Campylobacter insulaenigrae bacteremia with meningitis: a case report.

BACKGROUND: The bacterium Campylobacter insulaenigrae was first isolated from marine mammals of Scotland in 2004. Only one case of C. insulaenigrae infection in humans has been previously reported. CASE PRESENTATION: An 89-year-old Japanese man without dementia was admitted to our hospital, because he presented with a fever of 38 °C and weakness in right leg since 5 days. He had organized chronic subdural hematoma (CSH), and no history of pre-infection. At the time of admission, he had paralysis of the extraocular muscle, ataxia, and low manual muscle test score of the right side. He was suspected to have Miller Fisher syndrome; however, these symptoms improved without any treatment. On day 22 in the hospital, the patient presented a fever of 38.8 °C, left cranial nerve disorder, and hemiplegia. On day 25, the patient presented with signs of meningeal irritation; cerebrospinal fluid examination indicated an increase in the number of apocytes and a low glucose level. A contrast magnetic resonance imaging (MRI) scan of the patient's head indicated a contrast enhancement effect in his right meninges. The blood culture showed presence of spirillums; 16S rRNA gene sequencing confirmed that the spirillums in the blood culture were Campylobacter insulaenigrae (C. insulaenigrae). We started treatment with meropenem for bacteremia and meningitis. When the symptoms improved, meropenem was replaced with ampicillin, based on the result of the drug sensitivity test. The treatment continued for 4 weeks. CONCLUSIONS: We report the first case of meningitis caused by C. insulaenigrae bacteremia in humans, and the second clinical report of C. insulaenigrae infection in humans. The bacterial strains isolated from humans and marine mammals had different genotypes. This suggests that different genotypes could be responsible for differences in the hosts. Further case studies are needed to establish the reasons behind the difference in the manifestations of C. insulaenigrae infections reported so far.

Toxic shock syndrome with a cytokine storm caused by Staphylococcus simulans: a case report.

BACKGROUND: Exotoxins secreted from Staphylococcus aureus or Streptococcus pyogenes act as superantigens that induce systemic release of inflammatory cytokines and are a common cause of toxic shock syndrome (TSS). However, little is known about TSS caused by coagulase-negative staphylococci (CoNS) and the underlying mechanisms. Here, we present a rare case of TSS caused by Staphylococcus simulans (S. simulans). CASE PRESENTATION: We report the case of a 75-year-old woman who developed pneumococcal pneumonia and bacteremia from S. simulans following an influenza infection. The patient met the clinical criteria for probable TSS, and her symptoms included fever of 39.5 °C, diffuse macular erythroderma, conjunctival congestion, vomiting, diarrhea, liver dysfunction, and disorientation. Therefore, the following treatment was initiated for bacterial pneumonia complicating influenza A with suspected TSS: meropenem (1 g every 8 h), vancomycin (1 g every 12 h), and clindamycin (600 mg every 8 h). Blood cultures taken on the day after admission were positive for CoNS, whereas sputum and pharyngeal cultures grew Streptococcus pneumoniae (Geckler group 4) and methicillin-sensitive S. aureus, respectively. However, exotoxins thought to cause TSS, such as TSS toxin-1 and various enterotoxins, were not detected. The patient's therapy was switched to cefazolin (2 g every 8 h) and clindamycin (600 mg every 8 h) for 14 days based on microbiologic test results. She developed desquamation of the fingers on hospital day 8 and was diagnosed with TSS. Conventional exotoxins, such as TSST-1, and S. aureus enterotoxins were not detected in culture samples. The serum levels of inflammatory cytokines, such as neopterin and IL-6, were high. CD8+ T cells were activated in peripheral blood. Vß2+ population activation, which is characteristic for TSST-1, was not observed in the Vß usage of CD8+ T cells in T cell receptor Vß repertoire distribution analysis. CONCLUSIONS: We present a case of S. simulans-induced TSS. Taken together, we speculate that no specific exotoxins are involved in the induction of TSS in this patient. A likely mechanism is uncontrolled cytokine release (i.e., cytokine storm) induced by non-specific immune reactions against CoNS proliferation.

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.

Invasive Haemophilus influenzae type a infection and polyarthritis in a 72-year-old Japanese man: A case report.

