- Email: [email protected]
Lymphoepithelial cyst of oral cavity
Int. J. Oral SUfg. Surg, 1983: 12: 127-131 (Key words: cysl, cyst, lympiloepi,ilelial; lymphoepltheliai; surgery, ora/) ora/)
Lymphoepithelial cyst of oral cavity Report of a case case and review review of the literature SUMIO SAKODA, YOSHIKUNI KODAMA AND RYOSUKE SHIBA Department of Oral and MaXillofacial Maxillafacial Surgery, Miyazaki Medical College Hospital, Miyazaki, Japan ABSTRACT - A rare case of the oral ABSTRACT ora1lymphoepithelia1 lymphoepithelial cyst in the ventral surface of the tongue is reported. Histological findings findings show the same appearance as the cyst wall to the oral mucosa in the reported previously, and the continuity of the reviewed, and its specimen is suspected. Some aspects of the lesion are reviewed, pathogenesis, as reported in the literature, is discussed.
(Received for for publication 30 December 1981, 1981, accepted 8 October 1982)
The lymphoepithelial cyst of the oral cavity is a rare lesion which is microscopically identical to to the so-called branchial cyst or fistula of the neck. The floor ofthe mouth and ventral tongue are commonly involved. The histogenesis of the lymphoepithelial cyst is unknown, but there is a suggestion that the branchial or salivary gland epithelium is transformed into the cyst wall with aggregation of lymphoid tissue during embryogenesis. On the other hand, some have proposed that the lesion is formed by obstruction of the crypt of the oral tonsil. We report a case of the oral lymphoepithelial cyst in the ventral tongue, adding to to the 113 cases reported previously, which are are also reviewed.
Case report On May 26 1981, a 19-year-old female carne to Miyazaki Medical College Hospital Hospital with a complaint ofa small swelling in the ventral surface of the tongue. about I year ago, Though she had had noticed the lesion about she left it without treatment because there was neither
pain nor any remarkable remarkable change in size. size. Recently, Recently, she became aware of the lesion increasing in size. size. On systematic physical examination, no abnormality was detected, except for the intraoral lesion, which was a small round, circumscribed soft mass on ventral surface of the tongue, the right side of the ventral measuring approximately 5 mm in diameter. The lesion was slightly raised from surrounding mucous membrane, smooth, yellowish-white in color color and 1). freely freely movable from the underlying tissue (Fig. 1). The remaining oral mucosa was healthy. Neither swelling were detected in palpable lymph node nor swelling either side of the the neck. The lesion was was clinically diagnosed as mucocele, mucocele, Under local anesthesia, the lesion was totally and the underlining excised. excised. It lay only superficially and tissue tissue seemed to be normal. normal. The wound was closed and healed without without complication. complication. The excised excised cyst was was discus-like in shape and was filled by a light grey grey viscous fluid. Microscopically, the inner surface of the cyst wall was covered by stratified squamous epithelium, and a dense lymphocytic infiltration was observed in the dense surrounding connective tissue. The content of the cavity revealed desquamated epithelial cells, cells, Some duct-like epithelial islands were were observed in the covering submucosal connective tissue, which was was suspected of forming a continuity of the cyst wall to
128
SAKODA, KODAMA AND SHIBA SHeBA
Fig. 1. Clinical Clinical appearance appearance of of the tbe lymphoepitbelial Iyrnphoepirhelial cyst the tongue. cyst in the ventral surface surface of the
the the oral mucosal epithelium. The The diagnosis of oral Iymphoepithelial cyst lymphoepithelial cyst was established histologically. (See Fig. 2.)
Discussion The The lymphoepithelial cysts of the the oral cavity, which have been reported previously, supown case, are summarized in plemented by our own the Table 1. l. Clinically, most lymphoepithelial Iymphoepithelial cysts are are ac<:ompanied accompanied by no subjective symptom, and detected by dentists during usual are often detected dental lesion usually usually appears as a dented practice. The lesion soft, non-ulcerated and movable mass under the oral mucous membrane. The cystic cavity is is
Fig. 2. Photomicrograph Fig. Photomicrograph showing the tbe cystic cavity cavity covered with with squamous sqlllimous epithelium, surrounded by by lymphoid tissue containing no germinal centres. lymphoid tissue containing germinal centres. Some duct-like duct-like epithelial islands islands are are observed in the the Some is visible in epithelium isvisiblein covering submucosa. Mucosal Mucosalepithelium the portion. the upper-right upper-right portion.
