ORAL
ROENTGENOLOGY
American Academy of Oral Roentgenology Arthur H. Wuehrmann, Editor
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THE ROLE OF SIALOGRAPHY CHRONIC
OBSTRUCTIVE
Ileon Eisenbud, Oral Pathology The Nowat
Sinai
D.D.S.,”
Laboratory
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IN THE DIAGNOSIS
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AND THERAPY
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SIALADENITIS and Norman
ant1 Ilcpartment
C’ranin, of llcntal
D.D.X.,“” and Oral
New Yo,rk, N. Y.
Surycry,
Hospital
INTRODUCTION
of the salivary glands long have commanded the attention of pathologists and surgeons. Surgical parotitis, which Robinson1 calls a “vanishing disease” was, until the advent of fluid t,herapy and chemotherapy, a “morbid and lethal postoperative complication.” Tumors of the salivary glands, because of their serious and unpredictable character, continue to be a great challenge despite accomplished surgical management, and pathologists continue to expend much effort in unrewardin g attempts to codify histologic patterns and establish prognostic criteria. Possibly because of the emphasis on tumors, scant attention has been paid to the diagnosis and treatment of a fairly common, though less dramatic, disease of the major salivary glands. This condition is characterized by the onset of swelling in the parotid or submaxillary region, sometimes accompanied by pain and low-grade fever. Usually only one gland is involved. The pain is moderate, with periods of remission. The swelling is slight to moderate and is not to be compared with that acen in casts of mumps or surgical parotitis. Indeed, in some cases it is possible to detect enlargement only by means of manual examination. Salivary output from the affected gland is markedly diminished, and often it is totally absent. A few drops of mucoid, scmipurulcnt exudate may be recovered in some cases by compressing the gland and milking the duct. Acute signs
D
ISEASES
*Associate **Assistant
Attending Attending
Pathologist (Oral Dental and Oral
Pathology), The Mount Sinai Hospital. Surgeon, The Mount Sinai Hospital.
1181
As CiIrl? ils l!)lY A2lWtlill injcctccl t)ismuth ilit il s~~hmaxillar~~ clllc*t to dclllonstt’ttte ii calculus. In I926 I’slcnghi.’ (1arIstcq~,+ .Jac~ol)o\%4illId associates,’ ~‘ayncx,‘, and \\‘iskovskyv” rclport,ed indrpmulcntly on their work ill siaIogral)~~y. in 1931, initiat,cd the use ol’ iodized oil, ancl in 193X I%lady ;Illtl llock(ar’ rc~portrd their findings in nint3y-ninc> (*ases in which this typca 01’ c*ontrast rncdilutr \+riis used. The latter authors attcmptc4 to establish diagnostic cdritclri;l rc>Iatcbtl to the sialographic pictures. Although a few more recent works WV wf~:~wd to in appropriate scJc*tions OF this articlc, in general the litc>ratur(l has 1~~11fount1 to IW lacking in spec4firit) with rcl’c~rence to the diagnosis ;lrrd treatment of chronic obstructive sialatlc~nitis. ln presenting this scrics 01’ ciglrty-sevc>n cils~s, we intchntl to itlli1l~Z(’ tltc> tratut*v of the disclase tLntit,v in some detail, to illustrate its oc~urrcncc and sitiiilarit~ in both glands, and to corrclatc certain possible t1tiological factors with th(b clinical and radiographic findings. In addition, an ilttclrll)t will 1)~ made to delintatc thcl cxaet role of sialographp, namc~I+v,that of a thc>rapeutic tncasut7~ with incidental diagnostic VillUC. (‘oiitrary to Iirost opinions esI)rcasscyl in thch IitPl'ilturc that no thcraJ)y for chronic. v>Is(‘s hils t)(l(Jn c~st:rblish(~tl, \VC’ t)rlicbvc, that dilatation and sialography represent ;I clear-cut thrrapelltic ~~~cc~dtlrc~ \vhicll is bcncficial in thv ~l~ajorit~~ of casc~ ot’ chronic obstructive sialntl~~nitis.
A,mamentcrriu1)1.-Thc following materials arc c~~ployrd in this procedure (Fig. 1) : 1. Soft silver probes, Xos. 0 to 5. 2. Ureteral whalebone bougies with brrlled tilt, adapted for sialographic use by shortening to 12 inches in length. These are tough but pliable probes which can be made to follow the course of tortuous ducats without trauma or pcrforat,ion. They are available (Sklar Nfg. C”o., Long Island City, I\;. I-.) in five sizes: 0 t,o 1. Since introducing these instruments, we hart been able to perform sialograms which might otherwise have been impossible. The bougies have brcn especially hclpful in serial dilatation of the submaxillary duct. 4. Cannulas, gauges 18, 1.9, 20, ~1, and ~2. Those arc prepared 1)~ 11/2 inchblunting, rounding, and polishing the points of ordinary injection needles. A 15 degree bend l/h inch from the hub will produce a more convenient shape for handling, particularly for insertion in t,hc submaxillary duct. 5. Iodized oil. We have use Lipiodol and Iodochlorol, both oil based media, for years without evidence of side effects or toxicity. 1%‘~ believe that the viscosity and lubricating rffrct of the oil may provide some therapeutic advantages in the freeing of mucous plugs and small soft, calculi. For this reasoq WC hare not turned to the water-soluble
SIALOGBAPHP
contrast media. Nevertheless, certain reports of deleterious effects from oily media deserve mention. Mandel and Baurmash” described granulematous changes in the buccal soft tissues after inadvertent deposit. of the medium. Epsteen and Bendix” reported that poppy seed oil (the vehicle of Lipiodol) injected into the submandibular glands of ten healthy dogs produced marked foreign-body reactions, as seen when the glands were removed and examined histologically at 1 to 4 week intervals. Thackray”’ has demonstrated a granulomatous and fibrotic rcaction to Lipiodol in a parotid gland removed some weeks after sialograph,v; he concludes that “in time a marked fibrous reaction develops
Fig. l.-Armamentarium ureteral whalebone bougies, and cut-down set.
