Hemosuccus Pancreaticus

(Hemoductal Pancreatitis) Gastrointestinal Hemorrhage Due to Rupture of a Splenic Artery Aneurysm Into the Pancreatic Duct Brack A. Bivins, MD; Charles R. Sachatello, MD; Vincent P.

Chuang, MD;

\s=b\ A patient with recurrent upper gastrointestinal bleeding was found to have pancreatitis and a pseudoaneurysm of the splenic artery that communicated with the pancreatic duct. Similar pathology noted in ten other patients found in an extensive review of the literature suggest that this rare entity must be considered in the diagnosis of gastrointestinal hemorrhage of obscure origin. In this collected experience, the combination of recurrent left upper quadrant pain, a history of pancreatitis, and recurrent bouts of gastrointestinal bleeding of obscure origin were usually present in those patients who were found to have a splenic artery aneurysm as the source of the blood loss. Distal pancreatectomy with resection of the splenic artery aneurysm is curative.

(Arch Surg 113:751-753, 1978) duct is

pancreatic through Hemorrhage gastrointestinal (GI) bleeding. Sandly termed pancreaticus" the

rare cause

an

extreme¬

of

in this syndrome "hemosuccus the name and Rose2 while Longmire 1970, suggested "hemoductal pancreatitis" in 1973. This article describes a patient in whom we were able to make this diagnosis preoperatively and presents an exten¬ sive review of the literature, from which we were able to collect ten other cases.

blom1

REPORT OF A CASE Clinical History

Patrick

Brady,

MD

that she had been drinking in excess of 0.5 liter per day of bourbon. A lesser curvature gastric ulcer was seen on upper GI x-ray series, while extensive pancreatic calcification was visible on plain films of the abdomen. The gastric ulcer was treated by wedge excision. Postoperatively the patient did well and was discharged. Four months later, in July 1976, she again was seen for anemia. On physical examination she was found to have moderate epigas¬ tric tenderness and a stool positive for blood. Workup during this admission gave normal results. A transfusion brought the hema¬ tocrit value to 34%, and the patient had no further bleeding throughout her hospitalization. Her fourth admission, in March 1977, was prompted by upper GI bleeding requiring four units of blood at her local hospital. On admission she described left upper quadrant pain followed by hematemesis and melena. On physical examination she was found to be cachetic, with mild epigastric tenderness and a stool positive for blood. Shortly after admission she underwent fiberoptic upper endoscopy, with the findings of mild gastritis and fresh blood in the duodenum. No source of bleeding was seen. Over the succeeding three days, she had several episodes of hypotension accompanied by bloody bowel movements and a fall in hematocrit value. Repeated endoscopy and upper GI x-ray series were normal. Because of the obscure nature of her bleeding, she underwent visceral arteriography, which disclosed vascular changes consis¬ tent with chronic pancreatitis and a splenic artery aneurysm. The

Fig 1.—Splenic arteriogram, arterial phase. Solid arrow indicates splenic pseudoaneurysm. Small pancreatic arteries (open arrows) are irregular and beaded, consistent with chronic pancreatitis.

A 67-year-old, gravida 0, black woman was first admitted to the University of Kentucky Medical Center in September 1973 for treatment of a lung abscess. She was found to have normochromic anemia, with a hematocrit value of 20% on admission and a reticulocyte count of 6.8%. Her lung abscess responded to anti¬ biotic therapy and she was discharged improved, although no

was determined. Three years later, in February 1976, she was readmitted for evaluation of anemia (hematocrit value, 32%) and rectal bleeding. She complained of epigastric pain and weight loss and admitted

etiology of her anemia

for publication Nov 23, 1977. Departments of Surgery (Drs Bivins and Sachatello), Radiology (Dr Chuang), and Medicine (Dr Brady), University of Kentucky, Albert B. Chandler Medical Center, Lexington. Reprint requests to Department of Surgery, University of Kentucky Medical Center, Lexington, KY 40506 (Dr Bivins).

