ANAPLASTW
ADENOCARCINOMA
LOUIS M ANDEL, D.D.S., AND HAROLD BAURMASH, D.D.S., BRONX, N. Y.
H E opportunity to observe and conduct an unusually thorough study of a malignant tumor metastasizing to the oral region, with resultant oral manifestations, is not frequently made available to the stomatologist. Those cases in which the primary focus of malignancy is not in the oral area but metastasizes to this region, with all the accompanying appropriate symptoms, are usually in the latent spreading phase. A vivid illustration is presented in the following case report of an anaplastic adenocarcinoma of unknown origin with metastasis involving the parotid area. An interesting facet of this case is the fact that the patient’s initial symptoms of distress were of an oral nature. Case Report The patient, a GSyear-old Negro man, presented himself with the complaint that for the
T
past six months he had noticed a swelling involving his right cheek and some associated pain during mandibular opening. Within the last two months, he had developed occasional hemoptysis, but had sought no medical treatment. Additional questioning failed to reveal any further pertinent medical history.
Present Illness.-When the patient first noticed the swelling of his right cheek he attributed the condition to dental problems. In due course, two periodontally involved upper right molars were extracted. Following the extractions, some increase in the size of the swelling developed. This was most marked in the area immediately anterior to the tragus of his right ear. At this time, he became aware of a limitation of mandibular opening. More recently, the pain had become most severe during mastication and led the patient to seek further care. Present Illness.-When the patient first noticed the swelling of his right cheek, he served involving the soft tissues anterior to the right ear and outlining the area normally occupied by the parotid gland (Fig. 1). Upon examination, the mass was found to be nonfiuctuant, hard, and somewhat fixed t.o the surrounding soft tissues. Some tenderness to palpation was noted along the superior aspect of the swelling in the region of the right temporomandibular joint. This tenderness was most marked during the opening and closing movements of the mandible. No cervical lymph nodes were palpable in the area. An intraoral examination disclosed a moderate degree of limitation of mandibular excursions with a slight deviation to the right during the opening movement. The site of the recent extractions appeared to ha.ve healed satisfactorily. A normal flow of saliva was visible from the patent orifice of the right Stenson’s duct. Full-mouth periapical films, as well as right lateral and posteroanterior views, were The intraoral films revealed no related pathology and showed the sockets of the recently extracted upper right first and second molars to be healing normally. The right taken.
From the Department of Oral Surgery, School of Dental and Oral Surgery, Columbia
University.
401
402 a decalcification of the rj ight lateral (Fig. 2) and posteroanterior views ~lemc~nstrated condple. A moth-eaten type of radiolucency~ indicaating a destructive process, (‘011 Id he discerned involving the head anal nec~k of 111~ condylv.
Fig. &-Lateral view of right mandible ren?aling
bone dnntniction
in condylar
regi
L.
In view of the clinical antI radiographic findings, tenlporortlantlibular j o i n t :-ra) pictures also were taken (Fig. 2). Although the left condgle was visualized, no defin i t i v e outline of the right condyle wultl he ascrrtainetl. I,anlinograrrt?: of thP right an d left temporomandibnlar joints ser\-etl tci (*onfirm the findings that Ihe heat1 ant1 neck Of the right condyle were involved extensivrly t)>, an osteol,\-tiv ~~I’OCCS~. Clinical Course.-Because the hwelliqg ill\-olved the parotid area, it was deci d e 11 t o perform a sialographic study of the right parotid gland. Todochlorol (2 c.c.) was il3troduced i n t o the orifice of the right Stenson’s tluvt and s-ray pictures (right later: al and
ANAPLASTIC
ADENOCARCINOMA
403
poste;roanterior views) were taken (Fig. 4). The sialograms exhibited several interes :ting facts . It was quite obvious that the course of Stenson’s duct had been char Iged. The duct, as it cro ssed the masseter muscle, had been displaced grossly downward and si what forward from its normal course. .Uthough compressed, the arborizati on of the duct was with in normal limits. The body of the parotid gland, as represent ,ed b y the radiropaque oil in the secondary and finer ducts, also was displaced markedly, but ii I an
B. F i g . 3.- - A ,
R i g h t temporomandibu1a.r
joint.
R, L e f t tcmporomandibular
josint.
infer .ior and slilghtly posterior direction. In addit,ion, the duct to an accessory p w a s visualized branching from Stenson’s duct as it crossed the ramus of the ma is of’ interest i to note that, although this duct normally takes a vertical course, been displaced posteriorly and inferiorly so that it now occupied an almost posit.ion.
arotid lobe ndible . It it too h a d horizcmtal
T w o days l a t e r , follow-up x-ray pictures revealed the absence of residual oil in the ductal system, indicative of a normally functioning gland (Fig. 5). The sialograms disclosed a gross displacement, without destruction, of the right parotid gland by a superi orly situated extrinsic mass. In view of both the clinical signs observed at the time of exam ination and this sialographic appearance, a preliminary diagnosis of a neoplastic pro cess
was made. A malignant type was indicated by the severe osteolgtic involvement of the CO,1dyle and the fixation of the mass to the surrounding tissues. The patient then was admi tted to the hospital, one week after first havirig been seen in the dental clinic. Hospital Course.--Upon shaving, during his first hospital day, the patient notic, ed a paresthesia of his lower right lip and the right side of his chin. Percussion of the ren iaining right mandibular teeth gave evidence of a definite uumlmcss.
