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Cystadenoma of the pancreas
CYSTADENOMA A.
A.
OF THE PANCREAS MOZAN, M.D.
Chicago, Illinois HE pancreas, with its pathological probIems, &II remains recessed and to a T large degree unnoticed behind the cloak of the stomach, Iesser omentum and adnexal viscera.” These are the words of Dr. AIexander Brunschwig. The reviewer finds not onIy a maze of conflicting histopathoIogic interpretations, differences of CIassification and meager and inconsistent descriptions but aIso disagreement as to what constitutes maIignant tendency or change. Some authors in their pathoIogic descriptions have used such terms as cystadenoma, muItiIocuIar cysts, proIiferative cysts and true pancreatic cysts either synonymousIy or interchangeabIy in discussing the same Iesions. Etiology. Christian Harms36 in 1932 aIIudes to Priesel’s’l concept as to the genesis of these tumors stating that he attributes these growths to a retention of secretion with subsequent abnorm&I proIiferations of the smaIIer ducts and pressure atrophy of the intervening acinar tissue. Hans Schmidt% attributes the origin to an accessory pancreas stating that the growth starts in a congenita1 anomaIy; others have suggested dispIaced portions of the primitive urogenita1 foId, mesonephros and primordia of the aIimentary canal. PrieseI makes the additiona statement that a number of authors believe cystadenoma arises in isoIated, aberrant pancreatic tissue. ArchibaId40 considers them true tumors arising by proIiferation from parenchyma1 ceIIs. Judd41 states that they form Iike the cystadenoma of the thyroid or ovary and are papiIIary ingrowths. Carter and SIattery18 concIude that those tumors with ceils resembIing pancreatic tissue originate from aIimentary cana primordia whereas to cystadenomas bearing no resembIance pancreatic tissue probabIy arise from the misplaced genita1 ceIIs of the urogenita1 foId or the mesonephros. Trauma, which is a factor in the development of the hematic or pseudocyst, cannot be considered here. It has been disproven that infiammation plays any incipient role in the growth of the cystadenoma. To concIude, “
204
IittIe is known of the etioIogy of this condition and its rarity of occurrence is a factor in Iimitation of any proIonged thorough research. Classijication. Mahorner and Mattson62 after a carefu1 study of 108 cases of cysts of the pancreas, twenty of which were necropsy studies, cIassifIed the cysts on an etioIogic basis as foIIows: (I) cysts resulting from defective deveIopment (cysts among infants, cysts associated with poIycystic disease of the kidney, dermoid cysts and incIusion cysts); (2) cysts resuIting from trauma (pseudocysts, hematic cysts); (3) retention cysts; (4) neopIastic cysts (cystadenoma, cystadenocarcinoma, teratomatous cysts) ; and (3) cysts resuIting from parasites. Brunschwig13 considers the cystadenoma as a Iesion occupying a midway position between soIid benign tumors and true cysts of the pancreas, but he further modified his position by saying, “This statement shouId not be construed to indicate that a11 soIid benign epitheIiomas (except isIet tumors) become cysts and that cystadenomas represent a stage in this metamorphosis.” Rabinovitch and Pines76 aIso refer to cystadenomas as true pancreatic tumors arising from the parenchyma of the organ. The oIder authors7g cIassified cysts of the pancreas as foIIows: (I) retention cysts, (2) proIiferative cysts, (3) congenita1 cystic disease, (4) hydatid cysts and (3) pseudocysts. Cystadenoma of the pancreas is markedIy rare in occurrence, and we find a variance as to its reported incidence. Patbology. GrossIy, cystadenomas are rounded, neopIastic masses with a coarsely lobulated surface. They vary from the size of a cherry to tumors as Iarge as a footbaII.73 They usuaIIy have a definite semi-translucent, fibrous-like, tense capsuIe. They exhibit a tense, hard, noduIar consistency to paIpation. Their gross appearance may be Iikened to a mass of gIass marbIes set in a dense, rubbery substance. Brunschwig Iikens them to a cIuster of grapes. These IocuIes or cyst spaces combine in a honeycomb and are numerous, American
Journal
of Surgery
Mozan-Cystadenoma varying in size from microscopic to severa centimeters in diameter. On section the fresh surfaces appear coarsely porous, and as the cut cyst spaces empty, the surface takes on a sponge-like appearance. In some cases there may be one, two or more spaces, Iarger than the rest, of a relativeIy sohd consistency. The cyst spaces are liIIed by cIear, or purpIish fluid with a yeIIow, brownish variabIe viscosity. The contents may be thin, turbid, hemorrhagic, viscid, mucoid clear, or gelatinous. The intervening librous stroma also varies in amount. There were nine cases reported in the Iiterature in which the stroma contained depositions of calcium. Hyalinization may occur, causing the resembIance to corpus aIbicans of the ovary. Smooth muscIe fibers may be present.45*67*71v104The tumors usuaIIy are we11 encapsuIated and consequentIy aIthough infrequently grow by expansion there is no encapsuIation but onIy a thin Iine of demarcation. The capsuIe is coursed by Iarge veins and arteries. OccasionaIIy (and it is here that opinions among pathoIogists differ) the tumors seem to inliltrate the adnexa1 structures and surrounding pancreatic parenchyma. Beust (in Brunschwig13) reports an infiltration through the duodena1 muscuIaris to the mucosa. Brunschwig aIso reports a case of a tumor mass measuring 7 cm. in diameter occupying the head of the pancreas and grossly infiItrating the waI1 of the duodenum. The common practice has been to classify such papiIIary cystadenomas of the pancreas as benign if there is a Iack of direct microscopic evidence of maIignancy. Dockerty (quoted by Benson and Gordon5) beIieves that in the presence of papiIIary projections, even though the microscopic criteria for malignancy are minimaI, they shouId be cIassilied as maIignant. SofranoD’ in 1906 reported a porta vein embedded in the cyst waI1 but with no infdtration of the vein wall by the tumor mass. This type of encroachment is unlike and apart from extension by inhltration. It is this characteristic of the cystadenoma of the pancreas, the ability to engulf with ameboid-like extension around adnexa1 organs and structures, to which this article wishes to caI1 particuIar attention. This type of extension and growth is compatible with benignancy whereas the infiltrative type may be questioned by some pathoIogists as a stage of premalignancy aIthough there is a lack of evidence of any February,
1951
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203
metastases. GrossIy, the cystadenoma does not attain the Iarger size of other pancreatic cysts. On section it is usuaIIy muItiIocuIar whereas the pseudocyst which is the most common among the pancreatic cysts is usuaIIy unilocular or bi1ocuIar. Characteristically the cysts are lined by a singIe Iayer of epitheIium. OccasionalIy there is a papillary-like piling up. The ceIIs vary from low to taI1, cIoseIy aligned, coIumnar epitheIium. They may be cylindrical simiIar to those of the pancreatic ducts but infrequentIy are cuboidal or flattened. The increased tension of the fluid causes the flattening of the ceIIs which may get so extreme as to cause a resembIance to Iymphoid ceIIs or endothelial cells. Diagnosis of Iymphangioma, therefore, has been made in some instances in the past. E. W. Thurston, in examining the photographs on the case reported herein, calted the author’s attention to this fact because of certain sections that had Iymphangioma-Iike or Iymphosarcoma-like appearance. The cytopIasm of the ceIIs is cIear. NucIei are basal in position, certain portions show single layer epithelium whereas other sections of the same specimen may show piIed up epithelium in the papilIary foIds. It was Gruber’sa5 contention that this indicated a malignant tendency. The stroma. between the cyst waI1s is variable in thickness and usually contains a thin layer of Ioose connective tissue. However, in many instances hyaIinization occurs in connective tissue septa. OccasionaIly smooth muscle fibers may be found. There were nine cases reported containing caIcification in the stroma. Harms35 was the first to note this characteristic of cystadenoma. Histopathologic differentiation of cystadenoma, cystadenocarcinoma, and multilocular cysts of the pancreas becomes at times quite compIex. Brunschwig and others caI1 attention to this difficulty. GeneraIIy speaking, nevertheless, the cyst spaces of the multilocular cyst are larger and scarcer whiIe in the cystadenoma they are smaIIer and multitudinous. After eliminating the malignant tumors and segregating them the present review discIoses a residuum of fifty-six cases of benign cystadenoma. Benignancy, a criterion in the differentiation, was adhered to rigidIy. Seven of these cases could not be differentiated from muItiIocuIar cyst and polycystic disease of the pancreas. These seven cases were therefore subtracted from the total of fifty-six cases
206
Mozan-Cystadenoma
Ieaving a baIance of forty-nine benign cystadenomas up to the present writing. Symptoms and Findings. The fact that lesions were found incidentaIIy at necropsy in patients who died from other causes and without any history of pancreatic disease indicates the insidiousness of the cystadenoma and deficiency of prodromas. UsuaIIy a paIpabIe tumor mass in the epigastric region is the first recognizabIe sign of the Iesion. These neopIasms may become Iarge enough to be discovered inadvertentIy by the patient or by the examining physician during the routine course of examination. Very often it is asymptomatic and painless. The symptomatoIogy usuaIIy is that of any variety of pancreatic cyst. Symptoms depend upon the invoIvement or encroachment, whether compIeteIy circumferentiaIIy or as an infringement, upon the Iumen of the surrounding viscera due to pressure or enguIfment of the growing tumor. The symptoms which are compIained of are anorexia, epigastric discomfort, (and in the Iarge tumors) premature satiation nausea, vomiting, beIching and gaseous distention. Jaundice was encountered in three cases.3g~72,g1 This was due to the fact that the cystadenoma invoIved the head of the pancreas adjacent to the common bile duct. Sharp, darting pains in the abdomen may occasionaIIy be observed prior to discovery of the mass. Pain when it occurs wiI1 vary in intensity and Iocation with the Iocation of the cyst and the organs and structures impinged upon by the tumor. Pain may be referred to the back and it may occur anywhere in the abdomen aIthough usuaIIy it is confined to the upper hypochondrium and most often in the Ieft upper quadrant. Severity and quaIity of the pain varies from a distensive sensation or a duI1, dyspeptic, regurgitant ache to a steady tugging sensation and even to a severe, shooting, cramp-like radiation. If the tumor Iies in the body, neck or head and overIies the Iarge vesseIs, a tremuIous, thriII-like, reguIar throb which is synchronous with the puIse can be ascertained by paIpation; if the patient is thin and the this can be observed tumor protuberant, visually. The nausea and vomiting, if present, together with the earIy satiation and chronic wiI1 of necessity be dyspeptic symptoms accompanied with a Ioss of weight and weakness due to interference with nutrition. Mobility of the mass is dependent upon the
of Pancreas degree of embedment in the pancreas and its Iocation in the pancreas. Those in the body and tai1 may be quite mobiIe. Deep embedment in the pancreas causes limitation of movement. Adherence by fibrous attachment or through enguIfment of other adjacent organs wiI1 aIso curtai1 movabiIity. TABLE LOCATION
OF
THE
I
THIRTY-THREE
Head, Neck. Body.
................... ................... ................... TaiI .....................
