Compound nevus of the buccal mucosa

Report

of a Case

A

SKVUS is a benign neoplasm dcrivcd from pigmented or dopa-positive cells1 The compound nevu$ has histologic features similar to those of the intradermal nevus (common mole) and the junctional ncvus (dermoepidermal ncvus or marginal ncvus) . A perusal of the literature indicates a dearth of case reports on oral nevi. Of the cases recorded, Bcrnicr and Tiecke” and Allen and Bruce4 reported cases similar to the intl*adrrmal type occurring in the gingiva. couch and Kaufman” reported a junctional nwus of tha palate. Two palat,al lesions of a ra.re type oi’ ncvus known as the blue (Jadassohn-Tieche) nevus were described by Scofield.” Field and Ackerman’ presented a case OS nonpigmentcd nevus of the gingiva. Pack, Lenson, and CcrbeP emphasixcd the “extraordinary rarity” of pigmctnted nrvi in the mucosa of the oral cavity after studyin, 1~their distribution over the entire hod?; in 810 patients. To my knowledge, the only case of a compound JW~IS of the oral cavity to date is that mentioned by Greene and associates” ((3~~: 8, Armed Forces Tnst.itntr: of Pathology Accession No. 268206). (‘oniponnd and junctional ncvi, bclcausc of so-called junctional activit> (,acrtirc? formation of ~WVUS cells in t.hc basal layer of the epidermis at the dcrmal-epidermal junction), are forerunners of the melanocarcinoma’~ l”y I1 (malignant melanoma, to others). Molanoca.rcinoma of the mucous membranes of the head a.nd neck, genitals, and anorectal regions is almost uniformly fatal.‘“~ I:{ (.Xaudhry, Burke, and Uorlin” report that these malignant t.umors of t.hc oral cavity hax a much worst prognosis t.han those of cutaneous origin. Thus, the potential malignancand thck rare occurrcncc of t.he compound iicvw in the oral ca.vity arc t honght to 1x1 ol’ sufficient int.cbnbst to justify publ.ication of the prcscnt report. ( : ME

REPORT

A YY-ywr-old unmarried woman wtd referred ou Sept. 15, 1959, for removal of an impaxted third molar. During the esamination, 5 scssilc, brownish black, pigmented lesion WHX seen iu the oral mucosa about 3 cm. from the left cornw of the mouth. It measured itbout 1 cm. in diameter ad ~5s loaded at the level of the owlusal line (Fig. 1). The overlying muoosu was smooth. There w:w no history of recent cnlargcmcnt., ulceration, or Submitted

for

publication

on July

30, 1960. 27

28

Fig.

I.-l5uccaI

lesion

loc,ztt?tl

at lcccl

of owlusnl

lint,

opposite

upper

IePt cuspid.

N&roscopio Bzamination.--The pathologist rcportcd that microscopic examination of the buccal mucosa revealed an nrca in which the supporting connective tissue contained dense collections of fairly uniform cells arranged in a mosaic fashion. Superficially, some of these were heavily pigmcuted and others had agglomerated nuclei. The junctional areas of the epidermis contained focal collections of heavily pigment,t:d cells which were separated from the surrounding epidermis by a wdl-defined shrinkage artifact. .I. wide margin of intact, un-involved tissue separxtcd this lesion from the cut edge of the specimcu. Diagraosis:

Compound ncvus, buccul mucosa (Figs. 2 ana 3).

DISCUSSION

Factors Influencing [email protected] dcgrcc of malignancy of melanocsrcinema is influenced by many factors.13 The factors of age and sex hormones produce the worst prognosis in youn, w adults and pregnant women. In patients under the a.ge of pubert.y a,nd in old momcn, the prognosis is better. Chronic infection and repeated t.rauma also contribut,e to malignant change.’ As far as location in the body is concerned, the mortalit,y is high in casesof melanoma of the genital, mucocutaneous, and head and neck regions.12*I3

This section Fig. 2.-Photomicrograph of compound ncvus of buccal mucosa. junctional (dermoepidermal junction) changes and intradermal cell nests characteristic compound nevus. Junctional ccl1 nests (C.N.) appear to be “dropping ofP” into the (Hcmatoxylin and cosin stain. Magnification, xi5 ; rc!duced $5. )

shows of the dermis.

