Two neurogenic tumors of the oral cavity

Two neurogenic tumors of the oral cavity

TWO NEUROGENIC TUMORS OF THE ORAL CAVITY (!HARLES M C K A Y , M . D . S . , BEI+FAST, NORTHERN IREI,~ND I.:M()RS of the nerve tissues are ~'are in th...

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TWO NEUROGENIC TUMORS OF THE ORAL CAVITY (!HARLES M C K A Y , M . D . S . , BEI+FAST, NORTHERN IREI,~ND

I.:M()RS of the nerve tissues are ~'are in the oral cavity. The i'olh)wing benign tyl)cs have been descr'ibcd: neurofibroma (solitary and multiple), ncur'inoma (neut'ilemmoma, s c h w a l l l l O l l l a ) , ganglioneurl/ma, and a m p u t a t i o n (traumatic) neuroma. A c a s e o f t P a l u t l a t i c llCUPOlllfl, presenting some unusual features, a n d o n e (ti' ncurinoma ave /]cscribed here.

T

Traumatic (Amputation) N e u r o m a . - - A w o m a n , aged 82 y e a r s , w a s r e f e r r e d by h e r m e d i c a l a d v i s o r to t h e D e n t a l D e p a r t m e n t , R o y a l V i c t o r i a H o s p i t a l , B e l f a s t , N o r t h e r n I r e l a n d , c o m p l a i n i n g of a p a i n f u l s w e l l i n g i n s i d e t h e m o u t h . She had been a w a r e of an i n t r a o r a l s w e l l i n g on t h e lower j a w for s e v e r a l y e a r s , b u t it h a d g r o w n r a p i d l y in t h e last two m o n t h s f o l l o w i n g t h e r e m o v a l of a root, a n d r e c e n t l y it had become e x t r e m e l y s e n s i t i v e on e a t i n g . All t e e t h h a d been e x t r a c t e d a b o u t fort)" y e a r s p r e v i o u s l y and a full u p p e r d e n t u r e only w a s worn. T h e p a t i e n t w a s f r a i l b u t h e a l t h y , w i t h no h i s t o r y of serious illness. T h e a l v e o l a r p r o c e s s e s were g r e a t l y resorbed. A fingerlike m a s s was seen a t t a c h e d to t h e crest of t h e b)wer r i d g e in t h e r i g h t m o l a r region. ~t m e a s u r e d a p p r o x i m a t e l y 3 cm. by .l cm., b u t w a s s o m e w h a t b r o a d e r at t h e f r e e e n d ; it w a s lh:m in c o n s i s t e n c y a n d purplered in color, w i t h u l c e r a t i o n at t h e tip. T h e t u m o r w a s f r e e l y mobile a b o u t its base on t h e s l i g h t e s t p r e s s u r e . I t s n o r m a l p o s i t i o n was u p r i g h t , b u t it was f o r c e d d o w n w a r d a n d f o r w a r d d u r i n g m a s t i c a t i o n a n d c a u g h t at t h e tip on closure of t h e j a w s . It w a s e x t r e m e l y s e n s i t i v e to t o u c h at t h i s point~ b u t not e l s e w h e r e . R a d i o g r a p h s r e v e a l e d no b o n y abn o r m a l i t y , a n d a p r o v i s i o n M d i a g n o s i s of f i b r o m a was m a d e . O p e r a t i o n c o n s i s t e d of r e m o v a l of t h e t u m o r , u n d e r r e g i o n a l a n e s t h e s i a , by disse(tting it f r o m its a t t a c h m e n t to t h e n J u c o p e r i o s t e u m . T h e m e n t a l f o r a m e n w a s n o t s e e n ; h e m o r r h a g e w a s a r r e s t e d h v s u t u r i n g arm t h e t i s s u e s healed n o r m a l l y , w i t h no a n e s t h e s i a of t h e lip. T h e p a t i e n t is now s5 y e a r s of a g e a n d ha,~ ]lad no recurre~lce of l h e s w e l l i n g . Microscopic cxaminotion: [n some respects t h e t a i n o r rcseml~le(1 a n e u r e i n o n m , as p r o l i f e r a t i o n w i t h p a l i s a d i n g of t h e cells of t h e p e r i n e u r a l she~tth w a s seen in one area. t I o w e v e r , t h e m a i n p a r t of t h e t i s s u e showed i n t e r l a c i n g | ) a n d s of n e r v e fibers s e p a r a t e d by fibrous septa, a n d t h e final d i a g n o s i s , t h e r e f o r e , w a s t r a u m a t i c ncuron~a.

(:(lmparatively few cases ()f traumatic neuroma have been reported in the literature and, so far as is known, none of these is eolnparable in size with the present case. It is generally agreed '+-' that it is not a true tumor, but an a t t e m p t at repai~', that is, hyperi)lasia of the nerve fibers. Thuma, TM however, describes a m p u t a t i o n neuroma as a small t u m o r which forms at the end of a nerve t r u n k after i n j u r y an(l section o|' the nerve. Ire states thai the swelling is (.ause
Voh,me to

Number I

NEUROGENIC TUMORS OF ORAL CAVITY

s t a t e d by T h o m a to be a n o u t s t a n d i n g f e a t u r e .

