Intestinal Tuberculosis with Hoarseness as a Chief Complaint due to Mediastinal Lymphadenitis.

A 68-year-old woman was admitted to our hospital complaining of hoarseness. A chest X-ray detected an abnormal shadow on the upper right lung. Bronchoscopic examination revealed that the left vocal cord was fixed in the paramedian position, and therefore left recurrent nerve paralysis was suspected. Lymphadenopathy was found in the left supraclavicular area. Chest computed tomography showed that the pretracheal and subaortic lymph nodes were swollen. Gastroendoscopy showed a 2-cm protruding lesion with ulceration on the upper esophagus. Histological examination of the supraclavicular lymph nodes and biopsy specimens from the esophagus revealed non-specific inflammation. PET-CT showed abnormal accumulations not only on the upper right lung but also on the lower right of the abdomen. Colonoscopy was performed and multiple erosions on the terminal ileum were found. Polymerase chain reaction analysis of a specimen biopsied from the erosion of the terminal ileum was positive for Mycobacterium tuberculosis and intestinal tuberculosis was diagnosed. The patient was then treated with anti-tuberculous therapy. After treatment, the erosions on the terminal ileum, the swelling of the mediastinal lymphadenopathy, and the esophageal ulcer were all improved. The hoarseness was subsequently relieved. This is the first report of intestinal tuberculosis with hoarseness as a chief complaint due to mediastinal lymphadenitis.

Catheter ablation for right ventricular outflow tract ventricular tachycardia induced by fever in a 7-year-old girl.

We report a 7-year old girl with ventricular tachycardia exacerbated by fever. She experienced her first symptom of palpitations during an influenza infection, and electrocardiogram showed wide QRS tachycardia of a left bundle branch block inferior axis pattern. We performed an electrophysiological study (EPS) for diagnosis, but the wide QRS tachycardia could not be induced. The wide QRS tachycardia recurred during upper respiratory tract infection with fever and ceased with lidocaine administration. Wide QRS tachycardia is characteristic of ventricular tachycardia (VT) exacerbated by fever. VT was not able to be induced at a normal temperature on EPS; however, when her temperature exceeded 38 degrees C, VT was easily induced and was accompanied by decreased blood pressure. Radiofrequency catheter ablation was performed in the right ventricular outflow tract (RVOT) just under the pulmonic valve, with a fractionated potential 29 ms before onset of V1 QRS. VT was terminated approximately 4 s after RF application, and then VT could not be induced with or without high fever. This is the first report of successful ablation treatment in RVOT VT exacerbated by fever in a young child.

Efficacy of ecabet sodium enema on intractable rectal ulcer after piecemeal endoscopic mucosal resection for rectal tumor.

We describe a case with rectal bleeding from a rectal ulcer after endoscopic mucosal resection (EMR), successfully treated with an ecabet sodium (ES) enema. A 44-year-old woman with a laterally spreading rectal tumor of a granular type, 60 mm in diameter, underwent piecemeal EMR. After the EMR, she suffered from rectal bleeding on several occasions over a period of 1 month. Although she was repeatedly treated with thermocoagulation by a heater probe to stop the bleeding, a rectal ulcer with visible vessels still remained at the resected site. Because the rectal ulcer was considered to be intractable, an ES enema was used twice a day (1.5 g) for 2 weeks, which improved rectal bleeding. Colonoscopic findings revealed that the ulcer improved with mucosal healing after the ES enema treatment. This represents the first report of an ES enema treatment in a patient with a rectal ulcer after EMR. Further studies are needed to determine the effectiveness and safety of using an ES enema in patients with EMR-related refractory colorectal ulcers.

Cheilitis granulomatosa as an early manifestation of Crohn's disease.

Cheilitis granulomatosa (CG) is a rare disease, which presents usually as a persistent swelling of the soft tissues in the orofacial region and is characterized histologically by a granulomatous inflammation. We report the case of a 19-year-old man who suffered from anal fistula. The patient had a 6-year history of asymptomatic and persistent swelling of the lower lip. Examinations for gastrointestinal lesions containing double-balloon total enteroscopy revealed erosions located longitudinally throughout the small intestine and the patient was diagnosed Crohn's disease (CD). Biopsy of the lower lip showed non-caseating granuloma and confirmed the diagnosis of CG. Despite an elemental diet and mesalazine therapy, the lip swelling persisted. The CG can be the first presenting symptom of CD. CG as a complication of CD is discussed.

Infliximab treatment for Crohn's disease in a patient with IgA nephropathy.

We describe herein a case of IgA nephropathy in a 34-year-old woman with Crohn's disease (CD) treated with infliximab. CD first appeared at the age of 15 years. An elemental diet was started for remission maintenance. Ten years later, the patient suffered from a recto-vaginal fistula and subtotal colectomy with stoma formation was performed. At the age of 33 years, the patient was investigated for painless macroscopic hematuria and proteinuria. Renal biopsy revealed IgA nephropathy. Mizoribine was started but proteinuria persisted. Due to diarrhea she was admitted to our hospital, and scheduled maintenance therapy with infliximab was initiated. After the first infliximab infusion, the patient presented significant clinical improvement in both diarrhea and proteinuria with concomitant decrease of C-reactive protein to normal levels and proteinuria ~1 g/day. This represents the first report of infliximab treatment in a patient with IgA nephropathy associated with CD and clarifies the importance of tumor necrosis factor-alpha (TNFα) in immunity to renal disease. Further studies are needed to draw firm conclusions for the safety of infliximab in patients with IgA nephropathy.

Selective decrease in colonic CD56(+) T and CD161(+) T cells in the inflamed mucosa of patients with ulcerative colitis.

AIM: To investigate the role of local colonic mucosal NK receptor-positive T (NKR(+) T) cells in the regulation of intestinal inflammation, we analyzed the population and function of these cells in ulcerative colitis (UC). METHODS: Colonic mucosal tissues were obtained from colonoscopic biopsies of the descending colon from 96 patients with UC (51 endoscopically uninflamed, 45 inflamed) and 18 normal controls. Endoscopic appearance and histologic score at the biopsied site were determined by Matts' classification. A single cell suspension was prepared from each biopsy by collagenase digestion. Two NKR(+) T cell subsets, CD56(+) (CD56(+)CD3(+)) T cells and CD161(+) (CD161(+)CD3(+)) T cells, were detected by flow cytometric analysis. Intracellular cytokine analysis for anti-inflammatory cytokine interleukin-10 (IL-10) was performed by in vitro stimulation with phorbol-myristate-acetate (PMA) and ionomycin. RESULTS: CD56(+) T cells and CD161(+) T cells are present in the normal human colon and account for 6.7% and 21.3% of all mononuclear cells, respectively. The populations of both CD56(+) T cells and CD161(+) T cells were decreased significantly in the inflamed mucosa of UC. In contrast, the frequency of conventional T cells (CD56(-)CD3(+) cells and CD161(-)CD3(+) cells) was similar among the patient and control groups. The populations of NKR(+) T cells were correlated inversely with the severity of inflammation, which was classified according to the endoscopic and histologic Matts' criteria. Interestingly, approximately 4% of mucosal NKR(+) T cells expressing IL-10 were detected by in vitro stimulation with PMA and ionomycin. CONCLUSION: Selective reduction in the population of colonic mucosal NKR(+) T cells may contribute to the development of intestinal inflammation in UC.