Bailoor D N

Gupta Mk

Mhaske S

Nahar S

Raghuvanshi V

Raju Ragavendra

CiteSeerX

IOSR Journal of Dental and Medical Sciences (IOSR-JDMS) 
e-ISSN: 2279-0853, p-ISSN: 2279-0861. Volume 6, Issue 1 (Mar.- Apr. 2013), PP 45-48 
www.iosrjournals.org 
www.iosrjournals.org                                                                45 | Page 
 
Juvenile Pleomorphic Adenoma of the Cheek - A rare case with 
review of literature 
 
Gupta MK
1
, Bailoor D. N.
2
, Mhaske S.
3
 , Raghuvanshi V.
4
, Nahar S.
5
, 
Raju Ragavendra T.
6
  
 
1. Professor & Head of Oral Maxillofacial Surgery, and Dean PDA, University, Bhanpur, Bhopal, India 
2. Professor & Head of Oral Medicine and Radiology PDA, University, Bhanpur, Bhopal, India 
3. Head of Oral and Maxillofacial pathology PDA, University, Bhanpur, Bhopal, India 
4. Post graduate Student, Oral Medicine and Radiology, PDA, University, Bhanpur, Bhopal, India 
5. Postgraduate Student Oral Maxillofacial Surgery, PDA, University, Bhanpur, Bhopal, India 
6. Reader, Oral and Maxillofacial pathology PDA, University, Bhanpur, Bhopal, India 
 
 
ABSTRACT : Pleomorphic adenoma in adolescence is relatively rare. In buccal pad fat area these tumors are 
even rarer. A case of Pleomorphic adenoma of buccal pad area of 5 yrs duration, painless & slow growing is 
reported. The mass was removed by wide local excision & a follow up period of 1 yr with no recurrence has 
been noted. Pleomorphic adenoma should be considered in differential diagnosis of mass of the cheek in 
youngsters. 
Keywords:  Juvenile, Pleomorphic, Adenoma, Salivary, tumors 
 
I. Introduction 
Pleomorphic adenoma is a benign salivary gland neoplasm that represents less than 5 % of benign 
tumors in childhood.1Palate is considered the most common area followed by upper lip & cheek.2 The salivary 
neoplasm found in most commonly in juveniles is pleomorphic adenoma. Haemangioma and lymphangioma are 
the mesenchymal tumors which are seen in prevalence after this tumor.1 Mucosa of cheek and buccal pad area 
are uncommon sites for intraoral pleomorphic adenoma.3 In children recently only 3 cases have been reported. 
Here we discuss a case of pleomorphic adenoma in 15 yr old girl and relevant studies are discussed.4,5,6 
 
 
Case report 
A 15 yrs old female patient reported to the department of oral medicine and radiology of our dental 
college with chief complaint of swelling on left cheek of five years duration. Patient noticed swelling 5 yrs back 
since then swelling was growing gradually in size, it was asymptomatic with no history of discharge,bleeding, 
pain, or any sensory changes. Patient had a history of fall 7 yrs back. Her past medical & dental history was 
noncontributory.On General examination she had normal gait and countenance, her BMI was 19.4  
 
                                          
Figure No. 1 : Showing the general examination of  the patient with BMI of 19.4 and Extra-oral examination 
showing the asymmetry due to the buccal swelling approximately 3 X 2 cms. 
Juvenile Pleomorphic Adenoma of the Cheek –A rare case with review of literature 
www.iosrjournals.org                                                    46 | Page 
On Extra Oral examination facial asymmetry due to swelling was noted on the right side. A solitary 
dome shaped, oval, swelling with smooth surface was present on right cheek region. (Please see figure No. 1) 
No abnormality detected with overlying skin. Swelling was approximately in mid cheek region 3 X 2 cm in size 
extending superior-inferiorly from ala-tragus line to 2 cm above the lower border of mandible. Antero- 
posteriorly its was extending 2 cms from angle of mouth to an perpendicular dropping down from  outer canthus 
of  right eye to lower border of mandible . 
On palpation inspectory findings were confirmed, additional findings noted were swelling was non 
tender, firm, non fluctuant, non reducible, non pulsatile & mobile in all planes. Local temperature over the 
swelling was not raised. Skin overlying the swelling was pinchable.  
 
 
Figure No 2: The swelling was non pulsatile, firm in feel and moving freely in all planes. 
 
 
 
Figure No 3: The Ultrasound report showing well defined hypo- echoic lesion anterior to the muscle plane.  
Figure No 4: An excision of the lesion in progress, it was localized easily and had a fibrous capsule. 
 
