Received: 20 February 2013 Accepted: 6 February 2014 Disponible en ligne 24 March 2014
Available online at
ScienceDirect www.sciencedirect.com
Review
Intra-mandibular adenoid cystic carcinoma Carcinome ade´noı¨de kystique intra-mandibulaire A. Bouaichia, S. Aimad-Eddinea, X.-A. Mommersa, B. Ellab, N. Zwetyengaa,* a Department of Oral and Maxillofacial Surgery, Plastic Esthetic and Reconstructive Surgery, Hand Surgery, Faculte´ de Me´decine, Universite´ de Bourgogne, Hoˆpital du Bocage, CHU de Dijon, 14, rue Gaffarel, BP 77908, 21079 Dijon Cedex, France b Department of Dentistry and Oral Health, Universite´ de Bordeaux 2, CHU de Bordeaux, Victor-Se´galen, place Ame´lie-Raba-Le´on, 33076 Bordeaux Cedex, France
Summary
Re´sume´
Intra-mandibular localization of adenoid cystic carcinoma is rare. This tumor is characterized by progressive local, regional, and distant aggressiveness. We reviewed the latest data on this rare type of cancer with a small number of reported cases, alack of consensus for its treatment, and its bad prognosis. ß 2014 Published by Elsevier Masson SAS.
Le carcinome ade´noı¨de kystique est une tumeur maligne dont la localisation intra-mandibulaire est rare. Il se caracte´rise par une agressivite´ progressive locore´gionale et a` distance. Nous faisons une mise au point sur ce type de cancer rare, avec un nombre re´duit de cas, l’absence de consensus pour son traitement et son mauvais pronostic. ß 2014 Publie´ par Elsevier Masson SAS.
Keywords: Mandible, Adenoid cystic carcinoma Mots cle´s : Mandibule, Carcinome ade´noı¨de kystique
Introduction Adenoid cystic carcinoma (ACC) is a rare malignant tumor of the salivary glands accounting for less than 1% of maxillofacial cancers [1]. The intra-osseous maxillo-mandibular location is very rare [2]. It often develops insidiously and its diagnosis is made at an advanced stage when management can be problematic [1,2]. Moreover, the risk of distant metastases and its neurotropic nature worsen the prognosis [2]. There is no consensus on the therapeutic management due to the rarity of cases. We had for aim to review the latest data regarding intramandibular adenoid cystic carcinoma (IACC).
Epidemiology Twenty-three cases of IACC were reported in the Englishlanguage and French-language literature since 1955 (table I) * Corresponding author. e-mail: [email protected] (N. Zwetyenga).
[1–10]. IACC generally occurs in the fifth decade of life (table I) at a mean age of 55.6 years (33–82 years) with a slight male predominance (13 men versus 10 women) [1,4–10].
Predisposing factors Alcohol consumption, smoking, and poor dental health are not predisposing factors for IACC.
