clinical analysis of 120 cases of intraoral lymphoepithelial cyst
TRANSCRIPT
Vol. 113 No. 4 April 2012
Clinical analysis of 120 cases of intraoral lymphoepithelial cystXi Yang, MD, PhD,a Andrew Ow, BDS, MDS, AdvDip(OMS),b Chen-ping Zhang, MD, PhD,a
Li-zhen Wang, MD,c Wen-jun Yang, MD, PhD,a Yong-jie Hu, MD,a and Lai-ping Zhong, MD, PhD,a Shanghai,China; and SingaporeSHANGHAI JIAO TONG UNIVERSITY SCHOOL OF MEDICINE AND NATIONAL UNIVERSITY OF SINGAPORE
Objective. The aim of this study was to analyze a single institution’s experience in the clinical management of intraorallymphoepithelial cyst (LEC).Study Design. From 1993 to 2010, a total of 120 consecutive patients with intraoral LEC underwent surgery and wereretrospectively investigated regarding its clinical appearance, clinical differential diagnosis, treatment, and prognosis.Results. Of the 120 patients, 37 were male and 83 female, their ages ranging from 2 to 75 years with a mean of 44.1years. The most common locations were the tongue (50%) and floor of mouth (38.3%). The course of disease rangedfrom 2 months to 10 years with a mean of 8.0 months, and 75.8% of the patients had a course of disease �6 months.Routine laboratory examinations were within normal limits. All patients underwent complete surgical removal of thelesions. During the follow-up period, no lesion recurrence occurred, and the quality of life of each patient was good.Conclusions. The clinical characteristics of intraoral LEC are not specific and may be confused with other intraoral lesions.The first choice of treatment is surgical excision, which results in a good prognosis. (Oral Surg Oral Med Oral Pathol Oral
Radiol 2012;113:448-452)The intraoral lymphoepithelial cyst (LEC) is a de-velopmental lesion.1-6 The name of “lymphoepithe-lial cyst” was first used in 1949 to describe thepathologic characters of an intraoral branchial cleftcyst.7 In 1984,8 owing to its different histologicorigin and pathogenesis, the intraoral LEC was rec-ommended to be distinguished from the branchialcleft cyst. Several theories on the pathogenesis ofintraoral LEC have been proposed, includingKnapp’s theory9 and Buchner and Hansen’s theory.10
However, the most accepted theory of its pathogen-esis involves the accumulation of desquamated epi-thelial lining, which results in a dilated obstructedcrypt of the oral tonsil. It occurs commonly in adultsbut rarely in children. In the English-language liter-ature, 150 of such cases have been reported (Table
Supported in part by the Program for Innovative Research Team ofthe Shanghai Municipal Education Commission and by researchgrants (10dz1951300, 1052nm04700, and 10140902200) from theScience and Technology Commission of Shanghai Municipality.aDepartment of Oral and Maxillofacial Surgery, Ninth People’s Hos-pital, School of Stomatology, Shanghai Jiao Tong University Schoolof Medicine, Shanghai Key Laboratory of Stomatology.bDepartment of Oral and Maxillofacial Surgery, Faculty of Dentistry,National University of Singapore.cDepartment of Oral Pathology, Ninth People’s Hospital, School ofStomatology, Shanghai Jiao Tong University School of Medicine,Shanghai Key Laboratory of Stomatology.Received for publication Jan. 25, 2011; returned for revision Feb. 17,2011; accepted for publication Feb. 24, 2011.© 2012 Elsevier Inc. All rights reserved.2212-4403/$ - see front matter
doi:10.1016/j.tripleo.2011.02.051448
I),1-23 and the most common site of occurrence is thefloor of mouth (70.7%), followed by the lateral mar-gin (10.7%), ventral surface (7.3%) of tongue, softpalate (6.0%), hard palate (2.0%), retromolar area(1.3%), side of glossodesmus (0.7%), and oral ves-tibule (0.7%). Because of its low clinical morbidityand nonspecific symptoms, diagnosis of intraoralLEC remains a challenge. In the present study, wepresent 120 consecutive cases of intraoral LEC, in-cluding its clinical characteristics, differential diag-nosis, treatment, and prognosis, as well as a reviewof the literature.
PATIENTS AND METHODSA total of 120 consecutive patients with intraoral LECtreated from 1993 to 2010 at the Department of Oraland Maxillofacial Surgery, Ninth People’s Hospital,School of Medicine, Shanghai Jiao Tong University,were retrospectively reviewed with all relevant clinicalinformation and pathologic slides. All patients receivedsurgical removal of the lesions with preservation ofimportant neighboring structures, such as Whartonduct, papilla of Stensen duct, lingual nerve, and hypo-glossal nerve. The patients were subsequently reviewedin follow-up visits.
RESULTSPatient demographicsOf the 120 patients with intraoral LEC, there 37 weremale and 83 female with a male-to-female ratio of1:2.24. Their ages ranged from 2 to 75 years with a
mean of 44.1 years. The majority of intraoral LEC
e; UK,
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patients (70%) presented in the fourth to sixth de-cades of life. Four lesions occurred in pediatric pa-tients (age �18 years). The age distribution is pre-sented in Fig. 1.
