Fistula of the Submandibular Gland Presenting as a Painful Mass in the Neck: A Rare Case

(1)

Brief Report

Vol. 32, N o. 1, 2020 81

Received July 9, 2018, Revised February 13, 2019, Accepted for publication June 11, 2019

Corresponding author: Hyun-Joo Lee, Department of Dermatology, Pusan National University Hospital, 179 Gudeok-ro, Seo-gu, Busan 49241, Korea. Tel:

82-51-240-7338, Fax: 82-51-245-9467, E-mail: sapphire81@hanmail.net ORCID: https://orcid.org/0000-0002-1088-0975

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/li- censes/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

https://doi.org/10.5021/ad.2020.32.1.81

Fistula of the Submandibular Gland Presenting as a Painful Mass in the Neck: A Rare Case

Dae-Lyong Ha

^1,2

, Hyang-Suk Ryu

¹

, Gun-Wook Kim

^1,2

, Hoon-Soo Kim

^1,2

, Byung-Soo Kim

^1,2

, Hyun-Chang Ko

^1,3

, Moon-Bum Kim

^1,2

, Hyun-Joo Lee

^1,2

1Department of Dermatology, Pusan National University School of Medicine, ²Biomedical Research Institute, Pusan National University Hospital, Busan, ³Research Institute for Convergence of Biomedical Science and Technology, Pusan National University Yangsan Hospital, Yangsan, Korea

Dear Editor:

Salivary gland fistula is an abnormal pathway involving the salivary gland or duct. Accurate diagnosis is achieved by observing clinical manifestations, radiographic findings, and surgical exploration. Dermatologists may be unfamiliar with salivary gland fistulas presenting as parotid fistulas or fistulas of the submandibular gland because there are no such case reports in dermatologic literatures. We report a rare case of fistula of the submandibular gland and provide a brief review of the literature.

We received the patient’s consent form about publishing all photographic materials. A 53-year-old female presented with erythematous plaques and a painful, tender subcuta- neous mass on the left side of the neck (Fig. 1A). She had been diagnosed with sialadenitis a week before and had a sore throat after undergoing endoscopy. We initially diagnosed her with pyoderma and performed a skin biopsy and computed tomography (CT) scan of the neck. Histopa- thologic results showed septal fibrosis with perivascular inflammatory infiltration in the mid-dermis and subcuta- neous fat layer and no evidence of bacterial infection, in- cluding tuberculosis, on tissue culture and special stain- ings of Ziehl–Neelsen, periodic acid Shiff and gram stain (Fig. 1B, C). The CT scan revealed fuzzy enhancing tubular structures from the submandibular gland to the anterior part of the platysma muscle, consistent with symptoms of inflammation and fistula (Fig. 1D∼F). Routine blood ex-

amination showed no significant findings. Based on these findings, we diagnosed her with fistula of the submandibular gland. It improved with conservative treatment using systemic antibiotics under close observation.

Salivary gland fistula usually originates from the parotid gland although it also rarely arises from the submandibular gland¹. Most previous reports were limited to parotid gland fistula; only 13 cases describing fistula of the submandibular gland were found by using keyword, ‘submandibular gland fistula’, ‘submandibular fistula’, ‘submandibular’ and ‘fistula’ and compatible with submandibular gland fistula on PubMed searching (Table 1)^2-5. These cases list- ed causes of trauma¹, sialadenitis², calculi³, or structural anomaly⁴. Salivary gland fistula is caused by mechanical obstruction to the drainage of the salivary duct via natural pathways such as calculi or tumors pressing on the duct.

Because our case showed no such anomaly, we thought it developed as a complication of sialadenitis. The precise pathogenesis of sialadenitis and fistula formation is cur- rently unknown. The fistula tract in most cases communi- cated with other space such as paranasal sinus through in- ternal openings or had external openings on the skin or or- al cavity. Here, it connected the infected submandibular gland to the anterior part of the platysma muscle, thus penetrating the muscle. Only two of the previously reported 13 cases described a fistula with skin lesions show- ing cutaneous openings¹. The opening in this case did not

(2)

Brief Report

82 Ann D ermatol

Table 1. Summary of case reports of submandibular gland fistula

Author (year) Sex/age (yr) Cause Skin lesion Treatment

Reuther and Hausamen² (1973) F/24 Chronic sialoadenitis ND Excision

De Geus et al. (1976) M/8 High-voltage electrical burn ND Excision

Knezevic et al. (1983) M/14 Foreign body ND Excision

Asfar et al.³ (1989) M/55 Salivary calculi Swelling with fistula opening Excision

Asfar et al.³ (1989) M/43 Salivary calculi Tender nodule Excision

Asfar et al.³ (1989) M/75 Salivary calculi Tender nodule Excision

Bhuvanesh et al. (1995) M/41 Gun shot wound ND Antibiotics

Avik et al. (2006) M/10 Trauma Fistulous opening Excision

Kulkarini et al. (2015) M/45 Malignant tumor Swelling with fistula opening Excision

Kieliszak et al.⁴ (2015) M/20 Structural abnormality ND Excision

Stanford et al. (2015) F/56 Salivary calculi ND ND

Kusunoki et al.⁵ (2017) M/72 Salivary calculi Erythematous nodule with skin defect Excision

Present case F/53 Sialoadenitis Erythematous nodule Antibiotics

F: female, M: male, ND: not described.

