Pediatr Surg Int (2004) 20: 155–157 DOI 10.1007/s00383-003-1112-4
CASE REPORT
George Sakellaris Æ Kyriakos Kakavelakis Eustathios Stathopoulos Æ Helen Michailidou Giorgos Charissis
A palpable right lower abdominal mass due to Yersinia mesenteric lymphadenitis Accepted: 18 February 2003 / Published online: 10 February 2004 Springer-Verlag 2004
Abstract Infection by Yersinia pseudotuberculosis has become of increasing pathological importance. This report describes the case of a 12-year-old female with mesenteric lymphadenitis due to Yersinia pseudotuberculosis. The patient presented with fever, abdominal pain, and a palpable right abdominal mass. Abdominal ultrasonic imaging and computerized axial tomography (CT) revealed a mass. An exploratory laparotomy was performed, followed by appendectomy and mesenteric lymph node biopsy. The diagnosis of Yersinia infection was confirmed by serology and bacterial culture of the biopsy material. This condition should be considered in patients with a right lower abdominal mass and symptoms similar to those of appendicitis.
more rarely presents as a palpable abdominal mass [4,6]. The mass consists of one or more inflamed or swollen mesenteric lymph nodes, and the infection is located most often in the ileocecal region. Often the diagnosis is made at laparotomy. We report a case of Yersinia mesenteric lymphadenitis with a palpable lower right iliac fossa mass. Imaging studies (ultrasound and computerized tomography [CT] scan) are presented, and their findings are discussed. We believe that with history, physical findings, and the methods of investigation now available, it is possible to avoid surgery in such cases. In this report, the main evidence that surgery could be avoided was the negative findings from the frozen section biopsy.
Keywords Yersinia pseudotuberculosis Æ Palpable abdominal mass Æ Childhood
Case report
Introduction Yersinia pseudotuberculosis is among the subgroups of Yersinia species [10]. Due to mesenteric adenitis, infection with this organism typically causes symptoms similar to those of appendicitis [2,8,10]. However, Yersinia mesenteric lymphadenitis is a rare condition, and even G. Sakellaris (&) 6, Daskalojianni Street, 70014 L. Hersonissou, Crete, Greece E-mail:
[email protected] G. Sakellaris Æ K. Kakavelakis Æ G. Charissis Department of Pediatric Surgery, University Hospital of Heraklion, University of Crete, 71110 Voutes, Crete, Greece E. Stathopoulos Department of Pathology, University Hospital of Heraklion, University of Crete, 71110 Voutes, Crete, Greece H. Michailidou Department of Pediatrics, University Hospital of Heraklion, University of Crete, 71110 Voutes, Crete, Greece
The patient was a 12-year-old girl referred from Chania General Hospital; she was admitted to our department on 7 April 2001, with a history of abdominal pain for 6 days, vomiting, headache, and a temperature of 38C. Physical examination revealed a patient in moderate distress secondary to abdominal pain. She weighed 50 kg. Her blood pressure was 120/66 mmHg, heart rate 80 beats/min, and respiratory rate 18 breaths/min. A mild degree of tenderness and muscular rigidity in the whole abdomen was present. She had pain localized to the right iliac fossa and flank, and a palpable right lower abdominal mass of about 9 cm. Bowel sounds were present, and there was no jaundice. Neurological and heart examinations were normal, and the lungs were clear. She had a history of G-6-PD deficiency. The laboratory data were as follows: white blood cell count 8,300/mm3 (75% neutrophils, 11.5% lymphocytes, and 12.3% monocytes), sedimentation rate 103 mm/h, and C-reactive protein level 19.5 mg/dl. Blood, stool, and urine cultures were negative. Laboratory results showed normal electrolytes, renal function, and glucose level. An abdominal CT and ultrasound examination revealed a mass 5.6 cm in diameter between the right psoas muscle and the bowel’s helices, which was thought to be mesenteric in origin. The density of the mass was similar to that of soft tissue (20–40 HU). There was moderate splenomegaly and a small amount of fluid in the pouch of Douglas (Fig. 1). An exploratory laparotomy was carried out, and an inflammatory mass 6 cm in diameter consisting of mesenteric lymph nodes was revealed in the right flank, surrounded by numerous smaller nodes 1–2 cm in diameter. The appendix, although
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Discussion
Fig. 1 Ultrasound revealed a hypoechogenic mass measuring 5.6·2.5 cm located in the mid-abdomen and to the right
Fig. 2 Part of granuloma consisting mainly of epithelial cells with cell necrosis, nuclear debris, and polymorphonuclears (HE stain, final magnification ·100) macroscopically normal, was removed. A biopsy specimen of the mass was taken for histology study, which revealed granulomatous lesions with an epithelioid cell layer and polymorphonuclear cell infiltration—typical features of Yersinia infection (Fig. 2). The frozen section biopsy excluded the possibility of malignancy. Pus was observed draining from the biopsy site, and a swab was taken for culture. This grew Yersinia pseudotuberculosis, serotype I. Histological examination of the appendix revealed periappendicitis with intramural inflammatory lesions surrounded by lymphocytes. The agglutination test for Yersinia pseudotuberculosis was positive at 1:400 in the girl’s serum. The patient was put on triple intravenous (IV) therapy with metronidazole, cefoxitin, and canamycin. On day 10 of therapy her pain had resolved, and the patient was discharged. She made a complete recovery. An additional abdominal ultrasound study showed a small amount of fluid in the pouch of Douglas and a remarkable minimizing of the mass (2.2 cm · 0.9 cm). At a followup visit 6 weeks later, the patient was feeling better; she had experienced no further abdominal complaints, and the laboratory evaluation showed normal values.