Course of diseaseThe mean course of disease was 8.0 months, rangingfrom 2 weeks to 10 years. The course of disease was�6.0 months in 75.8% of patients (91/120).
SymptomsAll patients reported a gradually growing and painless
Fig. 1. Age distribution of patients with intraoral lymphoe-pithelial cyst.
Table I. Summary of published descriptions of intraorAuthor Year Cases (n) Age (y)
Gold10 1962 1 32Calman7 1963 1 40Vickers11 1963 1 30Bhaskar1 1966 24 15-65Young12 1967 1 42Knapp9 1970 13 17-48Acevedo2 1971 9 20-46Merchant3 1972 1 21Giunta4 1973 21 7-65
Buchner13 1980 38 14-81
Toto PD14 1982 6 25-60Sakoda15 1983 1 19Chaudhry8 1984 24 12-74Iwase T16 1985 1 34McDonnel17 1990 1 5Kumara18 1995 1 29Ahn19 1996 1 56Flaitz5 2000 1 4Suzuki20 2000 1 30Flaitz6 2004 1 72Epivatianos21 2005 1 27Khelemsky22 2010 1 34Nonaka23 2011 10 16-60
BM, buccal mucosa; F, female; FM, floor of mouth; HP, hard palateretromolar area; SG, side of glossodesmus; SP, soft palate; T, tongu
mass without obvious symptoms. However, 1 patient
reported discomfort when swallowing for a period of 1month and another patient reported slight pain of themass for a period of 3 weeks.
Physical examinationThe size of the lesions ranged from 0.2 to 2.0 cm witha mean of 0.8 cm; 96.7% of the lesions (116/120) were�1.0 cm. The palpable lesion was clearly defined, wassoft to moderate (79.2%) in hardness without obvioustenderness, and exhibited mobility. Various colors ofthe lesion were reported, including yellow-white(51.4%) (Fig. 2), pink (27.5%), transparent (9.2%),translucent (4.2%), and purple (1.7%). The sites ofoccurrence are described in Table II, with the mostcommon sites being the tongue (50%) and floor ofmouth (38.3%).
Laboratory examinationIn all patients, routine laboratory tests were performed,and all results were within normal reference ranges.
TreatmentAll patients underwent complete surgical removal ofthe lesions under local anesthesia (1% lidocaine). Allspecimens were sent for pathologic examination. The
CGender Location
M FMM FMM FM17 M, 7 F FM (n � 15), LMT (n � 8), HP (n � 1)M FM� 12 M FM (n � 7), SP (n � 4), VT (n � 2)M FMM FM9 M, 12 F FM (n � 17), SP (n � 2), BM (n � 1),
RR (n � 1)23 M, 15 F FM (n � 19), VT (n � 7), LMT
(n � 7), SP (n � 2), HP (n � 2),RR (n � 1)
3 M, 3 F FM (n � 2), T (n � 3), SP (n � 1)F VT9 M, 15 F FMM FMM FMM FMF FMM VTM SGM LMTM FMM FM2 M, 8 F T (n � 4), OP (n � 2), FM (n � 2), P
(n � 1), UK (n � 1)
lateral margin of tongue; M, male; OP, oropharynx; P, palate; RA,unknown; VT, ventral of tongue.
al LE
; LMT,
lesions were enucleated or excised with overlying mu-
ORAL AND MAXILLOFACIAL SURGERY OOOO450 Yang et al. April 2012
cosa. Important anatomic structures in close proximityto the lesions, such as Wharton duct, papilla of Stensenduct, lingual nerve, and hypoglossal nerve were pre-served. Primary closure with wound healing wasachieved in all patients.
Preoperative clinical diagnosisThe preoperative clinical diagnoses of these lesionswere incorrect in all patients. These diagnoses includ-ed: mucinous cyst (n � 30), nonspecific benign tumor(n � 25), lipoma (n � 15), fibroma (n � 10), papilloma(n � 10), nonspecific cyst (n � 10), sublingual glandcyst (n � 6), dermoid cyst (n � 5), sialolithiasis (n �5), abscess (n � 2), and heterotopic sebaceous glands(n � 2).
Gross specimenGrossly, the lesions were mostly described as circum-scribed, encapsulated, and cystic with white-yellowsticky liquid gelatinous thick white content (74.2%;
Fig. 2. Front view of intraoral lymphoepithelial cyst locatedon the right side of the glossodesmus.
Table II. Site of occurrence of the 120 intraoral LECsSite No. of cases Percentage
Tongue 60 50%Ventral surface of tongue 41 34.2%Base of tongue 11 9.2%Lateral margin of tongue 7 5.8%Dorsum of tongue 1 0.8%
Floor of mouth 46 38.3%Palate 6 5%
Hard palate 3 2.5%Soft palate 3 2.5%
Buccal mucosa 3 2.5%Lateral wall of pharynx 3 2.5%Side of glossodesmus 2 1.7%Total 120 100%
Fig. 3) or firm nodular cut surface (22.5%).