On the basis of history given by the patient and clinical features a provisional diagnosis of organized 
hematoma was made; differentials considered were fibroma, antibioma, lipoma, fibrous hyperplasia (reactive to 
trauma) schwannoma & pleomorphc adenoma. Investigations advised were panoramic radiograph & ultra 
sonography. No abnormality was detected with OPG. USG revealed a well defined, hypoechoic lesion 
measuring 2.7 X 2.1 cm was present anterior to the muscle plane. No internal vascularity was noted, no bony 
involvement was seen. 
Excision biopsy was advised as an investigation and treatment plan.  Surgical excision was uneventful, 
excised tissue was sent for pathological examination. Macroscopically tissue was, round to oval in shape, 
measuring about 2 X 2cm, grayish pink  in color, soft to firm in consistency; when cut into two halves the cut 
surface was smooth, covered by well defined thin capsule. The cut surface was  uniformly creamy white in color 
showing a cut potato like surface.  
 
 
 
 
 
 
 
 
 
Figure No 5: Showing the gross specimen which was submitted to Oral Pathology department for HP 
examination. 
Juvenile Pleomorphic Adenoma of the Cheek –A rare case with review of literature 
www.iosrjournals.org                                                    47 | Page 
Histopathological examination revealed a well defined capsulated tumor mass with combination of 
epithelial & stromal components (fig 6). The epithelial constituent was arranged in large sheets & islands 
composed of squamous, basaloid & cuboidal cells, few mucous cells, plenty of plasmacytoid spindle 
myoepithelial cells and few duct like areas with cuboidal large cells were  noted. Areas of chondroid, myxoid, 
osteoid, were tissue also present with variable predominance (fig,7) .The  findings were confirmative for the 
diagnosis of pleomorphic adenoma 
 
  
Fig 6 - The tumor was showing a well defined overlying capsule with neoplastic tissue composed of epithelial 
and variability in stromal components. (low power view). 
 
 
 
 
 
 
 
Fig 7 - Epithelial proliferation was in the form of sheets and islands and background stroma was composed of 
myxoid , chondroid and osseous tissue (high power view). 
 
II. Discussion 
The mean age at initial presentation of 42 patients treated by Peter Zbären et al7 mentioned that age of 
recurrence observed was 34 yrs and the interval between initial surgery and tumor recurrence varied between 3-
15yrs.8,9  The surgical treatment of pleomorphic adenoma has  never been standardized and Renehan A et al 
1996 & Piorkowski RJ et al 1981 both suggested that enucleation is likely to result in recurrence as compared 
with   total gland excision.10,11 
In our juvenile patient since the surgery was simple and able to completely remove a well 
circumscribed lesion, the removal of associated gland was not an issue. This is in agreement with Leverstein H 
et al 1997 who stated that surgery of this neoplasm must be highly customized on the basis of histologic type, 
extension & patients age.12 
Damage to the peripheral branches of facial nerve is always a surgical risk, when surgical intervention 
is done 3 in the facial region. However in this case there was no neurological deficit. This was mainly due to the 
intraoral approach for the well circumscribed tumor.Response to  Radio therapy: 
Renehan A et al has suggested adjuvant radiotherapy for Pleomorphic multi nodular recurrent case .10 
On the contrary Glas AS et al 2001 have stated that only when surgery is contraindicated for medical reasons 
should radiotherapy be even considered.13 Samson MJ et al 1991have suggested that radiation may be justified if 
surgical risk of sacrificing the adjunct nerve is relatively high.14 In our case no such risk was evident and we are 
prognosticating a low level of recurrence in this well circumscribed & completely excised tumor. 
The differential diagnosis of adolescent Pleomorphic adenoma includes  abscess, foreign body reaction 
to substances like fish bone, fibroma, dermoid cyst, lipoma, & rare entities like rhabdo-mayo-sarcoma, muco-
epidermoid carcinoma & adenoid cystic  carcinoma.15  .Manjunatha BS et al 16 have mentioned about a rare 
variant termed as Fibrolipoma which is seen  in the buccal fat region of the face. Early diagnosis of such lesions 
in Juveniles is very critical. 
 
III. Conclusion 
To conclude, adolescent Pleomorphic adenoma of cheek is a rare neoplasm and not easily diagnosed at 
provisional level, wide surgical excision is the treatment of choice and regular follow up at least monthly for 
period of 24 months would give us a hint of recurrence when it occurs. In India a rare conversion of 
Pleomorphic adenoma to carcinoma has been reported by Kini Y et al 17 and this makes the early diagnosis of 
such seemingly benign lesions even more urgent.  
 