Fi . S.-Higher magnification of junctional cell nest. Clear space roughly outlines an oval ccl? nest. hote finely granular accumulations of melanin in cytoplasm of melanoblasts (N) comprikng cel1 nest. Cytoplasm of other melanoblasts is clear. Phagocytized melanin, some in the form of coarse granules, is seen in melanophorcs (M.P. ). (Hematoxylin and cosin stain. Magniflcatlon, x500 ; reduced J,,$)

A clinical sign indicating a possible change from bcnignit.y to malignancy is darkening of pigment4 nevi. Ulwration and bleeding arc late signs which probably indicate a hopeless prognosis.“, *X I2 Ucration in size, scaling, Crust-

iug, tenderness, and infhnmhm arc ott1c.r I;II(~ (.tI;II,;lc,trl,isti~s \vtlic-tl tll* indicate malignant transforniatioi~.‘.‘;’ Histologically, considerable and i rregu tar junction ac*tivity and atypica lit.>. 01: tumor cells arc strongly su ggcst.i\-c! of malignant changc.“~ ~‘h!rOSCOpiC! (‘videncc of malignancy includes (1) invasion :~f the tumor ccllls in1.o the ugpcr layers of the epidermis, (2) the prescncc 01: &x~ration, (3) t.hr ~~~rosenco OI mitotic figures, and (4) the prescncc of 811 infl;m~~~~t.o~y infiltml I:. It is Cillltioned that malignant melanoma may csist in spite of the absenc’c of these .COUI criteria of malignancy.“’ Allen nimitiow t tie loosening 01: the tolls of tlw wl(* pegs and t.heir incorporation in tti(l tIerma ncoplasn~. I’rogrcssiv(! ljartial dissolution results in the IJC~S. ISOhtd Yph(‘l’iCtlt Ct!llS Jllil?IJO I’OlUltl Up to l.tlV stratum corneum. These n~us cells powdered with melanin arc’ considcrcd hy .4llc?n to by the source of all l~lelaJ~o~;~rci~~o~~~as. Iliferentid [email protected] oral mucosa is ho~nologous t.o skin and is likcwise richly endowed with (~l~romatopllorcs ;Illd dopa-positi\-o ~clls.~~~ Ih Thercfore, the oral mucosa 1na.y be affected by ilisordcrs of pigmentation in a manner similar to t.hc skin. Variations in the? appearma: 01: skin and month lesions arc usually related to environnicntal ant1 1)hysiologic pcculiaritics of tlw respcctivct tissues. l’igmcnts which Inay 1Jc fount1 in Ihc OlXl mucosa’!’ include melanin, hemosiderin, deposits of metals, and tattoo of the mouth. Blood and circulatory changes may also influence tlic color 0C oral tissncs anal tlicrcIl0r.r must hc corisidered in the differential diagnosis. The following c*onditions art’ csamI)l(lti : oral rnclanosis of acanthosis niprans and Addison’s discasc: ~~~tllol* o I’ ora I mucosae in anemia, arsenical pigmentation, mucosal li?perpi~~entat.iori oli pcllagra and hypothyroidism, black tongue, blue gum of soinc PUCKScyanosis, jaundice, melanoma, metal deposits (bismut.11, lead, silrrr, and lncr~ury j ! mtll+ moglobinemia caused by drugs, spot.1y normal pigmentation, polycythcmia ~J*oducing purplish red mucosac? quinacrinc ( Atabrine) slaid~luc spois. ant1 tattoo ol tongue or hgums with ink or charcoal. Most of thcso condit.ions arc familiar to the reader. In the rare category is acanthosis nigrans, a disease of ehromaffin tissue (adrenal medulta or paraganglions) insufXciency. The constitutional s\-mptoms are similar to ttiosc of L4ddison’s disease. The skin sl~owsnwlanin hyporpigmcntation and papillomatous hypcrtrophy with a prcdilcction for the asilla, i.mroin, submammary region, and elbows, and knees. Addison’s disease?” is cha racterixd lay marked weakness, low blood prossm:c~, loss of weight, anorexia, nausea, yomitin g, constipation, and hypoglycemia. Tht! metabolic disturbances include dccrcascd SL’I’UJ~sodium and c*hloricle concentration, increased plasma potassium level, and elevated blood urea aud nonprotein nitrogen. Melanin pigmentation of the skin and mucous mcmbranc’s is common but is not always prcscnt. Thn int-ensity ol’ Itlc!l;tnin pigmentation ranges from light to almost black. Benign pigmcntcd skin c*onditions to bc cxmsidcrcd in differentiating ncl-i of the facial area (skin and mucosal) from malignant melanomas or mc!lanocarcinemas include t.he following :I3 pigmented seborrhcic keratosis or wart, senile kcratosis or ‘ ‘ sailor’s skin, ’ ’ histiocytoma, scelerosing hemangioma, pigmented