Location IACC location is very rare and the tumor is usually located in the posterior region of the mandibular body and in the angle (22 of 23 cases) [4–9], but the parasymphyseal and symphyseal regions may also be involved (fig. 1) [5,10]. Several hypotheses have been made on the mechanism underlying the atypical intra-osseous location [7]: it could be due to either ectopic salivary glands or embryonic remnants of submandibular or sublingual glands trapped within the bone;
2213-6533/$ - see front matter ß 2014 Published by Elsevier Masson SAS. http://dx.doi.org/10.1016/j.revsto.2014.02.003 Rev Stomatol Chir Maxillofac Chir Orale 2014;115:100-104
100
Intra-mandibular adenoid cystic carcinoma
Table I Literature review on mandibular adenoid cystic carcinoma. Authors [references]
Sex
Age (years)
Clinical data
Location in the mandible
Treatments
Follow-up
1 - Santos et al. [4]
M
48
Pain – swelling
Molar region
Mandibulectomy and radiotherapy
Unknown
2 - Grimm et al. [5]
W
45
Pain – paresthesia – swelling
Bilateral body and symphyseal
Mandibulectomy Cervical curettage and radiotherapy
Alive at 3 years with metastases
3 - Shamin et al. [6]
M
45
Swelling and metastases at diagnosis
Angle and body
Chemotherapy
Died (unknown delay)
4 - Chen et al. [7]
M
56
Pain
Molar region
Refused treatment
Unknown
5 - Al Shukhun et al. [8]
W
80
Pain
Premolar
Mandibulectomy
Alive at 3 years without recurrence
6 - Gumgum et al. [9]
W
61
Swelling – cervical lymph-nodes
Retromolar
Mandibulectomy cervical curettage and radiotherapy
Unknown
7 - Clark et al. [10]
M
54
Swelling – cervical lymph-nodes
Parasymphyseal
Mandibulectomy cervical curettage and radiotherapy
Alive at 4.5 years without recurrence
8 - Bumstead [11]
M
54
Pain – swelling
Body
Surgery Radiotherapy
Lung metastasis (unknown delay)
9 - Bradley [12]
M
64
Trismus – pain – swelling
Angle
Surgery
Unknown
10 - Hamori et al. [13]
M
43
Otalgia
Body
Surgery
Lung metastasis (unknown delay)
11 - Shin et al. [14]
W
59
Trismus – pain
Angle
Unknown
Unknown
12 - Dahwan et al. [15]
W
40
Swelling
Ramus
Unknown
Lung metastasis (unknown delay)
13-Slavin et al. [16]
M
35
Swelling
Angle
Surgery
Unknown
14 - Burkes et al. [17]
W
50
Pain
Body
Surgery
Lung metastasis (unknown delay)
15 - Yoshimura et al. [18]
W
47
Paresthesia – loose teeth
Body
Radiotherapy
Unknown
16 - Mushimoto et al. [19]
M
24
Swelling
Body
Surgery
Unknown
17 - Kaneda et al. [20]
M
47
Swelling Loose teeth
Body
Surgery
Unknown
18 - Gingell et al. [21]
W
72
Pain
Body
Surgery
Alive at 7 years without recurrence
19 - Hirota et Osaki [22]
M
82
Swelling
Body
Radio-chemotherapy
Died (unknown delay)
20 - Brookstone et al. [23]
W
33
Pain – trismus
Body angle
Surgery and radiotherapy
Unknown
21 - Favia et al. [24]
W
46
Pain – swelling
Body
Surgery
Alive at 14 years without recurrence
22 - Chen et al. [25]
M
56
Pain
Body
Surgery
Unknown
23 - Our case
M
57
Pain – swelling – loose teeth
Symphyseal Body
Mandibulectomy, cervical curettage and radiotherapy
Alive at 6 months without recurrence
101
A. Bouaichi et al.
Rev Stomatol Chir Maxillofac Chir Orale 2014;115:100-104
Figure 1. Intraoral view of swelling in the mandible.
or to the malignant degeneration of mucus secreting cells normally found within the odontogenic epithelia [7].
Clinical signs Clinical signs are not specific because the evolution is slow and insidious. Swelling covered by healthy mucosa is often observed. The swollen mass is fixed to the mandible and associated with tooth mobility. Pain in the mouth is often a marker of severity because it reflects involvement of the underlying nerves. Weight loss may also be observed because of eating difficulties.
Radiological signs A panoramic dental X-ray is the initial examination; it reveals a poorly defined osteolytic lesion (fig. 2). CT scan is currently the key imagery revealing the tumoral mass, and whether the pre-mandibular soft tissues are involved or not; it can also reveal cervical lymph-node involvement (fig. 3). The examination can also be extended to the thorax, abdomen, and pelvis.
Figure 3. CT scan showing mass extending from the left body of the mandible to the symphyseal region with involvement of the surrounding soft tissues.
MRI can also reveal the lesion, and show the state of surrounding tissues, notably the involvement of the inferior alveolar nerve (an important prognostic criterion), and possible locoregional or distant metastases.