RECORDED
-
SITES
No. of Cases
Per cent
9 3 9 12
27 9 27 36
-
-L
Thirty-three case reports Iisted the site of attachment of the tumor mass to pancreas; these assumed the distribution and arrangement found in TabIe I. In the Iiterature severa patients were cognizant of a tumor mass in the epigastrium for a Iong time before reporting to the surgeon. Some of these patients feIt an intra-abdomina1 movement of the mass upon rotation of the body in the supine position. In those cases in which the cystadenoma was found in the head or neck of the pancreas and in the rarer event when attachment or encroachment upon the porta or other Iarge vesseIs occurred, a distinct puIsation was compIained of or couId be elicited by paIpatory traction on the mass. An additional unreported finding of this Iesion is the ascites and transient ankIe edema found in the author’s case. This condition is a direct resuItant of the extrahepatic porta bed bIock syndrome21 produced by the partia1 occIusion and pinching off of the portal, superior mesenteric and spIenic veins and inferior vena cava by the movement of the enguIfing tumor mass present. This can be easiIy seen in the accompanying drawing of the tumor mass. The diagnosis rests to a Iarge extent on three important findings, nameIy, (I) presence of a paIpabIe intra-abdomina1 tumor usuaIIy in upper Ieft hypochondrium (occurred in fortysix of the fifty-six cases reviewed) ; (z) dyspeptic distress (occurred in forty-one of the fifty-six cases) and (3) pain, variabIe in character (occurred in thirty-two of the fifty-six cases). (Waiters and CIeveIand’OO report pain American
Journal
of Surgery
Mozan-Cystadenoma in over 85 per cent of their 139 patients with pancreatic Cysts.) It may be mentioned that the Iaboratory offers IittIe or no assistance and in the event there is an interference with externa1 secretion, the then positive Iaboratory findings tend to lead the examiner further away from the correct conclusion. The extent of assistance by the laboratory chiefly lies in ruIing out such other conditions as adenoma, carcinoma and pancreatic abscess. The isIet adenoma patient would, of course, present WhippIe’s triad with a h,ypoglycemia beIow $0 mg. per cent. Blood amylase or Iipase and blood sugar usuaIIy are found at norma IeveIs. The resuItant degree of malnutrition and loss of weight is hardIy ever severe enough to affect aIbumin-gIobuIin ratio or total protein IeveIs. BIood, urine and stoo1 analyses provide Iittle if any specific aid in the diagnosis of pancreatic cyst or cystadenoma. Laboratory investigation is only important in negating and eIiminating other pathoIogic disorders that must be considered tentativeIy in arriving at a fina deduction of pancreatic cyst or benign tumor. Roentgen Examination. In its growth the cystadenoma as a ruIe displaces either the stomach or coIon. If the growth starts on the superior portion of the pancreas, it most probably will present in the Iesser omenta1 cavity pushing the stomach down and forward and causing a buIge through the gastrohepatic ligament. If the mass finds root in the inferior pancreatic border, the coIon wiI1 probabIy be displaced and the tumor wiI1 be covered by the gastrocoIic omentum. An extension through the layers of the transverse mesocoIon or retroperitonealIy is found infrequentIy. This is one lesion, as shown by a perusa1 of former literature, that taxes and exacts the fuI1 and cooperative use of a11 avaiIabIe diagnostic armamentarium at our disposa1. The roIe that x-ray assumes in aiding the differentiation of cystadenoma of the pancreas from other tumor masses of the region depends soIeIy upon the thoroughness and zealousness of the examining roentgenoIogist. There are two main types of tumor distinguishabIe in the pancreas roentgenologically, namely, benign cysts and cancers. The cysts because of their usuaIIy Iarge growth produce signs that are more pronounced than those of cancer of the pancreas. Since the benign cysts grow expansiveIy and ev&Iy in a11 directions, F&??%UrJ’,
195 I
of
Pancreas
207
they wiI1 present smooth, rounded outiines which is in direct contrast to the irreguIar growth in variabIe directions of the maIignant growths causing an irreguIar outline. Cystic tumors are usuaIIy movabIe whiIe the cancerous tumors are usuaIIy fixed to adjacent structures producing anguIar, rigid contours. Some tumors can be recognized by distinct differences in the shadows they cast on the pIain fiIm: shadows simiIar to the kidney in contrast to the retroperitonea1 fat. This abnorma1 shadou on the pIain film may be the first evidence of a tumor in this vicinity. Ehrmann and Eisler, quoted by Borak, have emphasized its importance. Changes in the stomach and duodenum due to their cIose reIationship to the pancreas occur as a direct resuIt of three main types of comencroachment, nameIy, dispIacement, pression and invasion. Displacement. Tumors IocaIized in the head of the pancreas, if Iarge enough, produce the characteristic horseshoe widening of the duodena curve. The antrum of the stomach is dispIaced forward. Tumors of the neck and isthmus of the pancreas cause an upward dispIacement of the stomach. Tumors of the body of the pancreas cause a forward or a downward dispIacement of the stomach. Tumors in the tai1 of the pancreas will displace the stomach to the right.‘O Compression. Likewise, compression can cause pronounced derangements, especialIy that due to benign growth, such as incompIete obstruction, a constant fiIIing defect of the duodenum, proIongation of evacuation time and distortions as the hourgIass stomach. It may simuIate HIing defects which however can be differentiated by ff uoroscopic observation during positiona changes of the patient. Invasion. This type of growth is most often seen in the maIignant tumor and very rareIy in the benign. Because x-ray is the onIy heIpfu1 examination that can be reIied upon for any assistance in deriving our diagnosis, it should be stressed that the surgeon shouId insist on a most compIete and thorough investigative effort on the part of the roentgenoIogist. Evidence obtained from a review of the cIinica1 history and the presence of a tirm, rounded, upper abdomina1 mass, coupIed with roentgenographic findings of pressure distortion upon neighboring organs, especiaIIy the stomach and coIon, suggests usuaIIy pancreatic cyst or tumor. The other conditions
208
Mozan-Cystadenoma
that must be considered and differentiated are mahgnant tumors, retroperitonea1 Iymph nodes, retroperitonea1 cysts, Iipomas and mesenteric cysts, spIenomegaIy, cysts of the kidney and pyonephrotic kidney, aneurysms of abdomina1 aorta and hydrops of the gaIIbIadder. CASE
REPORT
On October 2, 1946, the patient compIained of feeling a Iump in the stomach. She had been perfectIy we11 up to her present iIIness except for occasiona epigastric distress after eating fatty, fried foods. About August, 1946, she became conscious of a Iump in the epigastrium. She stated she was conscious of an intermittent puIsation in this mass and that she feIt rather certain that it was free and movabIe. Whenever she wouId turn in bed it moved about. LateIy, she experienced a sIight tug substernaIIy on movement of the mass. Associated with this there was an obvious, rapid increase in size producing a visibIe swelIing in the epigastrium. With movement it wouId roII into the Ieft upper quadrant beneath the Costa1 margin. She was abIe to bring the mass into the epigastrium at wiI1 mereIy by rotating her trunk. In the Iast two weeks the puIsating and throbbing feeIing of the mass became quite pronounced. Concomitant with this increased size of the epigastric mass she noticed that she attained earIier satiation and in the past seven weeks had Iost about 345 pounds. There was a transient type of edema of both Iegs which wouId disappear when the patient was recumbent. There were no other symptoms. Upon further interrogation the patient stated the puIsation and throbbing were first noticed as far back as February, 1946. Her appetite was aIways good but the patient recently noticed an earIier sense of fuIIness and satiation. SeIective dyspepsia had been noticed, however, to fat and fried foods which evoked a rather marked distention and beIching. Nausea was slight, but vomiting never occurred. There had been transient edema of both ankIes for the past seven to eight weeks which wouId disappear on Iying down. EIectrocardiogram studies were negative. A searching history with the thought of a possible traumatic cyst was taken with reference to any abdomina1 injury, but a11 information elicited was negative. PhysicaI examination reveaIed a we11 deveIoped white, American-born femaIe, about
of Pancreas
sixty-five years oId, not acuteIy III and free of pain. There was a mass fIIIing the epigastrium, substernaIIy, noduIar and firm in consistency. This mass which approximated the size of a grapefruit was reIativeIy fixed and puIsating. The thriI1 could be feIt through the abdomina1 waI1 by pIacing the hand directIy over the mass or by dispIacing the mass and retaining a stationary position with tension. SIight tenderness was eIicited by movement of the tumor or by traction on it. The movement of the tumor from side to side due to rotation of patient whiIe Iying down caused no pain, only cognizance of the changing position by the patient. There was no other tenderness or rigidity. From an x-ray examination a11 findings were negative except for a fairIy Iarge stone in the fundus of the gaIIbIadder and a Iarge paIpabIe mass within the ring of the Iesser curvature which is not movabIe and apparentIy did not arise from the stomach. The character of the mass was not determined. From the ffuoroscopic examination we may infer that the mass may have its origin in the Iesser omenta1 cavity. There was a forward dispIacement of the pyIoric end with a widening of thegastrovertebral interva1. In view of the aforementioned findings it was reasoned that the intra-abdomina1 mass was most probabIy of pancreatic origin. Mesenteric cyst, retroperitonea1 tumor and gastric maIignancy were aIso considered. Operative intervention was decided upon. On October 29, 1946, a mid-epigastric paramedian incision was made. A Iarge tumor mass presented itself through a thinIy stretched hepatogastric omentum. The Iatter was spIit and the Ieft gastric artery was sacrificed and Iigated to aIIow for a sufficient aperture and access to the tumor. The presenting mass proved to be a muItiIocuIar cystadenoma, hard and non-compressibIe, and about the diameter of a Iarge grapefruit. This fiIIed the Iesser omenta1 cavity causing a dispIacement of the stomach anteriorIy and inferiorIy. UtiIizing sharp and bIunt gauze dissection the mass was mobiIZed down to a rather broad base, about 233 inches to 3 inches in width. At this stage it was noticed that the spIenic at its confIuence with the superior mesenteric vein and also the adjacent portion of the porta vein were enmeshed in the substance of the Iowermost portion of the cystadenoma. The
American Journal of Surgery
Mozan-Cystadenoma neck of the pancreas from which the tumor arose and the head of the pancreas were both compressed and to a degree disIodged by the mass. The exact origin of the tumor was at the posterosuperior surface of the neck of the pancreas. It was decided to remove the uppermost twothirds of the mass by a transverse section. Figure I shows the tumor mass at this stage with the pertinent anatomic structures involved. The invoIved region is sketched with portions of the stomach, duodenum and removed to illustrate better the pancreas invoIvement of the splenic, superior mesenteric and portal vein and the superior mesenteric artery by the tumor. The drawing is seIfexplanatory otherwise. The remainder was then sIowIy and carefuIIy morceIIated away from the invoIved bIood vessels. The discovery that a bIunt probe could readiIy Ioosen the bIood vessel waIIs from the engulfing tumor mass was a gratefu1 finding. At no point did we note any infiltration of vascuIar waI1 by the cystadenomn. The remaining remnant of the tumor attached to the posterosuperior border of the neck of the pancreas had a base about I inch in diameter which disengaged rather easiIy from the pancreatic substance. The site of attachment needed no suturing as no bIeeding occurred. The spIeen was enIarged moderateIy due to the encroachment of the spIenic vein. The gastrohepatic omentum was rent in the approximated by catgut. perChoIecystostomy was subsequentIy formed and an oIive-shaped stone was removed. GaIIbIadder was cIosed over a catheter. Two Penrose drains were inserted, one into the lesser omental cavity through the foramen of Winslow and one to Morrison’s pouch. Both drains and catheter were carried exterior through stab wound in the IateraI Ilank. The abdomen was closed with chromic (Finney) and three tension nyIon sutures. Dermic graft was used on the skin. The pathoIogic report was as foIIows: grossly, the specimen consisted of three pieces 14 by 13 by 7 cm. in aggregate measuring composed of a Iarger mass with bosseIated surfaces attached to which were adipose tags. This larger mass was we11 encapsulated. The coIor was grey with darker brown areas. The surfaces made on cutting were spongy, cystic tissue, the cysts fiIIed with cIear grey Iiquid.
February, 195 I
209
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FIG.
I.