High-power photomicrograph of section Fig. 3. High-power shown in Fig. Fig. 2. 2.
and creamy mafilled with yellowish-white and terial. The The size of the lesion, as previously reported, ranges between 1I and 20 mm in greatest diameter. The age of occurrence in the 97 accounted cases ranges widely between 7 and 81 81 years (Table 2). 2). Of Of the 101 101 calculated cases, 64 cases (63.4%) are male and 37 cases (36.6%) relative predilection female, which indicates a relative for males (Table (Table 1). As revealed in Table 3, of the oral cavity are located almost all lesions of in the floor of mouth, the tongue or the soft soft palate. of the oral With regard to histopathogenesis of Iymphoepithelial cyst, several hypotheses have lymphoepithelial been proposed. Classically, it was generally accepted accepted that the origin of of the epithelial component was from the embryonic remnant of the branchial cleft system, since the histologic and the findings showed some resemblance to it, and "b ranchial or branchiogenic cyst" cyst" was term "branchial
Table 1. Lymphoepithelial cysts described in the the literature Author
No. No. of of eases
Age (range)
Gotrr' Gorn"
1 1 1 24
32 40 30 15-65
1 13
42
CALMAN4 VICKERS et 01." BHASKAR'
YOUNG & CLAMAN"
KNAPp·
ACEVEDO & NELSON I MERCHART' GIUNTA & CATAIDO' CATAIDO'
HASEGAWA et of.' SuGIHARA et 0/. 10 BuQiNER BuCHNER & HANSEN'
SAKODA et of. (present paper)
9 1 21 21
20-46 21 7-65
2
19-27
1 38
41 41 14-81
1
19
Sex M M M 17M 7F M
9M M 9M 12 F 1M 1F F 23M 15 F
F
Location floor of mouth floor of mouth floor of mouth 15 floor of of mouth 8 posterolateral surface of tongue 1 anterior palatine pillar floor of mouth 7 floor of mouth 4 soft soft palate 2 ventral surface of tongue 9 floor of mouth floor of mouth 17 floor of mouth 2 soft palate 1 mandibular labial vestibule 1 retromolar pad 1 ventral surface of tongue 1 floor of mouth ventral surface of tongue 19 floor of mouth 7 ventral surface of tongue 7 posterolateral surface of tongue 2 soft palate 2 anterior palatine pillar I buccal vestibule ventral surface of tongue
Size (maximum)
10x6x4 mm 20mm 15mm
5mm lOmm
rt"'" 0-<:
's: "tI "tI
:r:
0
tIl tIl
8x4x2 mm 3mm 15 mm
." .....
5:J tIl rt"'" .....
>t"'"
5mm
Q en en >-l
6x4mm 10mm
5mm
......
t5
130 130
SAKODA, KODAMA AND SHIBA lesions 3 ,$. Knapp proposed that the lesion arises lesions':".
patients* Table 2. Age distribution of patients" Age group
No. of cases
% of cases
0-9 10-19 20-29 30-39 40-49 50-59 60-69 70-
1 12 30 18 18 16 14 2
4
1.0 1.0 12.4 30.9 30.9 18.6 18.6 16.5 16.5 14.4 14.4 2.1 4.1
97
100.0 100.0
Total
* 17cases 17 cases out out of 114cases 114 cases were omitted because of no description of age. age. introduced. BHASKAR , however, expressed the view that the lesion arose from inclusions of ectopic glandular epithelium into the lymphoid tissue of oral mucosa, and proposed that it it be called "oral lymphoepithelial cyst", which occurs in the same way as the development of similar cysts in in the area of the neck. neck. VICKERS & VON DER MUHLL MUHLL 12 12 proved Bhaskar's hypothesis experimentally by implantation of buccal pouch epithelium into submandibular lymph nodes in hamsters. On On the other hand, KNAPP' has has described the existence of a number of tonsillar tissues in the oral mucosa, where oral lymphoepithelial cysts are most likely involved. The continuity of the cyst wall to to the oral mucosa has been reported in many of these 2
through an an obstruction of the crypt of the oral tonsil and that it it should be called "pseudocyst" of the oral tonsil. Moreover, the facts: that the lesion occurs over a wide age-range; that it it is located in the superficial portion of oral cavity; that it is not as large as 2.0 2.0 em cm in diameter; and that the cyst wall connects very closely to the oral mucosa in many cases, contribute to our giving greater approval to the obstruction theory of Knapp.