Haemophilus influenzae is a small, nonmotile, non-spore-forming bacterium classified into 6 serotypes (a to f) and non-typeable strains that lack a capsule. Although H. influenzae serotype a (Hia) is prevalent in Canada, the United States, Brazil, Australia, across the African continent, and several other locations, it has not been reported in Japan thus far. Our case was of a 72-year-old Japanese man who sought medical consultation after presenting with chills, fever, and polyarthritis. Cultures of blood and synovial fluid from the left knee revealed H. influenzae infection. Diagnostic imaging showed poor contrast regions in both kidneys, fluid retention around both knee joints, the left shoulder joint, and both elbow joints. Subsequently, the patient was diagnosed with invasive H. influenzae infection accompanied by polyarthritis and renal infarction. 16S ribosomal RNA gene sequencing revealed that the bacterial strain was Hia. The patient was treated with antimicrobial agents and arthroscopic curettage. We present a case of invasive Hia infection accompanied by polyarthritis and renal infarction. To the best of our knowledge, this is the first case of Hia infection in Japan. The case is very rare considering that the disease occurred in an elderly patient who developed polyarthritis.

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.

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.

[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.

Two cases of pneumococcal spondylitis in the same household: a case report.

BACKGROUND: Pneumococci normally reside in the nasopharynx, and when individuals are in close contact with each other such as in a community or a family setting, it is transmitted from carriers and sometimes results in pneumonia. CASE PRESENTATION: Case 1: The patient was a 55-year-old woman who visited the hospital complaining of fever and headache. Lumbar pain occurred on hospital day 2, and purulent spondylitis was diagnosed using lumbar MRI. Blood culture results were positive for pneumococcus. Case 2: The patient was a 60-year-old male, and the husband of the woman in the Case 1. Fever and lumbar pain occurred on the same day similar to Case 1. Inpatient treatment was provided for pneumococcal bacteremia. Although no abnormalities were observed on the lumbar MRI scan taken on hospital day 2, purulent spondylitis was diagnosed by an MRI taken on hospital day 9. Both patients received appropriate antimicrobial treatment. When bacterial strain analysis was performed on samples from Cases 1 and 2, we noted that the capsule serotype was 12F, the drug sensitivity was similar, and the sequence typing matched completely, indicating that the causative bacteria for both cases were identical. CONCLUSIONS: Pneumococcal bacteremia and purulent spondylitis can occur in different members of a family simultaneously. Pneumococcal infection can transmit between two close family members; hence, whenever a close family member of an individual who has already been infected with pneumococcal infection, develops fever, the possibility of transmission must be considered.

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.

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.

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.

A case of epidemic myalgia with symptoms resembling acute purulent spondylitis and discitis.

BACKGROUND: Epidemic myalgia is a disease that presents with fever and extreme myalgia of the trunk due to an acute enterovirus infection. The trunk pain is mainly in the chest or in the epigastrium. We aimed to highlight a case of epidemic myalgia where initial diagnosis needed differentiation from acute purulent spondylitis and discitis. CASE PRESENTATION: A 33-year-old woman presented with fever, chills, and acute episodes of low back pain. The sole unusual finding was pain upon spinal percussion, limited to the 4th and 5th lumbar vertebrae. Spinal MRI showed no abnormality. Paired serum samples from disease days 4 and 15 showed a significant increase in coxsackievirus B3-neutralizing antibodies. Based on this course, we diagnosed epidemic myalgia. CONCLUSIONS: Epidemic myalgia should be considered when differentiating acute low back pain accompanied by fever.

Efficient open recruitment and perspectives of host families on medical student homestays in rural Japan.

We devised and assessed open recruitment of host families for medical student homestays in a rural area of Hyogo Prefecture, Japan, so that program organizers would not have to depend on professional and personal connections. The duration of the homestays was one night and two days, and they were conducted in August 2016, 2017, and 2018. The purpose of this community-based medical education program was to promote interactions between medical students and residents of Tamba area. The study asked one family member from each host family to complete a questionnaire after the homestay, and their experiences were evaluated in the study. The questionnaire results were analyzed using a visual analog scale (VAS; 0-100 mm). Thirty-three host families participated in the homestay program over three years. Results showed that VAS scores were high for enjoyment of homestays (VAS; 92.4 ± 13.0), continuation of the homestay program (91.7 ± 12.7), continuation of participation in the homestay program (89.2 ± 16.2), and desire for the homestay students to work in the area in the future (95.4 ± 6.3). The recruitment of host families through advertising was an efficient method for this community-based medical education homestay program. The results indicate that it is possible to attract more host families through open recruitment, which will contribute to the sustainability of the homestay program. Further research, including a follow-up of the students who participated and whether they chose a rural area or Tamba to practice is needed in the future. Since this is an ongoing program, further research in a similar format can be conducted in the future.