The smaI1 pieces resembIe the Iarger but were more fibrous. An olive-shaped smooth caIcuIus 23 mm. in Iength and diameter was present. (Figs. 2 and 3.) MicroscopicaIIy, the hematoxyIin-eosin stained sections of the formaIin fixed tissue reveaIed a narrow rim of normal-appearing pancreatic tissue. The remainder consisted of a Iarger amount of connective tissue surrounding many cystic spaces both moderateIy Iarge and smaI1. These were Iined with a low stratified, cuboida1 epitheIium in most instances and the Iumina were empty or contained fresh-appearing erythrocytes and pink-staining materia1 (secretion?). In addition to the simpIe cystic spaces there were others in which papiIIary projections were seen arising from the cyst Iinings. These were Iikewise covered with Iow stratified, smaI1, cuboida1 ceIIs with clear cytopIasm. In the remaining connective tissue were many coIIections of round cells, many fresh-appearing erythrocytes, hemosiderin and occasiona nerve fibers. Further cIose search revealed occasiona smaI1 ova1 or round coIIections of ceIIs with deepIy staining nucIei and light granuIar cytoplasm. The significance of these was conjectura1 but the possibiIity that they were ectopic isIets of Langerhans was considered. Three microphotographs are incIuded showing certain interesting aspects of the micropathoIogy. (Figs. 1, 3 and 6.) The patient was graduaIIy made ambulatory by the third postoperative day. Very IittIe drainage was noticed on the dressings after the fifth day. The first Penrose drain was
Mozan-Cystadenoma
of Pancreas
2 3 FIG. 2. This presents the gross specimens directIy after removal, cystadenoma choIeIith. FIG. 3. This shows the sea-sponge appearance of the transected cystadenoma.
and
4 5 FIG. 4. Pancreatic glands with adjacent fibro-adenomatous tissue. FIG. 5. Adenomatous area showing papiIIary character of growth. X 60.
FIG. 6. Structure of cystic areas. X 60.
removed on the sixth day and the second on the eighth day. RemovaI of the gaIIbIadder drain was effected on the eIeventh day which was two days after the patient’s discharge from the hospitai. This iiiustrates the uneventfui nature of the patient’s recovery. The sinus
tract Ieft by the drains continued discharging a biie-stained ffuid for nine more days before occluding compieteiy. Since convaIescence the patient has been recaiied for further cIinica1 observation and has been foiiowed up to the present time. She has gained g pounds, has no compiaints and one year and six months postoperativeiy her genera1 condition is excelient. The transient edema has not recurred since surgery. There is no evidence of recurring tumefaction anywhere intra-abdominaiiy. Her dyspeptic symptoms have entireiy subsided and she states she can now compIete a mea1 without a feeiing of overstuffring the stomach. The blood sugar, serum Iipase and differentiai count are within normai Iimits. SURGICAL
CONSIDERATIONS
Yamauchi is foremost stressing the importance
American
among the authors in of compiete extirpa-
Journal
of Surgery
Mozan-Cystadenoma tion no matter what the offending tumefaction may be. Sound surgica1 judgment demands compIete abIation of such Iesions. Drainage with or without partia1 excision was accompIished in sixteen cases. Of these there were five benign cystadenomas and one borderIine Iesion. One or more previous drainages had aIready been performed on eIeven of these cases which incIuded the five benign cystadenomas. Forty-two per cent of benign cystadenoma had been subjected to repeated drainage at the end of which procedure permanent cure was stiI1 doubtfu1 since none of the cases showed foIIow-up beyond one or two years. Extirpations were generaIIy accomplished in those instances in which the growth was smaI1, when it possessed a narrow base or when the tumor was found arising from the tail. A few instances are recorded in which grow-ths with broad bases or growths Iocated in the head and other inaccessibIe portions of the pancreas were compIeteIy extirpated. In a number of other cases the tumor was removed after much dif?icuIty depending upon the necessity of Iigating Iarge vesseIs and removing dense, fibrous attachments. In six cases the pancreas had to be partiaIIy resected. In the management of the malignant tumors in the head of the pancreas Brunschwig has elaborated technics and had described them in comprehensive detaiI. One of the stages of his operation \vas suggested by Marogna56 who demonstrated the feasibiIity of suturing the pancreas stump directIy into the bowe1 as an outIet of pancreatic juice after resection of the head of the pancreas. AcknowIedgment of the vascular supply to the pancreas is important. Of the anomalies the one to cause most mischief if overlooked is the abnorma1 origin of the middle coIic artery from the gastroduodenaI.*O The incidence of such an unusua1 origin is 8 per cent. An inadvertent or accidenta Iigation here wouId most certainly resuIt in transverse coIon necrosis. CompIete extirpation of certain broad-based cystadenomas and cysts, due to their Iocation, was considered by many surgeons to be hazardous because of the invoIved risk to the patient. With each new surgica1 intervention and experience in this region, coupIed with experimenta physioIogic work, a practica1 surgical technic has been evolved. Thus the hazard seems now to have been surmounted
February, I 951
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21 I
and this is very we11 iIIustrated in the comparative mortaIity statistics found in Table II. This review Iists forty-nine cases of benign cystadenoma of which thirty-one cases were expIicit as to the type of surgica1 intervention. Twenty-nine of the Iatter showed a postoperative follow-up in addition. TABLE II METHODS
OF
SURGICAL
TREATMENT
MORTALITY
Type of Operation
AND
COMPARATIVE
RATES
Recovered
I I Abdominal expIoratory 0nIy PartiaI excision
2
* ExpIoratory and biopsy. 3 Complete extirpation. 22 (Case herein incIuded)
/
’
~3linrcIcordedl
I
2
0
0 ‘4
cent)
IO0 ~
1: 1 .‘3” I ) 3 / 100
~ 3 ~ 13.6 I
I
Progress in surgery of the pancreas is beset with many hindrances two of which are outstanding. First is the inabiIity to prove the function, position and pathoIogic changes in the organ by any means at our disposa1 with any ease or assurance of correctness. The other is the inaccessibiIity and recession of the organ which make it prone to be disregarded by the surgeon in the performance of other abdomina1 surgery. SUMMARY
A complete extirpation of a benign cystadenoma of the pancreas was reported and a series of fifty-six cases coIIected from the medical Iiterature reviewed. 2. Attention is directed to a heretofore unIisted finding of transient edema found in this case. 3. PathoIogicaIIy, cystadenoma of the pancreas is a muItiIocuIar, cystic, adenomatous and neoplastic tumor, benign in character. 4. In its growth and extension it encompasses surrounding structures and organs without infiItration thus aIlowing facile dissection. Extension with infiltration is a rarity; encapsuIation is the ruIe. I.
Mozan-Cystadenoma
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3. CIinicaIIy, cystadenoma is characterized by an insidious sIow growth. Later a paIpabIe tumefaction deveIops and abdomina1 pain in the upper epigastrium, dyspeptic symptoms and weight 10s~. 6. Roentgen examination (utiIizing IateraI pIate technic) has proved a most vaIuabIe adjunct in diagnosis. 7. Eight of the fifty-six cases had associated diabetes meIIitus. 8. Ratio of occurrence in the sexes was seven femaIes to one maIe. 9. CompIete surgica1 extirpation is the method of choice in the opinion of most authorities and proffers Iowest mortaIity (13.6 per cent). IO. A pIea is made to the surgeon to keep in mind routine examination of the pancreas by paIpation in the course of surgery of the region as in doing gaIIbIadder, common biIe duct, gastric and duodena1 work. This procedure wiI1 afford him the opportunity to recognize the norma fee1 of the pancreas and thereby aIso insure the discIosure of a great amount of pathologic disorder that wouId otherwise remain hidden. Acknowledgment: I wish to thank Dr. E. W. Thurston, our pathologist, for his whoIe-hearted cooperation in the study of tissues and slides of the case and for his heIp in interpreting the transIated pathoIogic descriptions. I am aIso gratefu1 to Drs. T. M. Larkowski, G. ApfeIbach and N. Casciato for their assistance in transIating the foreign articIes. REFERENCES
cysts. Soutb. Med. Ed Surg., 100: 57-61, 1938. BABCOCK, W. W. SurgicaI affections of pancreas. S. Clin. Nortb Ame&, 15: 101-r ‘5, 1935. BALDWIN, J. F. Cystic tumors of the pancreas. Ohio State M. J., 4: 547, 1908. BELOFF, J. T. Cystadenoma; case successfuIIy treated by surgica1 extirpation. J. Mt. Sinai Hosp., 12: 817-820, 1945. Benson, R. E. and GORDON, W. Cystadenoma of the pancreas. Surgery, 21: 353-361, 1947. BERKMAN, J. M. and BANNICK, E. G. Huge muItiIocuIar cysts; 2 cases. Proc. Staff. Meet., Mayo Clin., 8: 185-188, 29, 1933. BEVAN, A. D. Discussion of pancreatic cysts and treatment employed for their relief. S. C&n. Nortb America, 3: 887-898, 1923. BOLAND, F. K. JR. Treatment of recurrent multiIocuIar cyst, case. Soutb Surgeon, I I : 126-131, ‘942. BOLT, R. F. A case of poIycystic disease of the pancreas. Brit. J. Surg., I: 142, 1913.
I. ANGEL, E. Pancreatic
2. 3. 4.
5. 6.
7.
8.
9.
of Pancreas examination. Radiology, 41: 170-180, 1943. BOWERS, R. F., LORD, J. W. JR. and MCSWAIN, B. Cystadenoma; 5 cases. Arch. Surg., 45: I I r-122, 1942. BOWMAN, H. A. H. Pancreatic cyst in Ieft hypochondrium-extirpated. Minnesota Med., 5: 697-706. 1922. BRUNSCHWIG, A. The Surgery of Pancreatic Tumors. St. Louis, 1942. C. V. Mosby Co. Radical Surgery in Advanced AbdominaI Cancer. Chicago, 1947. University of Chicago Press. BUCKSTEIN, J. New aid in diagnosis of tumor of head of pancreas. Surg., Gynec. ti Obst., 39:
IO. BORAK, J. Roentgen
I I.
12.
13.
14.
509, 1924.
15. CABOT CASE 27231. PapiIIary
adenocystoma of pancreas. New England J. Med., 224: 988-989,
1941. 16. CABOT CASE 27262. PapiIIary adenocystoma with hemorrhage. New England J. Med., 224: I I I21114, 1941. 17. CARLING, E. R. and HICKS, J. A. B. Cystadenoma-two cases and their probabIe reIationship to poIycystic conditions found in other viscera. Brit. J. Surg., 13: 238-246, 1925. 18. CARTER, R. F. and SLATTERY, L. Cystadenomata of pancreas. Am. J. Digest. Dis. ff Nutrition, 3: 705-707, 1936. 19. CASPER, M. Pancreatic cyst case; operation; recovery. Kentucky M. J., 28: 15-16, 1930. 20. CRAWFORD, F. B. Pancreatic cyst. Australian M. J., I: 552. 1926. 2 I. AARON, A. H. Cyclopedia of Medicine, Surgery and SpeciaIties, vol. I I. Philadelphia, 1942. F. A. Davis Co. 22. DAVEY, B. M. Surgery of pancreas. J. Michigan M. Sot., 20: 350, 1921. 23. DEAVER, J. B. Surgery of pancreas, principIes underlying. Boston M. +Y S. J., 176: 187, 1917. 24. DELAGENIERE, H. Le Mans Des Kystes gIanduIaires du Pancreas, une observation. Arch. prov. de cbir., 9: 209, 1900. Ibid., 15: 193, 1906. 25. DUNNING, L. H. Cystadenoma of the pancreas; removed by abdominal section. Am. J. Obst., pp. IOI-110, 1905. 26. EDLING. L. Zur Kenntniss der Cvstadenome des Pankreas. Vircbows Arch. j. path: Anat., 32: I IO, I905. 27. EINHORN, M. Pancreatic tumors malignant and benign. Am. J. M. SC., 172: 79G804, 1926. 28. FIESSINGER, N. and CATTAND, R. Cystadenoma of head of pancreas. Bull. et mkm. Sot. mkd. d. b8p. de Paris, 53: 497-504, 1929. zg. FINNEY, J. M. T. and FINNEY, J. M. T., JR. Resection of pancreas. Ann. Surg., 88: 584-592. 1928. 30. FRIEDENWOLD, J. and CULLEN, T. S. Pancreatic cysts, with report of seven cases. Am. J. M. SC., 172: 313-334, 1926. 3r. GILBRIDE, J. J. Tumors of pancreas; operations oerformed on twelve patients. J. A. M. A., 83: 984-989, 1924. 32. GOYENA, J. R. GIanduIar cyst in head of pancreas. Semanu m&d., I : 1258-1265, 1924.