References 1. 1. ACEVEDO, A. & NELSON, J. F. : Lymphoepithelial cysts of the oral cavity; report of 9 cases. Oral Surg. 1971: 31: 632-636. 2. 2. BHASKAR, S. N.: Lymphoepithelial cysts of the oral cavity; report report of 24 cases. Oral Surg. 1966: 21: 120-128. 3. BUCHNER, A. & HANSEN, HANSEN, L.: Lymphoepithelial 3. cyst of the oral cavity; a clinicopathologic study Surg, 1980: 1980: 50: 441-449, of 38 cases. Oral Surg. 4. CALMAN, H. J.: Sublingual branchiogenic cyst. Oral Oral Surg. Surg. 1963: 16: 333-338. 5. GRJNTA, GRJNTA, J. & CATALDO, E.: Lymphoepithelial cyst of the oral mucosa. Oral Surg, Surg. 1973: 35: 7784. 84. 6. GOLD, C.: Branchial cleft cyst cyst located located in the floor of the mouth. Oral Surg. Surg, 1962: 15: 1118-1120. 7. HASEGAWA, K., 7. K" MATUMURA, T., MlMURA, T., AOBA, T., ISHIDA, T., YAGl, T. & MASUDA, T.: Lymphoepithelial cysts of the oral cavity; report of 2 cases. cases. J. Dent, Osaka Univ. 1974: 20: 59-63 59-63 (in Japanese),
Table 3. Location of lesion Location Location
Floor of mouth Posterolateral Posterolateral surface of tongue Ventral surface surface of tongue Soft palate Anterior palatine pillar Mandibular labial vestibule Retromoler pad Buccal vestibule vestibule Total
No. of cases cases
%of cases
73 15 12 8 3 1 1
64.0 64.0 13.2 10.5 7.0 2.6 2.6 0.9 0.9 0.9
114
100.0
1
LYMPROEPITRELIAL LYMPHOEPITHELIAL CYST 8. KNAPP. M. J.: tonsils.. Oral J. : Pathology of oral tonsils Surg. 1970: 1970 : 29: 29: 295-304. 9. MERCHANT, N. N. E.: Lympho-epithelial cyst of the floor of the the mouth; a case report. report. Br. Dent. J. 1972: 132: 132 : 271-272. 10. SuGIHARA. HASuGIHARA. K., SAKU, SAKU , T., T.• MUKAI, R., HASHIMOTO. K. & SHIODA. S. : Lymphoepithelial cyst SHIMOTO, on ventral surface surface of of the tongue; tongue ; report of of a case. Japan J. J . Oral Surg. Surg, 1977: 1977 : 23: 643-648 (in Japanese). 11. 11. VICKERS, VICKERS, R. A., A., GORLIN, GORLIN. R. R. J. & SMART, E. E. A.: Lymphoepitheliallesion ofthe of the oral cavity; report of four cases. Oral Surg. Surg , 1963: 16: 1214-1222. 12. 12. VICKERS. VICKERS. R. A. & VON VON DIlR DER MUHLL, 0.: An investigation concerning inducibility of lymIym-
131
phoepitheliaI phoepithelial cyst in in hamsters hamsters by autogenous epithelial transplantation. J. J. Dent. Res. 1966: 4S: 45: 1029-1032. 13. YDUNG DUNG,, W. W. G. & CLAMAN. CLAMAN. S. N. N.:: A IymphoepitSurg , 1967: 23 23:: helia\ helial cyst of the oral cavity. Oral Surg. 62-67.
