A tuberculous granuloma of the oral mucosa and cervical lymph nodes

A TUBERCULOUS CERVICAL

GRANULOMA

LYMPH

OF THE

ORAL MUCOSA

AND

NODES

Report of a Case J. A. Gardner, D.D.S.,”

and R. J. llanft,

B.S., D.D.S.,w+ Pittsbwgh,

T

Pa.

T7BRRCT7LOSIB belongs to a group of diseases referred to as infectious granulomas, the term “granulomas” referring to the granulation-like appearance The group includes of the tissue reaction which characterizes these infections. syphilis, tuberculosis, sarcoidosis, leprosy, actinomycosis, and other mycotic and funguslike infections. Mthough the literature describes many definite and specific microscopic characteristics for each of these diseases, their clinical variations arc many and are multiplied even more by the presence of secondary infection. The lesion of tuberculosis is the result of an interplay of the forces of destruction and repair, plus the additional factors of the host’s sensitivity to the organism and the virulence of the organism itself. The tendency to repair, however, is commonly greater than t.he destructive process. The lesion can be seen in several stages of development. l’olymorphonuclcar leukocytes are the first ~11s to arrive in response to the tubercle bacilli. They arc actually phagocytic but arc unable to damage the organism they engulf. Within twenty-four hours the polymorphs are replaced by macrophagcs and monocytes. This is the body’s chief response to the bacillus; however, it is a response to the organism’s lipoid coating rather than to the bacillus itself. The monocytes engulf the polymorphonuclear leukocytes and their contained organisms, and a gradual dispersion of the lipoid material takes place within the cytoplasm of the monocytes, transforming them into so-called cpithelioid cells. Giant cells arc formed by th(? fusion of a number of cpithelioid cells, but, only in the presence of necrosis. They attain a large size and contain peripherally arranged nuclei or clumps of nuclei at one or both poles1 Involvement of the mucosa of the oral cavity or the cervical lymph nodes Thr! is usually found in patients suffering from active pulmonary tuberculosis. organisms are carried in droplets of sputum into the oral cavity from the trachcobronchial tree. There have been reported. howevrr, cases of localized tuherculous From the Department of Oral *Chief of the Department of Oral **Resident in Oral Surgery.

Surgery, Surgery. 406

Western

Pennsylvania

Hospital.

cervical adenitis2 without pulmonary disease. There is much speculation as to the possible portal of entry in such cases. The following is a case report of a localized tubcrculous granuloma of the oral mucosa and cervical lymph nodes without evidence of pulmonary disease. The additional factors of long-standing secondary infection and arteriovascular lues further complicated the differential diagnosis in this case. CASE REPORT

I&tory of Preseat Illness.-A 39-year-old Negro woman was seen in the oral surgery clinic of Western Pennsylvania Hospital on Jan. 7, 1959, with the chief complaint of a Two years earlier, following extraction of “painfully ulcerated mouth and swollen jaw.” all her remaining teeth, she noticed an enlargement of the submental nodes. These nodes had remained enlarged to this date. She stated that three months prior to her appearance at this clinic, a penny-sized ulcer developed in the left mandibular buccal fold. This had slowly progressed to ita present size. She also stated that six months previously she began experiencing fainting spells and that they had seemed to occur more frequently since the onset of her oral complaint. She had not experienced any of these so-called “spells” for the last three days.

Fig. I.--Ulcerated.

granular

lesion running

along the buccal surface ridge.

of the mandibular

alveolar

Clinical examination revealed an ulcerated, granulating, nonfungating lesion in the left mandibular buccal fold. It involved an arca extending from the third molar region anteriorly to the left lateral incisor. There was considerable evidence of secondary inf e&ion ; its borders were slightly indurated, and it appeared attached to the underlying bone at several points (Fig. 1). The patient was completely edentulous and had never worn a full denture prosthesis. The tonsillar area and oropharynx showed no evidence of ulceration but were slightly injected. The tongue and the remaining oral mucosa were within normal limits. The patient did not complain of trismus or dysphagia. There was