for sialographic Luer-Lok syringe,
technique. Graduated silver probes. assorted cannulas, iodized oil, local
graduated anesthetic,
around the extravasated irritant which may greatly complicate subsequent surgery.” However, his further conclusion that “the continued presence of an irritant may make thr difference between resolution and persistence of an inflammatory process ” is contrary to the findings in our study, which demonstrate beneficial effects from instillation of iodized oil. On the other hand, Sicard and Forestier,l’ working with dogs, found that 10 to 12 ml. of iodized oil could be injected intramuscularly every day, to a total of 100 ml., with no foreign-body reaction or harm. 6. Local anesthetic syringes and nnesthetic vial. 7. Cut-down set, including
a No. 11 Bard-Parker
blade.
~:I~b;xNl~l‘I) .\SI)
1184
rL,ohl,d,,l’ ,hl<
(‘K.iSIS
Instillation Y’echniqw----. Pulotid glc~tl: The orifc*cJ 01 tllr pat’otid tl11c:t is loc~alt~ti at the (*rest of ;I carunclc on the mneosal surl’ac~~ of I IW cheek oppositcl tlrcl nraxillar~~ first. molar. The orifice and duct contain muscular cl(bments hut a w gw?dly prohetl and dilated without difficulty. The adult t111c~t averages 15 10 2% mm. in lcngt~h a.nd runs a straight course in thcb c~licck cbsctyt for a turn ilround tllr ilIltP~iOr’ bortlw of the massctcr muscle, 8 to 10 nu11. hcahiritl the orificcu. Blthough anesthesia is uot essc>ntial for parot,itl sialography, the patient The ol)et*atol will he mor(’ con1fortablc il’ the Ion g I)uceal n(‘rv( is jnfiltrated. stands facing th(i seated patienl. ‘I‘h(, otifict~ is tlric~tl. ;Intl the glautl :I ntl tluc*t arc luilketl gently in a11 ;tut<‘riot* clircxc,tion. 111Itlost (‘ilses :L tl~cb~)01 t\vo of secretion, usually nlucoitl or Inllc,ol)nl.ulcIlt. will 1)~stlischargctl. At this momcnt the orificca will bc in c~vitl(~ttcc~;tntl a ~)rohc of the finest gilu#(L is l)itssetl until it rcachcs the turn of th(L tluct arountl th(b trlassctrr muscle. At this juncture the cdheck is grasptatl l)c~twee~l th(J thumb autl Cor~fitlgc~r at t,hv labial coltlmissuro autl pulled forwarcl. This si &chcs autL st.raightens t h(x tluvt and genc>rally will permit passage ol’ t,hta hougic to the hilus of the ,glantl. Serial dilatation is accompli&t1 by repetition of this proeedurc with probes of ineach bougic, to relnain in ~)lac~~ untlisturbc~cl fog creasing gauge, allowing s(b\reral minutes. M’h(an snficicnt tlilatatioll hils ~CCII achicvc~tl, ;I I)luntetl IIPCV~~CJ using iL llCUlO&t. nlay bt: inscYtec1 illl(1 immobilizc~tl ilt situ I)y all ilSSiStilllt. The syringe is now loadctl with warnt(atl (to rc~lucc~ viscosity) eont vast rnctliutrt and coupled to the needlt~. A mi~limtlm of 2 ml. ol’ oil should be injcctctl for alltl the parotid study. Strong thurnh ~~IWSU~~ must be used. i)llercnshaw 1t0se~~indicate that, a ~)rcssure ol’ at lcsast #MI mm, Hg is t~eyuircd. The gland will enlarge a,nd the pat,ient will ex1)(Jrien(*c some cl&comfort as t,hc n~at.~~Ai\Iis slowly but firmly instilled. The nrctllc and syringtb arc1 110~’ withdrawu. and a sponge is immediatcl~ cor~~p~ssc~tl against the orifice to pr(lvent lraka.gc~. Direct lateral and antcrol)ostc~l~iot~ ratliographs now may b(> taken. For the lateral views, the mouth shoultl t)(b opeu to avoitl sul)c’rilrll)ositiott of tctlth on the duct image. NubmmiZlary gland: The sialographic technique for the submaxillarygland presents greater problems than that for the parotid gland because of the fineness of the orifice and duct antI the highly rnotilc nat,ure of the carunclr. Th(l orifice of th(l submaxillary duct is located just lateral to the lingual frcnum at the medial t111tlof th(a I)licit sublingualis antI is often fc~untl sottIewhat inferior to the crest of thtl carunclc. Sphincterlike muscdular c~lcnlents surround the opening. the contraction of which combinrtl with its init,ial small diameter, may ma,kc cnt 1-y difficult ant1 ev(~n impossihlo in SOIII~ cases. The duct itself is lacking a muscle wall and is easily dilated, once it is entered. Tt runs from 25 to 40 mm. in a fairly straight posterior and inferior course. Anesthesia is essential for full cooperation of th(J patient, since the instrumentation is often lengthy and tedious. This is best achieved by administration of a lingual block or routine inferior alveolar with lingual block. Tdocal infiltration
tissues.
