The clinical significance of acquired jejunoileal diverticula

THE AMERICAN JOURNAL OF GASTROENTEROLOGY Copyright © 1998 by Am. Coll. of Gastroenterology Published by Elsevier Science Inc.

Vol. 93, No. 12, 1998 ISSN 0002-9270/98/$19.00 PII S0002-9270(98)00486-9

The Clinical Significance of Acquired Jejunoileal Diverticula E. de Bree, M.D., J. Grammatikakis, M.D., M. Christodoulakis, M.D., and D. Tsiftsis, M.D., Ph.D. Departments of Surgical Oncology and Radiology, University of Crete—Medical School, Herakleion, Greece

Objective: Because of the relative rarity of acquired jejunoileal diverticulosis, including its symptomatology and complications, diagnosis is often difficult and delayed, resulting in unnecessary morbidity and mortality. The purpose of the present study was to draw attention to jejunoileal diverticula and their complications as a site of gastrointestinal symptoms. Methods: The records of 10 patients with symptomatic jejunoileal diverticula treated in our departments were reviewed. Results: The clinical presentation was varying and nonspecific. Jejunoileal diverticula were diagnosed peroperatively in four patients operated on successfully for their acute complications. In one case the diagnosis was considered after a radiotargeted erythrocyte bleeding scan and in five other cases enteroclysis for chronic abdominal complaints demonstrated jejunoileal diverticula. The death of one patient operated on for massive hemorrhage from jejunal diverticula was probably related to delayed diagnosis and treatment. Conclusions: Jejunoileal diverticula should not always be dismissed as asymptomatic findings, as they may be the cause of vague, chronic symptomatology and acute complications, including intestinal obstruction, hemorrhage, and perforation. Awareness of the fact that jejunoileal diverticula may cause chronic nonspecific abdominal symptoms and serious acute complications may lead to earlier diagnosis and timely treatment with lower morbidity and mortality. (Am J Gastroenterol 1998;93:2523–2528. © 1998 by Am. Coll. of Gastroenterology)

careful exploration of the small bowel at laparotomy—may lead to earlier diagnosis and timely treatment, with lower morbidity and mortality. The purpose of the present report of 10 cases was to draw attention to primary acquired jejunoileal diverticulosis and its complications as a source of gastrointestinal symptoms. PATIENTS AND METHODS The records of 10 patients, four women and six men, with symptomatic jejunoileal diverticula, diagnosed in our departments from 1992 to 1998, were reviewed. The age of these patients at presentation varied between 57 and 85 yr, with a mean age of 70 yr. Clinical presentation, diagnostic approach, treatment, and outcome were analyzed to gain a better insight into this rare condition. The diverticula were located in the jejunum in seven patients, in the ileum in two patients, and in both jejunum and ileum in one patient. Four patients had a solitary diverticulum, which was located in the jejunum in three patients and in the ileum in one. In two patients (20%) associated diverticula were found in the duodenum and in another two patients (20%), in the colon. RESULTS The diagnosis of symptomatic jejunoileal diverticula was established in 10 patients. The clinical presentations were varied (Table 1). Five patients presented with acute complications, including free perforation with diffuse peritonitis, intraabdominal abscess, hemorrhage, and intestinal obstruction. In patient 3, who presented with upper gastrointestinal bleeding, upper gastrointestinal endoscopy and colonoscopy were negative and the patient was treated conservatively. Hemodynamic instability in spite of intensive support made exploratory laparotomy mandatory. In patient 4, who presented with massive gastrointestinal bleeding, after negative endoscopy a radiotargeted erythrocyte bleeding scan demonstrated an active bleeding site in the proximal part of the jejunum. One patient with no previous abdominal surgery presented with high small bowel obstruction. On laparotomy multiple jejunal diverticula with signs of previous inflammation resulting in adhesions and bowel obstruction were found. One patient presented with episodes of diffuse abdominal pain with vomiting, microcytic hypochromic iron-deficiency anemia, and malabsorption of vitamin B12. Four