Contribution of panendoscopy Panendoscopy is not useful until surgery, because the tumor occurs in salivary glands.
Diagnosis IACC should be managed like cancer, given the clinical and radiological signs of malignancy and the impossibility of performing an extemporaneous examination. The diagnosis is confirmed by histology.
Histology
Figure 2. Panoramic dental X-ray showing left parasymphyseal osteolysis with diffuse lysis of the periodontium.
102
ACC is classified among malignant tumors of the salivary glands [3] and was first described by Robin and Laboulbene in 1853. Theoder Biltroth called it cylindroma in 1856, because of its cylindrical aspect when observed with a microscope [4]. Macroscopically [8], it is a non-encapsulated well-defined solid tumor. In terms of histology, there are 2 cell lines: epithelial cells and myoepithelial cells with a hyperchromatic, angular nucleus and scant cytoplasm.
Intra-mandibular adenoid cystic carcinoma
Three architectural forms have been described [8]: the tubular form includes tubes lined by a double layer of cells, with epithelial cells making up the internal layer and basal cells the outer layer; the cribriform structure has a micro-cystic aspect with the presence of basophilic mucoid and/or hyaline material; the solid form includes uniform basal cells (without tubes or micro-cysts). It is mandatory to screen for perineural involvement since this is a key factor for the prognosis. Only Ki67 is useful to differentiate between adenoid cystic carcinoma and polymorphous adenocarcinoma, for immunohistochemistry.
Treatment We recommend a multidisciplinary meeting including surgeons, radiotherapists, dentists, and chemotherapists, as for any type of cancer. The indication and the choice of therapy are always controversial [5]. Nonetheless, for several authors, surgery is the mainstay of management [1,3–5,8–10]. It consists in a radical resection of the lesion with wide resection margins (macroscopic margins of at least 1 cm) [3,8]. It often involves segmental mandibulectomy, extended to the underlying and overlying structures, depending on the tumor extension. Systematiccervical lymphadenectomy isrecommendedbymost authors, whatever the lymph-node status, and should be bilateral if the tumor is symphyseal, or has reached or extended beyond the median line. It should be homolateral in other cases. Complementary postoperative radiotherapy is performed in accordance with the most recent recommendations, given the radio susceptibility, for better local control in terms of recurrence; it is indicated whatever the surgical margins and pathology results (histological grade) are. Its lack of susceptibility to chemotherapy can be explained by its slow evolution; this explains why chemotherapy has been abandoned as the first-line treatment. However, it is indicated for strictly palliative care in case of metastases.
Prognosis
perineural involvement, vascular emboli; cervical lymph-node involvement; metastasis, especially distant metastasis.
Disclosure of interest The authors declare that they have no conflicts of interest concerning this article.
References [1]
[2]
[3]
[4]
[5]
[6]
[7]
[8]
[9]
[10]
[11] [12]
The prognosis is bad and is mainly impacted by the risk of distant metastasis, occurring in 40 to 60% of patients [5], and the tendency to relapse. Involvement of the underlying nerves often leads to incomplete tumor resection and thus to frequent local recurrence [4]. Metastases were described in most of the reported cases, especially in the lung [2,4,8,9]. The survival rate is estimated at 77% at 5 years and 57% at 10 years [10]. It is related to: the size of the tumor; the involvement of surrounding tissues (muscles, submandibular glands);
[13] [14]
[15] [16] [17]
