APITlIARY eystadcnoma lymphomatosum, Wart,hin ‘s tumor, and the English term, lymphndenoma of’ the salivary gland, refer to a tumor of the parot,id gland which is of interest bccausc of its rarity and nnusual appearance.
This tumor was first described by Albrrcht and Arzt, in 1910. They dcscribed two cases of a tumor in the parotid region characterized by specific epiThey assigned to this t,helial structures supported by a lymphoid stroma. tumor the descriptive term, papillary cystadcnoma lymphomatosum. In 1929, Warthin reviewed the two previously described cases and added two of his own, ,Since that time it has been with an excellent description of their pathology. referred to as Warthin’s tumor, because of simplicity rather than precedence. ISCIDEKCE
There have been numerous cases reported since t,hat t,ime. Martin and I~lhrlich,7 in 1944, found sixty-six genuine rascs in the literature and added lwenty-two of their own. In 1946, McNecly* reviewed the literature and reljorted ninety-three cases, but, did not include lVlartin and Ehrlich’s. He added five cases of his own. In a somewhat hasty rev&v, I have found 104 cases. This indicates that it, is not as rare, perhaps, as was formerly- believed, but st,ill is not, a common tumor. 1\Tartin and Ehrlich observed 359 parotid tumors over ;I. period of twclvc years. Of thrsc, 6 per cent wcrc Warthin’s tumors. They comprised less Ihan two per cent of all the neoplasms of lhc hciltl ant1 ncek in lhis scrics. This is in kccpinhcr witjh the observat,ions of others. This neoplasm occurs in inidtlle-aged or elderly males. It is ten limes more frequent in males, with its highest incidence in t,he fitth decade of life. The ~oiuigest case ~~porlecl was two arid one-half years of age and the oldesl was 92 years. CI~INIC!AI>
The clinical history is not characteristic, so that the tumor is ~uually not clinically distinguishable from other parotid gland tumors. IXagnosis is almost n.lways made from biopsy. The chief complaint of the l)aticnt is usually one III.,
Read at the First Feb. 9, 1947. *Assistant Professor
of a papillary processes xvith
cystadenoma paic tolumnar
; reduced lyunphoid ing degeneration.
of facial disfigurement without an)- associated sy-ml)toms. The disfigurcmcnt or apprehension as to the Iruc nature of the growth is usually the reason the patient seeks medical advice. The tumor exhibits slow and stead- enlargement over a period of years. It usually does not, exceed 4 or 5 cm. in size and t,he average duration is three to four years. The tumor is usually in or attached to the parot,id gland and has never been reported in the substance of any of the other salivary glands. It is usually unilateral, but six cases of bilat~eral Nartin and I~hrlich rel)ortcd three ol Warthin’s tumor have been reported. their twenty-two cases as bilateral and in two of these the tumors were multiph~. The tumors usually occur as moderatcl,x- firm to slightly fluctuant, smoothly contoured masses in the preauricular or retromandihular areas. They are usually incapsulated, so are freely morable and do not I)roducc changes in the orcrlying skin. I’ATHOI.OGY
Gross YnSthoZogy-The tumor is usually incapsulated but in some instancrs the capsule may be absent and the t,umor map infiltrate the surrounding gland. The surface is usually smooth. The consistency varies from firm to fluctuant, On cutt,ing, varying amounts of serous to with variations in a single tumor. chocolate fluid escape. The rut surface shows the papillifrrous character, with small white nodules representing the lymphoid follicles. The papillae extend into small, irregular cystic spaces. Histolxctk //logy.-The Iumor is composed of a lymphoid slroma which is The papillae molded into papillae covered b\- stratified columnar cpithelium. may be simple vitli or may show complex patterns of arborization, which produces a cystic, alveolar, or tubular pattern characteristic of the tumor. The epithelium is of stratified columnar type; the outermost layer of cells is tall and cylindrical wit,11 deeply st.ained nuclei at the outer edge. Beneath the columnar cells there are two or three layers of polygonal or cuboidal cells with lighterstaining nuclei and a smaller amount of cytoplasm. There is often the appearThe cells arc pale and stain light,ly with ance of a clear cuticular border. eosin. Between the papillae and in the glandlike spaces there is a pink amorphous material in which there may be some cell debris. The cpithelium rests upon a very narrow basement membrane. The remainder of the stroma consists of lymphoid tissue varying from scant lgmphocytic infiltrations to dense, true germinal centers which are eslvxially prominent in the broad, blunt, rounded papillae. HIHTOWSESIS
Man\- hypotheses have been offered as to the origin of this tumor, nonc of which is entirely satisfactory or conclusive. Warthin” was of the opinion that they represented heterotopia of mucous membrane from pharyngeal endoderm, representing Eustachian tube, and were related to branchiogenic cysts. Ot,hers” have subscribed to the branchiogrnic theory bccar~se of the embryonal commonly type of epithelium associated with lymphoid stroma, an amlngenient found in branrhiogenic cysts.
