Cone beam computed tomography (CBCT) sialography—an adjunct to salivary gland ultrasonography in the evaluation of recurrent salivary gland swelling

Accepted Manuscript CBCT sialography – an adjunct to salivary gland ultrasound in the evaluation of recurrent salivary gland swelling Tobias Kroll, MD, Andreas May, DDS, Claus Wittekindt, MD, Christopher Kähling, MD, Shachi Jenny Sharma, MD, Hans-Peter Howaldt, MD, DDS, PhD, Jens Peter Klussmann, MD, PhD, Philipp Streckbein, MD, DDS PII:

S2212-4403(15)01190-6

DOI:

10.1016/j.oooo.2015.09.005

Reference:

OOOO 1299

To appear in:

Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology

Received Date: 13 May 2014 Revised Date:

14 February 2015

Accepted Date: 2 September 2015

Please cite this article as: Kroll T, May A, Wittekindt C, Kähling C, Sharma SJ, Howaldt H-P, Klussmann JP, Streckbein P, CBCT sialography – an adjunct to salivary gland ultrasound in the evaluation of recurrent salivary gland swelling, Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology (2015), doi: 10.1016/j.oooo.2015.09.005. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.

ACCEPTED MANUSCRIPT CBCT sialography – an adjunct to salivary gland ultrasound in the evaluation of recurrent salivary gland swelling

Department of Otorhinolaryngology
 (Head: Univ.-Prof. Dr. Jens Peter Klussmann) University Hospital Gießen, Justus Liebig University Klinikstraße 33, 35392 Giessen, Germany

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Department of Cranio-Maxillo-Facial Surgery
 (Head: Univ.-Prof. Dr. Dr. Hans-Peter Howaldt) University Hospital Gießen, Justus Liebig University Klinikstraße 33, 35392 Giessen, Germany

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Tobias Kroll, MDa, Andreas May, DDSb, Claus Wittekindt, MDa, Christopher Kähling, MDb, Shachi Jenny Sharma, MDa, Hans-Peter Howaldt, MD, DDS, PhDb, Jens Peter Klussmann, MD, PhDa, Philipp Streckbein, MD, DDSb

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Corresponding author: Tobias Kroll, MD Department of Otorhinolaryngology University Hospital Gießen, Justus Liebig University Klinikstraße 33 D-35392 Gießen, Germany Phone: +49 (0) 641 985-56784 Fax: +49 (0) 641 985-43709 E-mail: [email protected]

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Details about financial support and conflict of Interest: None

Number of words abstract: 163 Number of words manuscript: 1946 Number of References: 15 Number of tables: 1 Number of figures: 3

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Abstract:

Objective:

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Cone beam computed (CBCT) sialography could help improve the visualization of the duct system of salivary glands. The aim of this retrospective investigation was to follow up the use of CBCT sialography for diagnosis of pathologies within the

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intraglandular duct system when ultrasound was inconclusive. Study Design:

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14 consecutive patients suffering from recurrent swelling of a major salivary gland were evaluated. In 12 patients (8 female; 4 male; average age 46 years) a radiopaque contrast agent could be injected into the duct system followed by a routine CBCT scan. Four blinded examiners evaluated the acquired datasets

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retrospectively.

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Results:

CBCT scans revealed 7 stenosis, 2 salivary stones, 1 complete duct atresia, 1

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intraglandular duct ectasia and 1 regular duct system. Three of the detected pathologies were strictly intraglandular.

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Conclusion:

CBCT sialography shows promise as a supplementary non-invasive diagnostic tool for the visualization of the intraglandular duct system of the major human salivary glands. Controlled studies to further validate this method should be implemented.

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Introduction: Various radiological and non-radiological methods are available for the imaging diagnostics of salivary gland parenchyma and salivary ducts. Currently, because of

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its known advantages, sonography is the key non-radiological modality used for imaging the parenchyma of major salivary glands 1. However, salivary ducts can only be demonstrated when they are filled. This occurs either when they are obstructed

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through a salivary stone or a stenosis, or when a salivation-inducing substance, such as ascorbic acid, has been orally administered 2. Injection of an ultrasound contrast

duct system during examination 3, 4.

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agent into the duct system can improve visualization of the gland parenchyma and

A further, established, non-radiological imaging method for the visualization of salivary ducts is minimally-invasive sialendoscopy, which, since 2006, is increasingly

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being performed as a routine procedure in Otorhinolaryngology in Germany 5.

