Papilloma of the Gallbladder Yoto Aoki, MD, Izumiohtsu, Osaka Teruo Okamura, MD, Izumiohtsu, Osaka Hbfumi

Kurimoto, MD, Izumiohtsu, Osaka

Katsutoshi Taniguchi, MD, Izumiohtsu, Osaka

Benign tumors of the eatrahepatic biliary system are rare. Those of the gallbladder are mainly of three types; papillomas, cholesterol polyps, and inflammatory epithelial proliferations [I]. Other possibilities include adenomyomas, fibromas, lipomas, aberrantpancreas, gastrointestinal heterotopia, and so on. Papillomas are the most important type in that they may be malignant or precursors to malignant degeneration. The cause of papillomas is not yet clear despite the various theories that have been presented. In the majority of cases the condition is discovered at surgery or autopsy, and it is seldom diagnosed before surgery [2]. Cholesterosis of the gallbladder, however, is well known as “strawberry gallbladder,” and its incidence at autopsy is relatively rare, but this condition is a more common complication of papilloma of the gallbladder than is generally imagined. We are reporting a case of papilloma of the gallbladder associated with cholesterosis. Clinical and pathologic problems posed by this lesion are discussed with a brief survey of the literature. Case Report The patient, (PA), a thirty-one year old married woman, visited our clinic on September 20,1972, because of attacks of pain in the right upper quadrant of the abdomen, radiating to the right shoulder. The attacks had occurred over the past three years at approximately two month intervals, but she never had fever, chills, or jaundice at the time of attack of pain. There was no intolerance to fatty foods. History revealed nothing significant except nephritis. Physical examination demonstrated no abnormalities except for slight tenderness in the right upper quadrant.’ Laboratory investigation showed a hemoglobin of 80 per cent, an erythrocyte count of 3,750,OOOper mm3, and a leukocyte count of 3,800 per mms. Serum glutamic oxalacetic transaminase, lactic dehydrogenase, alkaline phosphatase, and cholesterol levels were within the normal range. There were no abnormal urinary findings.

X-ray films of the gallbladder showed it to be normal in size and shape. However, a thumb-head sized negative shadow was present on all views (Figure 1) and was thought to be a stone. An upper gastrointestinal series revealed no abnormalities. From these findings, a diagnosis of cholecystolithiasis was made. At operation on October 5, 1972, a thin walled gallbladder of normal size was found. No stones could be palpated; however, the surgeon was able to feel a pinky-head sized, soft, elastic growth attached to the fundus. The common duct was normal. Hence, the gallbladder was excised. When the gallbladder specimen was filled with contrast medium and examined radiologically, a seaweed-like projection measuring 0.5 by 1.0 cm at the fundus and some negative shadows at the neck were demonstrated. (Figure 2.) On cut section the lumen of the gallbladder was filled with bloody bile and some red gelatinous substance resembling a blood clot. The mucosal folds of the gallbladder were streaked with yellow, showing a strawberry appearance, and a papillary projection measuring almost 1 cm in length was attached to the fundic mucosa. (Figure 3.) These papillae were branching fronds. (Figure 4.) On bacteriologic examination, no microorganisms grew in the biie on 72 hour culture. Microscopic examinations of the bile showed erythrocytes and columnar cells but no malignant cells. Microscopic sections confirmed the diagnosis of papilloma with cholesterosis of the gallbladder. (Figure 5.) The patient’s postoperative course was uneventful and she was discharged on the sixteenth postoperative day. In the one and a half year follow-up period since surgery, she has been entirely asymptomatic.

Comments Papilloma of the gallbladder was first reported by Heschel [3] in 1852. Since then, there have been many descriptions of the disease, and in many cases, it has been associated with chronic cholecystitis, gallstones, or both. There is widespread disagreement on the incidence of papilloma of the gallbladder. Kirklin [4] reported an incidence of 8.5 per cent and Swinton

Reprint requests should be addressed to Yozo Aoki, MO, Department of Surgery, ltumiohtsu City Hospital, Izumi&tsu.Osaka 595. Japan.

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and Becker

[5] found

it in 0.1 per cent of

patients who had undergone cholecystectomy. Some of the discrepancy in these figures is the result of confusion in the nomenclature and interpretation of different

pathologic

entities.

Forexample,

Phillips

The Amwioan dawnat d Surgery

Papilloma of the Gallbladder

[6] in 1933 described 500 cases of papilloma of the gallbladder, but Kerr and Lendrum [ 71, reviewing the literature two years later, accepted only seven cases as true papillomas. In Japan, only sixteen true papillomas have been reported up to 1973.

