Abstract

We report a case of phlegmonous gastritis associated with Kaposi sarcoma in a 37-year-old, human immunodeficiency virus (HIV)–positive man who presented with an acute abdomen. Computed tomographic scan revealed free fluid in the abdominal cavity and a thickened gastric wall. A partial gastrectomy was performed. The resected portion of stomach had a hemorrhagic, necrotic thickened wall and showed extensive, acute suppurative inflammation, especially in the submucosa, with focal transmural involvement. Beneath an area of healing ulceration, a focus of Kaposi sarcoma was present. Group A β-hemolytic streptococcus was grown from peritoneal fluid, and treatment with numerous antibiotics was initiated. After a difficult postoperative course that responded to 8 weeks of antibiotic therapy, the patient was medically stable and discharged from the hospital on antiretroviral therapy for HIV. Phlegmonous gastritis is a rare and rapidly progressive bacterial infection of the gastric wall. Kaposi sarcoma is one of the most common malignancies in HIV-positive patients, and gastric involvement is relatively common in those patients with systemic Kaposi sarcoma. To our knowledge, this is the first reported case of phlegmonous gastritis associated with Kaposi sarcoma, and it represents a rare survival following surgical and antibiotic therapy.

REPORT OF A CASE

A 37-year-old man presented to the emergency department with a 2-day history of abdominal pain, nausea, vomiting, watery diarrhea, and severe weakness. He denied any significant medical history. On physical examination, his temperature was 38.9°C; blood pressure, 100/60 mm Hg; pulse rate, 130/min; and respiration rate, 28/min. The abdomen was tender in all 4 quadrants, with guarding and rebound tenderness. Rectal examination revealed heme-positive stools. Laboratory data showed a white blood cell count of 1900/μL; hematocrit, 41.4%; serum urea nitrogen, 36 mg/dL; creatinine, 2.4 mg/dL; and glucose, 125 mg/ dL. Results of liver function tests were normal. Serum amylase and lipase levels were within normal limits. Computed tomographic scan of the abdomen demonstrated free fluid in the abdominal cavity and a thickened gastric wall. Subsequently, the patient underwent emergency laparotomy with partial gastrectomy. The greater curvature of the stomach from cardia to mid antrum was marginally viable and exhibited cyanosis, thickening, erythema, and a mild phlegmonous reaction in the lesser sac. About 800 mL of purulent fluid in the peritoneal cavity was aspirated, and culture of this fluid grew Streptococcus pyogenes (group A). Postoperative complications included pleural effusion, bacteremia, pneumonia, and respiratory failure. Cultures from bronchoalveolar lavage grew methicillin-resistant Staphylococcus aureus and Escherichia coli. Serologic screening for human immunodeficiency virus (HIV) was positive, with a CD4 count of 14.88/μL. The patient was kept on a ventilator and treated with vancomycin, ceftriaxone, piperacillin/tazobactam, ampicillin, azithromycin, metronidazole, and antiretroviral medications. After 8 weeks of therapy, he was afebrile and eating, and was discharged from the hospital.

PATHOLOGIC FINDINGS

Gross Findings

The partial gastrectomy specimen measured 18.5 × 7.0 × 3.0 cm and consisted of a portion of the greater curvature. The external surface of the stomach appeared dark red and dull with foci of fibrinopurulent exudates. The lumen was filled with hemorrhagic fluid. The mucosa was dark red and hemorrhagic and had an irregularly shaped 1.7 × 1.5-cm ulcer located 1.0 cm from the resection margin. Cut sections revealed a yellow-tan, edematous, and extensively thickened submucosa. The wall varied in thickness from 0.7 to 1.3 cm (Figure 1).

Figure 1.