American
Journal
of Surgery
Mozan-Cystadenoma 33. HABERER, H. Operation for benign solid tumor case. Arch. j. klin. cbir., 148: 398-403, 1927. 34. HADLEY, E. C. MuItiIocuIar cyst of the pancreas. Brit. M. J., I: 112, 1914. 35. HARMS, C. Cystadenoma; case. Centralbl. J. a&. Patb. u. patb. Anat., 55: 53-56, 1932. 36. HEYMANN, E. Ueber einen gutartigen, Pankreastumor. Deutscbe med. Wcbnscbr., 15: 48, 1924. 37. HICK, F. K. Pancreatic cysts. M. Clin. North America, 16: 555-563, 1932. 38. HUEPER, W. C. Pancreatic cystadenoma. Arch. Path. w Lab. Med., 5: 261-266, 1928. 39. JANES, R. M. Cystadenoma; 2 cases. Brit. J. Surg., 23: 809-815, 1936. 40. JEMERIN, E. E. and SAMUELS,N. A. Cystadenoma of pancreas. Ann. Surg., 127: 158-169, 1948. 41. JUDD, E. S. and WALTERS, W. Abstract of 1931 report of surgicaI procedures for lesions. Proc. Staff. Meet., Mayo Clin., 7: I IO-I 12, 1932. 42. KERR, A. A. Cysts and pseudo-cysts of the pancreas. Surg., Gynec. @ Obst., 27: 40, 1918. 43. KIBBY, S. U. Cystic tumor of tail of pancreas, case, J. Connecticut M. Sot., 2: 331-332, 1938. 44. KOONTZ, F. L. Cyst of pancreas. Kentucky M. J., 15: 166, ,917. 45. KORTE, W. Die chirurgischen Krankheiten und die Verletzungen des Pankreas. Deutscbe cbir., 45-D: 234, 1898. Zur BehandIung der Pankreascysten und Pseudocysten. Deutscbe med. Wcbnscbr., 30: 536, 191 I. 46. KUHN, H. P., SCHUTZ, C. B. and HELWIG, F. C. Cystadenoma; case. J. Missouri M. A., 27: 570-572, ‘930. 47. LANG, F. J. Uber einige GeschwuIstbiIdungen des Pankreas. V&bows Arch. f. path. Anat., 257: 235-248, 1925. 48. LEPOUTRE, C. and BUSSER, F. Cystadenoma. Bull. Assoc. franc. p. l’etude du cancer, 22: 214-220, 1933. 49. LOCKWOOD, C. E. Tumors of pancreas. J. A. M. A., 77: 155+, 1921. 50. LORENZ, H. Cystadenoma, vo1. III, p. 1282. Wiener GeseII, der Artzte, 192 I. 3 I. MAHORNER, H. R. Cysts of pancreas; etiology and pathology. Proc. Staff Meet., Mayo Clin., 5: 101-103, and 1930. 52. MAHORNER, H. R. and MATTSON, H. EtioIogy and pathoIogy of cysts of pancreas. Arch. Surg., 22: 1018-1033, 1931. 53. MAINGOT, RODNEY. Post-graduate Surgery. New York, 1938. Appleton-Century. 54. MALCOLM, J. D. A case of compIete removal of a multilocular cystic tumor of the pancreas. Lancer, I: 1676, 1906. 5s. MARIANI, C. Adenoma cistico de1 pancreas; asportazione parziaIe, marsupium deIIa resante parte; quarigione. Clin. cbir., I I : 800-810, 1903. 56. MAROGNA, P. Tumors and cysts of pancreas. Arch. ital. cbir., 7: 113-120, 1923; ab., J. A. hf. A., 81: 425, ‘923. 5;. RIATTSON, H., JUDD, E. S. and I\IAHoRxEa, H. R. Pancreatic cysts., forty-seven cases. Arch. Surg., 22: 838-849, 1931. 58. MCKECHNIE, R. E., JR. and PRIESTLEY, J. T. MuItiIocuIar polycystic tumor. Canad. M. A. J., 36: 592-593, 1937.
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59. MCWHORTER, G. L. Cysts of pancreas. Arch. Surg., I I : 619632, 1925. 60. MCWILLIAMS, C. A. True pancreatic cyst. S. Clin. Nortb America, 3: 439-442, 1923. 61. METZLER, F. Tumor, cystadenoma of pancreas. Arch. j. klin. cbir., 134: 772-779, 1925. 62. MEYER, L. J. Pancreas-resection of body and tai1, recovery. Australian M. J., I : 396, 1916. 63. NEEF, F. E., RHODENBURG, G. L. and RITTEH, S. I-I. Cystadenoma; remova through vertical incision in gastrohepatic omentum. Am. J. Surg., 46: 377-386, 1939. 64. NOORDENBAS, W. Extirpation of cystadenoma. Nederl. tijdscbr. o. geneesk., 2: 5546-5556, 1929. 65. OPIE, EUGENE, L. Disease of the Pancreas, Its Cause and Nature, p. 266. PhiIadeIphia, 1910. Lippincott. 66. PARENTI, G. C. Diffuse cystadenomatosis. Patbologica, 25: 165-169, 1933. 67. P~scuccr, L. M. Pancreatic cysts and Iithiasis; classification and incidence; report of pseudo cyst associated with disseminated parenchymal calcification. Am. J. Roentgenol, $2: 80-87, 1944. 68. PETTIKARI, V. Cystic adenoma and acute pancreatitis; case. Arch. ital. di anat. e istol. pat., 7: 469-483. 1936. 69. PORTIS, M. MuItiIocuIar cyst of pancreas. M. Clin. Nortb America, 8: 641-645, 1924. 70. PRIESEL. Beitrag zur PathoIogie der Bauchspeicheldruse. Frankfurt. Ztvbr. ,j. Path., 26: 453, 1922. 71. PRIMIIOSE,A. Cysts and pxudo cysts; report of a case of total extirpation by an extraperitoneal method. Surg., Gynec. @ Obst., 431-436, 1922. 72. PRIXLE, S. Cystadenoma of Pancreas, complete rcmova1. Brit. J. Surg., 13: 180-182, 1925. 73. QIXMBY, A. J. and QUIMBY, W’. A. Roentgen findings in upper right abdomen with special reference to duodenum, gaI1 bladder and pancreas, New York State J. Med., ro5: 821, ‘917. 74. RABINOVITCH, J. and PINES, B. Cysts of the pancreas. Arch. Surg., 45: 3, 1942. 75. RANSOHOFF, J. Cystic adenoma of the pancreas; extirpation. Tr. Ohio M. Sot., 4: 225-234, r9o1. 76. REEKE, TH. Beitr;ige zur PathoIogie des Pankreas. Frunkjurt. Ztscbr. j. Path., 40: 444-451, 1930. 77. RIVAIIOLA, R. A. Tumors of pancreas. Rev. Asoc. med. argent., 34: 166g-1701; J. A. M. A., 78: 1766, 1922. 78. ROBSON, A. W. and MOYNIHAN, B. G. Diseases of the Pancreas. PhiIadeIphia, 19o3. \Y. B. Saunders Co. 79. ROSE, B. T. Encysted adenoma of’ head of pancreas. Brit. J. Surg., 33: 93, 1945. 80. ROTH, H. Cysts of pancreas. Report of two cases. Am. J. Surg., 39: 257-261, 1925. 81. SACON, J. I., BURLANDO, A. and CAMAROTTA, A. GIanduIar cyst; cIinica1 and roentgen diagnosis; recovery of case foIIowing surgica1 intervention. Hosp., Argentina, 5: 21~232, 1934. 82. SANTY, P., MALLET, P. and CROIZAT, Guy and P. Polycystic tumors of the body of pancreas; Ieft pancreatectomy, case. Lyon cbir., 33: 689. 692, 1936. 83. SCHLEGEL, A. SurgicaI Diseases of Pancreas, Beitr. z. klin. cbir., 133: 562-582, 1925.
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84. SCHLONVOGT, E. Cystadenoma. Nowotwory, 6: 145-158, 1931. 85. SCHMIDT, HANS. Uber ein Adenom des Pankreas. Zentralbl. j. patb., 31: 491, Igzr. 86. SCHMIEDEN, V. and SEBENING, W. SurgicaI anatomy and methods. Arch. j. klin. Cbir., 148: 319-387, 1927; Surg., Gynec. fl Obst., 46: 735751. 1928. 87. Scorer, G. Adeno-cistoma papiIIefero de1 pancreas. Arch. per le SC. med., 1906, abstr., Centralbl. j. allg. Patb. u. patb. Anat., 18: 844, 1907. 88. SIMON. H. Cvstadenoma of nancreas. Beitr. z. klin: cbir., 1”34: 410-412, 192;. 89. SKIBNIEWSKI,T. Zystisches adenom des Pankreas. Polska gaz. lek., 5: 2, 1926. Adeno-cistoma DaniIIifero de1 00. SOFRANO. F.. pancreas. A&b. per. le SC.med., 30:~184, 1906. 91. SPARKMAN, J. R. Cyst of pancreas. J. Soutb Carolina M. A., 14: 5, 1918. gz. SPEESE, JOHN J. Cystadenoma of the pancreas with extension to the abdomina1 ten years after drainage of a pancreatic cyst. Pbiladelpbia Acad. Med. Am. Surg., 61: 759. 1915. 93. STILLMAN, A., JR. Surgery of pancreas at RooseveIt HospitaI from 1918 to 1928. Am. J. Surg., go : 58-64, 1929. 94. TAVERNARI, A. PapiIIiferous cystadenoma; case. Patbologica, 23 : 207-2 14, I g3 I. 95. TOCHOWICZ, L. MobiIity of tumors. Polska gaz. lek., 8: 641-642, Igzg.
of Pancreas 96. VANDKVENTER, H. Cysts of Pancreas with report of a case. J. Michigan M. Sot., 24: 586, 1925. 97. VAUGHAN, G. T. PapiIIary adenocystoma of pancreas. Virginia M. Montbly, 50: 811-912,
‘924. 98. WAGENSTEEN, 0. H. Pancreas cysts. Journal Lancet, 50: 219-224, 1930. 00. WALTERS. W. and CLEVELAND. W. H. SurnicaI Iesions ‘of pancreas, review. ‘Arch. Surg.,-42: 819-838, 1941. IOO. WHITE, H. Pancreatic cysts and pseudocysts. Guy’s Hosp. Rep., 54: 17, Igoo. IOI. WO~EL, P. Pathogenesis of cysts of pancreas. Arch. j. klin. Cbir., 140: 483-500, 1926. 102. WRIGHT, A. DICKSON. Post-graduate Surgery (Rodney Maingot), Chapt. I. 1938. AppIetonCentury. New York. 103. YAMANE. Beitroge zur Kenntnis der Pankreascvsten. Bern. 102 I. PauI. HauntbuchhandIuns. 104. YAMAUCHI, S. MuItiIocuIar pseudomucinous cystadenoma; report of case successfuIIy extirpated, with discussion of surgica1 treatment. Hawaii M. J., 3: 67-70, 1943. 10s. YANOWSKIY, S. M. Cystadenoma; case. Vestnik kbir., 45: 83-86, 1936. 106. YOUNG. E. L.. JR. MuItiIocuIar cvstadenoma. New England J.‘Med., 216: 334-339, 1937. 107. ZIEGLER, H. Excision of head of pancreas for carcinoma with studies of its bIood suppIy. Surg., Gynec. c!? Obst., 74: 137-145. 1842. LI
,
/
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.