Chummun S, McLean NR, Kelly CG, Dawes PJDK, Meikle D, Fellows S, et al. Adenoid cystic carcinoma of the head and neck. Br J Plast Surg 2001;54:476–80. Duberge´ T, Be´ne´zery K, Resbeut M, Azria D, Minsat M, Ellis S, et al. Carcinomes ade´noı¨des kystiques ORL : e´tude re´trospective multicentrique de 169 cas. Cancer Radiother 2012;16: 247–56. Gondivkar SM, Gadbail AR, Chole R, Parikh RV. Adenoid cystic carcinoma: a rare clinical entity and literature review. Oral Oncol 2011;47:231–6. Santos T de S, Melo DG, Gomes AC, Andrade ES, de Oliveira e Silva ED. Adenoid cystic carcinoma of mandible. Braz J Otorhinolaryngol 2011;77:807. Grimm M, Henopp T, Hoefert S, Schaefer F, Kluba S, Krimmel M, et al. Multiple osteolytic lesions of intra-osseous adenoid cystic carcinoma in the mandible mimicking apical periodontis. Int Endod J 2012;45:1156–64. Shamim T, Varghese VI, Shameena PM, Sudha S. Primary intraosseous adenoid cystic carcinoma of the mandible with lung metastasis. J Oral Sci 2008;50:95–8. 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. Al-Sukhun J, Lindqvist C, Hietanen J, Leivo I, Penttila ¨ 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. Gumgum S, Oner B, Ustay C. Rapidly growing adenoid cystic carcinoma in mandible: a case report. Oral Oncology Extra 2005;41:142–5. Clark JM, Triana RJ, Meredith SD. Uncontrolled central adenoid cystic carcinoma: case report. Ear Nose Throat J 2000; 79:784. Bumstead WD. Cylindroma of the mandible. Oral Surg Oral Med Oral Pathol 1955;8:546–55. Bradley JC. A case of cylindroma of the mandible. Br J Oral Surg 1968;5:186–93. Ha ¨mori J, Krasznai G. A mandibula cylindromaja. Orv Hetil 1969;5:302–3. 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. Dhawan IK, Bhargava S, Nayak NC, Gupta RK. Central salivary gland tumours of jaws. Cancer 1970;26:211–7. Slavin G, Mitchell RM. Adenoid cystic carcinoma of the mandible. Br J Surg 1971;58:546–8. Burkes Jr EJ. Adenoid cystic carcinoma of the mandible masquerading as periapical inflammation. J Endod 1975;1: 76–8.
103
A. Bouaichi et al. [18]
[19]
[20]
104
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;94: 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.
Rev Stomatol Chir Maxillofac Chir Orale 2014;115:100-104 [21] [22] [23]
[24]
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. 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;12:760–3.
Available online at
ScienceDirect www.sciencedirect.com
Review
Intra-mandibular adenoid cystic carcinoma Carcinome ade´noı¨de kystique intra-mandibulaire A. Bouaichia, S. Aimad-Eddinea, X.-A. Mommersa, B. Ellab, N. Zwetyengaa,* a Department of Oral and Maxillofacial Surgery, Plastic Esthetic and Reconstructive Surgery, Hand Surgery, Faculte´ de Me´decine, Universite´ de Bourgogne, Hoˆpital du Bocage, CHU de Dijon, 14, rue Gaffarel, BP 77908, 21079 Dijon Cedex, France b Department of Dentistry and Oral Health, Universite´ de Bordeaux 2, CHU de Bordeaux, Victor-Se´galen, place Ame´lie-Raba-Le´on, 33076 Bordeaux Cedex, France
Summary
Re´sume´
Intra-mandibular localization of adenoid cystic carcinoma is rare. This tumor is characterized by progressive local, regional, and distant aggressiveness. We reviewed the latest data on this rare type of cancer with a small number of reported cases, alack of consensus for its treatment, and its bad prognosis. ß 2014 Published by Elsevier Masson SAS.
Le carcinome ade´noı¨de kystique est une tumeur maligne dont la localisation intra-mandibulaire est rare. Il se caracte´rise par une agressivite´ progressive locore´gionale et a` distance. Nous faisons une mise au point sur ce type de cancer rare, avec un nombre re´duit de cas, l’absence de consensus pour son traitement et son mauvais pronostic. ß 2014 Publie´ par Elsevier Masson SAS.
