Atypical multilocular follicular cyst of the mandible

Operative ATYPICAL ( IOI,ONEI, Ii~~)

Oral

MULTILOCULAR FREDERICK CAPTAIX

Fi.

FOLLICULAR

RICHARDSON,

BERNARD

Surgery

M.

* DENTAL COHEN,

**

CYST OF THE CORPS,

DENTAI,

ITNITED Cows.

STATES LILvITe~

MANDIBLE 111~

FOKC~:.

STATES

AIR FORCE RESERVR HIEE’ COMPLAINT.--On Sept. 19, 1951, the patient reported to the dental with the complaint of a progressively enlarging swelling, of about one year’s duration, in the body of the left side of the mandible. History of Chief Complaint and Mode of Onset.-Tn September, 1948, the No reason for remoynl was lower second and third molars were extracted. stated or known. In December, 1949, the upper third molar was extract,ed and a “ growt,h” was removed from the region of the lower left second and t,hird molars. No other symptoms were noted. Clinical Examination.-The patient was a W-year-old Negro in no obvious distress. Externally, a large nontender mass was risible in the left body of the mandible. Palpation of this region elicited crepitus nntl some feeling of There were no nodes palpable in fluid. The mass was fixed to the mandible. t,he neck. Slight trismus was noted. Intraorally, in the region of the lower left molars, there was an area of slight inflammation. The second and third molars were missing. There was a nodular mass (approximately 15 mtn. in diameter) at the junction of the body and ascending ramus. I’alpation in this area elicitecl a fluidlike COIIsistency with some crepitus. This region was slightly tender. The floor of the mouth on the left side was raised and a mass could be palpajted in the body of the mandible as well as in the buccal fold. The tongue and ~nuco14s membrane of the remainder of the mouth were ~lornl>11. There was no eyitlence of disease of the teeth or other structures wit,hin the mouth. and no evidence of paresthesia of the mandibular division of the fift,h nerve. X-ray Examination.-On Sept,. 20, 1951, aI1 esaminat,ion of the lcbft side of the mandible was made in multiple projections. I\ large I~lultilOCulilr cystlike lesion was revealed, extending from the base of the neck of the nondyle to the midportion of the body. The lesion extentle(1 well up into the coronoitl process and sigmoid notch, with considerable expansion of the bone. Very little of the bony cortex remained immediately distal to the angle. There was no evidence of direct invasion of the bone by the lesion and there was no evidence of calcification or tooth formation in association with it.

C clinic

with

*United **Chief, United

States Air Force Hospital. Scott Oral Surgery Section, Morrisonin States Air Force.

Air Force Base, Ill. City Hospital, New York

City:

on active

duty

E‘ig J.

1 and

2.-Radiographic

plates bone

taken loss and

on admission, size of the

Sept. lesion.

20,

1951,

showing

extensiw

Provisional Diagnosis.--l+onl ing provisional

the history diagnoses were made :

and the examination

the follou-

1. Ameloblas~oma. 2. I~ollicular cyst. :I. Sarcoma.

Physical Examination.-Kmninat no colltraintlicatiorl

Fip.

:3.--11raxving

sbowin

ion was essentially- negative. Thwc was

for surgery.

P extent

Medial of bony cleatrwtion. ing through the defect.

surfare

of opposite

sirle

shmr-

Progress in the Hospital.-The patient was adnlittctl to the hospital on The lalmratory Sept. 20. 1.951, for biopsy and removal of the cystic lesioll. report, on Sept. 20, 1951, was essentially negative r>sccpt for an elevated white count of 1.5,900. Biopsy.-It was decided that a biopsy should be done to determine the exact nature of the tumor. On Sept. 21, 1951, under local block anesthesia. a wedge-shaped section was removetl from the intraoral lesion. This included a portion of the normal mucosa. T;‘iye cubic centimeters of yellowish ClCill’

flnitl was aspirated. The ittckic,tt \C;IS ~~l~~cl wit tt So. 000 silk anal 1II(~ ttt;ttot*ial preserretl for pnthologiv es;ttltitt;ttiott itr ,> IWI’ c*catltt’ortr~alin.

Pathologist’s Report.-- ‘l’hv cliagttosis was sittll,It~ (*j,st. GROSSI~~~~~ti?t~/ti~~?~.--JI;It~~t~iiI1 cotrsistrtl nteasnring 2 cm. ill tli;ltttCt~~l’. II b\ ilS il tltitt hy a soft metnltratious strnvtltrc~.