Volume N\‘omher

l.i

I

COMPOUND

NEVUS

OF

BUCCAL

MUCOSri

3 I

basal-cell epithelioma and other pigmented nevi (such ax the intradermal MYUS~ blue nexus, Mongolian spots, nevus of Ota), and t.he benign melanomas, which should include the “juvenile melanoma,” and the melanotic freckle of Hutchinson or lentigo maligna. Histiocytoma occurs in patients beyond middle age as a discrete, firm, rounded nodule which is darker t.han the rest of the skin. The lesions are Histologically, these are fibromas located located primarily on the cstremitities. III the dermis, and they contain deposits of hemosiderin. The blue ncrus is a t.ype of pigmcntcd nevus related to the Mongolian spots of orientals. A type of blue ncvus that begins, in adults, on the conjunctiva as a bluish discoloration and la.tcr spreads to the pcriorbital tissues is called the nevus of Ota. Mongolian spots arc bluish pigmentations found at birth, most frcqncWlin the sacral area, which usually disappear with age. ‘ ‘ Juvcnilc melanoma’ ’ is a term given to those lesions seen in cliildhood which clinically and histologically resemble the malignant melanoma but which are cured by local cscision. In about 50 per cent of the WSW, giant ~11s formed by syncytial accumulation of melanoblasts is seen. The melanotic freckle of Hutchinson, or lentigo maligna, is charactcrizcd by a history of gradual cnlargemcnt of a pigmentctd macule which becomes somewhat thickened. It occurcs quite frcqucntly 011 tllc Pace. It, is a slowly growing, superficial type of melanoma. Ot.hcr caonditions to bc ruled out WC superficial forcipn bodies, such as localized argyria (amalgam tattoos) ,?I and superficial cmbcddcd warfare missilts, oral mclanosis associated with intestinal polyposis (I’eutz-Jrghers ~-IIdromcl) ,22 and traumatic and spont.ancous hcmatomas. Of the spontaneous hematomas, pctcchiac of purpura hemorrhagica, hemophilia, liver disease! and hypovitaminosis C have to be considcrcd. On a histologic basis, the int.radermal and the junctional ncvi are the lesions most frequently to bc distinguished from a compound ncvus. Clinically, the intradcrmal nevus’ of the skin may be flat or raised, papillary, and kerat.otic. These lesions may be present at birth but have a peak incidence at pubcrt... IIistologically, the tumor is romposed of nests and cords of cells in the dermis. The typical JHXUS cells are oval or cuhoidal with a homogcneons cytoplasm. The nuclei of the cells arc modcratcl- chromatic and round. Melanin pigment is usually found in the more superficial ~11s. Slitotic figures are rarely seen in ncri of ad&s. The ncvus cells usually trail off into the The cells here tend to be spindle dept.hs of the dermis without sharp limitation. shaped, surrounded by fibrous tissue which has t.he loose, wavy, pale appearance o-f a ncurofibroma. Occasionally, the IICVLIS cells are a~~lnged in columns snggcstivc! of neural sheaths ( ’ ‘ ncluroid tubes ’ ’ 01: Nasson j . ‘Ii In other areas, thr fibrous tissue may be in concent.ric arrangement (“lames foliacec” of Masson) resembling Mcissner’s tactile bodies.“~ The overlying epidermis is usually thinned and may bc fla.t or papillary, with or without hyperkcratosis. The junctional INWIS~ is a potentially malignant neoplasm. Fortunat.ely, ma.lignant transformation is rclativcly infrequent. In clinical appearance, t.he junctional nevus of the skin is a flat, smooth , gcncrally hairless, light to dark

32 lJromI

0,s.. O.hl. & O.P.

ARAMlNE Inde.

l-‘igIlIcIhx~

illOh

011

th!

Wilt

Ji,nu:,l.y.

I’d

SUl’l’itC(‘S

I’K?