American Journal of Surgery
A.
OF THE PANCREAS MOZAN, M.D.
Chicago, Illinois HE pancreas, with its pathological probIems, &II remains recessed and to a T large degree unnoticed behind the cloak of the stomach, Iesser omentum and adnexal viscera.” These are the words of Dr. AIexander Brunschwig. The reviewer finds not onIy a maze of conflicting histopathoIogic interpretations, differences of CIassification and meager and inconsistent descriptions but aIso disagreement as to what constitutes maIignant tendency or change. Some authors in their pathoIogic descriptions have used such terms as cystadenoma, muItiIocuIar cysts, proIiferative cysts and true pancreatic cysts either synonymousIy or interchangeabIy in discussing the same Iesions. Etiology. Christian Harms36 in 1932 aIIudes to Priesel’s’l concept as to the genesis of these tumors stating that he attributes these growths to a retention of secretion with subsequent abnorm&I proIiferations of the smaIIer ducts and pressure atrophy of the intervening acinar tissue. Hans Schmidt% attributes the origin to an accessory pancreas stating that the growth starts in a congenita1 anomaIy; others have suggested dispIaced portions of the primitive urogenita1 foId, mesonephros and primordia of the aIimentary canal. PrieseI makes the additiona statement that a number of authors believe cystadenoma arises in isoIated, aberrant pancreatic tissue. ArchibaId40 considers them true tumors arising by proIiferation from parenchyma1 ceIIs. Judd41 states that they form Iike the cystadenoma of the thyroid or ovary and are papiIIary ingrowths. Carter and SIattery18 concIude that those tumors with ceils resembIing pancreatic tissue originate from aIimentary cana primordia whereas to cystadenomas bearing no resembIance pancreatic tissue probabIy arise from the misplaced genita1 ceIIs of the urogenita1 foId or the mesonephros. Trauma, which is a factor in the development of the hematic or pseudocyst, cannot be considered here. It has been disproven that infiammation plays any incipient role in the growth of the cystadenoma. To concIude, “
204
IittIe is known of the etioIogy of this condition and its rarity of occurrence is a factor in Iimitation of any proIonged thorough research. Classijication. Mahorner and Mattson62 after a carefu1 study of 108 cases of cysts of the pancreas, twenty of which were necropsy studies, cIassifIed the cysts on an etioIogic basis as foIIows: (I) cysts resulting from defective deveIopment (cysts among infants, cysts associated with poIycystic disease of the kidney, dermoid cysts and incIusion cysts); (2) cysts resuIting from trauma (pseudocysts, hematic cysts); (3) retention cysts; (4) neopIastic cysts (cystadenoma, cystadenocarcinoma, teratomatous cysts) ; and (3) cysts resuIting from parasites. Brunschwig13 considers the cystadenoma as a Iesion occupying a midway position between soIid benign tumors and true cysts of the pancreas, but he further modified his position by saying, “This statement shouId not be construed to indicate that a11 soIid benign epitheIiomas (except isIet tumors) become cysts and that cystadenomas represent a stage in this metamorphosis.” Rabinovitch and Pines76 aIso refer to cystadenomas as true pancreatic tumors arising from the parenchyma of the organ. The oIder authors7g cIassified cysts of the pancreas as foIIows: (I) retention cysts, (2) proIiferative cysts, (3) congenita1 cystic disease, (4) hydatid cysts and (3) pseudocysts. Cystadenoma of the pancreas is markedIy rare in occurrence, and we find a variance as to its reported incidence. Patbology. GrossIy, cystadenomas are rounded, neopIastic masses with a coarsely lobulated surface. They vary from the size of a cherry to tumors as Iarge as a footbaII.73 They usuaIIy have a definite semi-translucent, fibrous-like, tense capsuIe. They exhibit a tense, hard, noduIar consistency to paIpation. Their gross appearance may be Iikened to a mass of gIass marbIes set in a dense, rubbery substance. Brunschwig Iikens them to a cIuster of grapes. These IocuIes or cyst spaces combine in a honeycomb and are numerous, American
Journal
of Surgery
Mozan-Cystadenoma varying in size from microscopic to severa centimeters in diameter. On section the fresh surfaces appear coarsely porous, and as the cut cyst spaces empty, the surface takes on a sponge-like appearance. In some cases there may be one, two or more spaces, Iarger than the rest, of a relativeIy sohd consistency. The cyst spaces are liIIed by cIear, or purpIish fluid with a yeIIow, brownish variabIe viscosity. The contents may be thin, turbid, hemorrhagic, viscid, mucoid clear, or gelatinous. The intervening librous stroma also varies in amount. There were nine cases reported in the Iiterature in which the stroma contained depositions of calcium. Hyalinization may occur, causing the resembIance to corpus aIbicans of the ovary. Smooth muscIe fibers may be present.45*67*71v104The tumors usuaIIy are we11 encapsuIated and consequentIy aIthough infrequently grow by expansion there is no encapsuIation but onIy a thin Iine of demarcation. The capsuIe is coursed by Iarge veins and arteries. OccasionaIIy (and it is here that opinions among pathoIogists differ) the tumors seem to inliltrate the adnexa1 structures and surrounding pancreatic parenchyma. Beust (in Brunschwig13) reports an infiltration through the duodena1 muscuIaris to the mucosa. Brunschwig aIso reports a case of a tumor mass measuring 7 cm. in diameter occupying the head of the pancreas and grossly infiItrating the waI1 of the duodenum. The common practice has been to classify such papiIIary cystadenomas of the pancreas as benign if there is a Iack of direct microscopic evidence of maIignancy. Dockerty (quoted by Benson and Gordon5) beIieves that in the presence of papiIIary projections, even though the microscopic criteria for malignancy are minimaI, they shouId be cIassilied as maIignant. SofranoD’ in 1906 reported a porta vein embedded in the cyst waI1 but with no infdtration of the vein wall by the tumor mass. This type of encroachment is unlike and apart from extension by inhltration. It is this characteristic of the cystadenoma of the pancreas, the ability to engulf with ameboid-like extension around adnexa1 organs and structures, to which this article wishes to caI1 particuIar attention. This type of extension and growth is compatible with benignancy whereas the infiltrative type may be questioned by some pathoIogists as a stage of premalignancy aIthough there is a lack of evidence of any February,
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metastases. GrossIy, the cystadenoma does not attain the Iarger size of other pancreatic cysts. On section it is usuaIIy muItiIocuIar whereas the pseudocyst which is the most common among the pancreatic cysts is usuaIIy unilocular or bi1ocuIar. Characteristically the cysts are lined by a singIe Iayer of epitheIium. OccasionalIy there is a papillary-like piling up. The ceIIs vary from low to taI1, cIoseIy aligned, coIumnar epitheIium. They may be cylindrical simiIar to those of the pancreatic ducts but infrequentIy are cuboidal or flattened. The increased tension of the fluid causes the flattening of the ceIIs which may get so extreme as to cause a resembIance to Iymphoid ceIIs or endothelial cells. Diagnosis of Iymphangioma, therefore, has been made in some instances in the past. E. W. Thurston, in examining the photographs on the case reported herein, calted the author’s attention to this fact because of certain sections that had Iymphangioma-Iike or Iymphosarcoma-like appearance. The cytopIasm of the ceIIs is cIear. NucIei are basal in position, certain portions show single layer epithelium whereas other sections of the same specimen may show piIed up epithelium in the papilIary foIds. It was Gruber’sa5 contention that this indicated a malignant tendency. The stroma. between the cyst waI1s is variable in thickness and usually contains a thin layer of Ioose connective tissue. However, in many instances hyaIinization occurs in connective tissue septa. OccasionaIly smooth muscle fibers may be found. There were nine cases reported containing caIcification in the stroma. Harms35 was the first to note this characteristic of cystadenoma. Histopathologic differentiation of cystadenoma, cystadenocarcinoma, and multilocular cysts of the pancreas becomes at times quite compIex. Brunschwig and others caI1 attention to this difficulty. GeneraIIy speaking, nevertheless, the cyst spaces of the multilocular cyst are larger and scarcer whiIe in the cystadenoma they are smaIIer and multitudinous. After eliminating the malignant tumors and segregating them the present review discIoses a residuum of fifty-six cases of benign cystadenoma. Benignancy, a criterion in the differentiation, was adhered to rigidIy. Seven of these cases could not be differentiated from muItiIocuIar cyst and polycystic disease of the pancreas. These seven cases were therefore subtracted from the total of fifty-six cases
206
Mozan-Cystadenoma
Ieaving a baIance of forty-nine benign cystadenomas up to the present writing. Symptoms and Findings. The fact that lesions were found incidentaIIy at necropsy in patients who died from other causes and without any history of pancreatic disease indicates the insidiousness of the cystadenoma and deficiency of prodromas. UsuaIIy a paIpabIe tumor mass in the epigastric region is the first recognizabIe sign of the Iesion. These neopIasms may become Iarge enough to be discovered inadvertentIy by the patient or by the examining physician during the routine course of examination. Very often it is asymptomatic and painless. The symptomatoIogy usuaIIy is that of any variety of pancreatic cyst. Symptoms depend upon the invoIvement or encroachment, whether compIeteIy circumferentiaIIy or as an infringement, upon the Iumen of the surrounding viscera due to pressure or enguIfment of the growing tumor. The symptoms which are compIained of are anorexia, epigastric discomfort, (and in the Iarge tumors) premature satiation nausea, vomiting, beIching and gaseous distention. Jaundice was encountered in three cases.3g~72,g1 This was due to the fact that the cystadenoma invoIved the head of the pancreas adjacent to the common bile duct. Sharp, darting pains in the abdomen may occasionaIIy be observed prior to discovery of the mass. Pain when it occurs wiI1 vary in intensity and Iocation with the Iocation of the cyst and the organs and structures impinged upon by the tumor. Pain may be referred to the back and it may occur anywhere in the abdomen aIthough usuaIIy it is confined to the upper hypochondrium and most often in the Ieft upper quadrant. Severity and quaIity of the pain varies from a distensive sensation or a duI1, dyspeptic, regurgitant ache to a steady tugging sensation and even to a severe, shooting, cramp-like radiation. If the tumor Iies in the body, neck or head and overIies the Iarge vesseIs, a tremuIous, thriII-like, reguIar throb which is synchronous with the puIse can be ascertained by paIpation; if the patient is thin and the this can be observed tumor protuberant, visually. The nausea and vomiting, if present, together with the earIy satiation and chronic wiI1 of necessity be dyspeptic symptoms accompanied with a Ioss of weight and weakness due to interference with nutrition. Mobility of the mass is dependent upon the
of Pancreas degree of embedment in the pancreas and its Iocation in the pancreas. Those in the body and tai1 may be quite mobiIe. Deep embedment in the pancreas causes limitation of movement. Adherence by fibrous attachment or through enguIfment of other adjacent organs wiI1 aIso curtai1 movabiIity. TABLE LOCATION
OF
THE
I
THIRTY-THREE
Head, Neck. Body.
................... ................... ................... TaiI .....................
