JKIMSU, Vol. 5, No. 1, January-Marh, 2016 ISSN 2231-4261 CASE REPORT Multifoal Carinoma of Oral Cavity: A Case Report 1* 2 Anilkumar L Bhoweer, Sudarshan Ranpise 1 Consultant Oral Mediine & Dental Radiology and Dental Surgeon, Dadar-400028, Mumbai, 2 (Maharashtra), India, Department of Oral Mediine & Radiology, Bharati Vidyapeeth Dental College, CBD, Navi Mumbai- 400614(Maharashtra) India Abstrat: Oral Carinoma is most ommon among several other aners in our ountry (over 40%). The most ausative fator is tobao in various forms. It is very rare to have multiple foi or entres for aner in oral avity. The ase presents a rarity, having multiple (about 4) aner sites in the oral avity. Keywords: Multifoal Carinoma, Toludine Blue Staining, Field Canerization. Introdution: Ever sine Billworth in 1879 reported the ourrene of multiple primary malignant tumours [1], it has been no longer onsidered to be a uriosity. Multiple primary arinomas at different loations in the body have been reported by quite a few workers and their inidene has been quoted to be from 1 to 7 perent by Mortel [2] and Kuehn [3]. Cade and Lee [4] reported the inidene of multiple malignanies as 4.1%, Eirh and Kregh [5] 8.8%, Mortel and Foss [6] 9.7%, Warne and Gates [7] about 3.7% and Slaughter [8] as high as 18%. Multiple arinomas assoiated with tumours of the lung, gastro-intestinal trat and urinary system frequently ourred in the head and fae [9-11]. Oral avity has been the site of many of these malignanies. Berg et al [12] found that respiratory and upper digestive trat aners ourred six times as often as expeted in the patients with oral aner. In their study, tongue aner patients developed exess aners never often than did the other mouth aner patients with exessive number of laryngeal and lung aners following oor of mouth aner. Horawitz [13] studied the relationship of oral arinoma to the other primary lesions in the body. He found, amongst 116 patients who had oral aner, that about 19 of them had multiple arinomas. Werthamer [11] and his olleagues, in the study of multiple primary lesions, laid down de nite riteria for multientri arinoma. Lund [14], in the study of aner of the bual muosa, reported that statistially the development of a seond arinoma of the same type in the mouth was approximately 15 times as ommon as would be expeted by hane ourrene alone. Tan [15] and his assoiates desribed a ase of arinoma of right and left heek and laimed the multientri origin of the arinoma. It is still a rarity to have more than two different foi or entres in oral avity alone. Though the inidene of oral arinoma is as high as 40% in our ountry (India), multifoal arinoma is rarely observed. The patient who reported to us had about four primary foi at different loations in the oral avity, whih is extremely rare. Journal of Krishna Institute of Medial Sienes University 120
JKIMSU, Vol. 5, No. 1, January-Marh, 2016 Case Report: A 45 year old male patient reported with the main omplaint of a fairly large ulerative lesion on the lower lip, whih did not respond to the loal appliation of various drugs and systemi antibiotis. He had no other serious omplaint. The physial ondition was normal. He was a hroni tobao hewer for over 9 years. There was nothing signi ant about past and family history. On examination, the patient was having a poor oral hygiene and all the teeth were heavily stained. The ulerative lesion on the lower lip, for whih the patient ame for advie and treatment, was very extensive, involving more than 2/3rds of the lip (Fig.1). The uler was not very deep, but had indurated margins and it bled on the slightest provoation. The hroni nature of the lesion whih failed to respond to the treatment by drugs, unhealthy appearane and the indurated edges of the uler, were indiative of a malignant nature and, therefore, it was linially labelled as arinoma of the lower lip. There was a small lesion present on the upper lip whih again was indurated and therefore was also suspeted to be malignant (Fig.1). Fig 2: Multientri Oral Caner Image 2 Intra-oral examination then revealed an extensive lesion on the dorsum of the tongue whih was hyperkeratoti, raised and had a pebbled surfae. The lesion was ulerated at plaes and was indurated (Fig. 2). This lesion was also suggestive of a malignany. The patient did not bother about this lesion as it was not painful, though it was present for more than 6 months. Further examination then dislosed a large leukoplaki path on the right bual muosa, whih showed erythematous pathes intervening the lesion. The patient had a little disomfort (burning) in this region sine last two years, but he did not seek advie for it. This was also suggestive to be an early hange of malignant nature in the leukoplakia (Fig.1). Similarly, there was leukoplakia on the left heek but without any evidene of uleration and hene it was labelled as 'leukoplakia'. Fig 1: Multientri Oral Caner Image 1 Fig 3: Multientri Oral Caner Image 3 Journal of Krishna Institute of Medial Sienes University 121
