|Year : 2015 | Volume
| Issue : 3 | Page : 101-104
Adenoid cystic carcinoma of the mandible with lung metastasis
Eren Yildirim1, Mesut Sivri2, Omer Demirtas3, Mehmet Ertugrul Çiftçi4, Gulen Kamak5
1 Department of Oral and Maxillofacial Radiology, Faculty of Dentistry, Kirikkale Univrsity, Kirikkale, Turkey
2 Department of Radiology, Faculty of Medicine, Selcuk University, Konya, Turkey
3 Department of Oral and Maxillofacial Radiology, Faculty of Dentistry, Katip Celebi University, Izmir, Turkey
4 Department of Oral and Maxillofacial Radiology, Faculty of Dentistry, Akdeniz University, Antalya, Turkey
5 Department of Periodontolgy, Faculty of Dentistry, Kirikkale University, Kirikkale, Turkey
|Date of Web Publication||27-Nov-2015|
Dr. Mehmet Ertugrul Çiftçi
Department of Oral and Maxillofacial Radiology, Faculty of Dentistry, Akdeniz University, Antalya
Source of Support: None, Conflict of Interest: None
Adenoid cystic carcinoma (ACC) is a malignant epithelial tumor of salivary glands. A 53-year-old man with intraosseous ACC in the mandible presented to our Faculty of Dentistry. Clinical examination revealed the swelling and enlarged local lymph nodes in the right mandibular area. After histopathological and radiological examination, ACC in the right mandibular molar area with lung metastasis was reported. We also review the literature.
Keywords: Adenoid cystic carcinoma, lung metastasis, mandible
|How to cite this article:|
Yildirim E, Sivri M, Demirtas O, Çiftçi ME, Kamak G. Adenoid cystic carcinoma of the mandible with lung metastasis. J Oral Maxillofac Radiol 2015;3:101-4
|How to cite this URL:|
Yildirim E, Sivri M, Demirtas O, Çiftçi ME, Kamak G. Adenoid cystic carcinoma of the mandible with lung metastasis. J Oral Maxillofac Radiol [serial online] 2015 [cited 2020 Oct 26];3:101-4. Available from: https://www.joomr.org/text.asp?2015/3/3/101/170623
| Introduction|| |
Adenoid cystic carcinoma (ACC), which was formerly known as cylindroma, is a malignant epithelial tumor characterized by slow growth, late onset of metastasis, and poor prognosis. It commonly affects adults, with a peak incidence in the fourth to sixth decades.  Palatal minor salivary glands, parotid, and submandibular glands are generally affected.
Very rarely, ACC may arise centrally within the jawbones, usually in the posterior mandible of adults, presenting with pain due to the perineural infiltration.  The mandible is more commonly involved than the maxilla. About 40-60% of patients develop distant metastases to lung, bone, and soft tissues. 
The aim of this study is to report the histopathological, clinical, and radiological features of a case of ACC occurring within the mandible of an adult Turkish man who also showed lung metastases, to examine the differential diagnostic features and also review the literature [Table 1]. ,,,,,,,,,,,,,,,,,,,,,,,
|Table 1: Published data on 24 patients with primary intraosseous adenoid cystic carcinoma of the maxillo-mandibulary region|
Click here to view
| Case Report|| |
A 53-year-old man was referred to Ataturk University, Faculty of Dentistry, Department of Oral and Maxillofacial Radiology of Ataturk University, Faculty of Medicine with swelling in the right molar region that started 6 months ago. Physical examination of the face revealed facial asymmetry and enlarged local lymph nodes. The swelling was nonpulsatile and nonfluctuating. On the intraoral examination, the vestibular mucosa along the right lower molar area and oropharynx were hyperemic and painful upon palpation. The patient had paresthesia in the right mandibular molar area. We were informed that the tumor region was edentolous for a long time. Orthopantomography revealed a radiolucent image in the area of the right lower molar involving the mandibular canal [Figure 1]. To make detailed imaging, computer tomography (CT) with contrast was performed. CT showed an irregular osteolytic lesion which had caused bone destruction and expansion [Figure 2] and [Figure 3]. The lesion was measured as 79 × 70 × 65 mm in the right mandibular region and bilateral cervical lymphadenopathy that are approximately 14 × 6 mm in diameter were detected in the neck and submandibular area on CT scans. An incisional biopsy was performed. The tumor cells had hyalinized eosinophilic material and numerous cyst-like spaces showing a classical Swiss cheese pattern. ACC of the mandible was reported on histopathological examination. Positron emission tomography CT scans examined for metastatic assessment and lung metastasis was revealed in the right part of the lung [Figure 4]. The patient was referred to the medical oncology department where the surgery and radiotherapy treatment planned.