Keywords: Mandible, Adenoid cystic carcinoma Mots cle´s : Mandibule, Carcinome ade´noı¨de kystique
Introduction Adenoid cystic carcinoma (ACC) is a rare malignant tumor of the salivary glands accounting for less than 1% of maxillofacial cancers [1]. The intra-osseous maxillo-mandibular location is very rare [2]. It often develops insidiously and its diagnosis is made at an advanced stage when management can be problematic [1,2]. Moreover, the risk of distant metastases and its neurotropic nature worsen the prognosis [2]. There is no consensus on the therapeutic management due to the rarity of cases. We had for aim to review the latest data regarding intramandibular adenoid cystic carcinoma (IACC).
Epidemiology Twenty-three cases of IACC were reported in the Englishlanguage and French-language literature since 1955 (table I) * Corresponding author. e-mail: [email protected] (N. Zwetyenga).
[1–10]. IACC generally occurs in the fifth decade of life (table I) at a mean age of 55.6 years (33–82 years) with a slight male predominance (13 men versus 10 women) [1,4–10].
Predisposing factors Alcohol consumption, smoking, and poor dental health are not predisposing factors for IACC.
Location IACC location is very rare and the tumor is usually located in the posterior region of the mandibular body and in the angle (22 of 23 cases) [4–9], but the parasymphyseal and symphyseal regions may also be involved (fig. 1) [5,10]. Several hypotheses have been made on the mechanism underlying the atypical intra-osseous location [7]: it could be due to either ectopic salivary glands or embryonic remnants of submandibular or sublingual glands trapped within the bone;
2213-6533/$ - see front matter ß 2014 Published by Elsevier Masson SAS. http://dx.doi.org/10.1016/j.revsto.2014.02.003 Rev Stomatol Chir Maxillofac Chir Orale 2014;115:100-104
100
Intra-mandibular adenoid cystic carcinoma
Table I Literature review on mandibular adenoid cystic carcinoma. Authors [references]
Sex
Age (years)
Clinical data
Location in the mandible
Treatments
Follow-up
1 - Santos et al. [4]
M
48
Pain – swelling
Molar region
Mandibulectomy and radiotherapy
Unknown
2 - Grimm et al. [5]
W
45
Pain – paresthesia – swelling
Bilateral body and symphyseal
Mandibulectomy Cervical curettage and radiotherapy
Alive at 3 years with metastases
3 - Shamin et al. [6]
M
45
Swelling and metastases at diagnosis
Angle and body
Chemotherapy
Died (unknown delay)
4 - Chen et al. [7]
M
56
Pain
Molar region
Refused treatment
Unknown
5 - Al Shukhun et al. [8]
W
80
Pain
Premolar
Mandibulectomy
Alive at 3 years without recurrence
6 - Gumgum et al. [9]
W
61
Swelling – cervical lymph-nodes
Retromolar
Mandibulectomy cervical curettage and radiotherapy
Unknown
7 - Clark et al. [10]
M
54
Swelling – cervical lymph-nodes
Parasymphyseal
Mandibulectomy cervical curettage and radiotherapy
Alive at 4.5 years without recurrence
8 - Bumstead [11]
M
54
Pain – swelling
Body
Surgery Radiotherapy
Lung metastasis (unknown delay)
9 - Bradley [12]
M
64
Trismus – pain – swelling
Angle
Surgery
Unknown
10 - Hamori et al. [13]
M
43
Otalgia
Body
Surgery
Lung metastasis (unknown delay)
11 - Shin et al. [14]
W
59
Trismus – pain
Angle
Unknown
Unknown
12 - Dahwan et al. [15]
W
40
Swelling
Ramus
Unknown
Lung metastasis (unknown delay)
13-Slavin et al. [16]
M
35
Swelling
Angle
Surgery
Unknown
14 - Burkes et al. [17]
W
50
Pain
Body
Surgery
Lung metastasis (unknown delay)
15 - Yoshimura et al. [18]
W
47
Paresthesia – loose teeth
Body
Radiotherapy
Unknown
16 - Mushimoto et al. [19]
M
24
Swelling
Body
Surgery
Unknown
17 - Kaneda et al. [20]
M
47
Swelling Loose teeth
Body
Surgery
Unknown
18 - Gingell et al. [21]
W
72
Pain
Body
Surgery
Alive at 7 years without recurrence
19 - Hirota et Osaki [22]
M
82
Swelling
Body
Radio-chemotherapy
Died (unknown delay)
20 - Brookstone et al. [23]
W
33
Pain – trismus
Body angle
Surgery and radiotherapy
Unknown
21 - Favia et al. [24]
W
46
Pain – swelling
Body
Surgery
Alive at 14 years without recurrence
22 - Chen et al. [25]