RECORDED
-
SITES
No. of Cases
Per cent
9 3 9 12
27 9 27 36
-
-L
Thirty-three case reports Iisted the site of attachment of the tumor mass to pancreas; these assumed the distribution and arrangement found in TabIe I. In the Iiterature severa patients were cognizant of a tumor mass in the epigastrium for a Iong time before reporting to the surgeon. Some of these patients feIt an intra-abdomina1 movement of the mass upon rotation of the body in the supine position. In those cases in which the cystadenoma was found in the head or neck of the pancreas and in the rarer event when attachment or encroachment upon the porta or other Iarge vesseIs occurred, a distinct puIsation was compIained of or couId be elicited by paIpatory traction on the mass. An additional unreported finding of this Iesion is the ascites and transient ankIe edema found in the author’s case. This condition is a direct resuItant of the extrahepatic porta bed bIock syndrome21 produced by the partia1 occIusion and pinching off of the portal, superior mesenteric and spIenic veins and inferior vena cava by the movement of the enguIfing tumor mass present. This can be easiIy seen in the accompanying drawing of the tumor mass. The diagnosis rests to a Iarge extent on three important findings, nameIy, (I) presence of a paIpabIe intra-abdomina1 tumor usuaIIy in upper Ieft hypochondrium (occurred in fortysix of the fifty-six cases reviewed) ; (z) dyspeptic distress (occurred in forty-one of the fifty-six cases) and (3) pain, variabIe in character (occurred in thirty-two of the fifty-six cases). (Waiters and CIeveIand’OO report pain American
Journal
of Surgery
Mozan-Cystadenoma in over 85 per cent of their 139 patients with pancreatic Cysts.) It may be mentioned that the Iaboratory offers IittIe or no assistance and in the event there is an interference with externa1 secretion, the then positive Iaboratory findings tend to lead the examiner further away from the correct conclusion. The extent of assistance by the laboratory chiefly lies in ruIing out such other conditions as adenoma, carcinoma and pancreatic abscess. The isIet adenoma patient would, of course, present WhippIe’s triad with a h,ypoglycemia beIow $0 mg. per cent. Blood amylase or Iipase and blood sugar usuaIIy are found at norma IeveIs. The resuItant degree of malnutrition and loss of weight is hardIy ever severe enough to affect aIbumin-gIobuIin ratio or total protein IeveIs. BIood, urine and stoo1 analyses provide Iittle if any specific aid in the diagnosis of pancreatic cyst or cystadenoma. Laboratory investigation is only important in negating and eIiminating other pathoIogic disorders that must be considered tentativeIy in arriving at a fina deduction of pancreatic cyst or benign tumor. Roentgen Examination. In its growth the cystadenoma as a ruIe displaces either the stomach or coIon. If the growth starts on the superior portion of the pancreas, it most probably will present in the Iesser omenta1 cavity pushing the stomach down and forward and causing a buIge through the gastrohepatic ligament. If the mass finds root in the inferior pancreatic border, the coIon wiI1 probabIy be displaced and the tumor wiI1 be covered by the gastrocoIic omentum. An extension through the layers of the transverse mesocoIon or retroperitonealIy is found infrequentIy. This is one lesion, as shown by a perusa1 of former literature, that taxes and exacts the fuI1 and cooperative use of a11 avaiIabIe diagnostic armamentarium at our disposa1. The roIe that x-ray assumes in aiding the differentiation of cystadenoma of the pancreas from other tumor masses of the region depends soIeIy upon the thoroughness and zealousness of the examining roentgenoIogist. There are two main types of tumor distinguishabIe in the pancreas roentgenologically, namely, benign cysts and cancers. The cysts because of their usuaIIy Iarge growth produce signs that are more pronounced than those of cancer of the pancreas. Since the benign cysts grow expansiveIy and ev&Iy in a11 directions, F&??%UrJ’,
195 I
of
Pancreas
207
they wiI1 present smooth, rounded outiines which is in direct contrast to the irreguIar growth in variabIe directions of the maIignant growths causing an irreguIar outline. Cystic tumors are usuaIIy movabIe whiIe the cancerous tumors are usuaIIy fixed to adjacent structures producing anguIar, rigid contours. Some tumors can be recognized by distinct differences in the shadows they cast on the pIain fiIm: shadows simiIar to the kidney in contrast to the retroperitonea1 fat. This abnorma1 shadou on the pIain film may be the first evidence of a tumor in this vicinity. Ehrmann and Eisler, quoted by Borak, have emphasized its importance. Changes in the stomach and duodenum due to their cIose reIationship to the pancreas occur as a direct resuIt of three main types of comencroachment, nameIy, dispIacement, pression and invasion. Displacement. Tumors IocaIized in the head of the pancreas, if Iarge enough, produce the characteristic horseshoe widening of the duodena curve. The antrum of the stomach is dispIaced forward. Tumors of the neck and isthmus of the pancreas cause an upward dispIacement of the stomach. Tumors of the body of the pancreas cause a forward or a downward dispIacement of the stomach. Tumors in the tai1 of the pancreas will displace the stomach to the right.‘O Compression. Likewise, compression can cause pronounced derangements, especialIy that due to benign growth, such as incompIete obstruction, a constant fiIIing defect of the duodenum, proIongation of evacuation time and distortions as the hourgIass stomach. It may simuIate HIing defects which however can be differentiated by ff uoroscopic observation during positiona changes of the patient. Invasion. This type of growth is most often seen in the maIignant tumor and very rareIy in the benign. Because x-ray is the onIy heIpfu1 examination that can be reIied upon for any assistance in deriving our diagnosis, it should be stressed that the surgeon shouId insist on a most compIete and thorough investigative effort on the part of the roentgenoIogist. Evidence obtained from a review of the cIinica1 history and the presence of a tirm, rounded, upper abdomina1 mass, coupIed with roentgenographic findings of pressure distortion upon neighboring organs, especiaIIy the stomach and coIon, suggests usuaIIy pancreatic cyst or tumor. The other conditions
208
Mozan-Cystadenoma
that must be considered and differentiated are mahgnant tumors, retroperitonea1 Iymph nodes, retroperitonea1 cysts, Iipomas and mesenteric cysts, spIenomegaIy, cysts of the kidney and pyonephrotic kidney, aneurysms of abdomina1 aorta and hydrops of the gaIIbIadder. CASE
REPORT
On October 2, 1946, the patient compIained of feeling a Iump in the stomach. She had been perfectIy we11 up to her present iIIness except for occasiona epigastric distress after eating fatty, fried foods. About August, 1946, she became conscious of a Iump in the epigastrium. She stated she was conscious of an intermittent puIsation in this mass and that she feIt rather certain that it was free and movabIe. Whenever she wouId turn in bed it moved about. LateIy, she experienced a sIight tug substernaIIy on movement of the mass. Associated with this there was an obvious, rapid increase in size producing a visibIe swelIing in the epigastrium. With movement it wouId roII into the Ieft upper quadrant beneath the Costa1 margin. She was abIe to bring the mass into the epigastrium at wiI1 mereIy by rotating her trunk. In the Iast two weeks the puIsating and throbbing feeIing of the mass became quite pronounced. Concomitant with this increased size of the epigastric mass she noticed that she attained earIier satiation and in the past seven weeks had Iost about 345 pounds. There was a transient type of edema of both Iegs which wouId disappear when the patient was recumbent. There were no other symptoms. Upon further interrogation the patient stated the puIsation and throbbing were first noticed as far back as February, 1946. Her appetite was aIways good but the patient recently noticed an earIier sense of fuIIness and satiation. SeIective dyspepsia had been noticed, however, to fat and fried foods which evoked a rather marked distention and beIching. Nausea was slight, but vomiting never occurred. There had been transient edema of both ankIes for the past seven to eight weeks which wouId disappear on Iying down. EIectrocardiogram studies were negative. A searching history with the thought of a possible traumatic cyst was taken with reference to any abdomina1 injury, but a11 information elicited was negative. PhysicaI examination reveaIed a we11 deveIoped white, American-born femaIe, about
of Pancreas
sixty-five years oId, not acuteIy III and free of pain. There was a mass fIIIing the epigastrium, substernaIIy, noduIar and firm in consistency. This mass which approximated the size of a grapefruit was reIativeIy fixed and puIsating. The thriI1 could be feIt through the abdomina1 waI1 by pIacing the hand directIy over the mass or by dispIacing the mass and retaining a stationary position with tension. SIight tenderness was eIicited by movement of the tumor or by traction on it. The movement of the tumor from side to side due to rotation of patient whiIe Iying down caused no pain, only cognizance of the changing position by the patient. There was no other tenderness or rigidity. From an x-ray examination a11 findings were negative except for a fairIy Iarge stone in the fundus of the gaIIbIadder and a Iarge paIpabIe mass within the ring of the Iesser curvature which is not movabIe and apparentIy did not arise from the stomach. The character of the mass was not determined. From the ffuoroscopic examination we may infer that the mass may have its origin in the Iesser omenta1 cavity. There was a forward dispIacement of the pyIoric end with a widening of thegastrovertebral interva1. In view of the aforementioned findings it was reasoned that the intra-abdomina1 mass was most probabIy of pancreatic origin. Mesenteric cyst, retroperitonea1 tumor and gastric maIignancy were aIso considered. Operative intervention was decided upon. On October 29, 1946, a mid-epigastric paramedian incision was made. A Iarge tumor mass presented itself through a thinIy stretched hepatogastric omentum. The Iatter was spIit and the Ieft gastric artery was sacrificed and Iigated to aIIow for a sufficient aperture and access to the tumor. The presenting mass proved to be a muItiIocuIar cystadenoma, hard and non-compressibIe, and about the diameter of a Iarge grapefruit. This fiIIed the Iesser omenta1 cavity causing a dispIacement of the stomach anteriorIy and inferiorIy. UtiIizing sharp and bIunt gauze dissection the mass was mobiIZed down to a rather broad base, about 233 inches to 3 inches in width. At this stage it was noticed that the spIenic at its confIuence with the superior mesenteric vein and also the adjacent portion of the porta vein were enmeshed in the substance of the Iowermost portion of the cystadenoma. The
American Journal of Surgery
Mozan-Cystadenoma neck of the pancreas from which the tumor arose and the head of the pancreas were both compressed and to a degree disIodged by the mass. The exact origin of the tumor was at the posterosuperior surface of the neck of the pancreas. It was decided to remove the uppermost twothirds of the mass by a transverse section. Figure I shows the tumor mass at this stage with the pertinent anatomic structures involved. The invoIved region is sketched with portions of the stomach, duodenum and removed to illustrate better the pancreas invoIvement of the splenic, superior mesenteric and portal vein and the superior mesenteric artery by the tumor. The drawing is seIfexplanatory otherwise. The remainder was then sIowIy and carefuIIy morceIIated away from the invoIved bIood vessels. The discovery that a bIunt probe could readiIy Ioosen the bIood vessel waIIs from the engulfing tumor mass was a gratefu1 finding. At no point did we note any infiltration of vascuIar waI1 by the cystadenomn. The remaining remnant of the tumor attached to the posterosuperior border of the neck of the pancreas had a base about I inch in diameter which disengaged rather easiIy from the pancreatic substance. The site of attachment needed no suturing as no bIeeding occurred. The spIeen was enIarged moderateIy due to the encroachment of the spIenic vein. The gastrohepatic omentum was rent in the approximated by catgut. perChoIecystostomy was subsequentIy formed and an oIive-shaped stone was removed. GaIIbIadder was cIosed over a catheter. Two Penrose drains were inserted, one into the lesser omental cavity through the foramen of Winslow and one to Morrison’s pouch. Both drains and catheter were carried exterior through stab wound in the IateraI Ilank. The abdomen was closed with chromic (Finney) and three tension nyIon sutures. Dermic graft was used on the skin. The pathoIogic report was as foIIows: grossly, the specimen consisted of three pieces 14 by 13 by 7 cm. in aggregate measuring composed of a Iarger mass with bosseIated surfaces attached to which were adipose tags. This larger mass was we11 encapsulated. The coIor was grey with darker brown areas. The surfaces made on cutting were spongy, cystic tissue, the cysts fiIIed with cIear grey Iiquid.
February, 195 I
209
of Pancreas
FIG.
I.