INTRODUCTION Acquired jejunoileal diverticula are uncommon and asymptomatic in the majority of patients. However, they should not be dismissed as an incidental finding, for they may be the underlying cause of vague, chronic symptomatology and acute complications, including obstruction, hemorrhage, and perforation. Due to the relative rarity of these lesions and their complications, diagnosis is often difficult and delayed. Awareness of the fact that they may cause chronic nonspecific abdominal symptoms and serious complications—as well as the wide use of enteroclysis and Received Apr. 17, 1998; accepted July 15, 1998. 2523

TABLE 1 Clinical Presentation, Diagnostic Tests, Treatment, and Outcome in 10 Patients With Symptomatic Jejunoileal Diverticula Patient Age (yr)/Gender

Symptoms, Signs, and Laboratory Tests

Diverticulitis and perforation Pt 1 Diffuse abdominal pain with 74/M guarding, fever, and leukocytosis Pt 2 Diffuse abdominal pain with 65/F tenderness without guarding, fever, leukocytosis, and anemia Hemorrhage Pt 3 Melena and rectal loss of bright 70/M red blood

Pt 4 60/M

Melena and rectal loss of bright red blood

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Intestinal obstruction Pt 5 Acute small bowel obstruction 79/F Pt 6 85/F

Frequent colicky abdominal pain with changes of bowel habits

Pt 7 70/F

Episodes of colicky abdominal pain with abdominal distension and obstipation Pt 8 Episodes of diffuse abdominal 69/M pain with RUQ tenderness, abdominal distension, and nausea Chronic abdominal pain Pt 9 Chronic vague abdominal pain 57/M with constipation Pt 10 66/M

Episodes of diffuse abdominal pain, microcytic hypochromic anemia, iron deficiency, and decreased serum vitamin B12

Diagnostic Tests

Findings

Treatment

Outcome

X-ray abdomen Exploratory laparotomy

Dilated small bowel loops Multiple jejunal diverticula with perforation

Segmental resection

Alive

X-ray abdomen CT abdomen Exploratory laparotomy

Air-fluid levels small bowel Abscess between small bowel loops Solitary perforated ileal diverticulum with abscess

Drainage of abscess, segmental resection

Alive

X-ray abdomen Upper GI endoscopy Colonoscopy Exploratory laparotomy X-ray abdomen Upper GI endoscopy Colonoscopy Erythrocyte bleeding scan Exploratory laparotomy

Normal Normal Normal Jejunal diverticula, one with active bleeding Normal Normal Normal Bleeding from proximal part of jejunum Active bleeding from solitary jejunal diverticulum

Segmental resection

Multiple organ failure, death

Local excision of the diverticulum

Alive

X-ray abdomen Exploratory laparotomy

Air-fluid levels in small bowel loops Multiple jejunal diverticula with adhesions

Segmental resection

X-ray abdomen Colonoscopy CT abdomen Enteroclysis X-ray abdomen Colonoscopy Enteroclysis X-ray abdomen US abdomen Colonoscopy Enteroclysis

Dilated small bowel loops Normal Normal Multiple duodenal and jejunoileal diverticula Normal Normal Solitary duodenal and jejunal diverticulum Normal Normal Multiple colonic diverticula Large solitary jejunal diverticulum (Fig. 1)

Metoclopramide

Relaparotomy for adhesive ileus, alive Cured

Metoclopramide

Improvement

Metoclopramide

Improvement

X-ray abdomen Colonoscopy Enteroclysis X-ray abdomen Uper GI endoscopy Colonoscopy Enteroclysis

Normal Multiple colonic diverticula Multiple small ileal diverticula Normal Normal Normal Multiple diverticula in the entire jejunum (Fig. 2)

Analgetics, diet advises

Improvement

Antibiotics Iron and vitamin B12 substitution, analgesics, and metoclopramide

No change, Improvement

CT 5 computed tomography; GI 5 gastrointestinal; RUQ 5 right upper quadrant; US 5 ultrasonography.