Yersinia pseudotuberculosis infection is rare, occurs worldwide, and most commonly presents with acute mesenteric lymphadenitis, which may mimic acute appendicitis in presentation [2, 4, 6, 9, 10]. The organism is a Gram-negative bacillus capable of growth at 4oC [2, 3]. The majority of Y. pseudotuberculosis cases (75%) have involved 5- to 20-year-old individuals. The mode of transmission in humans is by the oral route. Modes of presentation include mesenteric adenitis, gastroenteritis, peritonitis, or intestinal ulceration [6]. Life-threatening septicemia with Yersinia species is rare and usually occurs in patients with diabetes, cirrhosis, or immune deficiency. Moreover, the bacteria may cause other symptoms, such as scarlatiniform rash, arthritis, conjunctivitis, uveitis, erythema nodosum, and sterile pleural effusion [5, 6, 7, 9]. Mesenteric lymphadenitis, which is the most common clinical presentation, includes right lower abdominal pain, fever, and vomiting; it typically presents with a history similar to that of appendicitis [2, 8, 10]. However, there have been very few reports of Yersinia pseudotuberculosis infection presenting as a palpable abdominal mass that is due to pathological enlargement of the mesenteric lymph nodes in the ileocecal region [4, 6]. The differential diagnosis of right abdominal mass in childhood includes Yersinia infection, neuroblastoma, Wilms tumor, appendix abscess, Hodgkin’s disease, nonHodgkin’s lymphoma, dermoid tumors, inflammatory pseudotumors, metastases, and rare primitive mesenteric tumor [1, 4, 6]. The diagnostic clues that point toward Yersinia pseudotuberculosis infection as a cause of an abdominal mass are obtained from the following [4, 5, 6, 7, 9]: 1. Clinical findings, especially clinical signs in the skin, joint, or eyes, as well as abdominal pain and fever. 2. A suggestive epidemiological history. 3. CT scans, MRI, and ultrasound examination. With the primary enteric presentation of a mesenteric lymphadenitis or, possibly, with ultrasound visualization of the abdomen and pelvis, enlarged mesenteric lymph nodes and/or peritoneal findings, including appendiceal inflammation, periappendiceal fluid, and/or terminal ileitis, may also be revealed. 4. Serological studies. Agglutinations tests or ELISA are supplementary investigations in the diagnosis of yersiniosis because they take time, and antibody may be absent or in low titer in an acute phase sample. 5. Bacterial culture. Because this is a bacterial infection and ought not to be present in sterile fluids, the acquisition by culture from sources such as blood, cerebrospinal fluid, peritoneal fluid, synovial fluid, or other organ-based biopsy (e.g., intestinal tissue, skin) may be confirmatory. Positive blood cultures are rare. Isolation and subsequent identification of the organism is possible from stool cultures, although it
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is difficult given the slow growth pattern and overgrowth of normal fecal flora. 6. Histology and bacteriological culture of the specimen if laparotomy is carried out and a lymph node biopsy is taken. These steps are essential because although the affected appendix may appear normal, involved lymph nodes (mesenteric) typically show epithelioid granulomatous changes, lymphoid hyperplasia, coagulative necrosis, and histiocytic cell hyperplasia. Enteric lesions may be associated with crypt hyperplasia, microabscesses, and villus shortening. The histological findings of frozen section biopsy could rule out the possibility of malignancy. The relevant and important diagnostic clues in the present case came from clinical findings and imaging studies. The abdominal mass was revealed with CT and ultrasound visualization of the abdomen, which revealed typical characteristics of the mass (hypoechogenic, well demarcated, oval, and accompanied by numerous smaller mesenteric nodes and peritoneal thickening in the region of the terminal ileum and cecum). The laboratory tests (bacterial culture and histological and serological studies) supported the clinical and radiological diagnosis. We noted the coexistence of periappendicitis. The triple IV antibiotic therapy was successful.
Conclusion Although very rare, yersiniosis should be considered in the differential diagnosis of an abdominal mass that is accompanied by abdominal complaints and fever. High clinical suspicion and careful assessment of imaging and laboratory findings are required for making the diag-
nosis. Early diagnosis of Yersinia mesenteric lymphadenitis and appropriate treatment with antibiotics could in some cases prevent the need for surgery. If complications such as severe abdominal pain, acute abdominal presentations, peritoneal findings, or, uncommonly, intussusception occur, then exploratory laparotomy may be warranted.
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