The smaI1 pieces resembIe the Iarger but were more fibrous. An olive-shaped smooth caIcuIus 23 mm. in Iength and diameter was present. (Figs. 2 and 3.) MicroscopicaIIy, the hematoxyIin-eosin stained sections of the formaIin fixed tissue reveaIed a narrow rim of normal-appearing pancreatic tissue. The remainder consisted of a Iarger amount of connective tissue surrounding many cystic spaces both moderateIy Iarge and smaI1. These were Iined with a low stratified, cuboida1 epitheIium in most instances and the Iumina were empty or contained fresh-appearing erythrocytes and pink-staining materia1 (secretion?). In addition to the simpIe cystic spaces there were others in which papiIIary projections were seen arising from the cyst Iinings. These were Iikewise covered with Iow stratified, smaI1, cuboida1 ceIIs with clear cytopIasm. In the remaining connective tissue were many coIIections of round cells, many fresh-appearing erythrocytes, hemosiderin and occasiona nerve fibers. Further cIose search revealed occasiona smaI1 ova1 or round coIIections of ceIIs with deepIy staining nucIei and light granuIar cytoplasm. The significance of these was conjectura1 but the possibiIity that they were ectopic isIets of Langerhans was considered. Three microphotographs are incIuded showing certain interesting aspects of the micropathoIogy. (Figs. 1, 3 and 6.) The patient was graduaIIy made ambulatory by the third postoperative day. Very IittIe drainage was noticed on the dressings after the fifth day. The first Penrose drain was
Mozan-Cystadenoma
of Pancreas
2 3 FIG. 2. This presents the gross specimens directIy after removal, cystadenoma choIeIith. FIG. 3. This shows the sea-sponge appearance of the transected cystadenoma.
and
4 5 FIG. 4. Pancreatic glands with adjacent fibro-adenomatous tissue. FIG. 5. Adenomatous area showing papiIIary character of growth. X 60.
FIG. 6. Structure of cystic areas. X 60.
removed on the sixth day and the second on the eighth day. RemovaI of the gaIIbIadder drain was effected on the eIeventh day which was two days after the patient’s discharge from the hospitai. This iiiustrates the uneventfui nature of the patient’s recovery. The sinus
tract Ieft by the drains continued discharging a biie-stained ffuid for nine more days before occluding compieteiy. Since convaIescence the patient has been recaiied for further cIinica1 observation and has been foiiowed up to the present time. She has gained g pounds, has no compiaints and one year and six months postoperativeiy her genera1 condition is excelient. The transient edema has not recurred since surgery. There is no evidence of recurring tumefaction anywhere intra-abdominaiiy. Her dyspeptic symptoms have entireiy subsided and she states she can now compIete a mea1 without a feeiing of overstuffring the stomach. The blood sugar, serum Iipase and differentiai count are within normai Iimits. SURGICAL
CONSIDERATIONS
Yamauchi is foremost stressing the importance
American
among the authors in of compiete extirpa-
Journal
of Surgery
Mozan-Cystadenoma tion no matter what the offending tumefaction may be. Sound surgica1 judgment demands compIete abIation of such Iesions. Drainage with or without partia1 excision was accompIished in sixteen cases. Of these there were five benign cystadenomas and one borderIine Iesion. One or more previous drainages had aIready been performed on eIeven of these cases which incIuded the five benign cystadenomas. Forty-two per cent of benign cystadenoma had been subjected to repeated drainage at the end of which procedure permanent cure was stiI1 doubtfu1 since none of the cases showed foIIow-up beyond one or two years. Extirpations were generaIIy accomplished in those instances in which the growth was smaI1, when it possessed a narrow base or when the tumor was found arising from the tail. A few instances are recorded in which grow-ths with broad bases or growths Iocated in the head and other inaccessibIe portions of the pancreas were compIeteIy extirpated. In a number of other cases the tumor was removed after much dif?icuIty depending upon the necessity of Iigating Iarge vesseIs and removing dense, fibrous attachments. In six cases the pancreas had to be partiaIIy resected. In the management of the malignant tumors in the head of the pancreas Brunschwig has elaborated technics and had described them in comprehensive detaiI. One of the stages of his operation \vas suggested by Marogna56 who demonstrated the feasibiIity of suturing the pancreas stump directIy into the bowe1 as an outIet of pancreatic juice after resection of the head of the pancreas. AcknowIedgment of the vascular supply to the pancreas is important. Of the anomalies the one to cause most mischief if overlooked is the abnorma1 origin of the middle coIic artery from the gastroduodenaI.*O The incidence of such an unusua1 origin is 8 per cent. An inadvertent or accidenta Iigation here wouId most certainly resuIt in transverse coIon necrosis. CompIete extirpation of certain broad-based cystadenomas and cysts, due to their Iocation, was considered by many surgeons to be hazardous because of the invoIved risk to the patient. With each new surgica1 intervention and experience in this region, coupIed with experimenta physioIogic work, a practica1 surgical technic has been evolved. Thus the hazard seems now to have been surmounted
February, I 951
of Pancreas
21 I
and this is very we11 iIIustrated in the comparative mortaIity statistics found in Table II. This review Iists forty-nine cases of benign cystadenoma of which thirty-one cases were expIicit as to the type of surgica1 intervention. Twenty-nine of the Iatter showed a postoperative follow-up in addition. TABLE II METHODS
OF
SURGICAL
TREATMENT
MORTALITY
Type of Operation
AND
COMPARATIVE
RATES
Recovered
I I Abdominal expIoratory 0nIy PartiaI excision
2
* ExpIoratory and biopsy. 3 Complete extirpation. 22 (Case herein incIuded)
/
’
~3linrcIcordedl
I
2
0
0 ‘4
cent)
IO0 ~
1: 1 .‘3” I ) 3 / 100
~ 3 ~ 13.6 I
I
Progress in surgery of the pancreas is beset with many hindrances two of which are outstanding. First is the inabiIity to prove the function, position and pathoIogic changes in the organ by any means at our disposa1 with any ease or assurance of correctness. The other is the inaccessibiIity and recession of the organ which make it prone to be disregarded by the surgeon in the performance of other abdomina1 surgery. SUMMARY
A complete extirpation of a benign cystadenoma of the pancreas was reported and a series of fifty-six cases coIIected from the medical Iiterature reviewed. 2. Attention is directed to a heretofore unIisted finding of transient edema found in this case. 3. PathoIogicaIIy, cystadenoma of the pancreas is a muItiIocuIar, cystic, adenomatous and neoplastic tumor, benign in character. 4. In its growth and extension it encompasses surrounding structures and organs without infiItration thus aIlowing facile dissection. Extension with infiltration is a rarity; encapsuIation is the ruIe. I.
Mozan-Cystadenoma
212
3. CIinicaIIy, cystadenoma is characterized by an insidious sIow growth. Later a paIpabIe tumefaction deveIops and abdomina1 pain in the upper epigastrium, dyspeptic symptoms and weight 10s~. 6. Roentgen examination (utiIizing IateraI pIate technic) has proved a most vaIuabIe adjunct in diagnosis. 7. Eight of the fifty-six cases had associated diabetes meIIitus. 8. Ratio of occurrence in the sexes was seven femaIes to one maIe. 9. CompIete surgica1 extirpation is the method of choice in the opinion of most authorities and proffers Iowest mortaIity (13.6 per cent). IO. A pIea is made to the surgeon to keep in mind routine examination of the pancreas by paIpation in the course of surgery of the region as in doing gaIIbIadder, common biIe duct, gastric and duodena1 work. This procedure wiI1 afford him the opportunity to recognize the norma fee1 of the pancreas and thereby aIso insure the discIosure of a great amount of pathologic disorder that wouId otherwise remain hidden. Acknowledgment: I wish to thank Dr. E. W. Thurston, our pathologist, for his whoIe-hearted cooperation in the study of tissues and slides of the case and for his heIp in interpreting the transIated pathoIogic descriptions. I am aIso gratefu1 to Drs. T. M. Larkowski, G. ApfeIbach and N. Casciato for their assistance in transIating the foreign articIes. REFERENCES
cysts. Soutb. Med. Ed Surg., 100: 57-61, 1938. BABCOCK, W. W. SurgicaI affections of pancreas. S. Clin. Nortb Ame&, 15: 101-r ‘5, 1935. BALDWIN, J. F. Cystic tumors of the pancreas. Ohio State M. J., 4: 547, 1908. BELOFF, J. T. Cystadenoma; case successfuIIy treated by surgica1 extirpation. J. Mt. Sinai Hosp., 12: 817-820, 1945. Benson, R. E. and GORDON, W. Cystadenoma of the pancreas. Surgery, 21: 353-361, 1947. BERKMAN, J. M. and BANNICK, E. G. Huge muItiIocuIar cysts; 2 cases. Proc. Staff. Meet., Mayo Clin., 8: 185-188, 29, 1933. BEVAN, A. D. Discussion of pancreatic cysts and treatment employed for their relief. S. C&n. Nortb America, 3: 887-898, 1923. BOLAND, F. K. JR. Treatment of recurrent multiIocuIar cyst, case. Soutb Surgeon, I I : 126-131, ‘942. BOLT, R. F. A case of poIycystic disease of the pancreas. Brit. J. Surg., I: 142, 1913.
I. ANGEL, E. Pancreatic
2. 3. 4.
5. 6.
7.
8.
9.
of Pancreas examination. Radiology, 41: 170-180, 1943. BOWERS, R. F., LORD, J. W. JR. and MCSWAIN, B. Cystadenoma; 5 cases. Arch. Surg., 45: I I r-122, 1942. BOWMAN, H. A. H. Pancreatic cyst in Ieft hypochondrium-extirpated. Minnesota Med., 5: 697-706. 1922. BRUNSCHWIG, A. The Surgery of Pancreatic Tumors. St. Louis, 1942. C. V. Mosby Co. Radical Surgery in Advanced AbdominaI Cancer. Chicago, 1947. University of Chicago Press. BUCKSTEIN, J. New aid in diagnosis of tumor of head of pancreas. Surg., Gynec. ti Obst., 39:
IO. BORAK, J. Roentgen
I I.
12.
13.
14.
509, 1924.
15. CABOT CASE 27231. PapiIIary
adenocystoma of pancreas. New England J. Med., 224: 988-989,
1941. 16. CABOT CASE 27262. PapiIIary adenocystoma with hemorrhage. New England J. Med., 224: I I I21114, 1941. 17. CARLING, E. R. and HICKS, J. A. B. Cystadenoma-two cases and their probabIe reIationship to poIycystic conditions found in other viscera. Brit. J. Surg., 13: 238-246, 1925. 18. CARTER, R. F. and SLATTERY, L. Cystadenomata of pancreas. Am. J. Digest. Dis. ff Nutrition, 3: 705-707, 1936. 19. CASPER, M. Pancreatic cyst case; operation; recovery. Kentucky M. J., 28: 15-16, 1930. 20. CRAWFORD, F. B. Pancreatic cyst. Australian M. J., I: 552. 1926. 2 I. AARON, A. H. Cyclopedia of Medicine, Surgery and SpeciaIties, vol. I I. Philadelphia, 1942. F. A. Davis Co. 22. DAVEY, B. M. Surgery of pancreas. J. Michigan M. Sot., 20: 350, 1921. 23. DEAVER, J. B. Surgery of pancreas, principIes underlying. Boston M. +Y S. J., 176: 187, 1917. 24. DELAGENIERE, H. Le Mans Des Kystes gIanduIaires du Pancreas, une observation. Arch. prov. de cbir., 9: 209, 1900. Ibid., 15: 193, 1906. 25. DUNNING, L. H. Cystadenoma of the pancreas; removed by abdominal section. Am. J. Obst., pp. IOI-110, 1905. 26. EDLING. L. Zur Kenntniss der Cvstadenome des Pankreas. Vircbows Arch. j. path: Anat., 32: I IO, I905. 27. EINHORN, M. Pancreatic tumors malignant and benign. Am. J. M. SC., 172: 79G804, 1926. 28. FIESSINGER, N. and CATTAND, R. Cystadenoma of head of pancreas. Bull. et mkm. Sot. mkd. d. b8p. de Paris, 53: 497-504, 1929. zg. FINNEY, J. M. T. and FINNEY, J. M. T., JR. Resection of pancreas. Ann. Surg., 88: 584-592. 1928. 30. FRIEDENWOLD, J. and CULLEN, T. S. Pancreatic cysts, with report of seven cases. Am. J. M. SC., 172: 313-334, 1926. 3r. GILBRIDE, J. J. Tumors of pancreas; operations oerformed on twelve patients. J. A. M. A., 83: 984-989, 1924. 32. GOYENA, J. R. GIanduIar cyst in head of pancreas. Semanu m&d., I : 1258-1265, 1924.
