Multiple thrombosis associated with Cytomegalovirus enterocolitis in an immunocompetent patient: a case report.

BACKGROUND: Cytomegalovirus (CMV) is reported to have thrombogenic characteristics that activate factor X in vitro and stimulate the production of factor VIII and von Willebrand factor (vWF). Thrombosis associated with CMV infection is prevalent among immunocompromised patients and predominantly presents as a solitary large thrombus in the deep vein, pulmonary artery, splanchnic arteriovenous ducts, or other similar sites. Multiple thrombi, however, are rarely observed in such cases. Here, we report about an immunocompetent man with multiple microthrombi associated with CMV infection. CASE PRESENTATION: A 72-year-old Japanese man who complained of abdominal pain was hospitalized with multiple colonic stenosis. He was later diagnosed with CMV enterocolitis and treated with ganciclover from Day 27 post-admission. During hospitalization, the patient developed thrombi in his fingers. He was initially treated with anticoagulant therapy (rivaroxaban); however, the therapy was discontinued owing to a prolonged activated thromboplastin time and an elevated international normalized ratio of prothrombin time. Instead, vitamin K and fresh-frozen plasma were administered. Nevertheless, his coagulation profile remained abnormal. Eventually, he developed colonic perforation and had to undergo emergency surgery. An intraoperative specimen showed several microthrombi in the middle and small arteriovenous ducts of his small and large intestines. The patient's coagulopathy improved preoperatively, and his overall condition improved postoperatively. Since the activation of ADAMTS13 was reduced remarkably, the thrombotic tendency was determined to be a thrombotic microangiopathy-like condition owing to increased vWF. We could not attribute the coagulopathy to any other cause except CMV infection; therefore, we concluded that this was a case of multiple thrombosis associated with CMV. CONCLUSIONS: We present an extremely rare case of a patient with multiple thrombotic microangiopathy-like microthrombosis caused by CMV infection. Our findings suggest that CMV infection may be considered as a differential diagnosis for immunocompetent individuals who present with thrombosis of unspecified cause.

Methotrexate-associated lymphoproliferative disorder with an osteolytic vertebral lesion in an elderly patient with rheumatoid arthritis: A case report.

WHAT IS KNOWN AND OBJECTIVE: Methotrexate-associated lymphoproliferative disorder (MTX-LPD) is a rare complication that develops in patients treated with methotrexate (MTX). CASE SUMMARY: A 76-year-old male patient had been taking MTX for his rheumatoid arthritis. Computed tomography (CT) revealed masses in the liver, right adrenal gland and T6-T7 vertebra, including an osteolytic lesion. FDG-PET scan showed increased uptake in each lesion. MTX was discontinued, and CT showed complete remission of the tumours after three months. The disease course confirmed MTX-LPD diagnosis. WHAT IS NEW AND CONCLUSION: Bone lesions in LPDs mimic those of metastatic cancer. MTX-LPD should be considered in patients on MTX presenting with mass lesions.

Encephalopathy Induced by Preventive Administration of Acyclovir in a Man with Symptomatic Multiple Myeloma and Renal Dysfunction.

BACKGROUND: Acyclovir (ACV) neurotoxicity is a neuropsychiatric condition induced by the anti-herpetic drugs ACV and valacyclovir (VACV). It is presumed that elevated blood levels of ACV and its metabolite 9-carboxymethoxymethylguanine are involved in the development of ACV-induced encephalopathy; age and renal dysfunction are risk factors. Here, we report a case of encephalopathy caused by the administration of VACV for herpes zoster prophylaxis in a patient with renal dysfunction owing to multiple myeloma. CASE PRESENTATION: Renal dysfunction was diagnosed in a 70-year-old man visiting our hospital for a medical checkup. His creatinine clearance rate was 8 mL/min. He was diagnosed with symptomatic multiple myeloma, and bortezomib/dexamethasone (BD) therapy for multiple myeloma and VACV for herpes zoster prophylaxis were initiated. We administered 500 mg/day of VACV three times a week, a lower dosage than recommended, after adjusting for his renal impairment. His renal function was monitored twice per week during therapy. During the second course of BD therapy, 6 weeks after starting treatment, he was hospitalized owing to impaired consciousness (Glasgow Coma Scale score: E2, V4, M4), and his BD and VACV therapy were suspended. Brain magnetic resonance imaging and cerebrospinal fluid analysis showed no abnormalities. Three days after discontinuing BD and VACV therapy, his consciousness recovered completely, and impaired consciousness did not recur after resuming BD therapy. His clinical diagnosis was thus ACV-induced encephalopathy. CONCLUSION: VACV is often prescribed to patients with multiple myeloma receiving BD therapy to prevent herpes zoster. ACV-induced encephalopathy is commonly observed in patients with renal dysfunction; especially among patients with multiple myeloma with Bence-Jones proteinuria, renal tubules are easily damaged and plasma ACV concentrations are likely to increase and induce ACV-induced encephalopathy. Careful monitoring of the level of consciousness is necessary during preventive ACV therapy in patients with renal dysfunction.