M
56
Pain
Body
Surgery
Unknown
23 - Our case
M
57
Pain – swelling – loose teeth
Symphyseal Body
Mandibulectomy, cervical curettage and radiotherapy
Alive at 6 months without recurrence
101
A. Bouaichi et al.
Rev Stomatol Chir Maxillofac Chir Orale 2014;115:100-104
Figure 1. Intraoral view of swelling in the mandible.
or to the malignant degeneration of mucus secreting cells normally found within the odontogenic epithelia [7].
Clinical signs Clinical signs are not specific because the evolution is slow and insidious. Swelling covered by healthy mucosa is often observed. The swollen mass is fixed to the mandible and associated with tooth mobility. Pain in the mouth is often a marker of severity because it reflects involvement of the underlying nerves. Weight loss may also be observed because of eating difficulties.
Radiological signs A panoramic dental X-ray is the initial examination; it reveals a poorly defined osteolytic lesion (fig. 2). CT scan is currently the key imagery revealing the tumoral mass, and whether the pre-mandibular soft tissues are involved or not; it can also reveal cervical lymph-node involvement (fig. 3). The examination can also be extended to the thorax, abdomen, and pelvis.
Figure 3. CT scan showing mass extending from the left body of the mandible to the symphyseal region with involvement of the surrounding soft tissues.
MRI can also reveal the lesion, and show the state of surrounding tissues, notably the involvement of the inferior alveolar nerve (an important prognostic criterion), and possible locoregional or distant metastases.
Contribution of panendoscopy Panendoscopy is not useful until surgery, because the tumor occurs in salivary glands.
Diagnosis IACC should be managed like cancer, given the clinical and radiological signs of malignancy and the impossibility of performing an extemporaneous examination. The diagnosis is confirmed by histology.
Histology
Figure 2. Panoramic dental X-ray showing left parasymphyseal osteolysis with diffuse lysis of the periodontium.
102
ACC is classified among malignant tumors of the salivary glands [3] and was first described by Robin and Laboulbene in 1853. Theoder Biltroth called it cylindroma in 1856, because of its cylindrical aspect when observed with a microscope [4]. Macroscopically [8], it is a non-encapsulated well-defined solid tumor. In terms of histology, there are 2 cell lines: epithelial cells and myoepithelial cells with a hyperchromatic, angular nucleus and scant cytoplasm.
Intra-mandibular adenoid cystic carcinoma
Three architectural forms have been described [8]: the tubular form includes tubes lined by a double layer of cells, with epithelial cells making up the internal layer and basal cells the outer layer; the cribriform structure has a micro-cystic aspect with the presence of basophilic mucoid and/or hyaline material; the solid form includes uniform basal cells (without tubes or micro-cysts). It is mandatory to screen for perineural involvement since this is a key factor for the prognosis. Only Ki67 is useful to differentiate between adenoid cystic carcinoma and polymorphous adenocarcinoma, for immunohistochemistry.
Treatment We recommend a multidisciplinary meeting including surgeons, radiotherapists, dentists, and chemotherapists, as for any type of cancer. The indication and the choice of therapy are always controversial [5]. Nonetheless, for several authors, surgery is the mainstay of management [1,3–5,8–10]. It consists in a radical resection of the lesion with wide resection margins (macroscopic margins of at least 1 cm) [3,8]. It often involves segmental mandibulectomy, extended to the underlying and overlying structures, depending on the tumor extension. Systematiccervical lymphadenectomy isrecommendedbymost authors, whatever the lymph-node status, and should be bilateral if the tumor is symphyseal, or has reached or extended beyond the median line. It should be homolateral in other cases. Complementary postoperative radiotherapy is performed in accordance with the most recent recommendations, given the radio susceptibility, for better local control in terms of recurrence; it is indicated whatever the surgical margins and pathology results (histological grade) are. Its lack of susceptibility to chemotherapy can be explained by its slow evolution; this explains why chemotherapy has been abandoned as the first-line treatment. However, it is indicated for strictly palliative care in case of metastases.
