In nine patients who were subjected to lacrimal probing, the fluid reflux was from the upper canaliculus while irrigating from the lower one. All patients were found to have obstruction distal to the common canaliculus. Seven of nine patients accepted surgery, and dacryocystorhinostomy was performed in those cases. Discussion A dacryocele is swelling of a lacrimal sac from distal nasolacrimal duct blockage at the inferior meatus. It results in a proximal ball-valve effect (functional obstruction) at the

ACCEPTED MANUSCRIPT canalicular junction to the lacrimal sac and allows fluid to enter the sac, but not to leave [1, 2]. If distal nasolacrimal duct is obstructed, secretions may accumulate in it, the lacrimal sac swells and presses on two canaliculi, causing them to fold upon themselves and resulting in

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proximal obstruction. Among the other proposed mechanisms for mucocele formation is

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kinking of the canalicus due to swollen lacrimal sac and malfunctioning of Rosenmuller valve, the entrance of the common canaliculus into the lacrimal sac, following edema and inflammation [2]. Frequency of dacryocele is 0.08 to 0.1% in newborns with congenital

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nasolacrimal duct obstruction. About 25% of the cases are bilateral, although initial presentation may be unilateral [1]. Acquired dacryoceles may develop as a result of various

dacryocystomucopyoceles

are

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etiologies and are rarer compared to congenial dacryoceles [2-4]. Dacryocystomucoceles and dacryocystocele

types

acquired

at

any

age,

dacryocystomuco(pyo)cele type being more common. A blockage of the nasolacrimal duct by any of the foregoing local factors is required for all dacryocystomucoceles [3]. Bartley grouped secondary acquired lacrimal drainage obstructions in 5 categories:

infectious,

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inflammatory, neoplastic, traumatic, and mechanical [5, 6]. The mechanisms and etiological factors contributing to acquired dacryocystoceles are numerous: herpetic, chlamydial,

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mycotic, and bacterial infections; midface fractures; nasal or sinus surgery; topical drops (antivirals, antibacterials, and ocular anti-hypertensives) or systemic medications; dacryoliths;

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idiopathic inflammations (peripunctal lichen planus, Stevens-Johnson syndrome, Wegener granulomatosis); sarcoidal granulomas; and some carcinomas and lymphomas [3]. Infections were mainly responsible for the etiology of dacryocystoceles in the present study and no

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dacryocystoceles of tumoral or systemic disease origin were observed in any patient. Adult dacryocystoceles tend to be much firmer to palpation compared with the fluctuant infantile cases. The thicker overlying tissue planes in older individuals lead to the camouflaging of the mucoid contents that may exist in the sac. Contrary to congenital form which involves only dilation of the lacrimal sac, the acquired form may have enlarged nasolacrimal duct and the sac. In the absence of a neoplastic process, persistent inflammation or infection within the duct or an adjacent sinus can promote reactive bone remodeling of the canal by gradual obstruction upon the soft tissues of duct, cause narrowing and complete blockage at final stage [3]. In our study, duct enlargement in nasolacrimal duct without any pathological density or intensity was detected in three patients, while foreign objects of metallic density were observed in ductus along with enlargement. Although the patients with metallic density in lacrimal ducts provided no information about the use of drugs containing

ACCEPTED MANUSCRIPT heavy metal salts or application of dacryocystography in their history and rejected the operation, we consider the role of these factors in the etiology of these metallic densities. In general, diagnostic tools such as CT scanning, MRI, ultrasonography (US), and

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dacryocystography are highly revealing in diagnosis. CT and MRI studies of simple

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dacryocystoceles and dacryocystomucopyoceles show fluid-filled cysts without solid components or projections into the lumen along with a thin rim of enhancement of the wall,

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which is most obvious on MR examination with contrast injection. The cyst’s fluid content has low signal intensity on T1-weighted MRI images, but high signal intensity in T2-weighted images, resembling the appearance of a vitreous body [3]. CT and MRI enable the correct

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anatomic diagnosis by demonstrating the lacrimal sac mucocele adjacent to an enlarged nasolacrimal duct and showing an intranasal submucosal cystic mass [2, 3]. In addition, contrasted CT and MRI techniques play a significant role in differentiating orbital and nasolacrimal sac tumors located in medial canthal area and the vicinity from dacryocystoceles. CT and MRI examinations reveal the orbital tumors in the medial canthal

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region, such as hemangioma, neurofibroma, lymphangioma and rhabdomyosarcoma, as solid soft-tissue masses with variable degrees of enhancement [2, 3]. Although US is one of the

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best methods to distinguish solid and cystic lesions, they can be distinguished by CT using density value of the lesion. In our study, density values of dacryocystoceles varied from 2 to

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65 HU. Density values ranged from 2 to 20 HU (mean 11.8±7.8) in non-infected group, while from 32 to 65 HU (median 35) in infected group. Despite the low number of cases and lack of

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contrasted CT examinations, these findings suggest that HU values of 32 and over point to dacryocystoceles with infection (dacryocystomucopyocele). In a study by Kim et al. [7] dacryocystocele was detected in a patient who had allergic fungal sinusitis and purulent discharge in left eye, manifesting itself with a clear hyperdensity in CT which indicated fungal content despite lack of a density measurement. Similarly, our CT examination found homogenous high densities (64 and 65 HU) in bilateral dacryocystoceles with fungal content in a patient with fungal sinusitis, possibly as a result of fungal content. This means that except for a possible hemorrhage history due to trauma, high HU density values of 65 and over in dacryocystoceles should be a warning for fungal content. Bony fossa region of lacrimal sac is usually not enlarged since much of the enlargement take place above the orbital rim [3]. However, in most of our cases (in eight dacryocystoceles of seven patients), there was enlargement in lacrimal bony fossa. In four of these patients, accompanying nasolacrimal duct enlargement was observed.

