Successful Conservative Management of Emphysematous Cystitis With Pneumoperitoneum: A Case Report and Literature Review.

Emphysematous cystitis is a rare type of urinary tract infection that is characterized by the accumulation of gas within the walls and lumen of the urinary bladder. In rarer instances, pneumoperitoneum may accompany emphysematous cystitis. When pneumoperitoneum is suspected through imaging studies in patients with emphysematous cystitis, surgical abdominal exploration is frequently performed considering the possibility of bladder perforation or coexistence of intestinal perforation. We successfully managed a case of emphysematous cystitis accompanied with pneumoperitoneum conservatively. A 90-year-old woman hospitalized with a gastric ulcer developed abrupt lower abdominal pain and hematuria. Contrast-enhanced CT revealed gas within the bladder wall, which was consistent with emphysematous cystitis, and pneumoperitoneum. No obvious bowel or bladder perforation was observed in the CT scan. Regarding her high surgical risk and clinical stability, surgical abdominal exploration was not performed, and she was managed conservatively with urethral catheter placement and antibiotics. She recovered with the treatment, and CT imaging obtained on day 18 demonstrated resolution of the bladder wall emphysema and no signs of pneumoperitoneum. We performed a literature review using MEDLINE and Japana Centra Revuo Medicina Web and confirmed 13 previously reported cases of emphysematous cystitis and pneumoperitoneum. Based on the review of these 13 cases and our case, it is difficult to predict the presence of bladder perforation solely based on peritoneal signs in physical findings or ascites on CT scans. Therefore, it would be preferable to perform surgical abdominal exploration to make a definite diagnosis and select an appropriate treatment. However, the fact that at least eight out of the 10 cases managed conservatively survived suggests that there is a specific clinical entity among patients who present with emphysematous cystitis and pneumoperitoneum that can be safely managed conservatively. Further accumulation of cases and research is necessary to determine which cases can be treated conservatively.

Spontaneous Recovery of Hemophagocytic Lymphohistiocytosis Due to Primary Epstein-Barr Virus Infection in an Adult Patient.

BACKGROUND Hemophagocytic lymphohistiocytosis (HLH) is a rare life-threatening systemic hyperinflammatory condition. Most adult HLH cases are secondary to infection, malignancy, and rheumatic diseases. Epstein-Barr virus (EBV) infection is the most frequent cause of infection-induced HLH. Early treatment with dexamethasone, etoposide, and cyclosporine is generally recommended for adult patients with HLH. However, this treatment regimen was established based on pediatric clinical trial data; thus, its efficacy and validity in adults remain unclear. Because little is known about the disease course of untreated adult EBV-associated HLH (EBV-HLH), we report a case of an adult patient who recovered from EBV-HLH spontaneously without specific treatment and discuss potential treatment strategies. CASE REPORT A 34-year-old man presented to the emergency department with a 7-day history of fever, headache, and sore throat. The main laboratory test abnormalities were elevated liver enzymes, hyperbilirubinemia, hypertriglyceridemia, and hyperferritinemia. Serologic tests confirmed acute primary EBV infection. He was diagnosed with EBV-HLH based on the HLH-2004 diagnostic criteria and the HLH probability calculator (HScore). Because he was clinically stable, we did not initiate immunosuppressive/cytotoxic treatment targeting HLH. High-grade fever persisted, but the abnormalities in his laboratory data improved spontaneously, and he did not develop major organ failure. His fever resolved on day 29 without HLH-specific treatment. CONCLUSIONS In clinically stable adult patients with EBV-HLH without major organ failure, it might be an acceptable alternative to observe the patient for several weeks before initiating HLH-specific treatment. Further research is required to better predict the subset of patients who can be safely observed without treatment.

Portal and Mesenteric Vein Thrombosis Associated with Hyperthyroidism.

BACKGROUND Non-malignant and non-cirrhotic portal and mesenteric vein thrombosis is rare. It has been reported that the hyperthyroid state is associated with increased risks of venous thrombosis due to increases in levels of various coagulation and anti-fibrinolytic factors. Particularly, changes in levels of these factors are also reported in cases of portal and mesenteric vein thrombosis. Although hyperthyroidism is not known as a risk factor for portal and mesenteric vein thrombosis, it might be an underlying pathogenesis of hyperthyroidism-associated portal and mesenteric vein thrombosis. CASE REPORT A 59-year-old Japanese man with a history of Grave's disease presented with acute portal and mesenteric vein thrombosis and hyperthyroidism. Anticoagulation therapy was initiated and the dose of antithyroid drug was increased. He underwent various tests to identify causes of portal and mesenteric vein thrombosis. However, all test results were within normal range except for hyperthyroidism. Therefore, we discontinued anticoagulation therapy after normalization of thyroid hormone status. After 3 years, he experienced recurrence of portal vein thrombosis concomitant with hyperthyroidism. CONCLUSIONS Hyperthyroidism might be associated with portal vein thrombosis. Thyroid function tests should be performed in cases of portal and mesenteric vein thrombosis in the absence of other risk factors.