ariestlicsia
is
to
be
ilVOid(%l
IWcauSC
Of
tilt>
WSUltaIlt
distortion
of
1185
SIALOGRAPHY
Volume 16 Number IO
The patient is seated with the chair in a low position, and the operator stands behind him. The index finger and third finger of the operator’s left hand are placed externally to support the jaw and exert gentle upward pressure on the contents of the floor of the mouth. The thumb is simultaneously in position to retract and immobilize the tongue. The procedure described for the parotid gland now may be followed, with the exception that 1.5 ml. of oil should be adequate for full visualization of the duct system. Also7 lateral oblique rather than direct lateral views are preferred. Often the orifice to the submaxillary duct is indiscernible. If milking of the gland and duct is unrewarding7 the patient, may be asked to suck a wedge of lemon, which may stimulate sufficient excretion to permit location of the opening. In some cases it has been found helpful to prescribe pilocarpine nitrate, two drops of 0.5 per cent solution three times a day and at bedtime, for a few days before a second attempt is made. Often removing the ball from the finest whalebone bougic will help. Xcvcrthcless, despite patience and skill, it will be found that in some instances the duct cannot, be entered at all. Occasionally, in the case of the submaxillary duct, the finest bougie may be insert4 but no larger one may be introduced. Tn such instances the duct is slit lengthwise over the probe from the orifice for 3 to 1 mm. The larger bong& then may be introduced without difficulty. It should be mentioned that sialography is contraindicated in the presence of acut,e obstruction or acut,c infection. The procedure may be performed safely IO days to 2 weeks after the subsidence of an acute episode. CHRONIC ANALYSIS
OBSTRUCTIVE
SIALADESITIS
:
OF DATA
The relative incidence of parotid and submaxillary involvement is shown in Table I, and Table II shows the sex incidence of chronic obstructive sialTABLE
I. RELATIVE
IKCIDEKCE
Total
OF PARoTin
cases:
hKn
~I'BMAXILLARY
INVOLVEMENT
87
Parotid: 59 (68%) Submaxillary: 28 (32%) Bilateral parotid involvement: 5 Bilateral submaxillary involvement : Combined parotid and suhmaxillary:
1 2
adenitis. The finding of a preponderance of female patients has led to speculation regarding the possible relationship of hormone-induced changes in tho salivary glands. This possibility is borne out further when age incidence is analyzed (Table III). TABLE
II.
SEX
INCIDENCE
--_-
-SEX
Male Female
PAROTID
21 38
SUBMAXILLARY
21
TOTAL
It is evident that chronic obstructive sialatlcnitis is a dineasc, whicah OVWI~S more freqnclntly in adult life than during childhood ant1 t,hat t’c~n~;llw arc’ Ifectcd more frequently than males. In the parotid c+atcigory th(b1.c was. in this series, an especially high incidence in the -IO- to T,%ycar age group. (‘asc~s involving the submaxillary glantl showcat a somc>wh;rt I)I*oatl(‘t~ spr~atl. with rnatuw less specific concentration in the I~OW itgti ~IYJUJ). ‘l’h(‘r<~ wtlt*(‘. however, no submaxillary cases in child Ron, wh(~r(~as MM’S involving thca l)arotitl showed a 2% pcbr cent incitlcncc below thr age of 20 )‘(‘a t’s. I’at*otitl illvol\:rtnclnt in children thus is not, uncon~mon ; rcllatc4 ~~~i’c~ren~s;IIY’ fount1 in th(l lit(lrxturc. including it suggwtcvl possible rcblationship to ceystic fibrosis oi’ t h(b pall(‘,‘(‘i,s.‘:’ il
Parotid
8
3
1%
:!I)
4
Submaxillnw
II
I
II)
11
ti
The preponderance of parotid cases found in the JO- to S-pear age group lends support to the possible association with hormonal changes, previously suggrsted by the finding of a high incidence in female paticuts. Among these patients, coincidental menopausal symptoms were encountered frequently. Additional possible contributory factors associated with aging arra thr reduced salivary output and incrcasctl salivary viscosity seen in some persons. lndectl. many of these patients may be manifesting early signs of SjGgren’s disease, especially since salivary gla.ntl involvements in this caondition often antcdatcs &her symptoms by years. I+ Hist,ologic examination of submaxillary salivary glands removed because c)f rrpcatctl acute obstructive episodes has shown a pattern of acinar atrophy and fibrosis consistent with that clcsc~ribetl for Sjiigren’s disease. Related Medicnl Background.-A complet,e medical history was rc~rded in each case. No significant correlations wcrc cncountc~red except, for a high incidcncc of arthritis ( 18 patients out of 73 in the group over 20 years of age). If it were not for the known high incidence of arthritis in the general l)o~)ulation, one might consider this another indication of a possible relationship to Sjiigrcn ‘s disease in a segment of the group under study. Eighteen of t,hc eighty-seven patients had a known history ol’ ~nurnps. Of the cahildrcn under 14 years of age, nine had mumps during the year preceding the onset, of obstructirc> symptoms. Three adults similarly ha.cl contracted mumps during the year l)rectrtling the onset of obstructive symptoms. On the basis of these figures, one may spcculatc about a possiblr caausal relationship in somt cases. Partial testicular atrophy is known to follow in some instances of mumps orchitis; it is not unreasonable t,o suggest that some permanent damage to salivary glands may 1x1 incurred, which might prcbtlispose to subsequent. obstruct,ivc involvement.