Accepted

From the

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splenic vein was thrombosed and the patient had gastric and esophageal varices (Fig 1). Although no contrast material was seen passing into the pancreatic duct, the radiologist (V.P.C.) suggested Fig 2.—Histopathologic section through splenic pseudoaneu¬ Disruption of inferior wall of splenic artery (SA) at junction (open arrows) with pseudoaneurysm (AN). Note lack of intima layer in pseudoaneurysm. Pseudoaneurysm protrudes into major pancreatic duct (solid arrows). Fat necrosis, calcification, squa¬ mous metaplasia of ducts, and acinar tissue fibrosis are seen, rysm.

consistent with chronic pancreatitis.

that this

artery

patient's bleeding

could be

originating in the splenic entering the GI tract

aneurysm in the pancreas and

through the pancreatic duct. At operation she was found to have acute and chronic pancrea¬ titis with multiple small abscesses and intense peripancreatic inflammation, which precluded identification of the exact position of the aneurysm. Distal pancreatectomy, splenectomy, and prox¬ imal ligation of the splenic artery were performed. She was discharged home four weeks after the operation and has been followed up for six months postoperatively with no evidence of further GI bleeding.

Pathologic Findings The specimen consisted of spleen and distal pancreas. On cut section the pancreatic tissue appeared fibrotic, with numerous areas of calcification and microabscesses. Within the midportion of the pancreas there was a 1-cm saccular aneurysm of the splenic artery. Microscopic examination showed acute and chronic pancreatitis with fat necrosis, calcification, and squamous meta¬ plasia of the pancreatic ducts. Sections through the site of the aneurysm showed thinning of the arterial wall with evidence of recent hemorrhage and communication with the pancreatic duct

(Fig 2).

COMMENT

This case is the 11th reported case with upper GI bleeding due to erosion of a splenic artery aneurysm into the pancreatic duct.210 A summary of the pertinent clinical Reported Cases of Hemorrhage Through

the Pancreatic Duct From

Splenic Artery Aneurysm

Case/

Source Lower & Farrell,5 1931

Age, yr 1/16

Hentel,e1966

2/80

Jones & 1968

3/59

Finney,7

Medical

4/51

1969

Sandblom,' 1970

5/51

6/69

Definitive Operation Resection of aneurysm; distal pancreatectomy None

Recurrent upper GI bleeding; gastric resection Recurrent upper GI bleeding; abdominal pain; blind gastric resection No history of bleeding

Distal pancreatectomy;

Pancreatitis; splenic artery

Distal pancreatectomy;

Splenic artery aneurysm communicating with pancreatic

Distal pancreatectomy;

splenectomy

7/68

Koehler et al,3 1976

8/60

Bowers et al,'° 1976

9/62

Upper

GI

bleeding

splenectomy

splenectomy

Distal pancreatectomy;

3 times in 10

mo

Distal

pancreatectomy;

splenectomy Melena; colon diverticula and splenic artery aneurysm noted preoperatively; colon resection; rebled 3 mo later Melena; colon diverticula and splenic artery aneurysm noted preoperatively; rebled 10

Walter et al,4 1977

10/57

Present report

11/62

pancreatic cyst Pancreatitis; 7-cm splenic artery aneurysm

bleeding

splenectomy

Bowers et al,' 1976

Pathologic Findings Pancreatic fibrosis & atrophy with dilated ductal system & splenic artery aneurysm Pancreatitis with communication of splenic artery aneurysm with

Gastrointestinal (GI) bleeding 3 times in 2 yr before death from rupture of aneurysm Recurrent upper GI bleeding; oversewing of duodenal ulcer; subtotal gastrectomy 6 mo later for recurrent

Boijsen & Efsing,"