33

C a h n 4 r e p o r t s a case oeeu|'-

r i n g on the m a n d i b l e of a n e d e n t u l o u s m i d d l e - a g e d woman. I t was a movable, p a i n f u l n o d u l e a b o u t t h e size of a pea, w h i c h , on o p e r a t i o n , w a s f o u n d to be a t t a c h e d to t h e m e n t a l n e r v e .

T h e h i s t o l o g i e p i c t u r e w a s of n e r v e b u n d l e s

s e p a , ' a t e d b y f i b r o u s s e p t a a n d growing" i n m o r e or less d i s o r d e r l y f a s h i o n . A f u r t h e r ease is r e p o r t e d b y G r e e n . s

I t c o n s i s t e d of a s m a l l m o v a b l e

n o d u l e i n the r e g i o n of t h e r i g h t m e n t a l f o r a m e n i n a 27-year-old w o m a n . H i s t o l o g i c a l l y , the s p e c i m e n c o n s i s t e d of a m y e l i n a t e d n e r v e t r u n k w h i e h cont a i n e d a localized a r e a of e n l a r g e m e n t d u e to i n g r o w t h of fibrous tissue w i t h sepa r a t i o n a n d d i s t o r t i o n of the n e r v e b u n d l e s . ( ) r i n g e r ~ has d e s c r i b e d a n a m p u t a t i o n n e u r o m a w h i c h arose a f t e r ext r a c t i o n of a l o w e r m o l a r . O n o p e r a t i o n , a p e r f o r a t i o n t h r o u g h the b u e e a l p l a t e was f o u n d to be filled w i t h the f i b r o u s mass of the n e u r o m a .

Fig.

l.--Mierophotograph of neuroma showing interlacing b a n d s o f n e r v e f i b e r s in ( ' l o s e r e l a tion to a major nerve trunk. (Hematoxylin and eosin stain. )JIagniflcation, X70.)

()ne of the d i s t i n g u i s h i n g f e a t u r e s b e t w e e n t r a u m a t i c n e u r o m a a n d n e u r i n o m a ( s e h w a n n o m a ) is t h a t no n e r v e fibers are i n v o l v e d i n the l a t t e r . I n s t e a d , the n e r v e fibers r u n a r o u n d the t u m o r . Neurinoma (Neurilemmoma, Schwannoma) (Dr. A. S. Prophet's Case).--A patient, aged 19 years, reported to the Royal Victoria Hospital, on Sept. 15, 1955, complaining of a painful swelling in the labial sulcus in the lower incisor region. The swelling had commenced two weeks previously as a small, hard lump, which had grown rapidly but had not caused pain until the previous day, when an incision had been made in an attempt to drairt the swelling. This had not been successful and the lump was now very sensitive to touch. There was no history of serious illness, and the patient appeared to be in good health.

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MC K A Y

(). S., o. M., a o. 1~ Janumy, b.~5;-

()n i n t r a o r a I examination~ a iirm, sessile swellil~g 1/) inch i~t d i a m e t e r was seen i~ the mi,lline in the labial sulcus. The color of the o v e r l y i n g m u c o s a w a s n o r m a l and the s ~ r f a c e w a s s m o o t h except w h e r e a recent shallow incision had divided it. and revealed w h a t appearecl to be u n d e r l y i n g fibrous tissue. The lower incisor i.eeth were p r e s e n t and firm. and w e r e not t e n d e r on percussion. R a d i o g r a p h s revealed no abnormalit.~ of the te~lh or t~oue. O p e r a t i o n consisted ()f removal of the tlHnor~ w h i c h w a s ell{tapslllatet[ afl,I shelh~(I ,)u! ~deanly. P o s t o p e r a t i v e healing w a s u n e v e n t f u l . The |)iopsy report indicated t h a t the t u m o r was a neurinoma.

r; (

N

Fig. ').----Microphotograph of neurinoma sho~ing- proliferating Schwann cells, forlnillg palisa(les. (Hematoxylin and eosin stain. Mag'riification, XM).)

It is of intert~st to eonlpat,c the clinical findings in these two rare tumors of nerve tissue. Each had a short history---not more than lwo months in either c a s e - - a n d had g~,ow~t to a considerable size in that tim~,. N(@her had been painful until traumatized, the ncuronm dm'ino' mastication and th(~ neurinoma by a t'eccnt incision. Aside fronl these points, however., thor( ~ was little similarity. 3!he etiology was appar(,ntly t r a u m a in the nem'oma, but it was u n k n o w n in the ease of the neurinoma. The diffccenee in age of th(~ patients was extreme, and the shape and color of the tumors were also dissimilar. As already mentioned, the provisional diagnosis in the ease of amputation neu~'oma was fibrom;k altho~gb lhe ~'a.I)id g~'owl.h was a fa(d.o~' a,,,'ainst this. In tim (~asc of m~m'ino,ml, lh~' (lil't:'(,r~mlial (lia~'nosis in(.lu(h,(I :llv(,~,lar abscess, dental cyst, mucous ('yst, and fibcoma. The s]mH, hislm.y lmiut(,d l;o alveolar abscess but, on the oth('r h;m~l, the l(,(d h w(q'~ nol l(,nder to ])~rcussion and vitality Iests gay(, normal results. No a b n o r m a l i t y w~ls ~'evealed