Duration of Symytoms.-There was great, variation in the duration of thr disease from the onset of symptoms to t,he time of our initial examination of the patient (Table IV). No conclusions may be drawn, other than the obvious one that. the condition may he long standing and the symptoms persistent.
--
DIJRATIOK
Less 1 to 6 to More -
than 1 month 6 months 12 months than 1 year
NO. OF CASES (PAROTID)
11 8 6 34
NO. OF CASES (SUBMAXILLARY)
10 h 15
Clinical Observations.-External swelling over the affected gland usually was perceived only in association with an acute exacerbation. At such times varying degrees of enlargement of the gland and edema of the surrounding tissues were encountered. Fever was not a. serious complaint. Temperature elevations to more than 100’ F. occurred in only eleven of the fifty-nine cases of parotid involvement and in only five of the twenty-eight cases in which the submaxillary gland was involved. When the temperature did rise above 100 degrees, it remained below 101 degrees in all but two cases, in which it rose to 102 degrees. Lymphadenopathy was an inconstant finding, accompanying submaxillary involvement in roughly the same proportion of cases (25 per cent) as parotid involvement (20 per cent). Output of saliva was markedly diminished in many cases, with complete shutdown in seventeen (61 per cent) of the submaxillary cases and nineteen (32 per cent) of the parotid cases. Purulent drainage was obtainable from the duct orifice in nine (32 per cent) of the submaxillary cases and sixteen (27 per cent) of the parotid cases. A variety of organisms were grown in cultures of this exudate, including alpha and beta streptococcus, Staphylococcus aureus, Streptococcus pyogenes, Neisserin catarrhalis, Bacillus proteus vubgaris, and Bacterium coli. Evidences of acute infection, when present, invariably were controlled quickly by the administration of penicillin or one of the broad-spectrum antibiotics. With reference to dentures, fifteen (53 per cent) of the patients in the submaxillary category wore full or partial appliances, as did fift,een (25 per cent) of the patients with parotid involvement. There was no clear-cut association with the presence or absence of dentures. RIALOGRAPHIC
PATTERNS
Certain assumptions help one to understand the otherwise confusing pleomorphism of radiographic patterns. Essentially, the radiopaque medium visualizes the duct system. Acini cannot be seen individually, since they represent microscopic entities, but they may be seen when coalesced. Tumors are revealed
bg circumscribed anatomic displacement ac~ompanic~l hy ow or
tilling defects. Fibrosis is indicated by a less unified filling dc&icncy, without, anatomic displacement. The frcyucnt,ly cncountercd patterns of c(+tasia, pooling, and disorientat,ion may be explained by rstraductal diffusion of the dpc into thr parnnchyma and traversal of minor and major interlobular pathwa.vs. The patSterns may appear singly or in combination. \Yt: have separated t,he often superimposed and simultaneous variants into categories which are tabulated for the purpose of clxplaining thcair occurrence on an anatomic or clinical basis. Thus, it will hc noted that most, 01’ the illustrative rascs demunstrate more than one of the listed pattern abnormalities. In advanced obst,ructire parotid disease, for example, the intraglandular areas may manifest, part,iaJ lack of filling with regions of void, an even snowflake arrangement, or a comhinat,ion of the two. IIepcnding on the naturtr and degree of involvement, various combinations of extraductal diffusion, cctasia. nonfilling defcct,s, and duct disorientation will be noted. The Normal Parotid Sialogram.--Extrnglandular patterlt, direct lateral Gel@ (Pig. 2): The orifice or dist,al end of the duct is noted in the region of the maxillary first molar. There often appears to be ii loop in the last few millimeters of the duct, but, actually this represents the turn of the duct about the anterior border of the masseter muscle. The duct varies in width from 1 to 3 mm.; it has even contours and a consistent, diamctcr to the hilus.
Fig,
S.-Normal
parotid
sialogram. Note classic arborial of major and minor duct
*Anteroposterior views were found to provide routine use in studies of obstructive sialadenitis. For however, such views may provide important additional
pattern structure.
with
even
i~10w
contours
insuffkient additional data the localization and evaluation information.
and
filling
to warrant of tumors,
SIALOGRAPHY
1189
IntragZanduZar pattern: Behind the hilus of the gland an even, arborial pattern is evidenced. The larger ducts are visible as specific entities. Smaller ducts and their terminal acini are represented by a hazy, uniform, cloudlike appearance. When properly filled, the full gland contours are visualized somewhat like a dense soft-tissue shadow. The Normal Submaxillary Xialogranz (Pig. 3).Extraglandular pattern: The submaxillary duct follows a lazy “S” route in its long course through the floor of the mouth from hilus t,o orifice. It varies from 25 to 10 mm. in length, with even contours and a consistent diameter. While normally it is nearly collapsed with walls touching, under the pressure of the contrast. medium it expands to a width of 3 to 5 mm. Accessory ducts from the sublingual gland may join the submaxillary duct in its distal portion. In several cases a major connection (Bartholin’s duct) was encountered, and in one inst,ance the sublingual gland itself was visualized (Fig. 4). Intraglandular pattern: The minor duct. structure of t,he submaxillary gland is less coarse than that of the parotid. In the normal sialogram little or no arborization may be evident, and a soft, lobulated cloud effect is produced.
