www.journalomp.org

pISSN 2288-9272 eISSN 2383-8493

J Oral Med Pain 2018;43(2):52-55

https://doi.org/10.14476/jomp.2018.43.2.52

The Therapeutic Value of Ultrasound-Guided Irrigation in Sialodochitis: A Case Report

Chi-Hyuk Ahn, Q-Schick Auh, Yeon-Hee Lee

Department of Orofacial Pain and Oral Medicine, Kyung Hee University Dental Hospital, Seoul, Korea

Received May 24, 2018

Revised June 19, 2018

Accepted June 19, 2018

In the present case study, we reported a female patient with sialodochitis fibrinosa in which a conservative ultrasound (US)-guided irrigation method was used to relieve her symptoms. A 30-year-old female patient visited Kyung Hee University Dental Hospital with chief complaints of persistent swelling and discomfort in the left facial area. Her primary symptom was facial swelling that was accompanied by neuropathological symptoms, such as itching, tingling, and warmth that usually continued for 3 to 4 days. During clinical examination of orofacial area, mild swelling and fever were noted in the left face including parotid region, and mild indura-tion could be felt at the corresponding site; sialadenitis of the left parotid gland was tenta-tively diagnosed. Herein, we performed intraductal irrigation of the left parotid gland in three times per month and prescribed some medication. Thus, her signs and symptoms have been improved, and she did not experience a recurrence for 12-month follow-up to date. This study can support the usefulness of the US-guided irrigation method to treat the sialodochitis fibri-nosa without remarkable side effects.

Key Words: Conservative treatment; Irrigation; Medication; Sialodochitis; Ultrasound-guided

Correspondence to:

Yeon-Hee Lee

Department of Orofacial Pain and

Oral Medicine, Kyung Hee University

Dental Hospital, 26 Kyungheedae-ro,

Dongdaemun-gu, Seoul 02447, Korea

Tel: +82-2-958-9418

Fax: +82-2-962-8124

E-mail: omod0209@gmail.com

CaseReport

JOMP Journal of Oral Medicine and Pain

Copyright Ⓒ 2018 Korean Academy of Orofacial Pain and Oral Medicine. All rights reserved.CC This is an open-access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/), which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

INTRODUCTION

Sialadenitis is an inflammatory disease affecting the

structures of the salivary glands, such as the salivary gland

parenchyma and salivary ducts.1) Sialodochitis fibrinosa,

which is one type of sialadenitis, is unique and is diagnosed

when inflammation is localized to the salivary ducts.2)

Inflammatory changes of the salivary ducts usually leads

to duct stricture or expansion, which can cause abnormal

salivation. The parotid gland is commonly affected by in-

flammation in comparison with the other major salivary

glands.3) Although inflammation usually occurs in both the

salivary parotid gland parenchyma and the salivary ducts,

it also occurs exclusively in the salivary ducts. A previous

published case of sialodochitis that occurred only in the sal-

ivary duct was reported by Kussmaul. In that report, swell-

ing of the parotid gland, mucofibrinous plug excretion, and

a large number of eosinophilic leukocytes in the patient’s

saliva; accordingly, allergy was suspected as the cause of

the condition.4) As we know, there have been a few pub-

lished cases of sialodochitis fibrinosa. Since then, sialodo-

chitis occurring mainly in the salivary duct has been re-

ferred to as ‘Kussmaul disease’. When sialodochitis fibrinosa

is caused by salivary duct stricture, it can lead to degenera-

tive changes of the salivary glands and become severe.

Regarding the treatment and management of sialodochitis

fibrinosa, drainage, sialodochoplasty, and parotidectomy are

commonly used. As surgical procedures can lead to nerve

damage and permanent functional loss, surgical methods

are not recommended as the first line of treatment.5) For de-

cades, many clinicians and researchers have sought an ap-

propriate and more conservative approach to the treatment

of sialodochitis fibrinosa. This study reports a case in which

a conservative ultrasound (US)-guided irrigation method

53Chi-Hyuk Ahn et al. US-Guided Irrigation in Sialodochitis

www.journalomp.org

was used to relieve the symptoms of sialodochitis fibrinosa.

CASE REPORT

A 30-year-old female patient visited the department of

oral medicine at the Kyung Hee University Dental Hospital

with the chief complaints of persistent left facial swell-

ing and discomfort on the left side of the face. Her primary

symptom was left facial swelling that was accompanied by

itching, tingling, and warmth that had persisted for 3 to 4

days. She reported that she had suffered the symptoms for

the last 1 year, and that her symptoms were getting worse

over time. During oral and clinical examinations of the

oral cavity and face, mild swelling and warmth were noted

on the left side of the face, including in the parotid gland

area. Mild induration was felt at the corresponding site.

Therefore, we made a tentative diagnosis of sialadenitis of

the left parotid gland.