Phlegmonous gastritis. A, Hemorrhagic mucosa with an irregularly shaped, depressed area. B, Cut section of gastric wall with marked thickening of submucosa (formalin-fixed specimen). Figure 2. A, Hemorrhagic intact mucosa with dense cellular infiltrate in submucosa (hematoxylin-eosin, original magnification ×40). B, Mucosa with congestion and cryptitis (hematoxylin-eosin, original magnification ×200). C, Submucosa shows suppurative and necrotizing inflammation (hematoxylin-eosin, original magnification ×200). D, Numerous chains of cocci in the submucosa (Brown-Brenn, original magnification ×1000). Figure 3. A, Elongated, plump spindle cells with pleomorphic nuclei and slitlike spaces filled with red blood cells (hematoxylin-eosin, original magnification ×400). B, Immunohistochemical staining for CD34 is positive in the spindle cells (original magnification ×400)

Figure 1.

Phlegmonous gastritis. A, Hemorrhagic mucosa with an irregularly shaped, depressed area. B, Cut section of gastric wall with marked thickening of submucosa (formalin-fixed specimen). Figure 2. A, Hemorrhagic intact mucosa with dense cellular infiltrate in submucosa (hematoxylin-eosin, original magnification ×40). B, Mucosa with congestion and cryptitis (hematoxylin-eosin, original magnification ×200). C, Submucosa shows suppurative and necrotizing inflammation (hematoxylin-eosin, original magnification ×200). D, Numerous chains of cocci in the submucosa (Brown-Brenn, original magnification ×1000). Figure 3. A, Elongated, plump spindle cells with pleomorphic nuclei and slitlike spaces filled with red blood cells (hematoxylin-eosin, original magnification ×400). B, Immunohistochemical staining for CD34 is positive in the spindle cells (original magnification ×400)

Microscopic Findings

On light microscopy, the mucosa was hemorrhagic, mostly intact, and showed chronic inflammation and small foci of acute cryptitis. An ulcerated focus covered with regenerating epithelium was present. The submucosa showed necrosis and hemorrhage with a pronounced inflammatory infiltrate consisting mainly of polymorphonuclear leukocytes. There were also edematous areas and thrombosed submucosal veins. Numerous Gram-positive cocci arranged in chains were identified within the submucosa. The muscularis propria was hemorrhagic and focally involved with inflammation and necrosis (Figure 2). A microscopic nodule of Kaposi sarcoma (KS) underlying the ulcerated area was present. It was composed of spindle cells forming slitlike spaces with extravasation of red blood cells. The spindle cells were positive with CD31 and CD34 and negative for factor VIII and Ulex europaeus by immunohistochemical stains (Figure 3).

COMMENT

Phlegmonous gastritis is a rare, rapidly progressive, and potentially fatal bacterial infection. It was probably first recognized by Galen (second century AD), who called it “erysipelatous tumor of the stomach.” Up until 1994, about 500 cases had been reported in the literature, with the majority described in the preantibiotic era.1 

Phlegmonous gastritis, also called suppurative gastritis, is a diffuse cellulitis of the stomach wall, although few cases have been reported with focal abscesses. It is most often caused by hemolytic group A streptococcus (S pyogenes)2; however, it can also be caused by Staphylococcus, Pneumococcus, Enterobacter, and Actinomyces species, as well as by Escherichia coli, Corynebacterium diphtheriae, or gas-forming bacteria, and others.2,3 Several mechanisms have been implicated, such as direct invasion through an area of injury, hematogenous spread, and lymphatic spread.3 Predisposing factors recognized are mucosal injury (eg, chronic gastritis, peptic ulcer disease, and complication of endoscopic procedure), achlorhydria (eg, gastric carcinoma and gastric surgery), and an immunocompromised state (eg, chronic alcoholism, general debility, and HIV infection).4 

