Intra-oral minor salivary duct carcinoma

?linical RepoJ

INTRA-ORAL MINOR SALIVARY DUCT CARCINOMA Rajeev Sen ~, Prashant P u r w a r z, Bharat RekhP, Sunita Singh 4, B. Arora s J y o t s n a Sen 6, O.P. S a c h d e ~ a 7 : Salivary duct car~ ittoma (SDC) it a recently de~crlbed highly malignant tumor, seet~[ mo~t commoMv in the parot~d gland & occar~ in the 6 'h and 7 ~' decade o[ hfe. Rarely, it occur~ il~] the minor ~'alivary gland~. 7he ea~e ts presented because oJ tt~ rare occurrence and unttsl~ally I pre~etttit~g as a ~welling predominantly i n v o h u t g the maxillary bone. The tumor bear~ hi~'tologicaq homology with breast car~ tnoma, prostattc carcinoma and ~weat duct carcinoma. I Keu Words : minor sahvary duct carcinoma, comedo pattern.

INTRODUCTION Tumors of the minor salivary gland are rare and occurrence of Salivary Duct Carcinoma (SDC) is even rarer. These tumors are highly malignant and the prognosis is dismal. SDC should be differentiated from low grade salivary gland carcinoma because of its poor prognosis even with aggressive therapy.

CASE H I S T O R Y Mr. S. N. Aged 40 years and working as a post office clerk presented with swelling on right side of cheek for the last 7-8 months. He at first noticed it as a small hard, immobile and painful swelling over the right cheek bone with loosening of the teeth forcing him to seek a Dental consultation. Molars and premolars were extracted and

~Assoc Prof, ~ ~Demonstrator, 4Reader, SProf & Head, Dept of Pathology, 6Reader, Dept of Ra&ology, 7Prof & Head, Dept of ENT, Pt B D Sharma, PGIMS, Rohtak, Haryana

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lntra Oral Mmor Sahvar 3 Duct Carcinoma

Fig I

CT scan reveaN a hyper dense lesion seen m the region ol right maxillary sinus with extension me&ally into nasopharynx, superiorly into the orbit and lnferlofly eroding the hard palate

Fig lI

An 111 defined swelhng on the hard palate of the pat*ent

the wound had healed but the swelhng persisted and gradually increased tn s~ze, resulting m closure of the eyehd and the right nosml was blocked Patient was thereby referred to the Surgery Department of the Civil Hospital where F N A C attempted by the Surgeon &d not ymld satisfactory materml CT Scan s u g g e s t e d F~brous d y s p l a s m / O s t e o g e m c s a r c o m a extending medmlly up to the nasal conches, postenorily to the ethmoldal plate and upwards to the orbit ( Fig I) Local E x a m m a t t o n of swelhng over the right cheek m e a s u r i n g 6 x 5 cms r e v e a l e d a hard, fixed lump assocmted wtth shght oedema of the mower eyehd and crusted material m hts rtght nostril The other aspect of the swelhng on the hard palate had s~m~lm consistency (Fig II) A surgical biopsy was done from the hard palate which revealed eplthehal cells arranged m cnbnform pattern, with no defimte evidence of Fibrous Dysplasta A Caldwell Luc surgical biopsy was subsequently performed, which showed predominantly antra-ductal and mvaslve pattern of the neoplasm w~th extensive desmoplasnc response Epltbehal cells were arranged m cribntorm, roman bridges and at places glvmg a comedo'~Cpattern similar to the appearance of Ductal Carcinoma of Breast (Fig III) T u m o r cells were large with m o d e r a t e e o s m o p h l h c c y t o p l a s m and mnos~s was c o m m o n The report was c o n c l u d e d as SDC, highly a g g r e s s i v e t u m o r with

Fig llI

Micro section reveahng a comedopattern of sahvary duct carcinoma mfilteratmg the bone

propensity to have both loco-regtonal and dxstant spread Re-exammanon of the patient dxd not reveal involvement of reDonal lymph node, nor did the patient have any lump m rather breast and the prostate was found to be normal on P/R examination The patmnt subsequently underwent a local excision of the tumor and was g~ven radiotherapy m the neck and palate region