JKIMSU, Vol. 5, No. 1, January-Marh, 2016 Careful examination then further revealed an ulerated growth, lesion of gingiva in 36, 37 region. The margins of the uler were swollen and indurated. There was leukoplakia all over the marginal gingiva on the left side. This lesion was also linially suspeted to be a possible arinoma of the gingiva (Fig.3). However, the oor of the mouth and palate did not reveal any abnormality and the overlying muosa was normal. Oro-pharyngeal examination did not dislose any lesion. Examination of the nek for any enlarged nodes was negative. There was no indiation of any growth elsewhere in the body. Fig.6: Pre Staining Fig.7: Post Staining Fig. 4: Staining Proess with Toludine Blue Fig. 5: Post Staining All the lesions in the oral avity were subjeted for 'Toludine blue' [16, 17] test linially and all of them whih were linially suspeted to be malignant gave positive reation (Fig. 4, 5, 6, 7). Sine toluidine blue staining over the tongue surfae is not very reliable beause of roughness and debris and opening of muous glands, the stain does not go with even mild aeti aid solution. Therefore, only staining is not onsidered as a reliable method for early detetion of aner. Hene, biopsy of the suspeted lesion is mostly reommended. The linial impression was of multiple malignant lesions in the oral avity. Biopsies were performed from all these lesions to on rm the linial impression. Proper labelling was done for the site of the biopsy. Blood hemistry showed haemoglobin perentage as low as 9gm%. V.D.R.L. test was negative. Journal of Krishna Institute of Medial Sienes University 122
JKIMSU, Vol. 5, No. 1, January-Marh, 2016 Fig.8: Moderately Differentiated Malignant Squamous Cells forming Keratin Pearls in Lower Lip Fig.9: Setion shows Intraepithelial Malignant Transformation in Cheek. Ative In ltration within Underlying Connetive Tissue is not Evident, Carinoma-in-Situ Fig. 10: Setion shows Overlapping Features of Pseudoepitheliomatous Hyperplasia and Squamous Carinoma, Well Differentiated with Early Invasion in Tongue. Fig. 11: Gingival Squamous Cell Carinoma with Moderate Nulear Aplasia, Keratization and Pearl Formation In ammatory Cells in Gingiva. The report of the biopsies from the lower lip (Fig. 8), tongue (Fig.10) and gingiva (Fig.11) were all differentiated squamous ell arinoma and that from the heek was parakeratoti lesion with atypia of the ellular elements and hyperhromati nulei with numerous blood vessels. No in ltration into the deeper struture was evident (Fig.9). This was ompatible with arinoma in situ. The patient was then referred to the aner entre for treatment where he was treated with teleobalt radiation therapy. Disussion: An unusual ase having primary malignant lesions at four sites in the oral avity, i.e. lower lip, dorsum of the tongue, right bual muosa and the gingiva in the region of the left mandibular molar is desribed. The ases so far reported in the literature had two primary lesions and none had more than three different malignant entres in the oral avity. It seems to be really very unusual to have four primary foi in the oral avity. In the ase desribed the lesions were so widely separated from eah other, there was no doubt that these were different foi or entres for malignany Journal of Krishna Institute of Medial Sienes University 123
JKIMSU, Vol. 5, No. 1, January-Marh, 2016 to evolve. The intervening muosa was absolutely normal. The lesion on the upper lip was not onsidered to be a separate primary fous beause this ould be explained as the effet of "kissing" or "transplantation" from the lower lip due to ontinuous ontat. The fat that the patient was a hroni tobao hewer for a period of more than 9 years and the possibility of tobao extrat mixed with saliva spreading all over the mouth and ating as a arinogeni stimulant at different sites in the oral avity annot be denied. Horawitz and Chromat [13] used the word multientri when referring to arinoma arising in a single organ suh as oral avity sine it has been shown that dysplasti hanges annot be found in the tissue surrounding the linially observed aner. In the ase presented, the intervening muosa between the lesions was absolutely normal linially and it did not pik up toluidine blue stain. Caner generally spreads by multipliation of preexisting aner ells by invasion and destrution of surrounding tissues. This onept has been hallenged by several investigators [6, 19-21] who believed that there are other mehanisms involved in the growth and spread of arinoma. They believe that there is a proess of " eld anerization" involved, in whih a whole area of epithelium undergoes malignant degeneration at multiple points and it is not just a single or isolated small group of ells that is involved. It must be really true as, in the ase desribed, there were four lesions in