0I’ H;lt

ill1 pl;ltc>

IJ-~P

irt*vgtl;tr (*O\~?t’Ctl

rtttktto\vtt scvtiolt Ott

OII(

ot’

I)OIltJ

Slll*fil(a(‘

~lli~~~~,sco~ii~Escr?lliltnti/,)r.~--S~~~tiott iltror~gh the, sl,tdtttet~ snl)rttittrtl ITFl (@vSt With il fihTY~Us $Villl linetl illt~‘I~~lilll~ I,y sitttplr ~~OI~lttl?till~cJl,ithelinttt. l’he pathologist’s rel)ol’t W;IS Itot thought to 1,~ c~ottc~lnsive I)t~(~ilttse ot’ the small specimen provided 1:~ I:iopsy. It was felt thitt this rttipht Iw it cystic ~trttelol~l,7stotti~. A larger sl)cvittteti c~onltl be proritlrtl lt])olt opetxtiott itlltl il tttore c~onclnsive cliagitosis tttxtlr. If on olter;tti(,ll it was fount1 th:tt 1110Icsiott estelltletl further that1 the s-I*ity itltlicntetl, it wsectiolt >ttttl retttov:t t of the entire ttiass in hlocli sectiolt woaltl I)e Itecess:try. ()It the other h;ttt(l, if il(lrcltlitte ~NIIC remained after retttoval of ~ltc tuttror it l)ot~c anal (*hi], Kr:tFt ~0~1tl IW ~setl to fill the residual (Iel’ec*t ittl(l to hastelr I)otly regeneration. \-P,71Cd

Operation-The

I)reoprtxti\c orders were tttorl)hine sult’;tte. :q; gr.. itrttl sulfate, )(;(io gr., with 1,rov:tinc~ pe~ticillin. :XX),OOC) units tlilil~. The patient was nnesthetizecl wit,h l’entothd Sotlinm an(l clur~tre ;ttttl etrtlotracheal nitrous oxide ant1 os~~gett. ‘l’hc site of the iliac crest atttl thcl I~otly 01’ I Itc tttatidihlr wet-c pt+eparcd with Scptisol solittiott a11t1 iinct urc of Jlctapltc~tt. ‘I’he patient ~721stlraltc’tl SO i11;11ihe orthopedist vonltl rcrttove it portion OEthe iliilc crest itt the same tittle its the ttt;ttttlil)nl;tr lesion \V~S being t~~l)~~rcl. r\tl incision wits tttatle ill,c,ttt 5 tttttt. I:c~low the Iowc~r hortler OF the I,otly ot tllc ttlitfltlil~le extending frottt llte iltl~lt~ fOl’~‘ilI’(~ to tllr ~~rettiotar regiott, Ihis ittcisiott hcittg ;Il)l,r~osintatcl~. :I itlrhes long. ‘I’hv tlissectiori wits vi1rried through the suprrfici;t t I’:tsci;t ittttl the I)lee(ling \~rsscl~ were clitrtll)etl ~III~ lictl. The ltl:ltystrt:l tttt~sclr~ L\C;Stli\-itle(l, thus exposittp the f:tc~iitl itt*tet’v ittt0 vein. l’ltesc vessels wrist’ t icvl ;tlitl sevt~rt~tl. The ttiassctrr ttinsclc was tlissectccl 111)~;1rtl frottt the Itrrioslc~ut~t vo\.eriltg the ttttttor ttt:tss itlttl rett*i~(*t(~tl. The periostentti wits itic4scbcl itt thv si1111etlirection ;~ntl elevatetl from 1lit, caystic \Wll. Al thitt white ttteItIl~~.ittt( \zhicah votttaitted Ittatty lriahle sections ol’ l~,:l(~ \vits c~sltosetl. Thcl itttieriot. rs!c~~tsiorr of thtl tttitss ilt the t)otly 01’ the tltiltttliI)le \V;tll illIt tIlta l)ll(~c~~ll rcpion fWetl. ‘I’hC’ I)U(‘(‘il I \ViIS S(31)ill’Zlt~tl fl*Oltl tll? 1:01l? The ant1 lingual plales were crotletl with loss of the elitire li~lgUil1 l)t:itC. The lesion IVits f’ot~~ttl to t~xtrntl ascending rillllUS was c~sposctl posieriorty. At,ot~t, half ot’ 1hta Iilter;tI npwarcl irtto the contlgla r 11~1i it lltl si;rtrtoitl 1lOtCh. Ahout two-thirds of’ the width of surface of the ramus hit.tl l~rn tlestroyetl. ptrrygoid the inte1vxa.l plate was also c~rotletl. The lmlging 01 the itlterttitl muscle could be seen upott rettto\.itl ol’ the mrtlinl wall of the cyst. The sigm&l notch and the tip of tltc vorortoid pt~~cess were it Iso involved. The mentlnxne lining of the cyst was ~~stremrly thiu g?nd firmly ztttachecl. So lt~tt) i\tl*Opirlc