Pathologic diagnosisThe postoperative pathologic diagnosis was intraoralLEC in all patients (Fig. 4).
Follow-upThe follow-up period ranged from 1 month to 17 yearswith a mean of 5.2 years. During the follow-up period,no recurrence of the lesion occurred. In addition, thequality of life of all patients was reported to be good.
DISCUSSIONFrom the English-language literature, intraoral LEC isreported generally more frequently in male than femalepatients, with a male-to-female ratio of 1.42:1. How-ever, from the review of our patients, with a higheroccurrence in female patients, the male-to-female ratiois 1:2.24.
Generally, LEC occurs in all ages and is most fre-quently reported during the third and fourth decades oflife. Although LEC in children is seldom reported,2-5,17
the youngest age of its occurrence was reported in a4-year-old boy.3 In our series of 120 patients, the meanage of our patient sample was 44.1 years old, and thenumber of patients was most frequently (70%) in thefourth to sixth decades of life. The youngest patient inour sample was a 2-year-old girl, with the lesion at thefloor of mouth.
The intraoral LEC is frequently an incidental findingduring a routine dental examination. As such, it is a realchallenge for clinicians to make correct diagnosis at itsfirst presentation.
Laboratory examination results are always withinnormal limits in these patients. Imaging modalities,such as ultrasound, computerized tomography, mag-netic resonance imaging, and magnetic resonance an-
Fig. 3. The lesion was encapsulated and cystic with thickwhite content (arrow).
giography, do not demarcate the lesion clearly and as
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such are not helpful. This is because in most cases, thelesions are located in the deep mucosa and �97% oflesions are �1.0 cm in size.
Clinical characteristics of intraoral LEC, such as thecourse of disease, speed of growth of the lesion, andsymptoms, are not specific for such benign lesions.Therefore, it is always difficult to distinguish intraoralLEC from other lesions, such as mucinous cyst, lipoma,fibroma, sialolithiasis, sublingual gland cyst, and der-moid cyst.
In the case of mucinous cysts, they may present witha clinical history of an increase in size of the lesion,followed by rupture of the lesion and subsequent in-crease in size due to mucus accumulation. The surfacetexture of the mucinous cyst is always soft on palpa-tion. The most common site of occurrence is the lowerlip (60%-70%), with teenagers and children being mostcommonly affected. For intraoral lipomas or fibromas,these lesions are located in the deep mucosa, with their
Fig. 4. Lymphoid tissue and lymphatic follicle surroundingthe cystic squamous epithelium and the parakeratinized mat-ter in the cyst epithelium (hematoxylin and eosin staining;original magnification: A, �40; B, �100 (bottom).
surfaces exhibiting a smooth texture. The majority of
lipomas are soft on palpation, whereas fibromas aremoderate to hard on palpation. The most commonintraoral site of occurrence of lipomas and fibromas isthe buccal mucosa.
In most cases, sialolithiasis and sublingual glandcysts are more easily differentiated from intraoral LEC,except in cases when the lesion is very small in size andin the absence of a clear case history. The differentia-tion of these lesions from intraoral LEC is also impor-tant because of differences in selection of the surgicalapproach. The sialolith represents a circumscribed massin the salivary duct and is composed of calcium salts.This lesion presents as a yellow-white nodule in thedeep mucosa with a smooth surface. The complaint ofa submandibular gland swelling during meal times isuseful for differentiation. The mass is tender and hardon palpation, because it resides within the Whartonduct. Occlusal radiographs are helpful for the identifi-cation of this structure when it is located in the anteriorfloor of the mouth. Sublingual gland cysts always pres-ent as a painless, slow-growing, bluish, translucent,fluctuant, dome-shaped swelling in the floor of mouthregion. The patient may also report rupture of thelesion. The presence of a fluid of egg-white consistencyduring fine needle aspiration is the criterion for clinicaldiagnosis of the sublingual gland cyst.
Finally, dermoid cysts that are small sized shouldalso be considered in the differential diagnosis. Der-moid cysts almost occur in the midline of the floor ofmouth. They normally present as asymptomatic slow-growing swellings in the deep mucosa with a pale andrubbery consistency. These lesions are also prone tosecondary infection. Continued growth, sometimes �1cm in diameter, and propensity for secondary infectiousare important characteristics that distinguish dermoidcysts from intraoral LEC.
Complete surgical resection of the lesion is the treat-ment of choice for intraoral LEC, with a 100% controlrate in our study. Also as reported, the quality of life ofthese patients during the follow-up period is good.
CONCLUSIONSThe clinical characteristics of intraoral LEC are notspecific and may be confused with other intraoral le-sions. The first choice of treatment is surgical excision,which results in a good prognosis.
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Reprint requests:
Lai-ping Zhong, MD, PhDDepartment of Oral and Maxillofacial SurgeryNinth People’s HospitalSchool of StomatologyShanghai Jiao Tong University School of MedicineShanghai Key Laboratory of Stomatology639 Zhizaoju RoadShanghai 200011China.
[email protected]