111)loss of sensation aloug t.hc distribution of t.hc inferior alvcular ncrvc 01’ its m~~n~:~I I~ancbet;, but the pat&it. dill rxhibit :L slight Jegrcv nC motor wcakuess in the nrc:, rul,~&cri I3.v Lhc mandibular brant!h of the Envial rwrve. ‘l’hc submental nodes ww tn:irkedly VIII:~rgc~l, noutendcr, and oJ’ a rubburr wwsisteuc~. Therms\vas also a similarly l~largrd IIOIIV I.hat could be palpated in the left submaxillary rogiorl. The assaciatl.tecl soft I issucl (,I’ 1111’ :Irea was swollen and tender (Fig. 21. lAtera obliqur and al~l~~roposterior films ul 111~mnudibltr, as well :IC; iuiraor:\1 tiln~, \vtLre ordered at this t.imc. Wet, rcaadings gave no iodie:uion of bouy in\-olvemcntt. I\‘\‘ith 2 c.c. of Sylocainc l~~drochl~~ri~lo,a block of the left iul’c%rior alveolar I~C’~YI’\vns ol~t:ii~~wl. An additional 0.5 CA:. was iutiltrated in the vieinit.y of thn long bucral ncrvc’. .A wrdgt! III tissue was excised Gth a So. 15 bladr; this included part of the lesion, its indurntcd lwtltv~ and adjaecnt normal-appearing tissue. The wound WIS caloscd with three 0000 black sill; sutures. The specimen was submitted in 10 p” crnt formalin for histologic stully. Thr patient was thcu admittcd to the olnl surgery sorvicc! o-f this hospital.

Fig.

2.-Clinical

enlargement

of the submmtal

and submaxillary

n8)dcs.

Medical ZIGtory.-Because of some contradictions in the patient’s story and a lack of obtainable past medical records, there was some difficulty in obtaining a reliable history. The patient did remember several episodes of urinary tract infection two years earlier, “shots” she received about one year hefore for “ spasms,” and the fact that she had rceeived treatment for hypertension from a local physician and was on a salt-free diet. She revealed no history of product.ivc cough, dyspnca, angina, orthopnea, night sweats, inercascd thirst, frequency of urination, or swelling of the ankles. She did complain of a steady weight loss, frequent daily headaches, dizziness, and difficulty with vision ; she said that her eyea ran constantIy. There was no history of allergy, asthma, or hay fever. She had never experienced any episodes of scvcre prolonged bleeding. She denied any antiluetic therapy or a history of positive serology. Physical Esaminalion.--Physical examination revealed a fairly obese, confused, Negro woman who walked with a shuffling gait. Examination of her eyes revealed a slight ptosis There WRR a small, flattened, tanuish ncdul~~ of the left lid, with an a.ssociated conjunctivitis.

volurr~e

TURERCTJLOUS

I-t

Number I

GRANTJLOM:~

-IO!)

at the junction of the sclera and the iris. Other neurologic findings included indistinct optic disc margins and a slight left central facial nerve weakness. Other findings included a blood pressure of 130/95; pulse, 88; respirations, 20; and oral temperature, 99.20 F. The heart was slightly enlarged but within normal limits. The lungs and chest wore clear to percussion No palpable abdominal masses WIV found. There were no ot.her and auscultation. significant physical findings. Laboratory Findings and Diagsovtic Procctlu~s.-The results of tests ordered on admission were as follows: hemoglobin, 12.2 Gm.; hcmatocrit, 39 per cent; white blood ccllls, 7,000. The differential count was neutrophils 62 per cent, lymphocytes, 30 per cent, mouocytes 3 per cent, eosinophils 1 per cent, and 4 band cells were seen. Bleeding time, coagulation time, serum elect.rolytcs, nonprotein nitrogen, blooll sugar, alkaline phosphatasca, and urinalysis were all within normal limits. A dark-field smear taken from the oral lesion failed to demonstratc the presence of Twpowema paZZ,idum. Acid-fast examination of the sputum was negative. VIIRL studies exhibited a A plus reaction aud a positive Rolmer test. The serum electrophoresis revefilcd a marked inr.rcasc in beta and gamma globulins. The rc&port stated that this response could be due to active syphilis, sarcoidosis, or liver disease, but probably not to active tuberculosis. The clectroenccphalogrrtm~alogram revealed no significrtnt abnormalities. Films of the skull, chest, n.nd I~mcs of the hands and feet likelrise revralcd no significant abnormalities.

Pig.

X-Active

tuberculons nf

clustrrs

gmmuloma

of

epithelioid cells.

mwosa. Note fore&n-body giant cells an~n~: (Magniflcntion, xlri0 ; t’etluced !6.)

oral

A biopsy report of tho tissue scllt fuml the ~JiltllOlO~~ dapWtrtlent a.Iw*ribtvl :I specimen sulunittod in formaliu cousisting of a wc~lgo-shaped irreysular granular fragment of I issue rucasuririg 0.X mi. in it.s grcut.M diamc+er. The microscopic report tlrscribed clusters of cpitholioitl cells lhat were surrounded by a chrouic iuflrtmmatory type of clsudatc. Therr was no uvitleuce ol’ (aas(Miug uclc:ro&, I~ut t.hc:re were nuuI(~rous gSi:tut ~11s of tllca fuwiplIJody or lA:~nghn~~xtype iu 111cclusters OP epilhalioid cells. Sc??tions submitted for prriotlic :(.l*irl-S(:hiIT am1 acid-fast stains wore uctgativib. wlrs diagnosis itl~lienlc~(l I)? thcsc, fimliriys wits active grat~ul~~tru~of thr oral nnu~or(i~(Vig. :(,I. (&rirul CWWW.--On .Jauunry 1.1 the pal ic>ut W:LSs~h~~l111~1 I’or u rcl~iopsy of the oral Iosion. JIonever, she experienced a grand mu1 typo of scieurc whicah lasted for two or three