ACCEPTED MANUSCRIPT Among

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clinical

differential

diagnosis

of

dacryocystomucocele

and

dacryocystomucopyocele are dacryocystitis, lacrimal sac diverticulum, a supernumerary or isolated lacrimal sac with an ectopic caruncular punctum, lacrimal sac fistula to overlying

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skin, encephalocele, ethmoid or maxillary sinus mucoceles, dermoid or epidermoid cysts, and

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benign or malignant neoplasms of lacrimal sac [3].

In conclusion, acquired dacryocystoceles are rare masses of medial canthal area with

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different etiologies, whose radiological imaging findings may vary based on etiological factors and presence of infections.

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References

1. Wasserman BN, Schnall BM, Levin AV. Sequential bilateral dacryocele. Arch Ophthalmol. 2011 Jan;129(1):104-5.

2. Xiao MY, Tang LS, Zhu H, Li YJ, Li HL, Wu XR. Adult nasolacrimal sac mucocele. Ophthalmologica. 2008;222(1):21-6.

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3. Perry LJ, Jakobiec FA, Zakka FR, Rubin PA. Giant dacryocystomucopyocele in an adult: a review of lacrimal sac enlargements with clinical and histopathologic

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differential diagnoses. Surv Ophthalmol. 2012 Sep;57(5):474-85. 4. Bhaya M, Meehan R, Har-El G. Dacryocystocele in an adult: endoscopic management.

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Am J Otolaryngol. 1997 Mar-Apr;18(2):131-4. 5. Ali MJ, Gupta H, Honavar SG, Naik MN. Acquired nasolacrimal duct obstructions

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secondary to naso-orbito-ethmoidal fractures: patterns and outcomes. Ophthal Plast Reconstr Surg. 2012 Jul-Aug;28(4):242-5. 6. Bartley GB. Acquired lacrimal drainage obstruction: an etiologic classification system, case reports, and a review of the literature. Part 1. Ophthal Plast Reconstr Surg 1992;8:237–42. 7. Kim C, Kacker A, Chee RI, Lelli GJ. Allergic fungal sinusitis causing nasolacrimal duct obstruction. Orbit. 2013;32(2):143-5.

ACCEPTED MANUSCRIPT Figure Legends Figure 1. Nine year old male patient with bilateral dacryocystoceles developed as second to frontoethmoidal depressed fracture. A) Axial plane BT image of dacryocystoceles whose

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displaced anterior walls were calcified due to fracture (arrows) B) Rim style calcifications in

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medial canthal area (arrows) in coronal reformatted CT image of bone window C) Depression at the level of basal nose in volume rendering CT image D) Slightly heterogeneous

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hyperintense dacryocystoceles in coronal plane of T2 weighted MRI image. Figure 2. A-B) Left dacryocystoceles (arrow) and enlargement in nasolacrimal duct (arrows)

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in T2 weighted images in sagittal plane of 71-year-old woman subjected to MRI examination due to headache. C-D) Axial plane CT images of 72-year-old woman with the complaint of swelling in right medial cantus showing the dacryocystocele and enlargement in nasolacrimal duct (arrow).

Figure 3. Seventy-two-year-old female patient who had epiphora in right eye, swelling and

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purulent discharge in right medial canthal area A) Axial plane contrast-enhanced CT image showing infected dacryocystocele with metallic density and contrasted wall. B) Reformatted

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Sixteen-year-old

female

patient

with

periorbital

cellilutis

and

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Figure

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CT image in coronal plane showing foreign object of metallic density in nasolacrimal duct.

dacryocystomucopyocele. A-B) Axial plane contrast-enhanced CT images showing infectionbased contrasting in periorbital soft tissues on the right (arrows), on dacryocystocele wall

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(thick arrow) and on sclera (arrow head). C-D) CT images in axial plane bone window showing densities representing milimetric dacryoliths (arrows). Figure 5. Seventy-five-year old female patient with cerebrovascular disease, fever, bilateral mucopurulent discharge and fungal sinusitis. A) Dacryocystocele with bilateral homogenous hyperdense content (arrows). B-D) MRI of four years earlier. B) Axial T2 weighted image showing dacryocystoceles of different intensities on the right (arrow) and homogenous hypointense ones on the left (thick arrow). C-D) Sagittal T1 weighted images showing slightly hyperintense dacryocystocele on the right (arrow) and dacryocystocele with homogenous hyperintense content on the left (thick arrow).

ACCEPTED MANUSCRIPT Table. Age of the patients, and location, size, density, infection status and accompanying abnormalities of dacryocystoceles Location

Dimensions

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Additional Abnormalities

8x10x12

12

-

Bilateral wall calcification

9x10x13

6

-

10x14x17

32

+

(mm) Bilateral

Right

Right

14x14x20

-

+

50

Left

16x17x17

32

+

54

Left

7x8x9

70

Right

71 72

Periorbital cellulitis, dacrioadenitis,

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35

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Infection

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Age

Density

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Patient

dacryolithes

Enlargement in LF* Enlargement in LF

2

-

Metallic density in NLD*

15x17x24

40

+

Periorbital cellulitis

Left

7x11x23

-

-

Enlargement in NLD and LF

Right

11x15x15

35

+

Metallic density in NLD and LS*, enlargement in NLD and LF

Right

14x15x25

20

-

Enlargement in NLD and LF

75

Bilateral

12x16x20

64

+

Bilateral fungal content,

18x19x20

65

+

enlargement in LF

11x15x25

19

-

Enlargement in NLD and LF

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Left

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86

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72

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* NLD: Nasolacrimal duct, LF: Lacrimal fossa, LS: Lacrimal sac

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Figure 5