MALAYSIAN DENTAL JOURNAL<strong>Malaysian</strong> <strong>Dental</strong> Journal (2008) 29(2) 94-96© 2008 The <strong>Malaysian</strong> <strong>Dental</strong> <strong>Association</strong>Oral Granular Cell Tumour: A Clinicopathological Study Of 7 Cases And A BriefReview Of The LiteratureAjura Abdul Jalil. BDS, MClinDent (Mal) Stomology Unit, Institute for Medical Research, Kuala Lumpur.Lau Shin Hui. BDS, FDSRCS (Eng), Stomatology Unit, Cancer Research Centre, Institute for Medical Research, KualaLumpur.ABSTRACTOral granular cell tumour is a fairly rare lesion with a predilection for the tongue. Seven cases (6 females, 1 male)of oral granular cell tumour were seen during the 40 year period (1967-2006) in Stomatology Unit, Institute forMedical Research (IMR) in which 5 cases were located at the tongue. All the cases presented as a single swellingand excisional biopsies were carried out in all cases.Key wordsoral granular cell tumourIntroductionOral granular cell tumour is an uncommon lesionwith a predilection for the tongue. The aim of this article isto report seven oral granular cell tumour cases seen duringthe 40 year period from 1967 to 2006 of Stomatology Unit,Institute for Medical Research (IMR) and briefly discussedthe clinicopathologic features.MATERIALS AND METHODSThe records from the Stomatology Unit, IMR weresearched for granular cell tumour cases from 1967 to 2006.Congenital epulis cases, another known granular cell lesionwere excluded. Only 7 cases of granular cell tumour wereretrieved. The request forms and histological features werereviewed. Immunostaining for desmin, muscle specificactin and S100 protein using standard procedure werecarried out for all the cases selected.at the tongue, 1 in the buccal mucosa and 1 at the buccalsulcus. Most of the cases (n=5) presented as a painlessgrowth with durations ranging from 3 weeks to 2 years.The growths measured 0.5 to 1.5 cm in diameter. In allthe cases, excisional biopsy was performed. Histologicallyall the cases showed sheets of polygonal cells withgranular eosinophilic cytoplasm and small vesicularnuclei which extended from the subepithelial zone to themuscular layer. The cells appeared cytologically benign.Pseudoepitheliomatous hyperplasia of the epithelium wasobserved in 3 cases.Immunohistochemistry staining was only carriedout in 6 cases since in one case, there was not enoughtissue left in the wax block. Immunostaining with S100protein showed 5 cases positive for the antibody. In onecase, the immunostaining result was inconclusive. Allcases demonstrated negativity for desmin and also musclespecific actin.RESULTSThe patients’ characteristics are summarized inTable 1. There were 6 females and 1 male, with a mean ageof 29.4 years (range 11-62 years). The patients comprised of3 Malays, 3 Indians and 1 Chinese. Five cases were located94
Ajura / LauTable 1: Clinicopathological features of 7 cases of oral granular cell tumourNO RACE SEX AGE SITE SIGN & SYMPTOM1 Malay F 34 dorsum of tongue painless swelling2 Indian M 25 right lateral border of tongue painful swelling3 Malay F 11 right buccal mucosa painless swelling4 Indian F 62 left lateral border of tongue painless swelling5 Malay F 22 buccal sulcus adjacent to 14 and 15 painful swelling6 Indian F 33 anterior dorsum tongue painless swelling7 Chinese F 19 mid dorsum tongue painless swellingDISCUSSIONGranular cell tumour is a rare soft tissue lesionwith a predilection for the oral cavity although it hasbeen reported to occur in other parts of the body such asoesophagus 1 , thyroid 2 and colon 3 . Intra-orally, the mostcommon site is the tongue 4-8 . However, oral granular celltumour occurring in the parotid gland 9 , palate 10 , lower lip 11and floor of mouth 12 has been reported. The most commonsite in this study was the tongue (n=5, 71.4%) of which 3cases were located at the dorsal part and 2 on the lateralborder.In this case series, the granular cell tumour wasmore prevalent among the females (n=6, 85.7%). Thisfinding concurs with other studies 4, 6-8 . Oral granular celltumours have been reported in all age groups ranging from3 years 7 to 75 years 11 , however it frequently occurs in thethird and fourth decades of life 4, 6, 8 . The average age inthis study was 29.4 years and only one case presented in achild.Clinically it is usually presents as a small, firm,rounded painless swelling with normal mucosal colourwhich generally gives an impression of a fibrous polyp.Thus, oral granular cell tumours are usually excisedimmediately by the surgeons. In our study, the clinicaldiagnosis given by the surgeons included fibroma, lipoma,neurofibroma, neurilemomma and papilloma. Granularcell tumour is a fairly slow growing benign lesion. In aseries of 8 cases, Eguia et al 4 reported the duration of thelesion in the oral cavity ranging from 3 months to 2 years.Oral granular cell tumour usually happens singly althoughmultiple lesions occurring in the oral cavity have beenreported 11, 13 .Microscopically, the cells of granular cell tumourare rounded, polygonal with nuclei ranging from smalland dark to large with vesicular nuclei. The cytoplasmcontains fine to coarsely eosinophilic granules. The cellsare arranged in ribbons or nests divided by slender fibrousconnective tissue septa or in large sheets with no particularcellular arrangement 14 . Other histological features includepseudoepitheliomatous hyperplasia of the epithelium. Thisfeature may give a false impression of a squamous cellcarcinoma if a biopsy was taken superficially and thus willaffect the mode of treatment to the patient. In this study,only 3 cases were observed to have pseudoepitheliomatoushyperplasia. In a study done by Eguia et al4, 87.5% ofthe cases were noted to have pseudoepitheliomatoushyperplasia in the overlying epithelium. The cause ofpseudoepitheliomatous hyperplasia is unknown. Granularcell tumours with pseudoepitheliomatous hyperplasiaexhibited increase in Ki-67 staining in the basal cells ofthe overlying epithelium and it may represent an inductionphenomenon mediated by unidentified molecules producedby the granular cells 8 .Although Abrikossoff first described granular celltumour (granular cell myoblastoma) 80 years ago andconsidered it as a muscle tumour 14 , it is currently believedto be of neural origin. Immunohistochemistry demonstratesthe cells are positive to S100 protein 6, 8, 15-19 and neuronspecific enolase 16, 17 , but do not react with chromogranin 16 ,desmin 20 , actin 20 and myoglobin 20 . In our study, five caseswere positive for S100 protein. This finding concurs withother studies 6, 8, 15-19 .A number of tumours have a granular appearancewhen seen microscopically. The differential diagnosisincludes alveolar soft part sarcoma and rhabdomyoma.Alveolar soft part sarcoma is characterized by thepseudoalveolar arrangement of large, oval to polyhedralcell usually with distinct cell boundaries 21 . Rhabdomyomais composed of eosinophilic polygonal cells containinggranular cytoplasm with presence of cross striation 14 .Another lesion which contains granular cells is thecongenital epulis that happens in a newborn14. It is abenign lesion occurring usually on the maxillary alveolarridge of the newborns. Although histologically congenitalepulis and granular cell tumour have similar features,congenital epulis does not exhibit immunoreactivity forS100 protein 18, 22 .Generally, the treatment for oral granular cell tumouris straightforward by simple excision. The prognosis isgood and recurrence is rare. Eleven cases of oral granularcell tumours were treated by excisional resection usinglaser with no evidence of recurrence 5 . Granular celltumours can undergo malignant transformation. Lesionsare classified as malignant granular cell tumours if three ormore criteria are met: necrosis, spindling, vesicular nucleiwith large nucleoli, increased mitotic activity, high nuclearto cytoplasmic ratio and pleomorphism 23 . In a review doneby Aksoy et al 24 , out of 52 reported cases of metastaticgranular cell tumour, 4 cases originated from oral region.95