Address Address:: Sumio Sakoda Sakoda Department of Oral and Maxillofacial Surgery Miyazaki Medical College Coi/ege Hospital 5200 Kihara Kiyotake Miyazaki 889-16 Japan Japan
Lymphoepithelial cyst of oral cavity Report of a case case and review review of the literature SUMIO SAKODA, YOSHIKUNI KODAMA AND RYOSUKE SHIBA Department of Oral and MaXillofacial Maxillafacial Surgery, Miyazaki Medical College Hospital, Miyazaki, Japan ABSTRACT - A rare case of the oral ABSTRACT ora1lymphoepithelia1 lymphoepithelial cyst in the ventral surface of the tongue is reported. Histological findings findings show the same appearance as the cyst wall to the oral mucosa in the reported previously, and the continuity of the reviewed, and its specimen is suspected. Some aspects of the lesion are reviewed, pathogenesis, as reported in the literature, is discussed.
(Received for for publication 30 December 1981, 1981, accepted 8 October 1982)
The lymphoepithelial cyst of the oral cavity is a rare lesion which is microscopically identical to to the so-called branchial cyst or fistula of the neck. The floor ofthe mouth and ventral tongue are commonly involved. The histogenesis of the lymphoepithelial cyst is unknown, but there is a suggestion that the branchial or salivary gland epithelium is transformed into the cyst wall with aggregation of lymphoid tissue during embryogenesis. On the other hand, some have proposed that the lesion is formed by obstruction of the crypt of the oral tonsil. We report a case of the oral lymphoepithelial cyst in the ventral tongue, adding to to the 113 cases reported previously, which are are also reviewed.
Case report On May 26 1981, a 19-year-old female carne to Miyazaki Medical College Hospital Hospital with a complaint ofa small swelling in the ventral surface of the tongue. about I year ago, Though she had had noticed the lesion about she left it without treatment because there was neither
pain nor any remarkable remarkable change in size. size. Recently, Recently, she became aware of the lesion increasing in size. size. On systematic physical examination, no abnormality was detected, except for the intraoral lesion, which was a small round, circumscribed soft mass on ventral surface of the tongue, the right side of the ventral measuring approximately 5 mm in diameter. The lesion was slightly raised from surrounding mucous membrane, smooth, yellowish-white in color color and 1). freely freely movable from the underlying tissue (Fig. 1). The remaining oral mucosa was healthy. Neither swelling were detected in palpable lymph node nor swelling either side of the the neck. The lesion was was clinically diagnosed as mucocele, mucocele, Under local anesthesia, the lesion was totally and the underlining excised. excised. It lay only superficially and tissue tissue seemed to be normal. normal. The wound was closed and healed without without complication. complication. The excised excised cyst was was discus-like in shape and was filled by a light grey grey viscous fluid. Microscopically, the inner surface of the cyst wall was covered by stratified squamous epithelium, and a dense lymphocytic infiltration was observed in the dense surrounding connective tissue. The content of the cavity revealed desquamated epithelial cells, cells, Some duct-like epithelial islands were were observed in the covering submucosal connective tissue, which was was suspected of forming a continuity of the cyst wall to
128
SAKODA, KODAMA AND SHIBA SHeBA
Fig. 1. Clinical Clinical appearance appearance of of the tbe lymphoepitbelial Iyrnphoepirhelial cyst the tongue. cyst in the ventral surface surface of the
the the oral mucosal epithelium. The The diagnosis of oral Iymphoepithelial cyst lymphoepithelial cyst was established histologically. (See Fig. 2.)
Discussion The The lymphoepithelial cysts of the the oral cavity, which have been reported previously, supown case, are summarized in plemented by our own the Table 1. l. Clinically, most lymphoepithelial Iymphoepithelial cysts are are ac<:ompanied accompanied by no subjective symptom, and detected by dentists during usual are often detected dental lesion usually usually appears as a dented practice. The lesion soft, non-ulcerated and movable mass under the oral mucous membrane. The cystic cavity is is
Fig. 2. Photomicrograph Fig. Photomicrograph showing the tbe cystic cavity cavity covered with with squamous sqlllimous epithelium, surrounded by by lymphoid tissue containing no germinal centres. lymphoid tissue containing germinal centres. Some duct-like duct-like epithelial islands islands are are observed in the the Some is visible in epithelium isvisiblein covering submucosa. Mucosal Mucosalepithelium the portion. the upper-right upper-right portion.