Fig. 3. Fig. 4. Fig. 3.-Normal submaxillary sialogram. Note wide, even extraglandular duct contour with amorphous. cumulus-cloud appearance of intraglan(lular structure. Fig. 4.-Sublingual gland and accessory duct in a ST-year-old man with history of a calculus in submaxillary duct 7 rears prior to this film, as well as recurrent infections, flbrosis, and blockage of major duct. Patent accessory duct permittwl sublingual gland filling.
Abnornml Parotid Pattems.Extraglandular: The bifd duct (Fig. 5) is a variant, of the normal pattern, rather than a true abnormality. About 1 cm. from the orifice an accessory duct may be seen joining the major duct, presenting a bifid appearance. This accessory duct, which is a normal anatomic structure, is quite threadlike in appearance, having a diameter of 1 mm. or less. It is not visualized in all parotid sialograms, but when it does appear the diamet,er of the main duct often seems to be reduced. In such instances, predisposition to obst,ructive involvement might be anticipated in view of the reduced opportunity for egress of mucus plugs and viscous saliva.
Jlrwf sterwsis i Fig. 6) III;IJ. 1~ss(3.11I~artic*liliii*l~~ iii tlrr rcqioli of tliv hilal, junction. This cstremv narrown~~ o I’ I hv tluct nl;ly rvprc~~t vieat ri(‘iaI st cnosis. or it may hart a congenital basis. It has bc~n suggclstcd” that all~~rpic~edema OF the periductal tissurs may bc responsihlc in some instances. Whatavc~r tlrca basis, the narrowed duct prevents adequate cmptyin, v of ttir irffcrkd gland and causes rcsult,ant hark pressure, stasis, acinar bnbakdown. and I~ltirnatcly ret rogradtb infection. This hypothesis is in accord with tlic ohsc~r~ations of Winstcn and (Iould,‘” who attribut.ed all inflammator\. procures in their scrics of fifty-three cases to various abnormalities of the duct system. Mandcl and Raurnlash’7 add further support to this concept, indicating that infection is secondary to partial or complete duct obstruction. An obstruction is shown in Fig. 7. Although such :III orcurrencr is r’arv, a calcified concretion may bc encountered in the parotid duct. It will appear as a round or ovoid shadow in the control film which is routinctly taken hcf’orc sialography. Since sialoliths are often poor1.v calcified, careful inspection of the radioFig.
Fig.
5.
A.
Fig.
i.
6.
K.
Fig. S.-Bifld parotid duct in a 33-year-old woman with 3 year history of recurrent pain and swelling. Note thin threadlike appearance of major and accessory duct. Eighteenmonth follow-up subsequent to dilatation and sialography showed patient to be asymptomatic. Fig, K-Parotid duct stenosis in a 36-year-old woman with recurrent swelling of * years’ duration. Stenosis at the hilus has caused constant back pressure, resulting in secondary proximal dilatation and breakdown. Four-year follow-up after sialopraphy has shown patient to be free of symptoms. Fig. 7.-Parotid calculary obstruction in a 37-year-old wonmn with history of pain, swelling, and infection of several years’ duration. Recovery was complete Pollowing removal of stone (A). Sialogram (B) showed marked duct dilation and disorientation of intrsglnndular duct structure. Patient has been asymptomatic (luring a 3 year follow~up period.
Fig.
9.
Fig. &-Canalized stone. This may cause partial obstruction at first, which becomes more in relation to degree of closure of lumen. Fig. S.-Sacular dilatation in a 40-year-old woman with a 6 month history of recurrent swelling of the left parotid gland. Sialogram shows sacular dilatation of the duct with sialangiectasia and extensive disorientation of the minor duct structure. Following sialography, patient has remained asymptomatic for 6 years. severe
graph may be necessary to detect their prewncc. Since a stone may he canalized during the early stages of its development (Fig. 8)) partial obstruction to the flow of saliva or to the instillation of dye may result at first. Obstructive sympt,oms become more severe in relation to the degree of closure. The passage of the oily medium into the gland ultimately may be blocked completely. Sacular dilatation (Fig. 9) presents a pattern resembling a string of sausages and is reminiscent of Hirschsprung’s disease (congenital idiopathic dilatation of the colon). This disorder has been attributed t,o a neurogenic defect, such as a decrease in the number of ganglionic cells. I8 Smooth-muscle atonicity may similarly account for this picture in the parotid duct. Sacnlar dilatation is never
Fig.
10.
Fig.
11.
Fig.
12.