V o l u m e IO Number 4
ANAPLASTIC >4DENOCARCINOMA
Fig.
5.-Right
Fig.
lateral view demonstrating ductal
6.-Arrows
indicate
area
of
405
emptying of Iodochlo NC01
increased
density.
d
ANAPLASTIC ADENOCARCINOMA
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Except for an elevated sedimentation rate (48 mm. per hour), the laboratory findings at the time of admission were within normal limits. Routine chest films disclosed a large area of increased density in the lower left lung field (Fig. 6). In due course, this was interpret,ecl as being a malignant lesion. Papanicolaou examinations of the sputum and saliva from the right parotid gland were negative. Ihiring
the patient’s second hospital day, a surgical biopsy of the lesion in the
parotid area was performed for diagnostic reasons. rnder local anesthesia, an incision was made directly over the tumor mass and a section was removed for pathologic study. The surgeon’s impression, gained at the time of biopsy, was that the tumor was of an infiltrating and malignant type.
Fig.
8.--.krrow
indicates collapsed rertebra.
The pathologist’s report was as follows: GIYISS :
The specimen submitted. consists of several fragments of tissue measuring 0.7 cm. to 1.5 cm., said to be removed from the area of the parotid gland. Sections show in one area a dense fibrous tissue invaded by a poorly differentiated adenocarcinoma of the tubular type (Fig. 7, A and 8). There also is an invasion of the bone of the mandible to which the fibrous tissue is attached (Fig. 7, C). The mitotic rate is 1 TV. The stroma is densely fibrous. In the tubes,
Microscopic:
necrotic material is often seen. Other areas show either fields of normal parotid tissue (Fig. 7, A) or fields of parot,id gland tissue infiltrated by plasma cells and lymphocytes (Fig. 7, D).
Volume IO Number 4 Diagnosis:
ANAPLASTIC ADENOCARCINOMA Anaplastic
409
adenocareinoma.
Toward the end of the first week in the hospital, the patient developed severe back pains associated with an inability to stand upright. X-ray studies of the vertebral column were made, revealing a complete collapse of the eighth thoracic vertebra (Fig. 8). The available clinical, radiographic, surgical, and pathologic evidence indicated an inoperable malignant condition. It was decided to institute a course of radiotherapy to the involved areas. Radiotherapy was administered but, despite this treatment plan, the patient followed a rapid downhill course marked by acute respiratory distress with hemop tysis and dyspma. Four weeks after his hospital admission, the patient died.
Autopsy Findings.-The autopsy disclosed the presence of malignant foci in the right parotid area, lower left lung, hilar lymph nodes, eighth thoracic vertebra, liver, and cortex of the right kidney. Examination of the parotid area revealed that a firm white tumor mass, measuring G cm. in diameter, was present overlying the area of the ramus of the right mandible and was invading the surrounding bone. The parotid salivary gland itself was displaced inferiorly and posteriorly and grossly showed no signs of tumor invasion, thus confirming the sialographic findings. Unfortunately, serial sections of the parotid gland to determine the possible presence of any minute malignant foci were not made. The lower left lung had been invaded (Fig. 9, A) by a mass measuring S cm. in diameter. This resulted in the increased density noted in the chest film and the patient’s respiratory discomfort. The adjoining hilar lymph nodes also were extensively involved by the tumor. Multiple sections of the lung lesion revealed it to be composed of essentially the same anaplastic adenocarcinomatous tissue, with the same tubular pattern, as the surgical biopsy specimen taken from the parotid area. The surrounding left lung parenchyma was compressed and fibrotic. The eighth thoracic vertebra was collapsed. Pathologic specimens testified to the extensive trabecular destruction. Some sparse bone trabaculae and areas of marrow were still present, but the vertebra was massively involved by the malignancy (Fig. 9, B). Several small nodules of the anaplastic adenocarcinomatous tissue were found in the liver (Fig. 9, C) and the cortex of the right kidney (Fig. 9, D). No other metastatic lesions could be demonstrated either grossly or histologically. In summary, all the microscopic sections of the lesion showed anaplastic tumor elements forming glandular structures iu a dense fibrous stroma.