Radiological imaging modalities available to visualize salivary gland parenchyma and

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salivary gland ducts are conventional sialography, computed tomography (CT), and magnetic resonance sialography (MRI) 6. With the introduction and increasing use of

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cone beam computed tomography (CBCT) in otorhinolaryngology and cranio-maxillofacial surgery, a new radiological imaging method appears to have become available for the visualization of salivary glands and, in particular, of salivary gland ducts, after injection of a radiopaque contrast agent into the duct system. This approach, called CBCT sialography, is, however, still in an early stage of development

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The aim of this retrospective follow up report is to demonstrate that CBCT sialography is a suitable imaging modality for the diagnosis of salivary duct pathologies, in particular, when sonography has failed to demonstrate pathology in 3

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from

recurrent

swelling

of

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gland.

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patients

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ACCEPTED MANUSCRIPT Material and Methods: Indication for CBCT-sialography were remained uncertain reasons of recurrent gland swellings after initial clinical examination. Contraindications were detected reasons for recurrent gland swellings such as stones, duct lesions or intraglandular lesions.

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All patients (n = 92) who presented from February 2013 to February 2014 at our institution with recurrent swelling of a parotid or submandibular gland were scanned for this retrospective evaluation. After a comprehensive anamnesis and clinical

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examination, a diagnostic sonography of the swollen salivary gland was performed routinely in all cases. In 14 patients the cause of the recurrent salivary gland swelling

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could not be established in initial examination or sialendoscopy. CBCT sialography was indicated for further imaging of the salivary duct system. Each patient signed an informed consent prior to routine CBCT sialography.

In preparation for the CBCT sialography, the efferent duct of the swollen gland was

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dilated to gauge 22 with salivary duct probes (Marchal Salivary Duct Probes, Karl

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Storz, Tuttlingen, Germany). Then, depending on the gland, the caruncle or papilla was cannulated with an indwelling venous cannula (Vasofix® Braunüle®, Braun,

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Melsungen, Germany) through which a radiopaque contrast agent (Ultravist®-370, Bayer Vital, Leverkusen, Germany) was injected into the duct system of the swollen

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gland until the patient reported fullness of the gland or contrast agent flowed past the probe into the oral cavity. Prior to scanning, patients were positioned such that they did not have to be repositioned after injection of the contrast agent. A radiological image was then acquired with a CBCT scanner (ProMax 3D Max, Planmeca, Helsinki, Finnland). The following operational parameters were used to acquire the scan: For the parotid gland, a field of view (FOV) with a width of 230mm, a height of 160mm, and a resolution of 0.4mm was chosen. The resultant tube voltage was 96kV, with a tube current of 12mA. The data acquisition time for this scan was 9.0 5

ACCEPTED MANUSCRIPT sec. For the submandibular gland, a field of view (FOV) with a width of 130mm, a height of 55mm, and a resolution of 0.2mm was chosen. The tube voltage for this scan was 96kV, with a tube current of 12mA, and a data acquisition time of 12.0 sec. The acquired CBCT datasets were visualized as multi-planar (MPR) and 3-

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dimensional reconstructions (3D recon) and analyzed by four independent blinded examiners qualified for radiological evaluations. Data analysis was performed with DICOM viewer software (OsiriX MD, v2.8.5 64bit, Pixmeo SARL, Bern, Switzerland).

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For every patients images the visualization of the duct system was individually optimized followed by an evaluation for certain pathologies such as stones, duct

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stenosis, duct ectasia or duct atresia.

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ACCEPTED MANUSCRIPT Results: 14 consecutive patients with indication for sialography were included into the retrospective follow up. Due to impenetrable salivary ducts the CBCT scan could not be performed in 2 cases. Of the remaining 12 patients, 8 (66.7%) were female and 4

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(33.3%) were male. The average patient age was 46 years. The parotid gland was examined in 8 (66.7%) and the submandibular gland in 4 (33.3%) cases. 5 (41.7%) patients underwent sialendoscopy before CBCT sialography. In these cases, the

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indication for CBCT sialography was unsolved pathology within the sialendoscopy. Evaluation of the acquired CBCT images (Table 1) revealed 7 (58.3%) salivary duct