For the precise investigation of the left parotid gland, si-

aloscintigraphy was performed following the patient’s first

visit (Fig. 1). From the sialoscintigraphic image, abnormal

salivation in the left parotid gland was observed. Because

abnormal findings were observed, we decided to perform

another imaging modality, sialography. Therefore, sialogra-

phy of the parotid gland was subsequently performed, and

showed severe stricture of the anterior region of the left

parotid duct bifurcation (Fig. 2A). A sausage-string shape,

which is a typical of sialodochitis, usually resulted from re-

peated stricture and expansion of ducts of salivary glands.

In addition, it was found on the posterior part to the stric-

ture site.6) Although we did not observe a mucus plug with

fibroblasts at the orifice of the left parotid gland using

pathologic diagnostic methods, we made a putative diag-

nosis of sialodochitis of the left parotid gland based on the

clinical and imaging findings.

Intraductal irrigation of the left parotid gland was per-

formed in duplicate for 9 months. For the first three

months, irrigation of the parotid gland was done at a fre-

quency of once per month. The irrigation was then repeated

three times at a frequency of once per two months. In total,

we performed intraductal irrigation 6 times. All of the irri-

gation procedures were performed under US guidance using

the following detailed procedures: an initial US scan was

conducted to evaluate the salivary gland duct and paren-

chymal states with a GE LOGIQ5 Expert device (GE Medical

systems, Milwaukee, WI, USA) equipped with a linear scan-

ner 12 L (frequency range: 12 MHz, footprint: 45×10 mm).

Next, a polyethylene catheter was cannulated through

the orifice of the left parotid gland under US guidance.

Intraductal irrigation of the parotid gland was performed

three times during every procedure using a normal saline

solution. After each procedure, amoxicillin (250 mg, three

times per day for three days) was prescribed as a prophy-

lactic antibiotic, and loxoprofen sodium (60 mg, three times

ANT Lt-Lat Rt-Lat

5 min

Post lemon

A B

C D

15 min

ANT Lt-Lat Rt-Lat

ANT Lt-Lat Rt-Lat ANT Lt-Lat Rt-Lat

30 min

Fig. 1. Results of sialoscintigraphy. Sialoscintigraphy results. (A) 5 min after injection. (B) 15 min after injection. (C) 30 min after injection. (D)

After stimulation with lemon juice. The images show the stored saliva 5, 15, and 30 minutes after contrast media (Tc99m) was injected, as well

as the release of saliva following lemon juice stimulation. ANT, anterior; LT, left; LAT, lateral; Rt, right.

54 J Oral Med Pain Vol. 43 No. 2, June 2018

www.journalomp.org

per day for three days) was prescribed as an anti-inflam-

matory analgesic drug. Although mild swelling was present

for the first 2 months after treatment, it gradually subsid-

ed. After 9 months, the diameter of the expanded sausage

string-shaped parotid duct had visibly decreased (Fig. 2B).

The warmth and induration disappeared after three months.

We followed-up with the patient after 12 months, at which

point she reported that there had been no recurrence of

symptoms.

DISCUSSION

Salivary gland disorders can originate from various

causes. Recurrent chronic sialadenitis is commonly caused

by infection of the oral cavity or blockage by salivary

stones.7,8) Inflammation of the salivary ducts is typically ac-

companied by inflammatory changes of the salivary paren-

chyma; this condition is defined as sialadenitis. However,

inflammation can occur only in localized salivary ducts.9,10)

In our case, the salivary gland parenchyma was not func-

tionally impaired, and there were no abnormal changes

in found on imaging. Therefore, when a patient visits the

clinic with swelling, discomfort, and other neuropathologic

symptoms in the facial area, the clinician should include si-

alodochitis as a differential diagnosis.

Although the pathogenesis of sialodochitis is not clearly

known, many researchers have reported that it is caused

by hyper-allergic reactions and inflammation caused by

autoimmunity.2,11) Because the systemic overexpression of

inflammatory factors was also not observed in our case,

3.86 mm2.89 mm

6 cm 5 cm

A B

Fig. 2. Results of sialography. (A) Before

treatment. (B) After treatment (9

months later). Sialography imaging of

the left parotid gland before and after

saline irrigation. The imaging shows

that the diameter of the duct decreased

after irrigation of the parotid gland.

Table 1. The results of laboratory testing

Parameter Value Normal range

WBC (103/μL) 5.26 4.0-10.0RBC (106/μL)a 3.95c 4.0-5.4c

Hgb (g/dL) 12.9 12-16

Hct (%) 37.5 37-47

MCV (fL) 95.2 81-99

MCH (pg)b 32.8c 27-31c

MCHC (g/dL) 34.5 33-37

Platelet count (103/μL) 254 150-350ESR (mm/h) 18 0-20

Differential count LUC (%) 1.9 0-4.5

Seg. neutrophil (%) 61.1 40-74

Lymphocyte (%) 28.5 19-48

Monocyte (%) 6.2 4-9

Eosinophil (%) 1.8 0-7

Basophil (%) 0.5 0-1.5

% Polymorphonuclear cell (%) 65.2

IgG (mg/dL) 1,530 694-1,618

IgA (mg/dL) 192 68-378

IgM (mg/dL) 121 60-263

IgE (IU/mL) 69.9 -100

C3 (mg/dL)a 73.2c 88-201c

C4 (mg/dL) 20.5 16-47

Anti-CCP (units/mL)

55Chi-Hyuk Ahn et al. US-Guided Irrigation in Sialodochitis

www.journalomp.org

autoimmunity was excluded as a possible cause of the dis-

ease. In addition, we confirmed the status of the patients

with non-allergic sialodochitis. As it was difficult to associ-

ate the disease with allergic reaction based on the patient’s

medical records and blood test results, allergic sialodochitis

was excluded secondarily (Table 1).