410

GARDNER

AND

OS.. O.M. & O.P. April. 1961

HANFT

minut.es. She w&8 placed on phenobarbital and Dilantin therapy, and convulsion precautions were taken. It ‘iv&9 thought wiser to delay the biopsy procedure until the patient’s present condition had become more stabilized. The patient was placed on a combined oral surgery, ueurosurgical, and medical service. On January 12 the patieut experienced two abortive seizures, at which time additional phenobarbital and Dilantin WCI’C administered. On Jauuary 14 the pat.icnt was given a liquid breakfast but was allowed to have nothing by mouth after 9 A.M. At noon she was given 50 mg. of Phenergan hydrochloride intramuscularly and a soapsuds enema. At I P.M. she received hypodermic injections of 75 She was placed on call to the mg. of Demerol hydrochloride and r/roe gr. of atropine sulfate. operating room for 2 P.M. Under Surital sodium and nitrous oxide and oxygen anesthesia, with endotracheal anesthesia administered via an oral route, the patient was prepared and draped in an appropriate manner. The oropharynx was packed, and two wedges of tissue were removed. Out specimen was sent for a repeat histologic study and periodic acid-Sehiff and acid-fast stains. Tho other section was sent for culture, smear, aud guinea pig inoculation. The patient was then turned carefully onto her left side, redraped, and prepared for a spinal tap. The fluid pressure measured 380 mm. of ccrcbrospinal fluid. A sample was seut for dark-field examination, s~ology, protein, and colloidal gold cure. The patient was then 811~ was very gently romovcd from placed again in a supine position and carefully evaluated. the operating table and sent to the recovery room, whcrc estubation was eventually done by the anesthesiologist. The patient tolerated the procedure very wall. She was placed on tilt% usual post-spinal tap precautions. On January 15 the patient was placed on antiluetic therapy of 3,000,OOO units of penicillin daily for ten days. On January 19 spinal fluid serology and acid-fast studies were found to be negative, and all other studies were within normal limits. An angiogram revealed a slight shift of the anterior cerebral vessels to the left. The high spinal fluid pressure made it inadvisable to attempt a pneumoencephalogram. The neurosurgical noto suggested the possibility of an expanding lesion in the right frontal lobe area. On January 21 a purified protein derivative gave,1 a negative tuberculin reaction. On January 23 a second purified protein derivative gave a positive tuberculin reaet.iorl which produced a quarter-sized pruritic whcal. On January 24 a vcntriculogram revealed not experienced any seizures since January l2.

no significant

abnormalities.

The paticut

had

Ope?Mive Procedzlre.-On January 27 the patient was taken to the operating room for excision of the submental node. The submaxillary node paIpatcd on admission had regressed under the antibiotic therapy, as had the associated secondary infection. The submental nodo failed to respond to treatment.. The patient was prepared and draped in an appropriate manner. After anesthesia had been obtained by the infiltration of I per cent procaine hydrochloride with 1 :lOO,OOO epinephrinc, a transverse incision was made over t.hc node. Several no&s wero then s&atc~l Hemostasis was sccurrd with multiple ties, and the and isolat.ed by blunt dissection and freed. wound was closed with interrupted 00000 chromic catgut sutures suhcuticularly, followed by 000000 interrupted black silk sutures to dose the skin. A small pressure dressing was applied and the patient was returned to her ward. The specimen was sulnnitted in two parts-mm placed iu IO per cent formalin for histologic study and periodic acid-Schiff and *acid-fast stains and the other sent for culture and guinea, pig inoculation. Summary of Pathology and Bacteriologic Smdic S.---Roth the initial biopsy on admission and the repeat biopsy of the mucosal lesion were found negative for fungi and acid-fast The only positive bacteriologic findings were the presence of penicillin-sensitive organisms. The histologic pictures were Streptococcus viridarus and hemolytic Stapiyrlocoooue albus.

Volume 14 Number 4

TUBERCULOUS

GRANULOMA

411

identical, both indicating an active granulomatous type of infection. The immediate smears did not reveal any fungi, spirochetes, or acid-fast organisms, and further culture and guinea pig inoculation were instituted. The pathology report on the submental nodes removed on January 27 was as follows: “ The specimen consists of two closely adherent, discrete, hemorrhagic, thinly oncapsulated lymph nodes. Microscopic examiuation reveals discrete and confluent nodules of cpithclioid cells which have obliterated the normal architecture of the node. These areas lack caseation Fig. 4.