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stenoses, of which 3 (42.9%) were found close behind the papilla whereas the papilla showed no signs of obstruction in prior clinical examinations or ultrasound, 3 (42.9%) were in the main efferent duct, and 1 (14.3%) was in an intraglandular duct (Figure 1). In addition, in 2 (16.7%) submandibular glands, salivary stones that had remained

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undetected during sonography could be demonstrated as the cause of recurrent

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swelling of the gland. 1 stone was found in an intraglandular duct (50.0%), while the other was found in the main efferent duct (50.0%). Complete atresia of Stensen’s

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duct was found in 1 (8.3%) parotid gland, and intraglandular duct ectasia was found in 1 (8.3%) other parotid gland (Figure 2). For 1 (8.3%) gland, no correlating

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pathology could be found with CBCT sialography (Figure 3). Based on the results of CBCT sialography, 3 (25.0%) patients were indicated for a subsequent sialendoscopy, or revision sialendoscopy. 1 (8.3%) patient was treated with a combined approach. Based on an individual concept a wait-and-watch strategy was chosen in 8 (66.7%) patients.

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ACCEPTED MANUSCRIPT Discussion: Visualizing the ductal system of the major salivary glands has always been a challenge for clinicians, although various imaging modalities are available. Currently, the four most frequently used imaging modalities in everyday clinical routine are

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sonography, sialography, CT and MRI. Compared to the first two methods, the two latter methods, however, play a subordinated role due to cost, possibly radiation

sialendoscopy is increasingly gaining use 5.

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exposure and availability issues 9. As a further imaging method, minimally-invasive

Currently, sonography is the imaging method of choice for salivary glands because it

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can be directly performed by the attending physician, does not incur a radiation burden, and provides imaging accuracy that is adequate for diagnosis in many cases 1

. However, to be adequately visible during sonography, the salivary duct system has

to be filled. Salivary ducts can either be intentionally filled by orally administering

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ascorbic acid to induce saliva flow, or may already be filled as the result of

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obstructive pathologies such as salivary stones or duct stenosis 2. The retrograde administration of a contrast agent, similar to the procedure for sialography, can aid 3, 4

. A limitation of sonography in

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visualization of the duct system during sonography

demonstrating the cause of recurrent salivary gland swelling is that pathologies other

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than obstruction by stones can be difficult to diagnose with this method

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Sialendoscopy is a further established procedure for the diagnosis and therapy of salivary gland disease. Koch and collaborators preserved over 97% of salivary glands with stenoses of the Wharton- or Stensen ducts by using this method along with a Combined Approach

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. However, the method can only be used for

extraglandular duct pathologies, or those located close to the hilum. While sialography, which was initially described in 1902, currently still holds a place in salivary gland diagnostics, this method is increasingly being replaced by modern 8

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CBCT has become an established diagnostic tool in cranio-maxilo-facial surgery and is rapidly gaining entry into otorhinolaryngology. This imaging method is being used for the diagnostics of the bony structures of the paranasal sinuses and petrous bone.

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Early studies have shown that CBCT is also a suitable modality for the visualization of salivary ducts that have been enhanced with a radiopaque contrast agent

7-10

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study that directly compared conventional two-dimensional sialography with three-

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dimensional sialography, Jadu et al. postulated that CBCT sialography outperformed conventional sialography for the visualization of salivary gland parenchyma and

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salivary stones 8. Our findings support the results reported by Varoquaux et al. that the salivary gland duct system can be visualized up to the 6 th branch with CBCT sialography after a contrast agent has been injected

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. This high imaging accuracy is

due to the isotropic voxel resolution afforded by the CBCT unit

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. In a first

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comparative radiation dose study, Jadu et al. found that, by choosing appropriate

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parameters, the effective radiation dose from CBCT sialography equaled that from conventional sialography 9.

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In the present retrospective follow up report, the three-dimensional visualization of the salivary duct system through CBCT allowed us to diagnose a total of 3 previously

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undetected intraglandular pathologies. Our findings thus support Varoquaux et al.‘s proposition that the underlying causes for recurrent swelling of a salivary gland, not due to obstruction through a stone, can elude sonographic diagnosis. It remains to be clarified if sonography can accurately image intraglandular duct pathologies after contrast medium has been injected into the salivary duct system. Comparative studies to shed light on this question are outstanding. While it must be stressed that CBCT sialography cannot be a substitute for sialendoscopy, the targeted use of CBCT sialography could, however, reduce the 9

ACCEPTED MANUSCRIPT indication for sialendoscopy for pathologies of the extraglandular duct system or the area around the hilum. In these cases, CBCT sialography could be helpful in accurately identifying patients who could profit from further gland-preserving

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interventions.