End-of-life home care of an interstitial pneumonia patient supported by high-flow nasal cannula therapy: A case report.

BACKGROUND: High-flow nasal cannula (HFNC) therapy and morphine continuous subcutaneous infusion (CSI) have been used to ameliorate dyspnea in non-cancer patients with end-stage respiratory diseases, including chronic obstructive pulmonary disease and interstitial pneumonia, primarily in hospital settings. However, it is rare to perform home-based medical treatment using these. We observe a case to assess the feasibility of this treatment strategy. CASE SUMMARY: Here, we report a case of a 75-year-old man who was diagnosed with interstitial pneumonia 11 years ago and was successfully nursed at home during his terminal phase for over 10 mo without hospitalization, by introducing domiciliary uses of HFNC and morphine CSI with a patient-controlled analgesia device. CONCLUSION: Active utilization of HFNC and morphine CSI with patient-controlled analgesia device would substantiate successful end-of-life palliative home care of idiopathic interstitial pneumonia patients.

[Curd Formation Resulting from a Reaction between Cranberry Juice and Enteral Formula].

White solids were observed in the esophagus of a patient with multiple system atrophy. The patient was receiving enteral nutrition with a polymeric formula and cranberry juice via nasogastric feeding. To test the assumption that the precipitates were formed from a reaction between the juice and the formula, a verification experiment was conducted usingformulae of differinging redients, pH and protein content. The results indicated that a precipitate was formed when formulae with lower pH values and higher protein content were used. Mixing a pH neutral enteral formula with cranberry juice, vinegar or their 2- fold diluted solutions may result in the formation of precipitates in the stomach and esophagus.

Adult-onset Still's disease with macrophage activation syndrome diagnosed and treated based on cytokine profiling: a case-based review.

Adult-onset Still's disease (AOSD) is a relatively rare systemic inflammatory disorder and is diagnosed using various sets of classification criteria, with the Yamaguchi criteria as the most widely used criteria. Herein, we present the case of a 21-year-old woman admitted with a high fever, lasting for over 1 month, who did not fulfill the Yamaguchi criteria. However, by analyzing the inflammatory cytokine profile, we defined this case as AOSD based on a greatly elevated serum interleukin-18 level. In addition, we predicted the occurrence of macrophage activation syndrome by a characteristic increase in the soluble tumor necrosis factor receptor II level, which allowed a timely intervention for this malicious complication. Therefore, we suggest that cytokine profiling will be useful for the diagnosis and management of AOSD.

A case report of myocarditis combined with hepatitis caused by herpes simplex virus.

BACKGROUND: Viral myocarditis presents with various symptoms, including fatal arrhythmia and cardiogenic shock, and may develop into chronic myocarditis and dilated cardiomyopathy in some patients. We report a case of viral myocarditis and hepatitis caused by herpes simplex virus. CASE PRESENTATION: A 20-year-old woman was admitted to our hospital with fever, fatigue, and anorexia. The initial investigation showed elevated liver enzyme levels and elevated creatine phosphokinase, and computed tomography showed diffuse swelling and internal heterogeneous image in the liver. These findings were consistent with acute hepatitis; therefore, we performed a liver biopsy, which showed parenchymal necrosis and lymphocytic infiltration. The night that the liver biopsy was performed, blood pressure gradually decreased and revealed cardiogenic shock. Electrocardiography showed diffuse ST-segment elevation, and echocardiography showed a dilated, spherical ventricle with reduced systolic function and pericardial effusion. An endomyocardial biopsy revealed lymphocyte infiltration of the myocardium, confirming acute myocarditis. After a few days, tests for immunoglobin M and immunoglobin G antibodies against herpes simplex virus were positive. CONCLUSIONS: We presented a rare case of myocarditis combined with hepatitis that was caused by herpes simplex virus. Acute myocarditis can occur concurrently with hepatitis, pancreatitis, nephritis, and encephalitis; thus, determining the presence of other infectious lesions is necessary to provide appropriate treatment for the patient.