History

Severe abdominal pain for 8 yr after whooping cough; melena for 6 mo

following colectomy Recurrent pancreatitis

Distal pancreatectomy;

splenectomy

Distal pancreatectomy;

splenectomy

mo

Anemic & upper GI bleeding 3 times in 4 yr; wedge excision of healing gastric ulcer

Not stated

Distal pancreatectomy;

splenectomy

aneurysm

duct; pancreatitis Splenic artery aneurysm with atheromatous plaque communicating with pancreatic duct; no pancreatitis 4 X 5-cm splenic artery aneurysm communicating with pancreatic duct; pancreatitis Aneurysmal branch of splenic artery communicating with pancreatic duct; pancreatitis Splenic artery aneurysm communicating with pancreatic duct; pancreatitis 1.6-cm splenic artery aneurysm communicating with pancreatic duct; pancreatitis 1-cm saccular splenic artery pseudoaneurysm communicating with pancreatic duct; Pseudomonas pancreatitis

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data from these patients is given in the Table. The first and youngest patient, reported by Lower and Farrell4 in 1931, was a 16-year-old boy who developed severe episodic abdominal pain and melena following an attack of whooping cough. All the subsequent cases have been in patients over 50 years of age with males predominating (7:4 male-female ratio). Generally these patients had histories of abdominal pain and episodic recurrent bleeding. Only in the case reported by Bowers et al1" (case 9) was pain not a prominent feature. The bleeding was misdiagnosed initially in ten of the 11 cases; it was diagnosed correctly preoperatively at the time of the first bleeding episode only by Sandblom2 (case 6). Six of the 11 patients had undergone one or more operative procedures for GI bleeding before the significance of the splenic artery aneurysm was realized (cases 3,4, 5, 8, 9, and 11). In three cases (No. 3, 8, and 9) the patient was known to have a splenic artery aneurysm at the time of operation for presumed other causes of GI bleeding. The lesion was diagnosed by angiography in eight of the 11 cases, two by exploration and one by autopsy. In only three of eight cases studied by angiography was contrast material seen extravasating from the splenic artery aneu¬ rysm into the pancreatic duct (cases 8, 9, and 10). The operative management and pathologic findings were similar in the reported cases. Resection of the aneu¬ rysm and distal pancreatectomy was carried out in all cases except the patient reported by Hentel" who refused opera¬ tion and represented the only death in the series. Ten of the 11 cases were associated with gross and microscopic evidence of pancreatitis; one patient (No. 6) had an atheromatous plaque at the site of aneurysm formation. Splenic artery aneurysms are second in frequency only to abdominal aortic aneurysms, with 600 cases reported by 1970. " The multiple etiologies of these aneurysms are gradually becoming clear. Approximately 13% of splenic artery aneurysms are associated with fibromuscular dysplasia; 10% are seen in patients with portal hyperten¬ sion; and 13% are associated with atherosclerosis.12 The largest group of splenic artery aneurysms (45%) are found in multiparous women, presumably because of gestational changes in the wall of a tortuous splenic artery.1112 The smallest yet-identified subgroup is associated with what Stanley and Fry12 term "focal inflammatory processes," which in their experience account for 5% of all splenic

artery aneurysms.

The incidence of rupture of splenic artery aneurysms varies from 6% to 46%, with general acceptance of the lower figure.8·1'1'' The multiparous woman has been thought to be at the highest risk for rupture.6·8·'1'4 Patients with pancreatitis may prove to be another group with a high risk of rupture of splenic artery aneurysms. Of the six

ruptured splenic artery aneurysms seen by Stanley et al," three were in their focal inflammatory group. This associa¬ tion of pancreatitis and splenic artery aneurysms is most likely due to enzymatic disruption of the arterial wall. In the experience of Walter et al,4 10% of patients with chronic pancreatitis will have pseudoaneurysms of adja-

cent vessels

develop.