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ACCEPTED MANUSCRIPT Conclusion: In this retrospective follow up report CBCT sialography was helpful in diagnosing unexplained cases of swelling of one of the major salivary glands with intraglandular pathologies when ultrasound was inconclusive. We could thus demonstrate that

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CBCT sialography shows promise as a supplementary non-invasive diagnostic tool for the visualization of the intraglandular duct system. Our encouraging findings suggest that performing a CBCT sialography prior to sialendoscopy may be justified

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in cases of unexplained, recurrent swelling of a salivary gland to rule out pathologies that are intraglandular and, therefore, unreachable by sialendoscopy. Patients could,

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thus, be spared the unpleasantness of an unsuccessful intervention. An advantage to other radiological imaging such as CT and MRI are lower costs for CBCT. The exposure to radiation is less than a CT and comparable with conventional sialography.

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Controlled studies with higher case numbers and follow-up examinations are planned

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pathologies.

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to further verify the value of CBCT examinations for the diagnosis of salivary gland

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ACCEPTED MANUSCRIPT Figure legends: Figure 1: Image A shows a three-dimensional reconstruction of the efferent duct system of the parotid gland, image B an axial section in a planar reconstruction. The

the duct system. The stenosis is indicated by a red arrow.

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images show a stenosis located behind the junction to the tertiary division branch of

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Figure 2: These images show intraglandular ectasia of the efferent parotid gland duct system. Ectasia can be seen to begin at the junction point between the primary and

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secondary duct divisions branches and to continue into the secondary division branch. Image A shows a three-dimensional reconstruction of the efferent duct system, image B an axial section in a planar reconstruction. In both images, the red

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arrows indicate the ectatic portion of the duct.

Figure 3: No correlating pathology could be found and the reasons for recurrent

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swellings remain uncertain. A normal parotid gland duct system is shown as a 3-

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dimensional reconstruction in image A, and as a planar reconstruction in image B.

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ACCEPTED MANUSCRIPT References: [1]

Onkar PM, Ratnaparkhi C, Mitra K. High-frequency ultrasound in parotid gland

disease. Ultrasound quarterly 2013 Dec;29(4):313-21. [2]

Bozzato A, Hertel V, Bumm K, Iro H, Zenk J. Salivary simulation with ascorbic

ultrasound : JCU 2009 Jul-Aug;37(6):329-32. [3]

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acid enhances sonographic diagnosis of obstructive sialadenitis. Journal of clinical

Kroll T, Helbig M, Klussmann JP, Wittekindt C. [Intraductal application of

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Levovist(R) in salivary glands of animals]. Laryngo- rhino- otologie 2012 Apr;91(4):229-32.

Zengel P, Berghaus A, Weiler C, Reiser M, Clevert DA. Intraductally applied

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contrast-enhanced ultrasound (IA-CEUS) for evaluating obstructive disease and secretory dysfunction of the salivary glands. European radiology 2011 Jun;21(6):1339-48.

Kroll T, Finkensieper M, Hauk H, Guntinas-Lichius O, Wittekindt C.

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[Sialendoscopy--learning curve and nation-wide survey in German ENTdepartments]. Laryngo- rhino- otologie 2012 Sep;91(9):561-5. Kraff O, Theysohn JM, Maderwald S, Kokulinsky PC, Dogan Z, Kerem A, et al.

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2009 Sep;44(9):518-24.

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beam computed sialography in diagnosing salivary gland lesions. Imaging science in dentistry 2013 Mar;43(1):17-23. [8]

Jadu FM, Lam EW. A comparative study of the diagnostic capabilities of 2D

plain radiograph and 3D cone beam CT sialography. Dento maxillo facial radiology 2013;42(1):20110319.

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ACCEPTED MANUSCRIPT [9]

Jadu F, Yaffe MJ, Lam EW. A comparative study of the effective radiation

doses from cone beam computed tomography and plain radiography for sialography. Dento maxillo facial radiology 2010 Jul;39(5):257-63. [10]

Varoquaux A, Larribe M, Chossegros C, Cassagneau P, Salles F, Moulin G.