|Figure 1: Orthopantomography showing a radiolucent lesion in the right lower molar area involving the mandibular canal|
Click here to view
|Figure 2: Axial computer tomography scan showing expansion, bone destruction, and facial asymmetry in the right mandibular molar region|
Click here to view
|Figure 3: Coronal computer tomography scan showing lytic lesion in right mandibular molar region|
Click here to view
|Figure 4: Positron emission tomography computer tomography view showing metastasis in the right lung field|
Click here to view
| Discussion|| |
The incidence of ACC varies according to the site. It comprises about 1.6-2.2% of all tumors and 16% of malignant tumors of the parotid glands.  It may rarely arise centrally within the jawbones.  Although lung metastases were frequently revealed in patient with salivary gland type ACC, there are only a few cases of ACC in the literature reporting lesions arising centrally within the mandible with lung metastases. ,
ACC was first described by Theodor Bilroth in 1856. He explained its histologic features and identified the long amorphous compartments as cylinders. Thus, the term "cylindroma" was first used for the lesion. It is only recently that the tumor has been renamed as ACC by Spies in 1930. , Until the 1940s, the tumor was thought to be a benign variant of the mixed salivary gland tumor. The malignant nature of this tumor was explained by Dockerty and Mayo in 1943. 
Some theories have been conceived to clarify the reason of malignant salivary gland tumors arising within centrally jawbones:
- Entrapment of ectopic salivary gland tissues in jawbones,
- Neoplastic transformation of cyst epithelium, and
- Neoplastic transformation of sinus epithelium.
Moreover, some histological patterns of growth have been described.  The ordinary ACC has a cribriform pattern. Besides, some have a predominantly tubular pattern and the other have a solid pattern. The cribriform and tubular growth patterns frequently display better prognosis. However, the solid areas indicate an aggressive clinical course. , Hence, it was thought that such histologic grading offers valuable prognostic information.  In the present case, the cribriform pattern was observed in histological examination of the lesion.
Although central lesions may occur at any age with no gender predilection, salivary gland lesions occur more commonly in middle-aged females.  Central lesions frequently arise in posterior mandible, but seldomly it occurs in the maxilla with pain, swelling, rarely numbness, and paresthesia being as the typical primary presentations. ,, The characteristic features include late onset, slow growth, insidious spread of tissues, perineural invasion, and distant metastasis.  In our case report, it was detected in a middle-aged male with the involvement of mandible and lung.
The regional lymph nodes were affected rarely in patients with ACC. Howbeit, distant metastases, particularly to lung and bone is more common and unpredictable.  In our case, a thorough metastatic work-up was carried out, which revealed both regional lymph node and distant metastasis.
Metastatic tumors in the lung which stems from the jawbones will frequently monitor as multiple radiopaque lesions on CT images. However, we monitored only a single tumor in the right lower lung field in our case. Hence, we inferred that the lesion may have been the initial stage of metastasis to the lung. This was consistent with the report of Grillet et al. 
The treatment includes radical surgery with postoperative radiotherapy. This approach appears to reduce the recurrence rates significantly compared with surgery alone, but tumors with distant metastasis usually have a poor prognosis. , Appropriate diagnosis is very important to prevent distant metastasis and provide more decent treatment prognosis. Exhaustive studies should be designed for prompt diagnosis and suitable treatment planning.
| Conclusion|| |
Primary or central salivary gland tumors located in mandible are infrequent, and the origin of this type of tumor is still unknown. To our knowledge, only a few reported cases were found in the literature which displays similar features of this type of lesions. , More reports should be presented for prompt diagnosis. ACC should be considered in the differential diagnosis in posterior mandibular lytic and destructive lesions characterized by swelling.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Al-Sukhun J, Lindqvist C, Hietanen J, Leivo I, Penttilä H. Central adenoid cystic carcinoma of the mandible: Case report and literature review of 16 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;101:304-8.
Capodiferro S, Scully C, Macaita MG, Lo Muzio L, Favia G, Mairoano E. Bilateral intraosseous adenoid cystic carcinoma of the mandible: Report of a case with lung metastases at first clinical presentation. Oral Dis 2005;11:109-12.
Shamim T, Varghese VI, Shameena PM, Sudha S. Primary intraosseous adenoid cystic carcinoma of the mandible with lung metastasis: A case report. J Oral Sci 2008;50:95-8.
Bumstead WD. Cylindroma of the mandible. Oral Surg 1955;8:546.
Bradley JC. A case of cylindroma of the mandible. Br J Oral Surg 1968;5:186-93.
Hämori J, Krasznai G. A mandibula cylindromaja [Hungarian]. Orv Hetil 1969;5:186.
Dhawan IK, Bhargava S, Nayak NC, Gupta RK. Central salivary gland tumors of jaws. Cancer 1970;26:211-7.