American
Journal
of Surgery
Mozan-Cystadenoma 33. HABERER, H. Operation for benign solid tumor case. Arch. j. klin. cbir., 148: 398-403, 1927. 34. HADLEY, E. C. MuItiIocuIar cyst of the pancreas. Brit. M. J., I: 112, 1914. 35. HARMS, C. Cystadenoma; case. Centralbl. J. a&. Patb. u. patb. Anat., 55: 53-56, 1932. 36. HEYMANN, E. Ueber einen gutartigen, Pankreastumor. Deutscbe med. Wcbnscbr., 15: 48, 1924. 37. HICK, F. K. Pancreatic cysts. M. Clin. North America, 16: 555-563, 1932. 38. HUEPER, W. C. Pancreatic cystadenoma. Arch. Path. w Lab. Med., 5: 261-266, 1928. 39. JANES, R. M. Cystadenoma; 2 cases. Brit. J. Surg., 23: 809-815, 1936. 40. JEMERIN, E. E. and SAMUELS,N. A. Cystadenoma of pancreas. Ann. Surg., 127: 158-169, 1948. 41. JUDD, E. S. and WALTERS, W. Abstract of 1931 report of surgicaI procedures for lesions. Proc. Staff. Meet., Mayo Clin., 7: I IO-I 12, 1932. 42. KERR, A. A. Cysts and pseudo-cysts of the pancreas. Surg., Gynec. @ Obst., 27: 40, 1918. 43. KIBBY, S. U. Cystic tumor of tail of pancreas, case, J. Connecticut M. Sot., 2: 331-332, 1938. 44. KOONTZ, F. L. Cyst of pancreas. Kentucky M. J., 15: 166, ,917. 45. KORTE, W. Die chirurgischen Krankheiten und die Verletzungen des Pankreas. Deutscbe cbir., 45-D: 234, 1898. Zur BehandIung der Pankreascysten und Pseudocysten. Deutscbe med. Wcbnscbr., 30: 536, 191 I. 46. KUHN, H. P., SCHUTZ, C. B. and HELWIG, F. C. Cystadenoma; case. J. Missouri M. A., 27: 570-572, ‘930. 47. LANG, F. J. Uber einige GeschwuIstbiIdungen des Pankreas. V&bows Arch. f. path. Anat., 257: 235-248, 1925. 48. LEPOUTRE, C. and BUSSER, F. Cystadenoma. Bull. Assoc. franc. p. l’etude du cancer, 22: 214-220, 1933. 49. LOCKWOOD, C. E. Tumors of pancreas. J. A. M. A., 77: 155+, 1921. 50. LORENZ, H. Cystadenoma, vo1. III, p. 1282. Wiener GeseII, der Artzte, 192 I. 3 I. MAHORNER, H. R. Cysts of pancreas; etiology and pathology. Proc. Staff Meet., Mayo Clin., 5: 101-103, and 1930. 52. MAHORNER, H. R. and MATTSON, H. EtioIogy and pathoIogy of cysts of pancreas. Arch. Surg., 22: 1018-1033, 1931. 53. MAINGOT, RODNEY. Post-graduate Surgery. New York, 1938. Appleton-Century. 54. MALCOLM, J. D. A case of compIete removal of a multilocular cystic tumor of the pancreas. Lancer, I: 1676, 1906. 5s. MARIANI, C. Adenoma cistico de1 pancreas; asportazione parziaIe, marsupium deIIa resante parte; quarigione. Clin. cbir., I I : 800-810, 1903. 56. MAROGNA, P. Tumors and cysts of pancreas. Arch. ital. cbir., 7: 113-120, 1923; ab., J. A. hf. A., 81: 425, ‘923. 5;. RIATTSON, H., JUDD, E. S. and I\IAHoRxEa, H. R. Pancreatic cysts., forty-seven cases. Arch. Surg., 22: 838-849, 1931. 58. MCKECHNIE, R. E., JR. and PRIESTLEY, J. T. MuItiIocuIar polycystic tumor. Canad. M. A. J., 36: 592-593, 1937.
February,
1951
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59. MCWHORTER, G. L. Cysts of pancreas. Arch. Surg., I I : 619632, 1925. 60. MCWILLIAMS, C. A. True pancreatic cyst. S. Clin. Nortb America, 3: 439-442, 1923. 61. METZLER, F. Tumor, cystadenoma of pancreas. Arch. j. klin. cbir., 134: 772-779, 1925. 62. MEYER, L. J. Pancreas-resection of body and tai1, recovery. Australian M. J., I : 396, 1916. 63. NEEF, F. E., RHODENBURG, G. L. and RITTEH, S. I-I. Cystadenoma; remova through vertical incision in gastrohepatic omentum. Am. J. Surg., 46: 377-386, 1939. 64. NOORDENBAS, W. Extirpation of cystadenoma. Nederl. tijdscbr. o. geneesk., 2: 5546-5556, 1929. 65. OPIE, EUGENE, L. Disease of the Pancreas, Its Cause and Nature, p. 266. PhiIadeIphia, 1910. Lippincott. 66. PARENTI, G. C. Diffuse cystadenomatosis. Patbologica, 25: 165-169, 1933. 67. P~scuccr, L. M. Pancreatic cysts and Iithiasis; classification and incidence; report of pseudo cyst associated with disseminated parenchymal calcification. Am. J. Roentgenol, $2: 80-87, 1944. 68. PETTIKARI, V. Cystic adenoma and acute pancreatitis; case. Arch. ital. di anat. e istol. pat., 7: 469-483. 1936. 69. PORTIS, M. MuItiIocuIar cyst of pancreas. M. Clin. Nortb America, 8: 641-645, 1924. 70. PRIESEL. Beitrag zur PathoIogie der Bauchspeicheldruse. Frankfurt. Ztvbr. ,j. Path., 26: 453, 1922. 71. PRIMIIOSE,A. Cysts and pxudo cysts; report of a case of total extirpation by an extraperitoneal method. Surg., Gynec. @ Obst., 431-436, 1922. 72. PRIXLE, S. Cystadenoma of Pancreas, complete rcmova1. Brit. J. Surg., 13: 180-182, 1925. 73. QIXMBY, A. J. and QUIMBY, W’. A. Roentgen findings in upper right abdomen with special reference to duodenum, gaI1 bladder and pancreas, New York State J. Med., ro5: 821, ‘917. 74. RABINOVITCH, J. and PINES, B. Cysts of the pancreas. Arch. Surg., 45: 3, 1942. 75. RANSOHOFF, J. Cystic adenoma of the pancreas; extirpation. Tr. Ohio M. Sot., 4: 225-234, r9o1. 76. REEKE, TH. Beitr;ige zur PathoIogie des Pankreas. Frunkjurt. Ztscbr. j. Path., 40: 444-451, 1930. 77. RIVAIIOLA, R. A. Tumors of pancreas. Rev. Asoc. med. argent., 34: 166g-1701; J. A. M. A., 78: 1766, 1922. 78. ROBSON, A. W. and MOYNIHAN, B. G. Diseases of the Pancreas. PhiIadeIphia, 19o3. \Y. B. Saunders Co. 79. ROSE, B. T. Encysted adenoma of’ head of pancreas. Brit. J. Surg., 33: 93, 1945. 80. ROTH, H. Cysts of pancreas. Report of two cases. Am. J. Surg., 39: 257-261, 1925. 81. SACON, J. I., BURLANDO, A. and CAMAROTTA, A. GIanduIar cyst; cIinica1 and roentgen diagnosis; recovery of case foIIowing surgica1 intervention. Hosp., Argentina, 5: 21~232, 1934. 82. SANTY, P., MALLET, P. and CROIZAT, Guy and P. Polycystic tumors of the body of pancreas; Ieft pancreatectomy, case. Lyon cbir., 33: 689. 692, 1936. 83. SCHLEGEL, A. SurgicaI Diseases of Pancreas, Beitr. z. klin. cbir., 133: 562-582, 1925.
214
Mozan-Cystadenoma
84. SCHLONVOGT, E. Cystadenoma. Nowotwory, 6: 145-158, 1931. 85. SCHMIDT, HANS. Uber ein Adenom des Pankreas. Zentralbl. j. patb., 31: 491, Igzr. 86. SCHMIEDEN, V. and SEBENING, W. SurgicaI anatomy and methods. Arch. j. klin. Cbir., 148: 319-387, 1927; Surg., Gynec. fl Obst., 46: 735751. 1928. 87. Scorer, G. Adeno-cistoma papiIIefero de1 pancreas. Arch. per le SC. med., 1906, abstr., Centralbl. j. allg. Patb. u. patb. Anat., 18: 844, 1907. 88. SIMON. H. Cvstadenoma of nancreas. Beitr. z. klin: cbir., 1”34: 410-412, 192;. 89. SKIBNIEWSKI,T. Zystisches adenom des Pankreas. Polska gaz. lek., 5: 2, 1926. Adeno-cistoma DaniIIifero de1 00. SOFRANO. F.. pancreas. A&b. per. le SC.med., 30:~184, 1906. 91. SPARKMAN, J. R. Cyst of pancreas. J. Soutb Carolina M. A., 14: 5, 1918. gz. SPEESE, JOHN J. Cystadenoma of the pancreas with extension to the abdomina1 ten years after drainage of a pancreatic cyst. Pbiladelpbia Acad. Med. Am. Surg., 61: 759. 1915. 93. STILLMAN, A., JR. Surgery of pancreas at RooseveIt HospitaI from 1918 to 1928. Am. J. Surg., go : 58-64, 1929. 94. TAVERNARI, A. PapiIIiferous cystadenoma; case. Patbologica, 23 : 207-2 14, I g3 I. 95. TOCHOWICZ, L. MobiIity of tumors. Polska gaz. lek., 8: 641-642, Igzg.
of Pancreas 96. VANDKVENTER, H. Cysts of Pancreas with report of a case. J. Michigan M. Sot., 24: 586, 1925. 97. VAUGHAN, G. T. PapiIIary adenocystoma of pancreas. Virginia M. Montbly, 50: 811-912,
‘924. 98. WAGENSTEEN, 0. H. Pancreas cysts. Journal Lancet, 50: 219-224, 1930. 00. WALTERS. W. and CLEVELAND. W. H. SurnicaI Iesions ‘of pancreas, review. ‘Arch. Surg.,-42: 819-838, 1941. IOO. WHITE, H. Pancreatic cysts and pseudocysts. Guy’s Hosp. Rep., 54: 17, Igoo. IOI. WO~EL, P. Pathogenesis of cysts of pancreas. Arch. j. klin. Cbir., 140: 483-500, 1926. 102. WRIGHT, A. DICKSON. Post-graduate Surgery (Rodney Maingot), Chapt. I. 1938. AppIetonCentury. New York. 103. YAMANE. Beitroge zur Kenntnis der Pankreascvsten. Bern. 102 I. PauI. HauntbuchhandIuns. 104. YAMAUCHI, S. MuItiIocuIar pseudomucinous cystadenoma; report of case successfuIIy extirpated, with discussion of surgica1 treatment. Hawaii M. J., 3: 67-70, 1943. 10s. YANOWSKIY, S. M. Cystadenoma; case. Vestnik kbir., 45: 83-86, 1936. 106. YOUNG. E. L.. JR. MuItiIocuIar cvstadenoma. New England J.‘Med., 216: 334-339, 1937. 107. ZIEGLER, H. Excision of head of pancreas for carcinoma with studies of its bIood suppIy. Surg., Gynec. c!? Obst., 74: 137-145. 1842. LI
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