Volume 16 Number 10
SIALOGRAPHP
1193
seen in the submaxillary duct, which seems logical since the submaxillary duct has no muscular coat, The obstructive symptoms in the parotid gland proper, seen in cases of sacular dilatation of the duct, are easy to understand on the basis of the obvious hydrodynamic failure. Intraglandular: In cases of duct cldatatiun (Fig. 10) the major intraglandular ducts may display irregular enlargements and outpocketings, possibly on the same basis as that described for sacular dilatation of the main duct. As a result of back pressure and stasis, smaller excretory and secretorp ducts may develop divrrticula as well. The functional arrangement may he intact, indicating less severe structural deterioration. XiaZa,ngiectasia, (Fig. 11) presents a pattern that is attributed to the breakdown and confluence of acini with resultant collection of the dye in macroscopically visible pools. It may be interpreted as an early change in the gland parenchyma in response t,o back pressure and stasis. This type of acinar coalescerise may be comparable to the retention phenomenon seen in the palatal mucous glands. or “cottonball!’ apThackray lo has suggested that the usual “snowflake” pearance is due to the surface tension of the oily medium which causes it to assume a globular form. In one of the few papers on this subject which contain histopathologic documentation, Thackray contends further that cxtraductal diffusion of the dye, induced by the pressure of the injection, is responsible for the ectatic pattern and that the picture is in reality an artifact. His excellent photomicrographs show round, clear spares corresponding to opacities seen in the roentgenogram. These spaces had no linin, u of cpithelial cells, and serial sections showed each space to be in contact with a duct, the break in the wall of the latter being occasionally demonstrable. 11~ thus interprets the globular sialectatic pattern as an indicator of chronic inflammation of the gland with weakening of duct walls and subsequent rupture during sialography. Suffice it to say that ectasia is not seen in studies of the normal gland and t.hat, at the very least, its occurrence indicates an inability on the part of the gland to confine the injected material within normal anatomic boundaries. Disorifantation (Fig. 12) is the result of long-standing obstructive disease that leads to bizarre sialographic charact,eristics. Duct pathways seem to be without direction, running into opaque pools, coiling back upon themselves, and blind-end loops. Sialbranching from large trunks into irregular, interrupted, angiectasia may or may not be in evidence, since acinar atrophy and fibrosis already may have ensued. Some aspects of this disoriented pattern can be explained only on the basis of the dye’s escape into irregular interlobular spaces. Fig. lO.-Parotid duct dilatation in a 55-year-old man with a 3 year history of recurrent pain and swelling. Major duct dilatation accompanied by partial fllling and minor duct dilatation and disorientation. Accessory ducts are seen distal to hilus. Patient remained asymptomatic 5 years after sialography. Fig. Il.-Parotid sialangiectasia in a 58-year-old woman with a 7 year history of panglandular symptoms, with concurrent uveitis and rheumatoid arthritis (Sjijgren’s syndrome). Sialogram showed pooling of medium and loss of normal duct structure. Initial treatment yielded temporary relief. Repeated instillations have kept patient comfortable for 5 years. Fig. 12.-Parotid dact disorientation in a 47-year-old woman with a 3 year history of pain and swelling. Pooling, irregular and abnormal duct patterns, and areas of underfllling are in evidence. Sialography was performed twice in 6 years, during which time the patient was entirely free of symptoms.
Fig. l:(.-Photomicrorrarh tion. Nearly complete acinar the fibrow par‘enchyma.
Fig.
14.-Parotid showed
of submaxillary glan~l removed atrophy has occurr~tl. The ducts
tumor in a 53-year-old man with an irregularly bordered Ming defect.
because of remain intact
chronic obstrucand Iliscrrtp in
a history of rectal carcinoma. Sialogratn Biopsy revealed metntastic tumor.
HIXLOGRAPHY
Volume I6 Number IO
matous fibrosis (Fig. 13). In the presence of these residual patent ducts, partial visualization of the gland is possible. A tumor may be suspected if the filling defect occupies one portion of the gland (Fig. 14). Benign tumors cause a generally rounded and smooth-bordered defect, and the filled portion of the gland surrounding the defect shows normal arborization. Malignant infiltratin, p tumors cause a more irregular and diffusely outlined filling defect. Abnormal Submaxillary Patterns.-The abnormal sialographic patterns described for the parotid gland occur in the submaxillaq gland as well, with rep*tain modifications engendered by the differences in anatomy. Fig.
Fig,
15.
17.
Fig.
Fig.
16.
18.
Fig. 15.-Submaxillary duct dilatation in a 58-year-old man with a 6 year history of recurrent submaxillary swelling. Sialogram showed widening of the major duct with abnormal intraglandular duct pattern and underfilling of the gland structure. Patient has been asymptomatic 8 months following sialograuhy. Fig. l&-Submaxillary intiaglandular disorientation in a 58-year-old woman with a 4 year history of intermittent pain with csntinuous swelling. Sialogram showed total disorien’ation of the intraglandular duct pattern, massive dilatation of the hilar portions, and a lack of peripheral filling. Patient has remained comfortable for 3 years after two treatments. Fig. li.-Submaxillary intraglandular disorientation in a 42-year-old woman with a 1 year history of swelling and pain. Superior portion of gland showed normal cumulus-cloud effect, but elsewhere pattern is ill deflned with pooling and duct disorientation. Patient has been asymptomatic during 7 year follow-up period. Fig. 18.-Submaxillary hilar stricture in a 34-year-old woman with a 1 week history of pain and swelling. Sialogram showed an interruption of ductal continuity at the hilus with a sparse stellate pattern displaying only a few of the larger intraglandular ducts. Patien. has been asymptomatic during a 2 srar follow-up per’iocl.
girl \yith a 2 J-eat‘ histon ’ of rePig. lY.--Paroti(l snowflake pattern in a .>-year-old showed anesthesia, ‘rent pain and swelling after mumps. Sialograrn, performecl umler general sialc ,gmphy, mwflake” or “buckshot” ectasia xvitb intraalandulxr (luct ~lilntation. Foil lowing ! child has been wymptomatic for I! J-enrs.