Fig.

I.-Drawing of operative graft; 2, internal pterygoid

proce(lurc showing muscle; 3. masseter

placement of bone musclr ; I. inferior

graft in defect. rlental alveolar

1, Bony nervcx.

The orthopedic team removed a portion of the iliac cl-est by means 01‘ iltt incision made over the iliac crest of al)otIt thr-ee inches in length. The super*ficial fascia was incised and the skin I)lecding cont~~olletl with clamps i\lltl ties. The peCstemo was nest in&et1 and the ct.est of the ilium exposed. X settion of the crest of ahout 2 by 6 cm. was r-emovetl. iis well as an are;1 of callcellous bone in the form ol’ chips. Enough material was taken to fill the cays1 The peGosteum was closet1 with No. 000 grit, ant1 the sul)eAei:\l I’i\s(*iil cavity. and skin were closed in layc~s. The bone gra.ft was I)laced in the cavity ant1 the remainder- of the defect filled with cancellous hone chips. The periosteum and platysma mnsc~le were closed with X\‘o. 000 gut. The superficial fasci;] was closed wit,h No. 000 gut, and the skin closed with No. 000 silk.

A

lZlrgfl

operation.

~~l’~SSlll’(’

I);lll(lilc((’

\fJ?ilS

[)l;lCt’tl

()\‘(‘I’

I)~1111

ClftC’l’tlti\.C’

SilC’S

Of

lh(

Thr Ijatierrt t~lr~t.r~(~l to th(b \vittatl iti ~ILOCIc~orr(lilic~tt.

Postoperative Course.-mI’ostopei~ativt~orders WI’C : 13~111cr~o1, 150 trig. ever)’ ; four hours if nectletl ; 1,000 (+.(A.5 per etirrf glucoses an(I saline irltl3~--erlonsly chloromycetirr, i‘)50 mg. evrf*p [out* hours; ant1 pr*o(~;rirrr I)c’nic4llirr. :100,000 units

daily. The patient, was returned to his ward from the operating room at 5 I’..v. o11 Sept. 26, 1951. The blood pressure was 124/80; pulse, 110: respirxtiorr, 20. at 5 r.nr. The patient reacted at 5 :25. There mas sorrrc bleeding l’rorrr the gr’a fl site on the iliac crest and the dressing was reenforced. At 10 P.M. the tempererture was 98O; pulse, 102; respiration was 20. nt, was given to cont.rol pain in the hip region. One hour later pro,jcc+ile vomiting of fluid and blood occurred. The patient complained of nausea which was c’ontrolled with 1 CC. Prost,igmine with l/loo gr. of atropine sulfate. The second postoperative day was slightly- better. Irrtcrmasillary mires were placed and the mandible wired into occlusion. Slight tlificulty in swirllowing was noted but, became less annoying as the day W’OI’Pon. ‘l’hc> ostrrn:ll dressing was changed. The third, fourth, ant1 fifth post,operati\-c tla\;s were uircventful escclpt for the pain in the region of the left. iliac crest which was c~ont~ollrd with Demerol. On the sixth day the dressing and sutures were r~rrrro~etl front the skill. The wound was slightly edematous lwt with no evidence of discharge. ‘I%~~ postoperative s-rays were takerr ant1 the rctmrt was as statetl helow :

1230.I 50rllg.of rh~ed

X-ray Report (Oct. 2. 1951 ).--ftrcIsarnin;rtiou of the left sitlc II~’ the malldible showed evidence of the rerrro\-al of cystic loculatiorrs with th(A irrser*tiorl ‘I’hert> \V;IS 110 evitlerrcc of I’r’irct 1rr.g’. ‘l’h(s trtat tI of a 2 by 6 cm. bone graft. were wired iii occlusion (Ii‘igs. .i alit1 6). Pathologist’s Report

((ht.