Fig. 1. Erythematous nodule on the left side of the neck (A). Histopa- thologic findings showed upper dermal edema with lymphocytes and neutrophils infiltration in the mid to deep dermis, hematoxylin- eosin (H&E) ×20 (B), H&E, ×400 (C). Computed tomography scan rea- vled fuzzy enhancing tubular structures (4 cm) from the left submandibular gland to the anterior aspect of the plastysma muscle (*), consistent with fistula and inflamation (D∼F).

(3)

Brief Report

Vol. 32, N o. 1, 2020 83

Received March 4, 2019, Revised May 15, 2019, Accepted for publication June 18, 2019

Corresponding author: Jin Park, Department of Dermatology, Chonbuk National University Medical School, 20 Geonji-ro, Deokjin-gu, Jeonju 54907, Korea. Tel:

82-63-250-2745, Fax: 82-63-250-1970, E-mail: airmd@jbnu.ac.kr ORCID: https://orcid.org/0000-0002-8830-5479

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons. org/li- censes/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

adhere to the skin, but was connected to the platysma muscle, presenting an erythematous nodule. It might be easily misdiagnosed, as it is extremely rare and the skin lesion may present without any cutaneous opening. Derma- tologists should keep in mind the possibility of fistula of the submandibular gland when examining painful masses in the neck.

CONFLICTS OF INTEREST

The authors have nothing to disclose.

ORCID

Dae-Lyong Ha, https://orcid.org/0000-0002-2268-4795 Hyang-Suk Ryu, https://orcid.org/0000-0002-1697-397X Gun-Wook Kim, https://orcid.org/0000-0003-1599-7045 Hoon-Soo Kim, https://orcid.org/0000-0002-7649-1446 Byung-Soo Kim, https://orcid.org/0000-0003-0054-8570 Hyun-Chang Ko, https://orcid.org/0000-0002-3459-5474

Moon-Bum Kim, https://orcid.org/0000-0003-4837-0214 Hyun-Joo Lee, https://orcid.org/0000-0002-1088-0975

REFERENCES

1. Jana AK, Jaswal A, Sikder B, Jana U, Nandi TK. Fistula of submandibular gland-a rare presentation. Indian J Otolaryngol Head Neck Surg 2006;58:393-394.

2. Reuther J, Hausamen JE. [Extraoral salivary fistula of the submandibular gland]. Zahnarztl Prax 1973;24:134-135. German.

3. Asfar SK, Steitiyeh MR, Abdul-Amir R. Giant salivary calculi:

an orocervical fistula caused by a submandibular gland cal- culus. Can J Surg 1989;32:295-296.

4. Kieliszak CR, Gill A, Faiz M, Joshi AS. Submandibular ductal fistula: an obstacle to sialendoscopy. JAMA Otolaryngol Head Neck Surg 2015;141:373-376.

5. Kusunoki T, Homma H, Kidokoro Y, Yanai A, Hara S, Kobayashi Y, et al. Cervical fistula caused by submandibular sialolithiasis. Clin Pract 2017;7:985.

https://doi.org/10.5021/ad.2020.32.1.83

Pityriasis Amiantacea: An Epidemiologic Study of 44 Cases in Korean Patients

Hyun-Bin Kwak

¹

, Seok-Kweon Yun

^1,2

, Han-Uk Kim

^1,2

, Jin Park

^1,2

1Department of Dermatology, Chonbuk National University Medical School, ²Research Institute of Clinical Medicine of Chonbuk National University-Biomedical Research Institute of Chonbuk National University Hospital, Jeonju, Korea

Dear Editor:

Pityriasis amiantacea (PA) is a unique clinical skin con- dition characterized by thick, asbestos-like, adherent scales that engulf the scalp hairs¹. It has been reported as a clinical manifestation, or sequela, of various inflammatory or

infectious diseases of the scalp^2-4. Although it is occasion- ally seen in clinical practice, the data on PA are scarce in the literature.

We investigated the epidemiologic and clinical character- istics of PA. We retrospectively analyzed a series of 44 PA