RS3PE syndrome developing during the course of probable toxic shock syndrome: a case report.

BACKGROUND: Remitting seronegative symmetrical synovitis with pitting edema (RS3PE) is a rare syndrome characterized by "remitting," "seronegative" (namely rheumatoid factor-negative), and "symmetrical" synovitis with pitting edema on the dorsum of the hands and feet. Recently, there have been reports that serum vascular endothelial growth factor (VEGF) is elevated in this condition. CASE PRESENTATION: An 85-year-old man visited our department with a rash that had appeared 2 days earlier and a fever that had developed on the day of his visit. Based on clinical findings of fever, erythema exudativum multiforme, transitory hypotension, conjunctiva hyperemia, elevated creatine kinase, and desquamation, we suspected toxic shock syndrome (TSS). Therefore, we started treatment with vancomycin (1 g/day) and clindamycin (600 mg/day), after which his fever rapidly remitted. However, pitting edema on the dorsum of his hands and feet appeared on day 7, and the patient also had painful wrist and ankle joints. Additional tests were negative for rheumatoid factor, and anti-cyclic citrullinated protein antibodies were < 0.2 U/mL. Further, serum matrix metalloproteinase-3 (199.6 ng/mL; reference value ≤123.8 ng/mL) and serum VEGF (191 pg/mL; reference value ≤38.3 pg/mL) levels were elevated, and human leukocyte antigen-A2 was detected. The patient was thus diagnosed with RS3PE syndrome, for which he satisfied all four diagnostic criteria: 1) pitting edema in the limbs, 2) acute onset, 3) age ≥ 50 years, and 4) rheumatoid factor negativity. He was treated with oral prednisolone, resulting in the normalization of his serum VEGF level to 34.5 pg/mL 1 month after starting treatment. It is currently 1 year since disease onset, and although the patient has stopped taking prednisolone, there has been no recurrence of RS3PE syndrome. CONCLUSIONS: To the best of our knowledge, this is the first reported case of a patient developing RS3PE syndrome during the clinical course of TSS. We propose that the onset mechanism involved an increase in blood VEGF due to TSS, which induced RS3PE syndrome. As serum VEGF becomes elevated with both severe infections associated with shock and RS3PE syndrome, awareness that these conditions can occur concurrently is essential.

An extremely rare case of tubo-ovarian abscesses involving corynebacterium striatum as causative agent.

BACKGROUND: We present an extremely rare case of tubo-ovarian abscesses involving Corynebacterium striatum (C. striatum) as causative agent in a 53-year-old woman. CASE PRESENTATION: The patient presented with stomach pain, chills, and nausea. Her medical history included poorly controlled psoriasis vulgaris and diabetes. Laboratory and imaging findings led to diagnosis of septic shock due to tubo-ovarian abscesses. She was treated with antibiotic therapy and surgery to remove the left adnexa. Various cultures detected Prevotella spp. and C. striatum. We concluded that C. striatum from skin contaminated by psoriasis vulgaris had caused the tubo-ovarian abscesses by way of ascending infection. CONCLUSIONS: This may be the first known case of tubo-ovarian abscesses due to C. striatum. In patients whose skin has been weakened by psoriasis vulgaris or other infections, Corynebacterium should be considered as causative microorganisms, and antibiotic therapy including vancomycin should be administered.

Delivery may affect arterial elasticity in women.