Fig. Altrating Fig. cell) with

Fig. 5. nodules inI.-Microscopic section from submental node show6 li hter epithelioid the normal architecture of the node. (Maa-niflcation, X d 0: reduced $6 1 L-High-power photomicrograph of eplthelioid clusters and giant ccl 8 (Langhans’ (Magnification. X300 : reduced $6.) pcrlphery of lymphocytes.

412

O.S..0.M. %0.P. April.

1961

and arc surromlded by :I how of chronic inflammatory cells. There arc many foreign-body giant cells scattered within the epithelioid zones. The microscopic diagnosis is compatible with .sarcoidosis.‘7 The periodic avid&hiff and acid-fast st:rins wow negative (Figs. 4 and 5). On January 30 tho patient, was frw of her seizures ant1 all swelling had su1xGlc~l; the oral lesion, although the same size, was 110~ free of secnndary infection. The Imtirnt war then discharged, to be follow4 ou an outpatient basis. O~tpaticnt FoZZowuy.OIL .?cbrunr~ > every other suturl- WY reu~ovwl. ‘I%! LVIIUII~~ appeared to be healing weU. All remaining sutures were removed on February 9. On February 20 the culture started from material taken from the submental node revealed the presence of tubercle bacilli, and a small nodule could be palpated in the guinea pig inoculated with material from the oral lesion.

On February 20 ihr guinos pig was killctl. :utd the node in quest,ilm 1~~s iaolatc~l :rrt~l ‘l’utwrclc lmcilli wro clwmnstr:~twl in this qwcimcn, awl :ntl.istudied microscopically. tuberculosis t,herapy was institutt~~l. On March 10 therr was nmrkrd rc!grc:ssion nail rrsolution of t.he muwsal lesion ( lpig. 6 1). DISCIJASION

From the time ol’ the first. biopsy pepor!, 211who welxt W~IW~II~(\willi this case realized that, WV wt’rt: dealing with ow (II’ t.1~ gra~~~lomatorts itI fcct.iorts, that is, mycotic infections, Iurs, t uherr~~~losis,0~ sarc!oidosis. The nl?:coi ic inf’ecThe tiona were eliminat.ed 1))~repeated cultures and periodic acid-Ychiff stil.ins.

elect tqthorelic~ ~)ilt.l-wil ill!C~!llt~ll~lt.d tIw ~tossibilil~~ Or’ ;I. luct.ie I’t7wess 01’ sttreoidosis and, in lact, masked our thinking in the sttticwtcnt that the rise itt beta and gamma globulins was not likely to have been.produced by a tuberculomct. The neurologic symptoms could he it1t ributcd to lues, sarcoidosis, and tuttrrettIosis. IIn t&i, i he literature contains cwc4lettt. exa.mplw in which these synqttottts of cpilcpt i tkJl1 seizttrcs, llPil(l~1CI1C, Py‘ signs. fitGal \ve;tkttess! and draggittg gait wc~e ~‘urunclin studies made uu each ol’ these diseases. ,111 WCX? W~~Oi?P(I t0 IlilV(’ ntwosal alit1 ilotlc ittcidcnces. The initial lack of aAd-I’ast or~attisttts is ttoi it wmark;thle finding. l+equent.ly the erg aiiism cilnnot lw iWliltd by direct-sttien 1’ twhniqncs. Prolonged culture on special media and gttitteit pig ittocnhttiott wt ticwl~d to wneciitratc? the lMCillW3 in sufficient nutttbcrs t’or it to be demotistratcd. The microscopic sections revealed a fairly typical sarcoid lesion. However, tuberculotnas arc clttite similar to this Itictnrc-so similar that thm’r are itivestigators who believe that the lesion of sarcoid is caused by an attenuat.ed tuherclc bacillus. Others believe that 30 pc’r cent 01‘ 111thpatients with sarcoid 1ln.v-eeoticiwrciit tuberculosis. The most. eottt’usittp c~lcttwttt in this ~ttse may b(a jJlfiTtioIls, IUW attributed to the coincidental prcsertcc of two ~JWJll~IOJllil~Ol~S imitator of ~llally chwsch and tuberculosis. S.yphilis has 10~1,w beat klJow~l as 1111 pl’ocesscs. The sus~wted brain lesion is most ~wtltably :I nl~tltifcstt~tiotl of il t1oW a.rrested ant1 Intent tertiary stitce et’ syphilis. sc’l*lJJll

A case of tubereulous granuloma of the oral mucosa and submental nodes, with additional findings of latettt cerebrospinal 1~s. has been presented.

w.: A Text-Book of Pat.hology, ed. 5, Philadelphia, 1951, Lea & Febiger, pp. lcil-170. 3. Thoma, K. II.: Oral Pathology, ed. 3, St. Louis, 1950, The C. C Mosby Company, p. 625. 1. Boyd,