(Jt: th(! h;lJl(\S :ltl(l fc!(q

on th(! grnit,als W.U~ll~ ;t.tY? ,iunctic)niil nvvi 01’ ii1 l(b:]st Ilij,~(! a jllll(:f ioll;ll cc)lllponent in thv form ol’ cottlpoutId 11v~llS. .Ilistologically, bhc: jnwtiottal itcvtis is charactorixcd lay ditstws 0I’ ctIlargc:tl, twtndd, ~OOSCIWC~ CC~IS (]I’ 1hb IJ;~.s;~;~Jtl prickle cells of tltc cpidcrtttis. The ncvus wlls arrattgcyl in ~NJII~IS :l~)pvat. as WlI nests (‘ ‘ thcyues~ ‘) sulT01utdr11 11). il. clr;t 1’ q,acc. Liitygw wll ticAs whiclt project, into the corium 11ta.v bc seen. Tllcg Illa). illJlJ(‘Hl’ to 1JC in ttlc’ Slapc 01 dropping off (‘ * abtropfuntg * ’ ) . ‘1; I I’ tttitot ic figures utld IlOt.iWit~lI(! t.Hl~pl~lSiil ill’c cvidetit, the lesion may IJo on t.hc wtyc of tnaligtiant 1 I~~IlSf~J~tttilt iwl. The term contpouad ~tct.‘11.).’ was SII ggeslcd 101, itttradcrntal Itcvi which ha\-c aI1 associated junctional CIlitIl#e. ~~1tOllt. 3 2 pet Wilt. otf iIlt.~ild~?tW1it1 ncvi in i1tlUltS alld ;lhOUt 98 per Wtlt. 0I’ iictvi iti c~ltiltlrcm priw lo pnltwty liar this junctional cliangc. Clinically, it is dificult t.0 (list inguish a cwtttltowitl ncvus I’twii a.n intradcrmal Iicvus. Ilistologic cwtminat.iort is t,ltc only ntwrts of nl;tking this distinction. Microscopic fcatuws o-f tlw ititratlcrnial news itl’(’ Scott in association with ;junCtiollill c~hatiges. Thcw charact.cristic.s were notrd in I he discussion of intradorntal and junctional ncvi. .I unctional activity makes the conq~ounti novw l~c~ltavct liktb the jnnctionttl Itwws, and thus it, is potctitiall~ malignant. Xonpigrncntcd ncvi itnisi also ba cotisiilcrcd in t,lic diffcrcntial. diagnosis. Thesr are difficult to diagnose clinically and somctimcs even ltist,ol(~~ically. “I IGnphasizing t.his problem is the fincling that in 50 ~JW cent. of t.ho ntelano(*arcinornas of mucous Itl(?llllJtYltlw, thwr is an absence of appreciable tttnomtts 0T mclitnin.L The clinical features of the various conditions, thr age of t.lw patient. the history and location of the l&on, subjcctiw symptoms, and examination of tltc I~owovw, t.lie final tliagnosis blood arc helpful in the differential diagnosis. often may be obtain&l only by microscopic CsUlllitlat.iott.‘” Ocassionally, ;I clctrMedical consultation conwrning hcnw matologist may haw to bc consulted. logic? metabolic, or cnclocrine disturbances will 1~ necessary for sotrlct ~LWS. 1’~scltmelzt.-~Iost authors favor rcnioval of all pigment-cd Icsions ttlilt arc subject to irritation. 1list~ologic exam inat.ion 0I’ cwiscd spccimcws is matidatory, for in most cases it is difficult. clinically lo iwopnizc heiiign ot* iiittlignant cliangc.l~ ’ z iUld

1. 3 case of a rare compound Ile~3ls of th bnccal IllLIIlCOSil has been prcsentcd. 2. Cellular activity at t.he dcrtnoepidcrmnl junction makes the c.omponnd nevus a potentially malignant neoplasm. 3. Excision and histologic examination of all pigmcntcd lesions that. arc subject to irritation is advised. RFFERBXCES A 1. Allen, A. C.: The Skin. In Anderson, W. A. D.: Pathology, C. V. Mosby Company, pp. 1166-1172.

cd. 3, St. Louis, 1957, The

C’OMI’OUNI) 2. Allen, 3. 4. 5. 6. 7. Y. 9. 10. 11. 12. 1.3. 14. 15. 16. 17. 18. 19. 20. 21. 22.