BACKGROUND: Estrogen is considered to be cardioprotective, but estrogen replacement therapy for postmenopausal women has not shown results for either primary or secondary cardiovascular event prevention. During normal pregnancy, women have significantly higher levels of estrogen and it may be endogenous estrogen that helps prevent atherosclerosis. METHODS AND RESULTS: The present cross-sectional study examined the association between pregnancy followed by delivery and clinical atherosclerosis using the brachial-ankle pulse wave velocity (PWV). A total of 2,560 women undergoing annual health screening at the Institute of Hyogo Prefecture Health Promotion Association in Japan were recruited. Pregnancy history (the age of menarche/menopause and the number of gravida/para), conventional coronary risk factors, and brachial-ankle PWV were recorded. Multivariate linear regression by stepwise selection analysis demonstrated that women who had 1 or more deliveries had a significantly lower PWV, independent of age and other conventional coronary risk factors. CONCLUSIONS: Pregnancy followed by delivery may decrease arterial stiffness and prevent the progress of atherosclerosis in women. The contribution of such a pregnancy followed by delivery-related decrease in arterial stiffness to the reduction of cardiovascular disease in women should be further evaluated.

Endothelial lipase gene polymorphism is associated with acute myocardial infarction, independently of high-density lipoprotein-cholesterol levels.

BACKGROUND: Endothelial lipase (EL) is a major determinant of high-density lipoprotein-cholesterol (HDL-C) metabolism and promotes monocytes recruitment. The local expression of EL could influence atherogenesis directly, in addition to its systemic role in HDL metabolism. The EL gene has a common 584C/T polymorphism, but it is unclear whether this polymorphism is associated with HDL-C levels or acute myocardial infarction (AMI). METHODS AND RESULTS: A case-control study of 107 AMI patients and 107 control subjects was conducted. T allele frequency was lower in the AMI group than in controls (0.18 vs 0.26, p<0.05). No significant association was found between the 584C/T polymorphism and HDL-C levels. Multivariate regression analyses showed that the association of the T allele with AMI was statistically significant and independent of other risk factors when age, sex, hypertension, hypercholesterolemia, and diabetes mellitus were included in the analyses (odds ratio (OR), 0.52; 95% confidence interval (95% CI) 0.28-0.98; p=0.04). However, when smoking status was included, the association of the T allele with AMI did not remain statistically significant (OR, 0.61; 95% CI 0.32-1.18; p=0.14). CONCLUSIONS: The 584C/T polymorphism of the EL gene was associated with AMI independently of HDL-C levels and thus may be involved in the pathogenesis of AMI.

High-grade neuroendocrine carcinoma of the lung presenting an unusual spread mimicking pleural mesothelioma associated with dermatomyositis.

Neuroendocrine tumors of the lung comprise a heterogeneous group of tumors that represents a spectrum of disease from typical carcinoid tumors to the high-grade neuroendocrine carcinomas (large-cell neuroendocrine carcinomas and small-cell carcinomas). The high-grade neuroendocrine carcinomas are characterized by early metastasis and poor prognosis. The peripheral location and especially the massive pleural spread are rare for a high-grade neuroendocrine carcinoma. We report a case in which a high-grade neuroendocrine carcinoma, associated with dermatomyositis, presented an unusual pattern of progression, mimicking malignant pleural mesothelioma on diagnostic imaging.

New methods to evaluate endothelial function: Non-invasive method of evaluating endothelial function in humans.

Evidence has accumulated that impairment of vascular endothelial function is the initial step in the development of atherosclerosis. One important finding is impairment of the release of endothelium-dependent relaxing factor, which is now thought to be nitric oxide or its related substances, from endothelial cells. Flow-mediated dilatation has been known to be endothelium-dependent, and this can be detected during reactive hyperemia by high-resolution ultrasound in superficial arteries. Several coronary risk factors have been reported to be significantly related with decreased flow-mediated dilatation. We studied the association between the accumulation of coronary risk factors (hyperlipidemia, diabetes mellitus, hypertension, and current smoking habitus) and vascular endothelial function. The lower incidence of atherosclerosis in women before the menopause than that in men is an established epidemiological observation. Short-term estrogen therapy improves endothelial function in postmenopausal women. However, there are few reports on its long-term effects on endothelial function. Furthermore, we determined whether a reduced dosage of estrogen may maintain its beneficial effects. A similar improvement was also observed while women were on hormone replacement therapy even at the reduced dosage. Our results indicate that even at half the dose of estrogen, hormone replacement therapy may improve endothelial function in postmenopausal women.