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diverticulum was performed in another case. In patient 5 the postoperative course was marked by small bowel obstruction. At relaparotomy multiple adhesions were found and a gastrojejunostomy was performed. This time, the postoperative course was uneventful. The recurrent colicky abdominal pain was treated effectively with metoclopramide in patients 6, 7, 8, and 10. In patient 9 analgesics and dietary measures improved the abdominal complaints. Delay in diagnosis and treatment probably contributed to the death of one bleeding patient. The remaining nine patients are in good condition 2 months to 6 yr after initial diagnosis. DISCUSSION

FIG. 1. Enteroclysis demonstrating a large solitary jejunal diverticulum (arrow, patient 8).

FIG. 2. Enteroclysis demonstrating multiple diverticula in the entire jejunum (patient 10).

patients reported recurrent episodes of small bowel pseudoobstruction or chronic vague abdominal pain. In five cases the diverticula were diagnosed during the investigation of their chronic abdominal complaints. Enteroclysis was able to demonstrate the jejunoileal diverticula in these cases (Figs. 1, 2). In one case the diagnosis of a bleeding jejunal diverticulum was considered after a radiotargeted erythrocyte bleeding scan. In the remaining four cases the diagnosis of jejunoileal diverticula was made peroperatively. Resection of the involved bowel segment with primary anastomosis was performed in four cases with acute complications, while local excision of a bleeding solitary jejunal

Acquired diverticula may be primary or secondary to conditions such as abdominal operations, tuberculosis, and Crohn’s disease. Primary acquired jejunoileal diverticula, which are the subject of this article, are formed by herniation of mucosa and submucosa through the muscular layer of the bowel wall (false diverticula). They are usually multiple and occur at the mesenteric border, in contrast to the true congenital Meckel’s diverticulum. They tend to be larger and higher in number in the proximal jejunum and smaller and fewer as one progresses caudally. An exception is the terminal ileum, where the diverticula are often multiple. Their size varies from a few millimeters to more than 10 cm. The earliest reports of jejunoileal diverticula were those of Sommering and Baillie in 1794 and Voigtel in 1804 (1). Gordnier and Sampson (2), in 1906, were the first to operate on a patient with jejunoileal diverticulosis; their patient had a partial small bowel obstruction caused by jejunal diverticulitis and was cured by resection of the involved jejunal segment. In 1920, Case (3) made the first preoperative diagnosis of jejunal diverticulosis, during an upper gastrointestinal radiological examination. The incidence of jejunoileal diverticula is 1.1% to 2.3% found at enteroclysis (4, 5), at postmortem by insufflating the intestine with air (6), or during major abdominal surgical procedures (7). They are observed four times less often than duodenal diverticula (8). Most patients with acquired jejunoileal diverticulosis are in the seventh decade of life or older (9 –13). A male:female ratio ranging from 2:1 to 1:2 has been reported in different studies (9 –17). Diverticula occur five to eight times more often in the jejunum than in the ileum (11–13). About two-thirds of acquired jejunoileal diverticula are multiple (9 –12, 15). Associated diverticula are found in the colon in 35% to 75%, in the duodenum in 15% to 42%, in the esophagus in 2%, in the stomach in 2%, and in the urinary bladder in 12% of cases (8, 11–17). In our series colonic and duodenal diverticula were each found in 20% of the cases. Jejunoileal diverticulosis probably occurs because of motor dysfunction of the smooth muscle or the myenteric plexus in the small bowel. Disordered contractions of the affected small bowel, called jejunoileal dyskinesia, generate