The episodic recurrent GI bleeding seen in these patients may be due to tamponade with clot formation within the pancreatic duct, followed by sealing of the aneurysm. The sudden distention of the duct by the arterial bleeding would explain the pain seen in ten of the 11 cases. With the aneurysm sealed, the pancreatic duct clot will lyse, thus clearing the duct and allowing the patient to be asymptom¬ atic until the next bleeding episode. Obviously the process may recur a number of times over a prolonged interval, as in our patient (four times in two years, and possibly over four years if her anemia in 1973 was due to bleeding through the duct). It is increasingly apparent that patients with GI bleeding of obscure origin are best evaluated by angiog¬ raphy prior to operation. Although it would be ideal if such studies could be routinely performed during a bleeding episode, contast studies performed on a semielective basis are of considerable value, as demonstrated in our patient. When presented with the patient with a history of recur¬ rent GI bleeding, the surgeon must weigh the relative disadvantage of letting the patient continue to bleed while a diagnosis is being sought against the uncertainty of operation for bleeding from an unidentified source. With the possible exception of an aortoduodenal fistula, more patients are likely to be helped by the delay necessary to obtain appropriate angiography than by immediate opera¬ tion for bleeding from an undiagnosed origin. Appropriate interpretation of the angiographie findings depends on the awareness that occult bleeding may be due to visceral aneurysms bleeding episodically into the GI tract. Obviously the presence of a visceral artery aneurysm in a patient with unexplained GI bleeding must be consid¬ ered as a possible source of the bleeding. References 1. Sandblom P: Gastrointestinal hemorrhage through the pancreatic duct. Surg 171:61-66, 1970. Longmire WP, Rose AS: Hemoductal pancreatitis. Surg Gynecol Obstet

Ann 2.

136:246-250, 1973. 3. Koehler PR, Nelson JA, Berenson MM: Massive extra-enteric gastrointestinal bleeding: Angiographic diagnosis. Radiology 119:41-44, 1976. 4. Walter JF, Chuang VP, Bookstein JJ, et al: Arteriography of massive hemorrhage secondary to pancreatic disease. Radiology 124:337-342, 1977. 5. Lower WE, Farrell JI: Aneurysm of the splenic artery: Report of case and review of the literature. Arch Surg 23:182-190, 1931. 6. Hentel W: Ruptured splenic artery aneurysm. Rocky Mt Med J 63:48\x=req-\ 50, 1966. 7. Jones EL, Finney GG: Splenic artery aneurysms: A reappraisal. Arch Surg 97:940-947, 1968. 8. Boijsen E, Efsing H-O: Aneurysm of the splenic artery. Acta Radiol 8:29-41, 1969. 9. Soeno T, Kakizak G, Fujiwara Y, et al: Massive hemorrhage into the upper digestive tract due to rupture of splenic artery aneurysm into the pancreas. Am J Gastroenterol 61:55-62, 1974. 10. Bowers J, Koehler PR, Hammar SP, et al: Rupture of splenic artery aneurysm into the pancreatic duct. Gastroenterology 70:1152-1155, 1976. 11. Stanley JC, Thompson NW, Fry WJ: Splanchnic artery aneurysms. Arch Surg 101:689-697, 1970.

12. Stanley JC, Fry WJ: Pathogenesis and clinical significance of splenic artery aneurysms. Surgery 76:898-909, 1974. 13. Babb RR: Aneurysm of the splenic artery. Arch Surg 111:924-925, 1976. 14. MacFarlane JR, Thorbjarnarson B: Rupture of splenic artery rysm during pregnancy. Am J Obstet Gynecol 95:1025-1037, 1966.

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aneu-

Hemosuccus pancreaticus (hemoductal pancreatitis): gastrointestinal hemorrhage due to rupture of a splenic artery aneurysm into the pancreatic duct.

Hemosuccus Pancreaticus (Hemoductal Pancreatitis) Gastrointestinal Hemorrhage Due to Rupture of a Splenic Artery Aneurysm Into the Pancreatic Duct Br...
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