Whipple's disease with normal duodenal histology diagnosed by ileal biopsy using balloon endoscopy.

A 34 year-old previously healthy Japanese woman presented with a 4 year history of migratory arthralgia, chronic diarrhea, mild fever, and 10 kg weight loss. She was initially misdiagnosed with seronegative arthritis, followed by eosinophilic gastroenteritis. Oral prednisolone was found to be ineffective. Upper endoscopy revealed normal duodenal mucosa. Duodenal biopsy specimen indicated no abnormalities. However, balloon endoscopy revealed white shaggy villi in the ileum, and a biopsy specimen from which revealed diffuse infiltration of foamy macrophages in the lamina propria. These macrophages contained diastase-periodic acid-Schiff positive granules, consistent with Whipple's disease. Polymerase chain reaction analysis of DNA extracted from the ileum was compatible with Whipple's disease. In most previously reported cases of Whipple's disease, the duodenum was affected and duodenal biopsy specimens led to a diagnosis of Whipple's disease. Whipple's disease with normal duodenal histology is extremely rare and balloon endoscopy might be a useful diagnostic tool in such a case. The patient was treated with intravenous ceftriaxone and oral trimethoprim-sulfamethoxazole, and her symptoms improved one month after treatment. Balloon endoscopy performed 32 months later revealed complete improvement in lesions of the ileum.

Early onset bioprosthetic tricuspid valve stenosis in a case with cardiac sarcoidosis: Pathological findings based on autopsy.

A 60-year-old woman with cardiac sarcoidosis developed recurrent and refractory right heart failure 26 months after tricuspid valve replacement. Echocardiography revealed thickened and immobile cusp with increased diastolic tricuspid gradient of 8-10 mmHg, consistent with bioprosthetic tricuspid stenosis (TS). Prolonged intravenous injection of dobutamine and carperitide, with intermittent intravenous furosemide, was necessary at multiple times. Despite treatment, the patient died of refractory right heart failure. The explanted tricuspid bioprosthesis on autopsy revealed marked pannus formation, resulting in stiff and immobile cusps while the same mitral bioprosthesis, which was implanted on the same day, was normal. Sarcoid granulomas were not present either in tricuspid or mitral bioprostheses. Chronic valve inflammation associated with prolonged use of intravenous drugs and multiple episodes of line-associated bacteremia may have caused early onset bioprosthetic TS. Learning objectives:1Early onset bioprosthetic tricuspid stenosis (TS) is rare.2Elevated jugular venous pulse and pan-diastolic rumble with the Rivero-Carvallo sign are keys to the diagnosis of TS which is confirmed using echocardiography.3Repeated episodes of bacteremia associated with prolonged infusion of intravenous drugs might have contributed to the development of early onset bioprosthetic TS.