Jt has hecn suggested4 that they are vestigial remnants of orbital saliva J3’ in some Paruivora. The cahief objection to this hppoi ;hgla1 Ids which are present Failure of is that orbital inclusions ha~c not hecu clcmonstrated in man. csis salivary gland tissue to fuse with the ductal sgsk :m rem nants of undifferentiated hElS also heen suggested.
of salivary glands present ; hydropic
by lymphoid epithelium.
dlbrwlit and Arzt’ p~~o~wctl that tlirj~ arise: I’Iv~ hctcrotol)ic gland rests sitIIampcrl” and uated in l\-mph nodes in 01’ adjacent IO ihc salivary gland. JaffeG arc oI’ the opinion Illat 11rcx~arise from ~11s \vhich may be differcntiatctl from botll aeinar and duct (Ipit holiunl. These ccl Is \vere wiled 011c0cyt cs a11tl give rise lo the tumor when I)twent in alwwant salivary tissue of lymph nodes near the yarot,id gland. They cnllctl the 1~1mor onwq~toma. All agree that this is ii lcraloitl Illinor, hilt, the c3act liistogenesis has not been establishctl.
Two cases have been selected. of irradiation. Case 2 is reportrcl the epithelium.
(‘ax 1 gives some information on the effwt, because? of the lwxuliar vacuolar change in
CASE I.--W. TV:.G., white malt, ilgl’cd 63: cwtcrcd the hospital with the chief complaint of snollcn glands in the ncclr. For six ItlOlltllS, the patient llnd 1KYll il\\‘il1’(’ 01’ ill1 ~Wli~~gCf~l~~llt Of thC left cw%cal glands. One massive gland uxs Ilotctl. It was Ilonttnder, nonadhercnt, and n-as progwssircly iiiwcasin~ in six. 11~ wnsult cd his local physician and xrs given “six s-ray tiwtmciils.” a l’ler wllich the upland decreased in size and almost coi[il)lctc~l\- disal)~w;iretl. The dcwcasc in six was temporary and soon thcrcx VW cJ\-idrncc of rwurrence. The patient thaw had five more *‘s-ray treatments ’’ which did not producct arip dccrc~isc in sizcl. IIe was admitted to t,hc hospital tIv0 \Veeks liIl?r. l’ll\-SiCiXl examination was essentially ncpative except fola a hickor!--nnt-siz(~d, firm, nont(~ndw, ~u~attachctd nodule ai the angle of the jiltv on t,hc anteGol* border of the stcrnoclciclornastoid muwlc. There was one small nodule in the suhma?tilliH8y rcsion. iz1 ol)cration, the nodule was removed and a suprahyoid and deep wrvical I)-mph II& resection was done.
Microscopic $~i:ciwiMf&~~: .\ cystic 1umor with numc’roi~s papillary projeccosinophilic stratified columnar cpithclinm; tions, Covc~Yd I,vl,~],alc-stililli,l~~ stronia composed.. of l!-mphoid tissi7c nitli soinc pC~Illi?li~l CerltcTs in the 1ilrgCr papillae. ” I1iag7iosis : Papillary q3tadcnoina l~~i~i~~tiomatosn~~~.In many areas the stroma was more compact than usual and there was onI?- a cliffusc sprcatl of lymphocytes. In sorn(’ awas thcrc was vacuolar dc#ciici~atioii and ncwosis of the stroma. The cl)ithclinnl in many of the c)-stic sp;1ws ills0 sllo\~cd evidence of degeneration. The tumor was markedly mow vusc*ular than usual. These c+hanges MYZWof a l)atchy charac*lcr and, while> 1IIC S-IX>. al~~~arcntl~ produced necrosis and dc~encration in sonic arcas, the tumor \ras not markedly affected.
C”.\SE2.-J. S., white male, aged 65, had as his chief vomlkint a sensation 0C prcssnrc in the left car of tight, months duration. On ph~xical examination. a firm, almost cptlilte, frwl;- movable tumor, the six of a hen’s egg was notcld .jllst posterior and inferior to the angle of the left mandible. The tumor was not fixed on twsing of mnsc~lesnor did it al)pear fixed to the parotid. The other physiwl findings wcrc not significant. This slowgrowing, nontender mass was first noted fi VCLyears ~~~~c~~iousl~-. Tt had slowly increased in size. The tumor was surgically remowl. E’nthdogic Report.-Gross I:‘strmim(tim: The spccGmw consisted of an encal)sulatcd, semicystic mass, 4 by 2 by 2.5 cm. Micro.swpic E~?JI iwc tiw : Cystic tumor composed of complex papillomatons processes covered by a stratified, columnar cpithelium. The epithelium ap~warctl in maii~- places as a single I;I~cI* of cells on a delicate basement membrane. There was iI \-rq- delicate stroma present which i1; bylaws was lymphoid in vhnractc>r. There were a few gwminal centers present-. The el)ithelinm formed iii~mcrous acinar or plandlilie spaces. lzlrtially filled with a pink granular suhMost of the epithclial cells shod-ccl a marked cystic degeneration. stance. T11c cytoplasm was ~acaolalcd and dispc~rscd throuyhout the wl I in fine granules. This produced a cell of \-cry pale-staining charncler. Diuposis: Papillary C~Sti~ll~‘llOlllil l~ln~~homi~t0s~~ln. showing cystic dcgWcration of cpithelial elements. SLJIRIARY
1\, brief review of the literature has hccn made, a teraioid t,umor of prcscnt,cd to demonstrate the effect show a peculiar degenerative change
on papillary cystadcnoma 1SlnphornatosunI the parotid gland reported, and one cast of s-ray. A second case \vi\s includccl to in the c~~itlielinni of the neoplasm.