Volume 10 Number 1

N E U R O G E N I C TUMORS OF ORAL CAVITY

35

vadiographically and the incision made the day before showed that the contents were n~t fluid. The provisimml diagnosis, ihercfo,'e, was fibroma. A study of the literatm'e shows disagl'eemenl as to lln, nomenclature and origin ,,f m~rve tumors involvin~ lhe pevineural sh~ath. They have been referred to as neurofibroma, neuroblastoma, nem'ogenie fibroma, schwannoma, neurilemmoma, and neurinoma. Vcroeay ~' advanced the theory that the tissue of nerve tumors, previously thought to be connective tissue, is really neurogenic tissue and arises from the Sehwann cell sheath, which is the inner covering of the peripheral nerve fiber and therefore ectodermal. Penfield ~2 felt that the tumor arose from perineural mesodermal connective tissue, the cells therefore being eonneetive tissue cells. Stout, 16 Murray and Stout, ~~ and Bernier and Ash ~ also discuss the origin of the cells forming neurogenie tumors. Bernier gives two types: (1) neurofibroma, arising from either endoneurium or perineurium, often containing neuroepithelial elements and, (2) schwannoma, purely neuro-epithelial elements. Willis 2~ states that neurinoma literally means " n e r v e fiber t u m o r , " and is therefore inappropriate. He uses the term neurilemmoma or schwannoma. In common with other authors (for example, Stones"~), he describes two types of tumor on microscopic examination: (1) faseiculated or type A tumor, which has an orderly arrangement of parallel cells with intercellular fibers forming interwoven bundles, this arrangement giving a distinctive palisaded pat t ern; (2) reticular or type B tumor, which contains a disorderly, loose meshwork of cells of various shapes with intercellular vacuoles. There are no parallel bundles. The present tumor is of type A. 1'his tumor is found fairly commonly in other parts of the body, but is rare in the mouth. Bruce a mentions that 130 cases of neurilemmoma were recorded at Columbia University from 1926 to 1944. Of these, only two involved the oral cavity and pharynx. Other cases have been recorded by several authors, including Goldman 7 and more recently by Sehroff,~3 Christiansen and Bradley, 5 Baetz and Shackelford, ~ Thoma, 's Spilka, ~ Couch, 6 and Hunter2 Of the tumors so recorded, six involve the mandible in the region of the inferior dental foramen or the ascending ramus, four involve the facial tissues, two involve the palate, and one each involves the pharynx, floor of the mouth, and mental foramen region.

Summary Two rare tumors of the nerve tissues (amputation neuroma and neurinoma) occurring in the oral cavity are described, and the differential diagnosis is discussed. The literature is reviewed. My t h a n k s are due Professor P. J. Stoy and Dr. A. S. Prophet, Royal Victoria Hospital. Belfast, N o r t h e r n Ireland, for permission to report these eases, and Dr. F. MeKeown of the I n s t i t u t e of Pathology, Royal V i e t o r i a Hospital, for her help.

References 1. Baetz, :P. O., and Shackelford, J.: Sehwannoma of I n f e r i o r Alveolar N e r v e ; Report of Case, J. Oral Surg. 9: 331-333, 1951. 2. Bernier, J. L., and Ash, J . E . : Atlas of Oral and Dental Pathology, ed. 4, Washington, D. C., 1948, A m e r i c a n Registry of Pathology.

'~6

.UC KAY

~) S.. O.M.,a o. P. .Ianuat~. Iel+;7

:~. ]',l'u('e, K. W.: ~ o l i i a r y N e u r o f i ] ) r o m a (Neurih~mmonta, Sehwannomal of the (h'al ('tt'r ()ItAL ~UtC(;., OI/AI. ~[EI).~ & ()I~AI~ PATII. 7:1150-1159, 11i54, I. ('ahn, I, ]C.: Traum+~tie ( A m | m t a t i . n ) NelJvoma. Am. d. ()rlhodonti('s and ()val NutS. 25: IDO, 1939. 5. (*hristiatisett, (~. \V., at,i }~ra,tle>..I. 1,.: .Ne~troma of l'~!tate; 1Cevort ut' ( morea), Am..[. Cancer 24: 751. 1935. 17. Thoma, K. 1-[.: ()ral Pathology, ed. 3, St. Louis, 195(1, The C. V. Mosby (~ompa,Jy. IS. Thoma, K. F[.: Oral Surgery, ed. 2, London, 1952, Henry Kimpton. l!t. Verocay, 3o.~e: Zur K e n n t n i s der Neurofibrome, Beitr. path. Anat. 48: l, 1!110. 20. Willis. R. A.: Pathology of TtnlJours, ],omlon~ l.q4,~, Butterworth & (~o., lad.