Acute kidney injury and acyclovir-associated encephalopathy after administration of valacyclovir in an elderly person with normal renal function: A case report and literature review.

INTRODUCTION: Acyclovir (ACV)-associated encephalopathy is related to an increase in plasma levels of 9-carboxymethoxymethylguanine, an ACV metabolite, and is often reported in patients with renal dysfunction. We report a case of ACV-associated encephalopathy with rapid progression of renal dysfunction after oral administration of valacyclovir (VACV) and review literature of previous ACV-associated encephalopathy cases. PATIENT CONCERNS: An 88-year-old man was diagnosed with herpes zoster. VACV (3000 mg/day) treatment was initiated. Serum creatinine (Cr) level was 0.80 mg/dL. However, irritability, memory impairment, and decreased responsiveness occurred after 3 days. The Cr level was 6.76 mg/dL on admission. DIAGNOSIS: He was diagnosed with ACV-associated encephalopathy with acute kidney injury. INTERVENTIONS: VACV was discontinued, hemodialysis was initiated on the day of admission, and then the signs and symptoms improved approximately 72 hours after the admission. CONCLUSION: Worsening of renal function and encephalopathy should be a focus when using VACV or ACV, regardless of age and original renal function. Acute kidney injury and ACV-associated encephalopathy may particularly occur in the elderly even when renal function is normal. Therefore, regular monitoring of renal function and consciousness is necessary during VACV treatment.

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.

Diagnosis of E. coli tricuspid valve endocarditis: a case report.

Escherichia coli is a rare cause of bacterial endocarditis. We report a case of an elderly man with cirrhosis who developed bacterial endocarditis due to E.coli. We review the literature that summarizes a total of 41 cases, with a predominance of elderly women, diabetics, and patients with urinary tract infections and prosthetic heart valves. E.coli bacteremia may be mistakenly attributed to a more benign source such as urosepsis and overlooked as an indication of endocarditis. A delay in diagnosis may lead to higher mortality as the opportunity for early valve replacement in appropriate cases and more prolonged courses of antibiotics may not be considered.

Late onset of hemolytic uremic syndrome after the appearance of prodromal gastrointestinal tract symptoms.

Hemolytic uremic syndrome (HUS) may occur late after the onset and improvement of gastrointestinal tract symptoms. Clinicians need to carefully monitor for the onset of HUS, even if the patients have few symptoms.

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.

Acceptance of a homestay program and attitude toward community medicine among medical students.

BACKGROUND: In community-based medical education, opportunities for medical students to interact with local residents are important. To facilitate such interaction, we aimed to evaluate acceptance of a homestay program and attitude toward community medicine among medical students. METHODS: The participants (n = 39) were allowed to stay in the local homes of residents for one night in August 2016, 2017, and 2018. Before and after the homestays, the students responded to a self-reported questionnaire using the visual analog scale (VAS; 0-100 mm). The questionnaire included four questions on homestay/community medical training and community medicine and four questions about attitude toward community medicine in the local areas of medical students. Then, we compared the VAS scores before and after training. RESULTS: The VAS scores for all questions about homestay/community medical training and community medicine significantly increased: "Is it worthwhile for you to have experience in the field of community medicine," "Did you find the homestay enjoyable," "Does the homestay add educational significance to the program," and "Is direct interaction with residents meaningful?" For the two questions about attitude toward community medicine, the VAS scores significantly increased: "Is there a challenge to practicing community medicine" and "In the future, do you want to work in Tamba area where you stayed?" CONCLUSIONS: The medical students were extremely enthusiastic about the educational program for community medicine involving residential homestays, which improved their attitudes toward practicing community medicine. Moreover, the students appreciated that their training sites could become their future workplaces.