the mouth, very well separated from eah other and the intervening muosa was normal. This is a good example of " eld anerization" sine the entire muosa appeared to be suseptible for malignant hange and the malignant hanges were observed at four different sites in the oral avity, i.e. lower lip, tongue, heek, gingiva and the upper lip. Slaughter [20] and his assoiates, Willis [21] and Brunshwing [22] and his olleagues have submitted linial and mirosopi evidene to support suh a onept of multientri origin of arinoma and its spread by lateral anerization. The lesions are usually not very deep, but their surfae extension is quite wide. In our ase also, the lesions were at but very extensive on the surfae. It is widely aepted that the patient who has one malignany has a greater than average hane of having a seond malignany in an adjaent or a separate area. This was explained by Mortel [6], Lund [15] and Chierii [23]. Therefore it is neessary to have a probing detailed history whether the patient had aner at any site in his body in the past or not. These patients have over average tendeny to have a seond primary lesion, possibly in the oral avity. Hene, all aner patients must be observed arefully at frequent intervals so that an early detetion of aner in the oral avity ould be made whih an have a better prognosis. The patients who were treated for oral aner previously must be thoroughly heked often as the seond lesion in the oral avity may our subsequently. Conlusion: An unusual ase of multiple arinomas of oral avity having primary lesions at four different sites has been desribed. A possible explanation of " eld anerization" and tobao extrat mixed with saliva serving as a arinogeni stimulant at different entres in the oral avity is disussed. The need of detailed history and areful examination is stressed. Journal of Krishna Institute of Medial Sienes University 124
JKIMSU, Vol. 5, No. 1, January-Marh, 2016 Referenes 1. Burkes EJ. Multiple Caners of the Oral Cavity. N. Carolina Dental J. 1973; 21, 23. 2. Mortel CG, Dokerty MD and Buhhenstoss AH. Multiple primary malignant Neoplasms III Tumours of multientri origin. Caner 1961; 14:238-248. 3. Kuehn DC, Bekett R, Reed JF. Tissue Spei ity in Multiple Primary malignanies, study of 460 ases. Am. J. Surg. III 1966; 164-167. 4. Cade S and Lee E. Caner of the tongue. A study based on 653 patients. Brit. J. Surg. Marh 1957; 44:433-446. 5. Erih JB and Kragh LV. Treatment of Squamous Cell Carinoma of the oor of the mouth. A.M.A. Arh. Surg. 1959; 79:94-99. 6. Mortel CG and Foss E.L. Multientri Carinomas of the Oral Cavity. Surg. Gyne. & Obst. 1958; 106: 652-654. 7. Waren S and Gates O. Multiple Primary Malignant Tumours, a survey of the literature and a statistial study. Am. J. Caner. 1932; 16: 1358-1403. 8. Slaughter DP. Multientri Origin of intra-oral Carinoma. Sug. 1946; 20: 133-146. 9. Cahan WC. Lung Caner assoiated with aner primary in other itias. Am. J. Surg. 1955; 89: 494-514. 10. Goodner JT, Waston WL. Caner of the Esophagus. Its assoiation with other primary aner. Caner. 1956; 9: 1248-1252. 11. Worthamer S, Jabus M and Shlman J. Multiple Primary Malignanies. J.A.M.A. 1961; 175:558-562. 12. Berg JW, Shottenfeld D and Ritter F. Inidene of Multiple Caners. III aners of the respiratory and upper digestive system as multiple primary aners. J. Nat. Caner Inst. 1970; 44: 263-271. 13. Horawitz R and Chromet B. Oral Carinoma and Multipliity of Carinoma. Oral Surg.1996. 26: 37-91. 14. Lund C. Seond Primary Caner in ases of aner of the bual muosa. New Ersl. J. of Ml. 1953; 209: 1144-1147. 15. Tan KN, Medak H. and Lawrene Cohan. Extensive simultaneous bilateral arinoma of the bual muosa, report of a ase. J. of Oral Med. 1972; Vol.27 No.2: 54-57. 16. Nihel HH, Chomet B. In vivo staining test for delineation of oral intraepithelial neoplasti hange: preliminary report. J.Am Dent. Asso.1964; 68: 801-816. 17. Bhoweer AL. Toluidine blue in Oral Caner diagnosis. Paper read at Conferene, Indian Dental Assoiation. Jan.1977. 18. Sharp GS, Bullode WK, Helsper JT. Multiple Oral Caner. Caner. 1961; 14:512-516. 19. Slaughter DP, Southnide DP and Smelkai W. Field anerization in the oral strati ed squamous epithelium. Caner 1953; 6:963-968. 20. Willis RA. The mode of origin of tumours, Solitary loalised squamous ell arinoma, growths of the skin. Caner 1914; 4:630-644. 21. Brunshwig A and Thernton T.F. An experimental study of the lateral spread of epidermoid (squamous ells) arinoma in men and to the relation of suh a lesion to the wound healing stimules. Caner. Aug. 1954. 4:515-518. 22. Chierii G., Silverman S.R. and Forsythe B. A tumour registry study of Oral Squamous Carinoma. J Oral Med 1968; 23:91-98. * Author for Correspondene: Dr. Anilkumar Bhoweer, 364/A, Bedekar Sadan, N C Kolekar Road, Above Lakari Saree Shop, Dadar, Mumbai 400028 Email: banil42@yahoo.o.in Cell: 9820288501 Journal of Krishna Institute of Medial Sienes University 125