The clinical implications of thaw \~wieci I)attcms in the snbmasillary gland are similar to those deduced for the parotid gland patterns, with one except,ion: In contradistinction to the rar(~ OCCLII’TCIICC of stones in thv parotid duct, concretions an: often encountcrcd in the subnraxillar~y duct, especially at the hilus, and vven in tht> gland itself. Surgical intt~rvcntion is essential for alleviat.ion of symptomx in sucll CRS~S.Subscqurntly, occasional dilatation and sialography may serve to prcvcnt. the recurvnco oI’ ohst r*nct ion by wrnovirrg sccdlinp calculi and mucus thrombi. Pediatric Sinlog~raphic I-‘nttcl,ns.--Eight patients between thtl ages of 6 rnonths and 12 years were’ studied. X11 had parot,id involvement, and all except one were seen less than a y-car aft,cr having had mumps. In each vase a “buckshot ” or “snowflake” pattern was displa.vcd (Fig. l!)). Studies of the normal child showed a sialogram identical to that of the normal adult. On this basis, it seems reasonable to suggest once again that mumps may 1~ a factor in the prt?disposit,ion to ohstructivc~ sialadcbnitis.
1197
SIALOGRAPHY
Volume 16 Number IO RESULTS
OF THERa4PY
Initial response to dilatation and sialography was favorable in forty-seven (80 per cent) of the parotid cases and twenty (‘72 per cent) of the submaxillary cases. Pain and swelling were reduced. Undoubtedly, some of the cures might have occurred spontaneously without, intervention. However, the chronic nature of this disease and the usual long-standin, 0‘ svmptomatology should preclude coincident spontaneous resolution as a strong factor. Long-term results were more difficult to assess because of diminished followup response as the period subsequent to initial treatment increased. Forty-seven patients (54 per cent) were followed from 1 to 5 years, and in this group thirtynine (83 per cent) reported no recurrence of disease. No significant difference in treatment results was noted between the cases involving the parotid gland and those of submaxillary gland involvement. The recurrence rate following trea!ment is shown in Table V, in which patients are divided into groups according to maximum follow-up period. TABLE --..-
\’ -
~ NO NO. OF CASES
FOLLOW-t’,’
37 8 10 29 Totals
Under 1 year 1 to 3 years 3 to 5 years Over 5 years
84* *Three
PER1011
patients
1 month to 5 years were not seen after
.~
PAROTII,
RECURRENCE
RECURREKCE SUBMAXILLARY
18 4 .1:
ii
46
PAROTID
SUBMAXILLARY
2 6
6 0 2 3
4 1 1 1
20
11
7
treatment.
Re-treatment in the eighteen cases of treatment failure was undertaken at varying intervals and with varyin, v degrees of success. Eight patients failed to respond despite repeated re-treatment. The sialographic patterns of these eight glands all showed ectasia; six showed duct disorientation and extraductal diffusion, indicating advanced structural derangement. LIMITATIONS
OF SIALOGRAPHP
It must be emphasized that decisions regarding treatment, cannot be based on information of the type afforded by sialographic studies alone. Prognostic significance may be attached to the degree of structural derangement revealed by the duct pattern, but no further clinical significance may be assigned to the correlated sialogram. Specific patterns are not as yet attributable to specific disease stages or characteristics. Reference has been made to the value of sialography in the diagnosis of tumors. Yet it is unlikely that the extent or nature of surgical intervention would be influenced to any degree by the type of information obtained through sialography, once the presence of a neoplasm has been determined by history and palpation. On the other hand, a tumor so small as to escape detection on digital examinat,ion would not cause obstructive symptoms that would lead to sialog-
raphy. Should such a clinically imperceptible lesion be presthnt coinrid~~nta1l.y in a gland which is being studied with contrast medium. it is caertain that the tlci’rct would go unnoticed in the film. THE
ROLE
OF 814LOGRAPMY
Having expressed the opinion that information obtained from sialography is of limited clinical significance, what, map we say is the role of sialography? We find that there are consistent benefit,s to be derived from its USCin the trcatment of chronic obstructive sialadenitis through its dilating, irrigating, and lubricating effects. The resultant film indicates whether the material has been instilled in the gland in sufficient quantity, a factor of importance if adequate therapeutic dilatation of ducts is to be assumed. The film serves one more fun(dtion which may not be so obvious-it shows whether or not the material has been instilled into the gland at, all. Thus, the appreciation of sialography as a clinically significant procedure depends on an undclr;tanding of its relationship to therap!: as well as to diagnosis. SUMMARY
A?iD
CONCLUSIONS
1. A series of eight,y-seven cases of chronic obstructive sialadenitis has been studied in an effort to correlate possible ctiological factors with the clinical and sialographic findings. The armsmentarium and technique Sor sialography ham be& described. (68 per cent) involved tlir 2. Among the eighty-seven cases, fifty-nine parotid and twentp-eight (32 per cent) involved the submaxillary gland. The fact that 70 per cent of the patients were females indicates a possible relationship 01’ hormone-induced changes in the salivarv glands. This possibility was borne out further in the analysis of agct incidence, which indicated an especially high incidence of parotid involvement in the JO- to 60-year age group. The submaxillary gland cases showed a broader age spread, but in children t,he disease involved only the parotid gland. No significant systemic correlat,ions were encountcretl in the medical backgrounds of the patients. A possible relationship to mumps is suggested in the group of fourteen children in the study, since nine of these had the disease during the year preceding the onset of obstructive symptoms. There was great, variation in duration of the disease, but in general the condition was long standing and the symptoms were persistent. External swelling was perceived only in association with acute exacerbations; fever wa,s not a serious complaint ; lymphadenopathg was an inconstant finding. I’urulent drainage containing a variety of organisms was obtainable in some cases. There was no clear-cut association with the presence or absence of dentures. 3. Kormal and abnormal sialographic patterns have been described, classified, and illustrated. The often superimposed and simultaneous variants have been separated into categories for the purpose of explaining their occurrence on an anatomic or clinical basis. The degree of struct,ural derangement revealed b> the duct pattern may have prognostic significance, but n(J furt,hcr significance may be assigned to the correlated sialogram on the basis of present knowledge.