In regard to the definition of papilloma, we agree with Ochsner and Carrera [8] who state: “The term papilloma should be restricted to those lesions which are truly neoplastic, with a vascular connective tissue stalk and covered by a single layer of tall columnar cells. Repeated branching and rebranching often occur, result in multiple villous processes, each covered by epithelium. Cellular infiltration and lipoid deposition may occur in these tumors.” Our case agrees with the foregoing definition. The cause of these tumors is speculative. Chronic inflammation, metabolic disturbances, and stones may play a significant role in their origin [6,9]. The majority of cases are reported to occur after the third decade of life, and 80 per cent of the patients are female which is in keeping with the sexual predilection of other gallbladder diseases [IO]. It is extremely doubtful that papilloma of the gallbladder ever causes symptoms per se. However, the patient’s complaints are generally similar to those seen in chronic cholecystitis with or without stones. Although Tabah and McNeer [II] described the symptoms of pain, belching, fullness, intolerance to fatty or fried food, dyspepsia, and rarely, jaundice, the question of whether these clinical manifestations are due to associated gallstones, cholecystitis, or both arises. For this reason, by excluding patients with votuma131.Msy1976

Figure 3. The opeded gallbladder: a papillary projection (arrow I), fokfs the of mucosa streaked with yellow (arrow 2), and some red substances gelatinous resembling a blood clot (arrow 3).

Figure 4. Macroscopic appearance of the papillary projection showing branching fronds.

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Aoki et al

Figure 5. Microscopic view of the lesion in Figure 4. (Hematoxylin and eostn stain; original magnification X 40.)

associated calculi or cholecystitis, Borgerson, DelBearo, and Callaghan [IO] reported that the most frequent symptoms were flatulent dyspepsia, cramping abdominal pain, epigastric and right upper quadrant distress, and other symptoms which generally resembled the clinical features of chronic gallbladder disease. Greenwald 1121 and Coate [I31 each reported some cases in which colicky pain surely existed although the gallbladder showed only papilloma without stones. Lund and Burman [14] described a case exhibiting symptoms of acute cholecystitis and obstruction of the cystic duct due to a pedunculated papilloma. Walters and Snell [Is] stated that most papillomas are soft and friable, so that fragments may be free in the lumen. They therefore postulated that these fragments may pass into the common bile duct and cause typical symptoms of colic and jaundice. Craziani et al [16] described a case of diffuse papillomatosis without stones in the gallbladder. They stated that these tumors were friable, well vascularized, and bled easily and that blood clots and small free fragments were found within the gallbladder. They concluded that symptoms can be caused by papillomas alone since in their patient colicky pain and jaundice were present in the absence of stones. From these facts and our own case, we believe that blood clots resulting

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from bleeding of the papilloma play a major role in causing abdominal pain. The gallbladder frequently shows associated pathologic disorders. Kirklin [I 7] found more than minimal evidence of microscopic infection in about half the cases studied. Shepard, Walters, and Dockerty [18] reported gallstones in 68 per cent of their patients and cholesterosis in 37 per cent. Kane, Brown, and Hoerr [19] described stones in 38 per cent of cases, cholesterosis in 13 per cent, and chronic cholecystitis in 25 per cent. In our case, cholesterosis was an associated cholecystic disease. The relation between papillomas of the gallbladder and cancer is a matter of controversy. Halpert [20] reported no known relationship between the two; however, there are several reports of papillomas that showed malignant changes. In his series of 500 cases, Phillips [6] reported one case associated with carcinoma within the gallbladder, but the carcinoma seemed unrelated to the papilloma. Kirklin [17] states that malignant degeneration of papillomas is extremely rare and that only one such case was noted among 1,400 cases of papilloma of the gallbladder. Shepard, Walters, and Dockerty [18] described only one case of associated carcinoma among forty-nine patients with papilloma of the gallbladder. Tabah and McNeer [11] found three cases of papilloma of the gallbladder with in situ carcinoma. Clinically, it may be unquestionably safer to regard papilloma as a premalignant lesion. It is noteworthy that all reported cases of malignant “papilloma” were in situ and not invasive carcinomas in both surgical and autopsy specimens. The preoperative diagnosis of papilloma of the gallbladder can be made only by cholecystography. Kirklin [21] described the roentgenographic characteristics of papilloma of the gallbladder and concluded that certain cholecystographic patterns can be anticipated. The significant radiologic findings are briefly outlined as follows: (1) The defect produced by papillomas on cholecystograms maintains a more or less constant position even on repeated examination and with the patient in different positions. (2) There are frequently two or more shadow defects and they should therefore not be mistaken for stones. (3) The size or location of the defects is, as a rule, not helpful diagnostically. Papillomas do not show a predilection for any particular portion of the gallbladder. The treatment of papillomas of the gallbladder is cholecystectomy since the disease is frequently associated with cholecystitis and cholelithiasis, causes various symptoms, and may be a premalignant lesion.