NTE:VUS

OF

l
MlJ(‘OS.\

33

A. C.: Reorientation on Histogeuesis and (‘linical Significance of C’utiltlcous Ncvi and Melanomas, Cancer 2: 28, 1949. Nevus of the Gingiva, J. Oral Surg. 8: 163-167. 1950. Bernier, J. L., and Tiecke, R. W.: Allen, R. R., and Bruce, K. W.: Nevus of the Gingivn: Report of Case, J. Oral Surp. 12. “57-269 1954 Couch, ‘C.-D., Jz, ;nd’Kaufman M.: Junctional Nevus of Palate: Report of Case, J. Oral Hurg. 14: 334.336,195;. The Blue (Jadassohn-Tieche) News; a previously IJnreportc~d IIIScoficld, II. II.: t.raoral Lesion, J. Oral Surg., Ants. $ Hosp. D. Hprv. 17: 4-14, 1959. Field, H. J., and Ackerman, H. A.: Non-pigmented Nevus of Labial Gingira, Am. J. Orthotlontiw & Oral Surg. 29: 180-181, 1943. Regional DisLriln~tion of Moles x1111 Pack, G. T., Lenson, N., and Gerber, D. M.: Melanomas, A. M. A. Arch. Surg. 65: 862470, 195’. Greene, G. IV., Haynes, J. W., Dozier, Y., Blumbcrg, J. M., and Scrnicr, a. L.: Primary Malignant ~~clanonia of the Oral Mueosa, ORaL tilx(i., ()RAI. 3bm. & ORAL PATII. 6: l&%-1-143, 1953. l3ccrman. II.. Lane. K. A. G.. and Shafier. B.: Pinmcntcd Nevi and Malienant. Melanoma 0 of ihe &in, Am. J. M.’ SC. 229: 444-465, 19i5. Becrman, II.. Lane, R.. A. G., and Chaffer, H. : Pi~mriitcd Nevi and Ma1 iglinnt Meianonia of the’ Skin. Am. ‘J. M. Sc. 229: 683-600. ‘i955. .411cn, ,I. C., and Spitz, ti. : Hiatogenrsis and Clinico;r:ltliologic Correlation of Sevi and Malignant Mela~~orrlas, A. M. A. Arch. Dcrmat. & Hyph. 69: 150-171, 1954. Andrcms, G. C., and Domonkos, A. N.: Warts, Moles au11 Malignancies, Texas J. Med. 55: 568-590. 1959. Chaudhry, A. P., Burke, Hogcr J., and Gorlin, R. J.: Xalignant Melanoma of t.lw Oral Cavity, ORAL Hum., OKAL MED. & OWL PATH. 13: 5fW688, 1960. Sachs, W., MacKee, G. M., Swartx, 0. D., and Pierson, .I{. S.: Juuetion Ncvus nllc’l Nevocarcinoma, J. A. M. A. 135: 216-218, 1947. Lever, TV. F.: Histopathology of the Skin, Philadcll~hia, 1919, J. 13. Lippincott Company, pp. 391-408. Laidlaw, G. I’., and Cahn, L. R.: Mclanoblasts in t.he Gum, J. I). Hen. 12: 534-537, 1932. Cattoni. M.: Melanoblawts in the Inthsmcd Ginriva. 01~41, MIEI). & ORAI, .A I ORAL SIW:.. I'iTH. 6: 1095-1110, 1953. Sutton, R. L., aud Sutton, K. L., Jr.: Handbook of Dist:asw of the Skin, St. Louis, 1949, The C. V. Mosby Company, chapter on Diseases of Mueosac Adjoining the Skin. Anderson, W. A. D.: Pathology, cd. 3, St. Louis, 1948, The C. 8. Monl)y Company, p. 1096. Bell, C. I)., Cookscy, 1). E., ant1 Nickel, W. R.: Amalgam Tattoo (Localized Argyria 1, A. M. A. Arch. Dcrmat. & Hyph. 66: 593 195’. Jeghers, H., McKusick, V. A., and Katz, k. II.: Generalized Intestinal Polyposis and Melanin Spots of the Oral Mueosa, Lips awl Digits, NIW I*:s(;I,AsI) J. MEI). 241: 993-1005, 1919. 181

8. Kiulim

ST.