High-power photomicrograph of section Fig. 3. High-power shown in Fig. Fig. 2. 2.
and creamy mafilled with yellowish-white and terial. The The size of the lesion, as previously reported, ranges between 1I and 20 mm in greatest diameter. The age of occurrence in the 97 accounted cases ranges widely between 7 and 81 81 years (Table 2). 2). Of Of the 101 101 calculated cases, 64 cases (63.4%) are male and 37 cases (36.6%) relative predilection female, which indicates a relative for males (Table (Table 1). As revealed in Table 3, of the oral cavity are located almost all lesions of in the floor of mouth, the tongue or the soft soft palate. of the oral With regard to histopathogenesis of Iymphoepithelial cyst, several hypotheses have lymphoepithelial been proposed. Classically, it was generally accepted accepted that the origin of of the epithelial component was from the embryonic remnant of the branchial cleft system, since the histologic and the findings showed some resemblance to it, and "b ranchial or branchiogenic cyst" cyst" was term "branchial
Table 1. Lymphoepithelial cysts described in the the literature Author
No. No. of of eases
Age (range)
Gotrr' Gorn"
1 1 1 24
32 40 30 15-65
1 13
42
CALMAN4 VICKERS et 01." BHASKAR'
YOUNG & CLAMAN"
KNAPp·
ACEVEDO & NELSON I MERCHART' GIUNTA & CATAIDO' CATAIDO'
HASEGAWA et of.' SuGIHARA et 0/. 10 BuQiNER BuCHNER & HANSEN'
SAKODA et of. (present paper)
9 1 21 21
20-46 21 7-65
2
19-27
1 38
41 41 14-81
1
19
Sex M M M 17M 7F M
9M M 9M 12 F 1M 1F F 23M 15 F
F
Location floor of mouth floor of mouth floor of mouth 15 floor of of mouth 8 posterolateral surface of tongue 1 anterior palatine pillar floor of mouth 7 floor of mouth 4 soft soft palate 2 ventral surface of tongue 9 floor of mouth floor of mouth 17 floor of mouth 2 soft palate 1 mandibular labial vestibule 1 retromolar pad 1 ventral surface of tongue 1 floor of mouth ventral surface of tongue 19 floor of mouth 7 ventral surface of tongue 7 posterolateral surface of tongue 2 soft palate 2 anterior palatine pillar I buccal vestibule ventral surface of tongue
Size (maximum)
10x6x4 mm 20mm 15mm
5mm lOmm
rt"'" 0-<:
's: "tI "tI
:r:
0
tIl tIl
8x4x2 mm 3mm 15 mm
." .....
5:J tIl rt"'" .....
>t"'"
5mm
Q en en >-l
6x4mm 10mm
5mm
......
t5
130 130
SAKODA, KODAMA AND SHIBA lesions 3 ,$. Knapp proposed that the lesion arises lesions':".
patients* Table 2. Age distribution of patients" Age group
No. of cases
% of cases
0-9 10-19 20-29 30-39 40-49 50-59 60-69 70-
1 12 30 18 18 16 14 2
4
1.0 1.0 12.4 30.9 30.9 18.6 18.6 16.5 16.5 14.4 14.4 2.1 4.1
97
100.0 100.0
Total
* 17cases 17 cases out out of 114cases 114 cases were omitted because of no description of age. age. introduced. BHASKAR , however, expressed the view that the lesion arose from inclusions of ectopic glandular epithelium into the lymphoid tissue of oral mucosa, and proposed that it it be called "oral lymphoepithelial cyst", which occurs in the same way as the development of similar cysts in in the area of the neck. neck. VICKERS & VON DER MUHLL MUHLL 12 12 proved Bhaskar's hypothesis experimentally by implantation of buccal pouch epithelium into submandibular lymph nodes in hamsters. On On the other hand, KNAPP' has has described the existence of a number of tonsillar tissues in the oral mucosa, where oral lymphoepithelial cysts are most likely involved. The continuity of the cyst wall to to the oral mucosa has been reported in many of these 2
through an an obstruction of the crypt of the oral tonsil and that it it should be called "pseudocyst" of the oral tonsil. Moreover, the facts: that the lesion occurs over a wide age-range; that it it is located in the superficial portion of oral cavity; that it is not as large as 2.0 2.0 em cm in diameter; and that the cyst wall connects very closely to the oral mucosa in many cases, contribute to our giving greater approval to the obstruction theory of Knapp.