Conservative treatments, including irrigation and medi-

cation, are generally accepted as first-line treatment meth-

ods and are recommended for patients with sialodochitis.

However, surgical methods, including sialodochoplasty and

parotidectomy, have also been performed and confirmed as

having advantages for symptom relief. Among the surgical

approaches, sialodochoplasty can widen the diameter of the

salivary ducts through which the saliva exits. However, par-

tial or complete parotidectomy can lead to permanent func-

tional loss of the salivary gland and can damage the associ-

ated nerves.12) For our patient, the conservative US-guided

irrigation method was used to find the site of duct stricture

and minimize the leakage that can occur during irrigation.

An advantage of US guidance is that the dilation status of

ducts can be checked in real time.13) In our case, the ducts

in the corresponding site and salivary gland parenchyma

were retro-irrigated using a saline solution. To prevent pos-

sible additional infections, antibiotics were prescribed pre-

ventively after the procedure. Our method was found to be

effective and safe for the patient.

Once a diagnosis is made for a disease of the salivary

gland, a clinician can consider the various approaches to

treatment and care. The US-guided irrigation method for

the treatment of salivary gland diseases is a conservative

approach for structures of the salivary glands and may be

considered before surgical procedures. The US-guided ir-

rigation method used in this study may be an excellent

method of treatment that can eliminate the possibility of

predictable and severe side effects. However, the insufficient

number of clinical cases and a poor understanding of the

steps and frequency of the procedure preclude such a con-

clusion from being drawn. Further confirmation and studies

are needed to establish accurate methods of diagnosis and

treatment.

CONFLICT OF INTEREST

No potential conflict of interest relevant to this article

was reported.

ACKNOWLEDGEMENTS

The authors extend their special thanks to Jung-

Pyo Hong at the department of Orofacial Pain and Oral

Medicine, Kyung Hee University Dental Hospital.

REFERENCES

1. Ugga L, Ravanelli M, Pallottino AA, Farina D, Maroldi R. Diag-nostic work-up in obstructive and inflammatory salivary gland disorders. Acta Otorhinolaryngol Ital 2017;37:83-93.

2. Ray A, Burgin SJ, Spector ME. A rare case of Kussmaul Disease (Sialodochitis Fibrinosa). J Case Rep Med 2015;4:235894.

3. Bialek EJ, Jakubowski W, Zajkowski P, Szopinski KT, Osmolski A. US of the major salivary glands: anatomy and spatial re-lationships, pathologic conditions, and pitfalls. Radiographics 2006;26:745-763.

4. Baer AN, Okuhama A, Eisele DW, Tversky JR, Gniadek TJ. Eo-sinophilic sialodochitis: redefinition of ‘allergic parotitis’ and ‘sialodochitis fibrinosa’. Oral Dis 2017;23:840-848.

5. Owen ER, Banerjee AK, Kissin M, Kark AE. Complications of pa-rotid surgery: the need for selectivity. Br J Surg 1989;76:1034-1035.

6. Koch M, Iro H. Salivary duct stenosis: diagnosis and treatment. Acta Otorhinolaryngol Ital 2017;37:132-141.

7. Epker BN. Obstructive and inflammatory diseases of the major salivary glands. Oral Surg Oral Med Oral Pathol 1972;33:2-27.

8. Marchal F, Dulguerov P. Sialolithiasis management: the state of the art. Arch Otolaryngol Head Neck Surg 2003;129:951-956.

9. Hayashi K, Onda T, Ohata H, Takano N, Shibahara T. Case of suspected sialodochitis fibrinosa (Kussmaul’s disease). Bull Tokyo Dent Coll 2016;57:91-96.

10. Flores RBJ, Brea AB, Sanabria SAA, et al. Sialodochitis fibrinosa (kussmaul disease) report of 3 cases and literature review. Medi-cine (Baltimore) 2016;95:e5132.

11. Chikamatsu K, Shino M, Fukuda Y, Sakakura K, Furuya N. Recur-ring bilateral parotid gland swelling: two cases of sialodochitis fibrinosa. J Laryngol Otol 2006;120:330-333.

12. Bates D, O’Brien CJ, Tikaram K, Painter DM. Parotid and subman-dibular sialadenitis treated by salivary gland excision. Aust N Z J Surg 1998;68:120-124.

13. Bartlett LJ, Pon M. High-resolution real-time ultrasonography of the submandibular salivary gland. J Ultrasound Med 1984;3:433-437.