4. On the basis of sialographic studies, it is suggested that two factors predispose to chronic obstructive sialadenitis : Reduced ductal patency with inadequate opportunity for salivary egress. This reduced patency may be due to congenital anatomic variations, t,o calculi, or to cicatricial stenosis. Reduced ductal tonicity with gradual dilatation, pooling, failure of evacuation, and retrograde infection. 5. Initial response to dilatation and sialography was favorable in 80 per cent of the parotid cases and 72 per cent of the submaxillary cases. Among forty-seven patients followed from 1 to 5 years after treatment., 83 per cent reported no recurrence of disease. No significant difference in the results of treatment was noted between parotid and sukmaxillary eases. It is concluded that dilatation and sialography represents a clear-cut therapeut,ic procedure which is benrficial in the majority of cases of chronic obstructive sialadenitis. REFERENCES
1. Robinsop, J. R.: Surgical Parotitis, a Vanishing Disease, Surgery 38: 703-707, 1955. 2. Uslenghl, J. P.. Neuve tecnica para la investigacibn radiblogxa de las glhndulaa salivales, Semana mBd. 32: 41, 1925. 3. Carl&en, D. B.: Lipiodol Injection in Duct of Salivary Glands, Acta radiol. 6: 220, 1926. 4. Jacobovici, J., Poplitzka, W., and Albu, L.: Sialographie, Presse m6d. 34: 1188, 1926. 5. Wiskovsky. B.: Sialodochografie, Zentralbl. Hals-, Nasen-u. Ohrenh. 8: 320, 1926. 6. Payne, R.” ‘T.: Recurrent Pyoge&c Parotitis, La&et 224: 348, 1933. 7. Blady, J. V., and Hacker, A. F.: Sialography, Its Technique and Application in Roentgen Study of Neoplasms of the Parotid Gland, Surg. Gynec. & Obst. 67: 777, 1938. 8. Mandel, L., and Baurmash, H.: Pathologic Changes From Sialographie Media: Report of a Case, J. Oral Surg., Anesth. & Hosp. D. Serv. 20: 341, 1962. Substances on Salivary Glands 9. Epsteen, C. M., and Bendix, R.: Effects of Non-volatile in Sialography, Plast. & Reconstruct. Surg. 13: 299, 1954. 10. Thackray, A. C.: Sialectasis, Arch. Middlesex Hosp. 5: 3, 1955. 11. Sieard. J. A.. and Forestier. J.: Iodized oil as a Contrast Medium in Radioscouv,1. I B;ll. et &Em. Sot. m6d. l&p. Paris 46: 463, 1922. 12. Ollerenshaw. R. G. W., and Rose, S. S.: Radiological Diagnosis of Salivary Glantl Disease, Brit. J. R$diol. 24: 538, 1951. 13. Anderson, D. H.: Cystic Fibrosis of the Pancreas. In Brennemann, J., Mcauarrie, I., and Kelly, V.: Practice of Pediatrics, Hagerstown, Md., 1960, W. F. Prior Company, voi. I, chap. 29 A, p. 4. Svndrome, a General Disease, Brit. J. 14. Morean. A. D.. and Raven. R. W.: Sjogen’s ” ;. -s&g. 40: i54, 1932. ’ 15. Ashley, R. E.: Salivary Gland Diseases. In Coates, G.! Schenk, H., and Miller, M. V.: Otolaryngology, Hagerstown, Md., 1955, W. F. Prior Company, vol. IV, chap. 9, p. 14. 16. Winsten. J., Gould, D., and Ward, 0. E.: ‘Sialography, Rurg. Gynec. 8: Obst. 102: 315, 1956. H.: Chronic Parotitis, ORAL SIXG., ORAL MED. & OKAT, 17. Mandel, L., and Baurmash, PATH.
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Phrslologic
Pathology
of Megacolon.
Ann.
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