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increased intraluminal pressures that herniate mucosa and submucosa through the weakest mesenteric site of the bowel wall, where it is penetrated by paired blood vessels from the mesentery (18 –20). Acquired jejunoileal diverticula remain asymptomatic in 60% to 70% of cases (9, 10, 12, 17). These asymptomatic diverticula may be found incidentally at laparotomy. Symptoms and complications that require surgical intervention occur in 10% to 19% of the cases (10, 12, 14, 15, 17). Chronic symptomatology includes vague chronic abdominal pain, malabsorption, functional pseudo-obstruction, and chronic low-grade gastrointestinal hemorrhage. Acute complications are diverticulitis, with or without abscess or perforation, massive gastrointestinal hemorrhage, and intestinal obstruction. Acquired jejunoileal diverticula are usually diagnosed by radiological small bowel examination, especially by double-contrast enteroclysis (4, 5), at surgical exploration, or at autopsy. The differential diagnosis includes intraabdominal inflammations, such as appendicitis and cholecystitis; complicated peptic ulcer disease; complicated colonic diverticulosis; irritable bowel syndrome; and causes of small bowel obstruction, such as adhesions, volvulus, intussusception, and pneumatosis cystoides intestinalis (12, 21–23). Most patients with symptomatic jejunoileal diverticulosis have vague, chronic abdominal pain, localized epigastrically or periumbilically, with a bloating sensation after food intake. This chronic pain syndrome is believed to be due to jejunoileal dyskinesia (9). If the pain does not respond to conservative treatment, resection of the involved small bowel segment might be indicated; the reported results are good (9 –12, 24). Stasis of intestinal flow with bacterial overgrowth, due to the jejunal dyskinesia and retention of intestinal contents in the diverticula, may lead to deconjugation of bile salts and uptake of vitamin B12 by the bacterial flora, resulting in steatorrhea and megaloblastic anemia, with or without neuropathy (19, 25–27). In the few instances where administration of broad-spectrum antibiotics and repletion therapy of nutritional deficiencies do not bring relief of the symptoms, resection of the involved bowel segment has been performed with good results (10, 14, 25). Functional small bowel pseudo-obstruction, chronic or acute, may also be caused by jejunoileal dyskinesia. Metoclopramide may be administered in an attempt to enhance aboral small bowel movement (28). In three of our patients with jejunal diverticula, administration of metoclopramide provided some relief of their symptoms. When severe functional pseudo-obstruction persists, resection of the jejunum bearing the diverticula is the treatment of choice (9 –11, 18). Mechanical intestinal obstruction occurs in 2.3% to 4.6% of cases of jejunoileal diverticulosis (10, 12, 14). This may be the result of pressure on the intestinal wall from distended diverticula, inflammatory mass associated with diverticulitis, stricture or adhesions from recent or past diverticulitis, intussusception at the site of the diverticulum,