 
 
Juvenile Pleomorphic Adenoma of the Cheek –A rare case with review of literature 
www.iosrjournals.org                                                    48 | Page 
References 
[1]. Rogerson KC: Salivary gland pathology in children.Oral Maxillofac Surg Clin N Am 7: 591–598, 1995 
[2]. Toida M, Shimokawa K, Makita H, Kato K, Kobayashi A, Kusunoki Y, Hatakeyama D, Fujitsuka H, Yamashita T, Shibata T: 
Intraoral minor salivary gland tumors: a clinicopathological study of 82 cases. Int J Oral Maxillofac Surg 2005, 34:528-32. 
[3]. Bablani D, Bansal S, Shetty SJ, Desai R, Kulkarni SR, Prasad P, Karjodkar FR: Pleomorphic adenoma of the cheek: a case report and 
review. J Oral Maxillofac Surg 2009, 67:1539-42. 
[4]. Yamamoto H, Fukumoto M, Yamaguchi F, Sakata K, Oikawa T: Pleomorphic adenoma of the buccal gland in a child. Int J Oral 
Maxillofac Surg 1986, 15:474-7. 
[5]. Cohen MA: Pleomorphic adenoma of the cheek. Int J Oral Maxillofac Surg 1986, 15:777-9. 
[6]. Kronenberg J, Horowitz A, Creter D: Pleomorphic adenoma arising in accessory salivary tissue with constriction of Stensen's duct. J 
Laryngol Otol 1988, 102:382-3. 
[7]. Zbären P, Tschumi I, Nuyens M, Stauffer E. Recurrent pleomorphic adenoma of the parotid gland. Am J Surg. 2005 Feb;189(2):203-
7. 
[8]. Naeim F, Forsberg MI, Waisman J, et al. Mixed tumors of the salivary glands. Growth pattern and recurrence. Arch Pathol Lab Med 
1976;100:271–5. 
[9]. Yugueros P, Goellner JR, Petty PM, et al. Treating recurrence of parotid benign pleomorphic adenomas. Ann Plast Surg 
1998;40:573–6. 
[10]. Sistrunk WE. Mixed tumours of the parotid gland. Minn Med 1921; 4:155–60 
[11]. Klopp CT, Winship T. Treatment of mixed tumors of the parotid gland by subtotal parotidectomy. Arch Surg 1950;61:477– 86. 
[12]. Leverstein H, Tiwari RM, Snow GB, et al. The surgical management of recurrent or residual pleomorphic adenomas of the parotid 
gland. Analysis and results in 40 patients. Eur Arch Otorhinolaryngol 1997; 254:313–7. 
[13]. Glas AS, Vermey A, Hollema H, et al. Surgical treatment of recurrent pleomorphic adenoma of the parotid gland: a clinical analysis 
of 52 patients. Head Neck 2001;23:311– 6. 
[14]. Samson MJ, Metson R, Wang CC, et al. Preservation of the facial nerve in the management of recurrent pleomorphic adenoma. 
Laryngoscope 1991;101:1060 –2. 
[15]. Baldus SE, Streppel M, Stennert E, Dienes HP: Pleomorphic adenoma (mixed tumor) of the external auditory canal. Differential 
diagnosis of the tumors of the ceruminal glands. Pathologe 1999, 20:125-9. 
[16]. B.S. Manjunatha,1 G.S. Deepak Pateel,2 and V. Shah3. Oral Fibrolipoma-A Rare Histological Entity: Report of 3 Cases and Review 
of Literature J Dent (Tehran). 2010 Autumn; 7(4): 226–231. 
[17]. Kini Y, Desai C, Mahindra U, Kalburge J.  Rare carcinoma ex pleomorphic adenoma of the buccal minor salivary gland causing a 
therapeutic dilemma. Contemp Clin Dent. 2012 Apr;3(2):209-11 
 
 
 


Juvenile Pleomorphic Adenoma of the Cheek -A rare case with review of literature

http://citeseerx.ist.psu.edu/viewdoc/download;jsessionid=2886E1A0CCE8B7DC23EBE95FCB059B7B?doi=10.1.1.1076.1023&rep=rep1&type=pdf

Juvenile Pleomorphic Adenoma of the Cheek -A rare case with review of literature

Abstract

Similar works

Full text

Available Versions

CiteSeerX