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[Cone beam 3D sialography: preliminary study]. Revue de stomatologie et de chirurgie maxillo-faciale 2011 Nov;112(5):293-9. [11]

Koch M, Iro H, Kunzel J, Psychogios G, Bozzato A, Zenk J. Diagnosis and

Laryngoscope 2012 Mar;122(3):552-8.

Koch M, Kunzel J, Iro H, Psychogios G, Zenk J. Long-term results and

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gland-preserving minimally invasive therapy for Wharton's duct stenoses. The

subjective outcome after gland-preserving treatment in parotid duct stenosis. The Laryngoscope 2013 Nov 23. [13]

Benson B. Salivary gland radiology. In: SC W, MJ P, editors. Oral radiology:

Rzymska-Grala I, Stopa Z, Grala B, Golebiowski M, Wanyura H, Zuchowska

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priciples and interpretation. 6th edn ed. St. Louis, MO: Mosby; 2009. p. 578-98.

A, et al. Salivary gland calculi - contemporary methods of imaging. Polish journal of

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radiology / Polish Medical Society of Radiology 2010 Jul;75(3):25-37. Shahidi S., Hamedani S. The feasibility of cone beam computed tomographic

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sialography in the diagnosis of space-occupying lesions: report of 3 cases Oral Surg Oral Med Oral Pathol Oral Radiol 2014 Jun;117(6):e452-7.

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Diagnosis after CBCT Comorbidities sialography

Procedure after Symptoms CBCT sialography Recurrent Combined swellings Approach with acute (Sialendoscopy 36 months None Duct stenoses and surgery) Parotid gland Right parotits Metabolic Recurrent swellings 15 months syndrome Duct atresia Sialendoscopy Parotid gland Left Recurrent Arterial Duct stenoses 18 months Hypertension Parotid gland Right swellings at papilla Wait-And-Watch Recurrent Duct stenoses 3 months None Parotid gland Right swellings at papilla Sialendoscopy Recurrent swellings with acute Intraglandular Parotid gland Left parotits 1 months Adiposity duct ectasia Wait-And-Watch Recurrent Arterial Regular 4 months Hypertension salivary duct Wait-And-Watch Parotid gland Right swellings Recurrent Intraglandular swellings 60 months None duct stenoses Wait-And-Watch Parotid gland Left Recurrent Submandibular swellings 30 months None gland left Sialolithiasis Sialendoscopy Recurrent Intraglandular Submandibular swellings 5 months None Left sialolithiasis Wait-And-Watch gland Recurrent Submandibular swellings 6 months None gland Left Duct stenoses Wait-And-Watch Recurrent Submandibular 18 months None gland Right swellings Duct stenoses Wait-And-Watch Recurrent Arterial Duct stenoses 2 months Hypertension Parotid gland Right swellings at papilla Wait-And-Watch Table 1: Patient list with patient history, diagnosis and procedure after CBCT sialography

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Duration of symptoms (anamnesis)

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Standort Giessen Klinik für HNO-Heilkunde Kopf-/Halschirurgie Plastische Operationen Direktor: Prof. Dr. med. J.P. Klußmann Klinikstrasse 33 35390 Gießen

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Dr. med. Tobias Kroll Department of Otorhinolaryngology (Head: Univ.-Prof. Dr. Jens Peter Klussmann) University Hospital Gießen, Justus Liebig University Klinikstraße 33, 35392 Giessen, Germany

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Statement of clinical Relevance

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Cone Beam Computed Tomography sialography allows visualization of ductal anomalies also in peripheral locations. It shows promise as a supplementary non-invasive diagnostic tool for the visualization of the intraglandular duct system.

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Dr. med. Tobias Kroll

Universitätsklinikum Gießen und Marburg GmbH

Geschäftsführung

Aufsichtsratvorsitzender

Sitz der Gesellschaft: Gießen Amtsgericht Gießen HRB 6384

Martin Menger (Vors.) Prof. Dr. Werner Seeger (stv. Vors.) Dr. Christiane Hinck-Kneip Prof. Dr. Jochen A. Werner

Wolfgang Pföhler

www.ukgm.de