Patients with the diffuse form of phlegmonous gastritis often present with symptoms of acute abdomen, including intense epigastric pain, guarding, nausea and vomiting, fever, and increased white blood cell counts. The most prominent histopathologic finding in phlegmonous gastritis is submucosal involvement. This shows an infiltration of polymorphonuclear leukocytes with foci of necrosis, thrombosis, and hemorrhage. The mucosa and muscularis propria are affected to a lesser degree. The treatment consists of combining surgery (total or partial gastrectomy) and appropriate antibiotics.1,5 However, despite aggressive treatment, mortality is still very high. The mortality rate before the advent of antibiotics was 92%.2 In 1975, Miller and colleagues6 reviewed a series of 25 cases of phlegmonous gastritis described since 1945 in the American literature. The overall mortality rate in their report was 67% after surgery and large doses of antibiotics. In 1994, Wakayama and colleagues1 collected 24 more cases from the English literature. The mortality rate with gastrectomy was 25%, nonresectional surgery (drainage) was 43%, and nonsurgical treatment with antibiotics was 88%. Early recognition and early therapy are important for survival. Iwakiri and colleagues7 reported a case of acute phlegmonous gastritis that was successfully treated with antibiotics in 1999. The patient was a previously healthy woman who had severe upper abdominal pain without signs of peritoneal irritation. Endoscopy showed red edematous gastric mucosa. Endoscopic ultrasonography showed a thickened antral wall with a mass in the gastric antrum. Acute phlegmonous gastritis, localized form, with an abscess was diagnosed. Cultures grew Streptococcus pneumoniae. The patient was treated with antibiotics alone and recovered.7 Hu and colleagues8 reported a similar case in 2000. Phlegmonous gastritis was diagnosed by contrast abdominal computed tomographic scan and endoscopic ultrasonography, and follow-up studies showed decrease in wall thickness. In our patient, there was not a definitive diagnosis prior to the emergency laparotomy.

Phlegmonous gastritis associated with acquired immunodeficiency syndrome (AIDS) was reported by Mittleman and Suarez3 in 1985; however, we are not aware of reports describing phlegmonous gastritis associated with KS. Kaposi sarcoma is a low-grade vascular neoplasm associated with human herpesvirus 8, also known as the KS-associated herpesvirus. There are 4 epidemiologic types of KS. Classic KS is an indolent cutaneous form mainly found on lower extremities and affecting older men of Mediterranean and Jewish origin. Endemic KS is found among black people in equatorial Africa and is not associated with immune deficiency.9 Immunosuppression-associated or transplantation-associated KS is often associated with KS-associated herpesvirus transmission after solid organ transplantation.10 Epidemic or AIDS-related KS is the most common tumor in people infected with HIV.9,11 

Acquired immunodeficiency syndrome–related KS has a high frequency of visceral involvement.12,13 This includes the oral cavity, gastrointestinal tract, and respiratory tract. Friedman et al14 found that the gastrointestinal tract is involved in approximately 40% of patients at initial diagnosis and in up to 80% at autopsy. Gastrointestinal tract involvement can occur in the absence of cutaneous disease.15 Because the KS lesions in the gastrointestinal system tend to be submucosal, the majority of gastrointestinal cases are asymptomatic and are only recognized by endoscopy. In the advanced stage, the lesions can cause hemorrhage, obstruction, ulceration, and perforation.

In the case we described, the patient was asymptomatic and with unknown HIV status until he presented with an acute abdomen due to phlegmonous gastritis diagnosed after partial gastrectomy. This was associated with a nodule of KS. A single layer of columnar epithelial cells that is consistent with recently healed ulcer covered the submucosal KS. This ulcer may have been the site of bacterial invasion that caused this immunocompromised patient to develop a severe life-threatening infection, namely, phlegmonous gastritis. Surgical partial gastrectomy and extensive antibiotic therapy, including vancomycin, ceftriaxone, piperacillin/tazobactam, ampicillin, azithromycin, and metronidazole during an 8-week period resulted in survival of this patient.

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The authors have no relevant financial interest in the products or companies described in this article.

Author notes

Reprints: Qing Qing Yu, MD, Department of Pathology and Laboratory Medicine, Mount Sinai Medical Center, 4300 Alton Rd, Blum Bldg 201, Miami Beach, FL 33140 (qingqyu@msmc.com)