Slavin G, Mitchell RM. Adenoid cystic carcinoma of the mandible. Br J Surg 1971;58:546-8.
Shin MS, Kim HP, Kim ZS, Yuh IH, Chang HR, Chung KK. Adenoid cystic carcinoma of the mandible. Taehan Chikkwa Uisa Hyophoe Chi 1972;10:241-5.
Burkes EJ Jr. Adenoid cystic carcinoma of the mandible masquerading as periapical inflammation. J Endod 1975;1:76-8.
Yoshimura Y, Hasegawa K, Wada T, Fujita K, Kawakatsu K. Metastasis of adenoid cystic carcinoma of the mandible to the gasserian ganglion. J Am Dent Assoc 1978;96:469-73.
Mushimoto K, Hashimoto Y, Tabuchi M. Central adenoid cystic carcinoma of the mandible: Report of a case. Jpn J Oral Surg 1978;24:973.
Kaneda T, Mizuno N, Takeuchi M, Yamashita T. Primary central adenoid cystic carcinoma of the mandible. J Oral Maxillofac Surg 1982;40:741-5.
Gingell JC, Siegel MA. Adenoid cystic carcinoma of the mandible. J Am Dent Assoc 1983;107:600-2.
Hirota J, Osaki T. Primary central adenoid cystic carcinoma of the mandible. J Oral Maxillofac Surg 1989;47:176-9.
Brookstone MS, Huvos AG, Spiro RH. Central adenoid cystic carcinoma of the mandible. J Oral Maxillofac Surg 1990;48:1329-33.
Clark JM, Triana RJ, Meredith SD. Uncontrolled central adenoid cystic carcinoma: Case report. Ear Nose Throat J 2000;79:784, 785-6.
Favia G, Maiorano E, Orsini G, Piattelli A. Central (intraosseous) adenoid cystic carcinoma of the mandible: Report of a case with periapical involvement. J Endod 2000;26:760-3.
Chen YK, Chen CH, Lin CC, Hsue SS, Lin YR, Lin LM. Central adenoid cystic carcinoma of the mandible manifesting as an endodontic lesion. Int Endod J 2004;37:711-6.
Mahomed F, Altini M, Meer S, Rikhotso E, Pearl C. Central adenoid cystic carcinoma of the mandible with odontogenic features: Report of a case. Head Neck 2009;31:975-80.
Carlos-Bregni R, Vidaurre EC, Carolina Netto A, León JE, Almeida OP. Primary intraosseous adenoid cystic carcinoma of the mandible: Histopathological and immunohistochemical analysis. Pathol Oncol Res 2009;15:659-64.
Grimm M, Henopp T, Hoefert S, Schaefer F, Kluba S, Krimmel M, et al.
Multiple osteolytic lesions of intraosseous adenoid cystic carcinoma in the mandible mimicking apical periodontitis. Int Endod J 2012;45:1156-64.
Deshpande PS, Chintamaneni RL, Sujanamulk B, Prabhat MP, Gummadapu S. Intraosseous adenoid cystic carcinoma of maxilla: A rare case report. Contemp Clin Dent 2013;4:239-42.
Vinuth D, Agarwal P, Dhirawani RB, Dube G. Atypical case of primary intraosseous adenoid cystic carcinoma of mandible. J Oral Maxillofac Pathol 2013;17:436-9.
Ren ZH, Chickooree D, Liu JB, Wu HJ. Primary intraosseous ACC of mandible of possible salivary origin: A rare clinical entity. Int J Surg Case Rep 2014;5:222-5.
Santos Tde S, Melo DG, Gomes AC, Andrade ES, de Oliveira e Silva ED. Adenoid cystic carcinoma of mandible. Braz J Otorhinolaryngol 2011;77:807.
Fordice J, Kershaw C, El-Naggar A, Goepfert H. Adenoid cystic carcinoma of the head and neck: Predictors of morbidity and mortality. Arch Otolaryngol Head Neck Surg 1999;125:149-52.
de Kerviler E, Bely N, Laccourreye O, Clément O, Halimi P, Frija G. The aryepiglottic fold as a rare location of adenoid cystic carcinoma. AJNR Am J Neuroradiol 1995;16:1375-7.
Li Y, Li LJ, Huang J, Han B, Pan J. Central malignant salivary gland tumors of the jaw: Retrospective clinical analysis of 22 cases. J Oral Maxillofac Surg 2008;66:2247-53.
Orhan K, Yuksel Y, Gorur D. Solid adenoid cystic carcinoma of maxilla: A case report. Clin Dent Res 2006;30:42-7.
Grillet B, Demedts M, Roelens J, Goddeeris P, Fossion E. Spontaneous regression of lung metastases of adenoid cystic carcinoma. Chest 1984;85:289-91.
[Figure 1], [Figure 2], [Figure 3], [Figure 4]