Prognosis
perineural involvement, vascular emboli; cervical lymph-node involvement; metastasis, especially distant metastasis.
Disclosure of interest The authors declare that they have no conflicts of interest concerning this article.
References [1]
[2]
[3]
[4]
[5]
[6]
[7]
[8]
[9]
[10]
[11] [12]
The prognosis is bad and is mainly impacted by the risk of distant metastasis, occurring in 40 to 60% of patients [5], and the tendency to relapse. Involvement of the underlying nerves often leads to incomplete tumor resection and thus to frequent local recurrence [4]. Metastases were described in most of the reported cases, especially in the lung [2,4,8,9]. The survival rate is estimated at 77% at 5 years and 57% at 10 years [10]. It is related to: the size of the tumor; the involvement of surrounding tissues (muscles, submandibular glands);
[13] [14]
[15] [16] [17]
Chummun S, McLean NR, Kelly CG, Dawes PJDK, Meikle D, Fellows S, et al. Adenoid cystic carcinoma of the head and neck. Br J Plast Surg 2001;54:476–80. Duberge´ T, Be´ne´zery K, Resbeut M, Azria D, Minsat M, Ellis S, et al. Carcinomes ade´noı¨des kystiques ORL : e´tude re´trospective multicentrique de 169 cas. Cancer Radiother 2012;16: 247–56. Gondivkar SM, Gadbail AR, Chole R, Parikh RV. Adenoid cystic carcinoma: a rare clinical entity and literature review. Oral Oncol 2011;47:231–6. Santos T de S, Melo DG, Gomes AC, Andrade ES, de Oliveira e Silva ED. Adenoid cystic carcinoma of mandible. Braz J Otorhinolaryngol 2011;77:807. Grimm M, Henopp T, Hoefert S, Schaefer F, Kluba S, Krimmel M, et al. Multiple osteolytic lesions of intra-osseous adenoid cystic carcinoma in the mandible mimicking apical periodontis. Int Endod J 2012;45:1156–64. Shamim T, Varghese VI, Shameena PM, Sudha S. Primary intraosseous adenoid cystic carcinoma of the mandible with lung metastasis. J Oral Sci 2008;50:95–8. 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. Al-Sukhun J, Lindqvist C, Hietanen J, Leivo I, Penttila ¨ 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. Gumgum S, Oner B, Ustay C. Rapidly growing adenoid cystic carcinoma in mandible: a case report. Oral Oncology Extra 2005;41:142–5. Clark JM, Triana RJ, Meredith SD. Uncontrolled central adenoid cystic carcinoma: case report. Ear Nose Throat J 2000; 79:784. Bumstead WD. Cylindroma of the mandible. Oral Surg Oral Med Oral Pathol 1955;8:546–55. Bradley JC. A case of cylindroma of the mandible. Br J Oral Surg 1968;5:186–93. Ha ¨mori J, Krasznai G. A mandibula cylindromaja. Orv Hetil 1969;5:302–3. 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. Dhawan IK, Bhargava S, Nayak NC, Gupta RK. Central salivary gland tumours of jaws. Cancer 1970;26:211–7. Slavin G, Mitchell RM. Adenoid cystic carcinoma of the mandible. Br J Surg 1971;58:546–8. Burkes Jr EJ. Adenoid cystic carcinoma of the mandible masquerading as periapical inflammation. J Endod 1975;1: 76–8.
103
A. Bouaichi et al. [18]
[19]
[20]
104
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