:I, 1951).

GYISS E~aminatiolz.--RIatc~ial consisted of several I’ragmerrts oI’ flat,tcncd Suhrtrittetl se[t:rsheets of hone, measuring al)l)r.osirrrately 1 cm. iir diartretrr. rately and partially attached were sections of white softened flattritcvl tissue. all of which appeared to t)r I)ortions of the cayst. ~~11. ‘1% irrIer.trirl surf;~c*t~ of this cyst was smooth escrl)t t’o~ one area where a small white rtotlule rrteasuring 3 mm. in dianietcr~ WilS sw~i. ( ‘Ut se&ion ttl IYjUgh the Will I l’t’\‘~ill~‘~l gray-tan somewhat nodular etlrmatous fibrous tissue. : Ameloblastoma, cystic. This tumor showed rrorre of t Ire histocharacteristics of malignancy. I lowrrcr, Ihew tumors have ir dcc+lrd tendency to recur if not, c*omplrtely removed. Diaposis

logic

Microscopic Ezuminatioll.-~-Se~tiorr through the cayst wall revealed it to be made up primarily of dense fil)rous tissue infiltrated Tvith a considerable number of plasma cells and lymphocytes. The cyst wall was lined with stratified squamolls epithelium which was quite thick in some areas. The section I hrough

.\‘I’\-I’I(1.\1.

51 ~~1,‘1’11,0(‘1~1..\1~

b’OI.l.l(‘l‘l..\l~

Fig.

6

(‘\‘S’I’

()I<’ Al \2131111,1~:

7

The area, irrigated wit,h a solution c*onlaining :100,000 nIlits of: penicillill. rigations wer(~ cotrtinut~tl c.cltlcnl.rc~llt,ly wit,h l)enicillill ;IHC~oral ~hlot,olll-rrtili.

ir-

X-ray Report (Oct. 9. 1’)51).--1’osteroautr~io~ examination of the ma11 dible failed to reveal evidence of fracture through the operative defect on the left. The graft appeared to be in satisfacbory position. There was JIO s-ray evidence of destructive bone infectZion although low-grade osteoltkyelitis might not visualize earlier than two weeks following surgery.

Report of Culture (Oct. 11, 7.951).--C iram-positive cocci in chains and seen singularly in direct smear ; beta hemolytic streptococci isolated on cdulturc media; occasional pneumococci ; sensitivity tests intlicated that the organisms were sensitive to penicillin. day;) nJJd The irrigations were continued until October ‘LT, (twenty-eighth ta.ken. a. new culture was Report of Culture (Oct. 25, 1951) .-Small gram-negative rods and moderate amounts of pus cells were seen in direct smear. ()rganisms tvfv*e culturally and morphologically characteristic of members of the coliform group (C’oli aeroqenes) ; isolated on differential and selective media (par*e culture). The penicillin irrigations were changed to tlihydrost.reptomycil~ t,wice daily; 0.5 Gm. of t.he drug was given hy mouth four times daily. Thr penicillin therapy was discontinued. Four days later the patient complained oi’ dizziness and tinnitus and the dihydrostreptonryvcin was reduced to 0.25 (:~a, four times daily. The intraoral drainage had markedly tlecreased 1)~ NOY~JIIbcr 1, the thirty-fifth postoperative day. X-ray Rehort (Eov. 9, 1951).-Reesaminat,ion of the left si4e of the mandible revealed evidence of remora1 of the loculated tumor. There was no evidence of fracture involving the very thin remaining bone cortex. Therr was a bone graft in the large defect which appeared to be uniting ant1 there was no evidence of gross osteomyelitic infection. The irrigations were continued until the forty-second day when a new smear was taken. Report of Culture (Nov. 8, 1951) .-Alpha hetnolytic st,reptococci were isolated on cultnre media; there were occasional gram-negative diplocoeci. Neisserifr. cntnrrhdis was also found. The dihydrostreptomycin was discontinued and the patient placed 011 Ou Noveml)er 20 all suppuration had ceased chloromycetin and penicillin. (fifty-fifth day). Blood and urine were found t,o be within normal lim& X-ray Report (Nov. 21, 19,‘,1).-Reexami~lntioll of the left sicle of the mandible showed no marked change since the previous examination. The thitt cortex remained intact, there was no evidence of active hone infretion. nnrl thf~ The in I’erior I)ortion of the bone graft remained in its previous relationship. bone graft showed moderate rarefaction as commend with I)rerious erarninaThis rarefaction may 1~ a transitions ; the superior end appeared healthy. tory change preceding firm l~ny union. l,ut sllclultl lw followc~tl W1~cfully.

