Intest Res.  2017 Jul;15(3):422-428. 10.5217/ir.2017.15.3.422.

Behçet's disease with multiple splenic abscesses in a child

• Lim KI
• Yang DH
• Ryoo E

• KMID: 2382388
• DOI: http://doi.org/10.5217/ir.2017.15.3.422

Abstract

We report the case of a 5-year-old male patient with multiple aseptic splenic abscesses associated with Behçet's disease. The patient visited Gachon University Gil Hospital with fever, abdominal pain, and acute watery and bloody diarrhea, and reported a 2-year history of chronic abdominal pain and intermittent watery diarrhea. He was treated with antibiotics at a local clinic for fever and cervical lymph node swelling. Additionally, he had recurrent stomatitis. A colonoscopy showed multiple well-demarcated ulcerations throughout the colon, and abdominal computed tomography showed multiple splenic abscesses. Pathergy and HLA-B51 tests were positive. Investigations did not reveal any infectious organisms in the aspirate obtained via ultrasound-guided fine needle aspiration. After steroid treatment, all symptoms and multiple aseptic splenic abscesses resolved. However, oral ulcers, genital ulcers, and abdominal pain recurred after tapering the steroids. Infliximab treatment improved the patient's symptoms. However, 5 months after the treatment, the symptoms recurred. The treatment was changed to include adalimumab. Subsequently, the patient's symptoms resolved and colonoscopic findings improved. No recurrence was noted after 3 months of follow-up.

Keyword

Behcet syndrome; Spleen; Abscess; Pediatrics

MeSH Terms

Abdominal Pain
Abscess*
Adalimumab
Anti-Bacterial Agents
Behcet Syndrome
Biopsy, Fine-Needle
Child*
Child, Preschool
Colon
Colonoscopy
Diarrhea
Fever
Follow-Up Studies
HLA-B51 Antigen
Humans
Infliximab
Lymph Nodes
Male
Oral Ulcer
Pediatrics
Recurrence
Spleen
Steroids
Stomatitis
Ulcer
Adalimumab
Anti-Bacterial Agents
HLA-B51 Antigen
Infliximab
Steroids

Figure

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  Fig. 1 Colonoscopic and abdominal CT findings at initial visit. (A) Multiple well demarcated ulcerations that covered with whitish plaques were noted on the cecum, A-colon, T-colon and D-colon. And friability was also noted near the ulcerative lesion. (B) Multiple abscesses were noted on the spleen (arrows).

• 

  Fig. 2 Abdominal ultrasonographic findings. (A) Before steroid therapy, multiple splenic abscesses were observed. The largest one is 0.88 cm sized. (B) After steroid therapy, multiple splenic abscesses were subsided at 1 month later.

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  Fig. 3 Pathologic findings. Erosion and nonspecific inflammation were found but no granuloma was observed in ileum and total colon. (A) Ileum (H&E, ×100), (B) ileum (H&E, ×200), (C) colon (H&E, ×100), and (D) colon (H&E, ×200).

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  Fig. 4 Re-aggravation of genital, perianal and intestinal Behçet's disease after steroid tapering. (A) Ulcerations were observed on the penis tip and perianal area. (B) Multiple round well demarcated ulcerations were noted on the T- and D-colon. And hemorrhagic erosive lesions were noted on the rectum.

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  Fig. 5 Follow-up colonoscopic findings after adalimumab administration. Some old scars of previous ulceration were found in terminal ileum, cecum, and colon.

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