The American Journal

of Surgery

Papilloma

Summary A case of papilloma of the gallbladder is reported. The gallbladder was associated with cholesterosis but not with stones. The causes of colicky pain in this case are discussed. Papillomas of the gallbladder exhibit no characteristic clinical picture; the symptoms are similar to those of any type of gallbladder disease. Papillomas of the gallbladder may be associated with malignant degeneration. Cholecystectomy is the treatment of choice. Acknowledgment: We are deeply indebted Doctor M. Katsumi for his encouragement.

2. 3. 4.

5. 6. 7.

Palmer ED: Benign tumors of the gallbladder, p 607. Clinical

Gastroenterology, 2nd ed. Hagerstown, Maryland, Harper 8 Row. 1963. Gagliardi RA, Gelbach PD: Papilloma of the gallbladder. Gastroenterology 32: 666, 1957. Heschel R: Cii by Shepard VD, Walters W, Dockerty MB [ IS]. Kirklin BR: Cholecystographic diagnosis of papilloma of gallbladder. Am J Roentganol Radium Thar Nucl Med 1: 46, 1931. Swinton NW, Becker WF: Tumors of tha gallbladder. Surg C/in North Am 28: 669.1948. Phillips JR: Papilloma of the gallbladder. Am J Surg 21: 38, 1933. Kerr AB, Lendrum AC: A chloride-secreting papilloma in the gallbladder: a tumour of heterotopic intestinal epithelium

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to

References 1.

8.

14.

15. 16.

17.

18. 19. 20. 21.

of the Gallbladder

containing Paneth cells and enterochromaffine cells and associated with massive chloride loss: with a critical review of papilloma of the gallbladder. /3r J Surg23: 615, 1936. Ochsner S, Carrera GM: Benign tumors of the gallbladder. Gastroenterology 31: 266, 1956. Arbab AA, Brasfield R: Benign tumors of the gallbladder. Surgery61: 535, 1967. Borgerson RJ, DelBeccaro EJ. Callaghan PJ: Polypoid lesions of the gallbladder. Arch Surg 85: 234, 1962. Tabah EJ, McNeer G: Papilloma of the gallbladder with in situ carcinoma. Surgery 34: 57, 1953. Greenwald W: Papilloma of the gallbladder. Surgery 16: 370, 1944. Coate JD: Roentgenologic demonstration of tumors of gallbladder. Am J Roentgenol Radium Thar Nucl A&d 46: 3 19, 1941. Lund NM, Burman CEL: A case of acute cholecystitis and obstruction due to pedunculated papilloms identical with attack of acute cholecystitis and gallstones. Br J Surg 38: 390, 1951. Walters W, Snell AM: Diseases of the Gallbladder and Bile Ducts. Philadelphia, WB Saunders, 1940, p 176. Graziani D, Marks LM, Lim R, Friedell M, Swerdlow M: Papillomatosis of the gallbladder: report of a casa with intermittent jaundice and pain. Am J Gastroenteroi 44: 565, 1965. Kirklin BR: Cholecys~ic dlagnosis of papillornas and other tumors of the gallbladder. &oc Staff Meet, lbtayoC/in 5: 336, 1930. Shepard VD, Walters W, Dockerty MB: Benign neoplasms of the gallbladder. Arch Surg 45: 1, 1942. Kane CF. Brown CH, Hoerr SO: Papilloma of the gallbladder: report of eight cases. Am J Surg 83: 161, 1952. ~alpert a: ~eoplasms, p 942. Pathology, 5th ed (Anderson KAD, ed). St. Louis, CV Mosby, 1966. Kirklin BR: Cholecystographic diagnosis of neoplasms of the gallbladder. Am J Roentganol Radium Thar Nucl Md 29: 8, 1933.

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Papilloma of the gallbladder.

A case of papilloma of the gallbladder is reported. The gallbladder was associated with cholesterosis but not with stones. The causes of colicky pain ...
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