AJG – Vol. 93, No. 12, 1998 enteroliths developed within the diverticula, or volvulus of the diverticula-containing segment (11, 22, 23). An enterolith formed in a diverticulum may cause obstruction of the bowel lumen at the level of the diverticulum, but usually by impaction more distally, often in the terminal ileum (29 –31). The treatment of choice is to milk the enterolith distally into the cecum at laparotomy. Alternatively, the enterolith may be milked proximally to a less edematous portion of bowel and extracted through an enterotomy. When the enterolith causes obstruction at the level of the diverticulum, bowel resection is necessary (30, 31). This complication is associated with a mortality rate of 15% (30). Another cause of intestinal obstruction is volvulus of the diverticula-containing segment. The involved segment, with its diverticula filled with fluid—which is considerably heavier than the noninvolved part—might be responsible for initiating the volvulus, with the loaded segment swinging like a pendulum (32). The surgical therapy consists of untwisting or resecting of the involved segment (32, 33). A mortality rate of 25% has been reported (33). Gastrointestinal hemorrhage is another well-documented complication; it occurs in 3.4% to 8.1% of the cases of jejunoileal diverticulosis and may be chronic or massive (10, 12, 14). Hemorrhage is usually manifested in the form of bright red blood loss or melena, although reverse peristalsis may result in hematemesis, suggesting a bleeding peptic ulcer or variceal bleeding (34, 35). Trauma to the mesenteric vessels, mucosal ulceration, and diverticulitis without ulceration have been mentioned as causative factors (34 –36). The preoperative diagnosis of massive bleeding from jejunal or ileal diverticula is difficult. The frequent coexistence of colonic diverticulosis contributes to this diagnostic problem. Radiotargeted erythrocyte bleeding scan and mesenteric arteriography may demonstrate a source of blood loss in the small bowel. Careful inspection of the small bowel is mandatory, but peroperative recognition of jejunal or ileal diverticula may be difficult, as they are frequently buried in the mesenteric fat. Resection of the involved segment of the small bowel with primary anastomosis is the procedure of choice (34 –36). Patients with jejunoileal diverticulosis that has not been proven to be the cause of this episode peroperatively must undergo resection of the involved small bowel segment (34, 36). Failure to remove diagnosed diverticula has led to recurrent massive gastrointestinal bleeding and death (9). The incidence of recurrent hemorrhage is high and mortality rates for conservative and operative management are 80% and 14%, respectively (35). The mortality rate is related to delay in diagnosis and treatment, the massive transfusions required, and the advanced age of the patients. These parameters were all present in our patient 3. Jejunal or ileal diverticulitis, with or without perforation, is an acute complication, which occurs in 2.3% to 6.4% of the cases of jejunoileal diverticulosis (10, 12, 14). Acute necrotizing inflammatory reaction is the most common

AJG – December 1998 cause of perforation (82%) (37). Alternatively, perforation may result from penetration of its wall by a foreign body (6%) or from blunt trauma to the abdominal wall (12%) (37). Usually a localized peritonitis results, because the diverticulum is walled off by adjacent small bowel mesentery (37, 38). The symptomatology varies widely and is nonspecific. Other complications of ruptured diverticula include fistulas between small bowel segments, colon and urinary bladder, suppurative pyelophlebitis and multiple hepatic abscesses, and abdominal wall abscess (37, 39 – 41). A plain upright abdominal film may demonstrate pneumoperitoneum, providing evidence of gastrointestinal perforation. However, pneumoperitoneum without perforation and peritonitis is a well-documented complication of jejunoileal diverticulosis (9, 14, 41, 42). This spontaneous asymptomatic pneumoperitoneum is usually caused by transmural escape of air through a thin-walled diverticulum and can be treated nonoperatively. Computed tomography may be helpful in establishing the diagnosis of small bowel diverticulitis preoperatively (43, 44). However, the diagnosis of complicated or uncomplicated jejunoileal diverticulitis is seldom made before exploratory laparotomy or diagnostic laparoscopy (45– 47). Patients with uncomplicated diverticulitis may be successfully managed conservatively, similar to the management of uncomplicated colonic diverticulitis. When laparotomy has to be performed, segmental small bowel resection is the treatment of choice. Lesser procedures, such as simple closure, excision, and invagination, are associated with an approximately three times greater mortality rate (48). Recently, these procedures were also performed laparoscopically (46, 47). The reported overall mortality rate is 24%, with a mortality rate of 14% for resection of the involved segment with primary anastomosis (48, 49). Although many of the cases date from many decades ago, before the use of antibiotics, the overall mortality has not changed significantly. The high mortality appears to be related to the advanced age of the patients as well as to delayed diagnosis and treatment (38, 45, 48, 49). In conclusion, jejunoileal diverticula should not be regarded as an insignificant finding, because it may lead to chronic and acute complications. Awareness of the fact that jejunoileal diverticula may cause these chronic nonspecific abdominal symptoms and serious acute complications, as well as the wide performance of enteroclysis and careful exploration of the small bowel at laparotomy, especially in elderly patients, may lead to earlier diagnosis, with adequate treatment and lower morbidity and mortality. Reprint requests and correspondence: E. de Bree, Department of Surgical Oncology, University General Hospital, P.O. Box 1352, 71110 Herakleion, Greece.

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