Discussion The autopsy findings allow us to explain with authority the reasons for the symptoms of distress experienced .by the patient. Temporomandibular joint pain and trismus resulted from metastatic invasion by the malignant neoplasm of the right condyle and its surrounding tissues. Naturally, with destruction of the right temporomandibular joint, opening of the mandible resulted in a deviation to the affected side. With the local spread of the metastatic lesion, the inferior alveolar nerve became infiltrated by malignant tissue. This involvement manifested itself as a paresthesia of the lower right lip and chin with numbness of the right mandibular teeth. Subsequently, marked respiratory distress demonstrated itself with t,he onset of dyspnea and hemoptysis. These clinical symptoms were determined by the location of the tumor and degree of bronchial obstruction, necrosis, and ulceration. The dypsnea undoubtedly can be related to bronchial obstruction, while the hemoptysis may be interpreted as the result of malignant necrosis and ulceration of a bronchus. Despite this, the Papanicolaou smear of the sputum was negative. However, the complete accuracy of this test has been
410
MAiYUEL
ASI)
HAURMAHII
0. s., 0. N., & 0. P. :\pril.
l9ii
questioned.’ Autopsy evidence disclosed that the collapsed vertebra, which caused the severe back pains, occurred as the result of a destructive metastatic process rather than by local extension. Because the lesion was that of a poorly differentiated adenocarcinoma, a difficult problem in diagnosin g and localizing the tissue of origin was apparent. The first thoughts on the subject WPI’C dir&cd toward the parotid gland. From a clinical st,andpoint,, t h i s w a s t h e l&on which appeared first. Two points of available evidcncc would seem to contradict the conception that the parotid salivary gland was the nralignancy’s primary site of origin. E’irst. the sialogram indicated that, there was no direct invasion or destruction of parot,itl gland tissue. A direct ma.lignant involvement of the parotid gland inevitably would lead t,o tluct,al tlcstruction or replacemrnt as a result of the canc(‘rous invasion. This picture usually woultl hc reflected in a sialograllt by il. puddling of the radiopaquc oil into the surrounding glandular parcnchyrna a t the point of tluctal pclrforation or a filling defect of t,he oil in th(h gland itself. Tnstcad. thcrr was a lrrark(ltl tlisplaccmc~nt of the parotitl tissue without any sialographic signs of ductal tlcstruction or replacclncnt by malignant cells. Tn atltlition, a normal salivary flow through the patent Stcnson’s d u c t w a s obscrvcd clinically. This was c~nfirmctl 1)~ the complete ductal emptying of thr radiopaquc oil withill a normal poriotl (forty-tight hours). In a puddling situation, with the oil in the glandu1a.r parcnchpma, rctcntion of the contrast media may be eridcnt, for a period of many months and yt~~‘s. Second, the surgical biopsy pcrformctl at the time of admission showc~tl no tumor tissue directly originatin g from or invading the parotid gland. All specimens of paroticl glantl tissue tak(ln at the tilntl of surgical biopsy OI* a u t o p s y e i t h e r wcrc normal in appcarancc or had been invaded by chronic inflammatory cells. This round-ccl1 infiltration was a rclsult of the irritation cserted by the adjacent extrinsic malignancy. This available testimony would S~YW to c~litninatc the right paroticl glantl as the primary site of tumor origin. It is c*onceivable, although highly inprobable, that the malignancy nlight have had its origin from accessory salivary gland tissue in the arca. of the right parotid gland. Lesions also wcw found in the liver and kidney, both of which arc capable of giving rise to an adenocarcinoma. In view of the autopsy findings, it can be assumed that these foci were in an early tlevclopmental stage. This was substantiated by the fact that no discernible clinical findings were evident. It is possible, although u11likely, that the bronchial tree of thr left lung might haye acted as the initiating tissue of the neoplasm. Nest lung ca,rcinomas arise from a malignant metaplasia of the columnar cpithelium linin g the bronchi.2 but, they usually are of the squamous carcinoma type. From a. microscopic study of the malignant ~~~11s and their pattern per SC. the parotid gland could have been interprctcd as being the ntost probable site of tumor origin. However, clinical and sialographic evidence prccludcd this tentative diagnosis. A primary site, nndiscovercd during the autopsy esaminntion, is within the realm of possibility. Theoretically, any glandular tissue,
ANAPLASTIC ADENOCARCINOMA
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containing a columnar type of epithelium could act as the primary site of origin of the malignancy. The anaplasia and widespread metastases impeded a definitive diagnosis as to the primary focus. As a result, a decision as to the initiating tissue could not be based solely upon histologic findings, and no final detcrrnination as to the positive origin of the malignancy could. be rcachcd.
Summary A case report of a metastasizing anaplastic adenocarcinoma of unknown origin was presented. Although the lesions were widely disseminated, oral symptoms were the initiating factors in the patient’s discomfort and precipitated his decision to seek medical care. The sialographic findings of the parotid gland were outlined and discussed. The visualization of the ductal tree of the parotid gland proved to be of definite aid in eliminating the right parotid gland as the primary malignant focus. The derivation of the malignancy was not ascertained.
References 1. Willis R A * Pathology of Turnours, ed. 2, St. Louis, 1953, The C. V. Mosby Company, ;p:35&376. 2. Anderson, W. A. D.: Pathology, ed. 2, St. Louis, 1953, The C. V. Mosby Company, pp. 688-694. 800
GRAND
CONCOURSE.