illl-

.-ra re\

itt

Fig. 9

an< 1 1 O.-Postopemtive

views,

10.

March 5, 1951, of bony graft.

Showing

~lrganisakion

and

<‘c

don

Pathologist’s Report (SOY. 26, I!)51 ; ,Ir’ltl(‘~l I~‘Ol’CeSIttstitute 01’ I’iltht~logy,)--It. is true the ~oerttgettogt,al~hi~ a~~~wat~~ttw is highly suggestive 01’ n multiloculer disease, but from the tissue ;tlonc we cannot tttake it positive be ;tsSUttlecl, Ott thtl basis of the diagnosis of smeloltlastoma. It ('ill1 olrly residual fragments of epitheliurn in the cyst wall, thitt the lesion is one of tht, rather rirye multilocular follicular cysts. These should not I)r W~I t’nscvl with the atttrloblastomas which ttttty t+rl)tvsertt n vysticd t,cl:tvtiott t.athet. thittt vystiv cleprneratioti in a solid tumor. Zhhg7tosis: E’ollicul;tr cyst, n~aritliltlr. The patient was followetl >It the clinic tlaily hut, no further suJtl)urxtiott occurretl. OIL T)ev. 19, 1951! Ihe patient wits tiischat*gcci fat‘ ;I three-week period ;rntl upon return showed IIO evidence of tltxinnge. The skin over thv were tiiketr ;IJ)J)t’osittli~teJ~ r\rt’) area was soft ant1 not7nal in feeling. S-pays two weeks: ;ttttl showetl it progressive healing antI orgatrizwtiott of the ct+ttft. X-ray Report (I)ec. 7, 1951) .--Beesarnirri~tiotr of the left sitle of the martdible reveuletl evidence of slowly increasing bone Ittduc%ioti. The gra Ft remained viable in its upper portion. but, the mid ant1 lower l)ortions were showing evidence of absoqttion and in some ;treils there w;ts suggedve evitlrnce 0I’ begirinittg disilrtegt~iltioI1. There was tto e\-itlerrcr of sequesttxticltt. (‘onclusion : The healing il]~~)~ilf*~tl to IIf? progressing satisfac.tot,il?l, atltl the dttgree of grii i’t raref’adictn w’i~s not nec*essarGl\- c‘\-itlrrtec of sc~riorts ostt~omyelitis.

cut’the rttitrrtlil)le X-ray Report (,Jan. 7, I~~~).--Kee~arnirrilfiott itl)prt~ei:rl)le change since the last es;lrttination (I~‘igs. 9 ittltl IO).

sholvocl no

Conclusion The first diagnosis of simple cyst, type unknown, m’its made on ;I srtritll biopsy specimen with inadequate nrat~erial for detittite pathologic maiysis. The necessity for a final Itiopsy report is clearly ittclicatetl by a secottd tli;tgnosis of ameloblastoma. This was f~llowetl by a third diagnosis of ft)llic+lrI;tt’ cyst of a multilocu1a.r type gi\:etr by the Armed I’orces Instit.ute of I’athology. A corrserva.tivr approach WLS useti in the treatment of it l:rt+gr t*;ttiioIu~t~rri The postoperative Itone healing \vtts destructive lesion of the mandible. hastened 1)~ the itrtl)larrtirtg of a bone graft and ~at~ellous chips. It is also interesting to note that the bone graft was not esl’oliatetl in the presence of oral eorrtitrttirtatiott. Antibiotics were chosen 1)~ culture of the suppurativc material at, W?VerilI different periods. Sf?\‘et’SI C~t’ll~S \Vt’t’t? USf?tI RS the OrilJ flor’it changed.