DISCUSSION Sahvary Duct Carcinoma is very rare neoplasm of the salivary glands and was first described by Klemsaqser and coworkers in 1968 They are seen to anqe m the parotids m some 80% cases while only 5% are known to occur in

lndlan Journal of Otolaryngolog) and Head and Neck Surger 3 Vol 56 No 2 Aprtl

June 2004

lntra-Oral Minor Sahvar) Duct Carcinoma

lntra-oral minor salivary glands. SDC are most frequently seen in the 6 ~h and 7 th decades of life with a 3.8 : 1, male preponderance I. SDC is believed to arise from the excretory ducts or as a result of malignant trasformation of ductal cells in a pleomorphic adenoma 23. These patients usually present with a swelling, which is occasionally, painful and may have facial nerve involvement. At times these tumors are clinically misjudged as an adenocarcinoma, adenoid cystic carcinoma, basal cells c a r c i n o m a , bone tumors like osteosarcoma and sebaceous cell carcinoma. Grossly, these tumors are small, mottled yellow white with minutely cystic spaces and measure 0.7-4cms in greatest diameter 4. Histologically, these tumors are composed of ducts filled and distended with cells having micro papillary, tufted, cribriform and solid pattern with central comedonecrosis. Cells are large and have powdery, eosinophilic cytoplasm with a sharply defined cell margin, pleomorphic nuclei and prominent nucleoli. Roman bridging and cellular atypia are o b s e r v e d . T h e s e tumors h a v e a p r o l i f e r a t i n g c o m p o n e n t , which m a y be p o o r l y d i f f e r e n t i a t e d . Infiltration is associated with hyalanlzed stroma. Vascular, perineural invasion and osseous metaplasia are also seen sometimes5 a4 Histologically, these tumors can be confused with metastasic deposits from breast carcinoma, sweat duct carcinoma and prostatic carcinoma. ~ Ultrastructurally, these tumors show mtracytoplasmlc lumina with microvilh. In addition, irregular cocoon shaped collection of tubular or membrane material, desmosomes, tight junction and basal lamina are seen 4. These tumors express EMA, Keratin, B72-3, ct -Lactalbumin, c-erb-2, GCDFP-15 and CEA. ER are not expressed, but one tumor was found to be positive for PR. 5 The positivity of these antigens do not m any way explain the microscopic homology seen in ductal carcinoma of breast, sweat duct carcinoma or prostate carcinoma. The outcome of SDC is unfavouable. Most of the cases reported m literature have an aggressive nature, but a few

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reports with low grade potential h a v e also been documented. There is loco regional spread as well as distant metastasts to the bram, liver, adrenals lung, bone (skull), skin and thyroid. Lymph node metastasis is seen in 60% cases ~. Recurrence is found in 33.3%. 6 These tumors are usually treated with extended excision of the primary site along with removal of the regional lymphnode f o l l o w e d by radiation therapy, v S o m e authors h a v e performed subtotal maxillectomy followed by radical neck dissectmn in cases when cervical lymph nodes were involved. ~ REFERENCES 1

Eplvatlanos A , Damgtrakopoulos J and Trlgomdls G (1995) Intraora] salivary duct carcinoma a chmcopatholog~cal study of four cases and review of hterature Ann Dent ,54 36-40

2

Grenko R T , Gemryd P. Tytor M , Lundqvlst P G and Boeryd B (1995) Sahvary duct carcinoma HlStopathology, 26 261266

3

Ersoz C, Cenk F, Aydln O, Cosar EF and Talas D U ( 1 9 9 8 ) Sahvary duct carcinoma ex pleomorphlc a d e n o m a , analysis of the findings in hne-needle aspiration cytolog 5, and histology Dlagn Cytopathol , 19 201-204

4

Delgado R , K h m s t r a D and Albores-Saavedra J (1996) Low grade sahvary duct carcinoma A distinctive variant with a low grade histology and mtradnctal growth pattern. Cancer ; 78 958-967

5

Lew~s l E , M c K m n e y B C , W~land L H . Felvmro J A. and Olsen K D (1996) 9Salivary duct carcinoma Chnlcopathologlc and lmmunohistochemlcal revmw of 26 cases Cancer , 77 223 230

6

Martlnez-Barba E , Cortes- Guar&ola J A.,Mlnguela- Puras A , Torroba-Caron A, Mendez-lYujfllo S and Bermejo-Lopez J (1997) Salivary duct carcinoma Chnlcopathologlcal and wnmunohlstochemlcal studaes I Cramomaxlllofac Surg, 25 . 328-334

7

lkeda K , Yoshlda F, SuzuM H, O~hlma T, Hlrano K, Takasaka T (1997) A case report of sahvary duct c m c m o m a Tohoku J Exp Med , 183 : 233 -8.

Address for Correspondence : Dr. Prashant Purwar 60, Engineer Enclave Pitampura, New Delhi - 110 034

lndtan Journal of Otolaryngolog2~ and Head and Neck Surgery, Vol 56, No 2, April - June 2004