References 1. 1. ACEVEDO, A. & NELSON, J. F. : Lymphoepithelial cysts of the oral cavity; report of 9 cases. Oral Surg. 1971: 31: 632-636. 2. 2. BHASKAR, S. N.: Lymphoepithelial cysts of the oral cavity; report report of 24 cases. Oral Surg. 1966: 21: 120-128. 3. BUCHNER, A. & HANSEN, HANSEN, L.: Lymphoepithelial 3. cyst of the oral cavity; a clinicopathologic study Surg, 1980: 1980: 50: 441-449, of 38 cases. Oral Surg. 4. CALMAN, H. J.: Sublingual branchiogenic cyst. Oral Oral Surg. Surg. 1963: 16: 333-338. 5. GRJNTA, GRJNTA, J. & CATALDO, E.: Lymphoepithelial cyst of the oral mucosa. Oral Surg, Surg. 1973: 35: 7784. 84. 6. GOLD, C.: Branchial cleft cyst cyst located located in the floor of the mouth. Oral Surg. Surg, 1962: 15: 1118-1120. 7. HASEGAWA, K., 7. K" MATUMURA, T., MlMURA, T., AOBA, T., ISHIDA, T., YAGl, T. & MASUDA, T.: Lymphoepithelial cysts of the oral cavity; report of 2 cases. cases. J. Dent, Osaka Univ. 1974: 20: 59-63 59-63 (in Japanese),
Table 3. Location of lesion Location Location
Floor of mouth Posterolateral Posterolateral surface of tongue Ventral surface surface of tongue Soft palate Anterior palatine pillar Mandibular labial vestibule Retromoler pad Buccal vestibule vestibule Total
No. of cases cases
%of cases
73 15 12 8 3 1 1
64.0 64.0 13.2 10.5 7.0 2.6 2.6 0.9 0.9 0.9
114
100.0
1
LYMPROEPITRELIAL LYMPHOEPITHELIAL CYST 8. KNAPP. M. J.: tonsils.. Oral J. : Pathology of oral tonsils Surg. 1970: 1970 : 29: 29: 295-304. 9. MERCHANT, N. N. E.: Lympho-epithelial cyst of the floor of the the mouth; a case report. report. Br. Dent. J. 1972: 132: 132 : 271-272. 10. SuGIHARA. HASuGIHARA. K., SAKU, SAKU , T., T.• MUKAI, R., HASHIMOTO. K. & SHIODA. S. : Lymphoepithelial cyst SHIMOTO, on ventral surface surface of of the tongue; tongue ; report of of a case. Japan J. J . Oral Surg. Surg, 1977: 1977 : 23: 643-648 (in Japanese). 11. 11. VICKERS, VICKERS, R. A., A., GORLIN, GORLIN. R. R. J. & SMART, E. E. A.: Lymphoepitheliallesion ofthe of the oral cavity; report of four cases. Oral Surg. Surg , 1963: 16: 1214-1222. 12. 12. VICKERS. VICKERS. R. A. & VON VON DIlR DER MUHLL, 0.: An investigation concerning inducibility of lymIym-
131
phoepitheliaI phoepithelial cyst in in hamsters hamsters by autogenous epithelial transplantation. J. J. Dent. Res. 1966: 4S: 45: 1029-1032. 13. YDUNG DUNG,, W. W. G. & CLAMAN. CLAMAN. S. N. N.:: A IymphoepitSurg , 1967: 23 23:: helia\ helial cyst of the oral cavity. Oral Surg. 62-67.
Address Address:: Sumio Sakoda Sakoda Department of Oral and Maxillofacial Surgery Miyazaki Medical College Coi/ege Hospital 5200 Kihara Kiyotake Miyazaki 889-16 Japan Japan