Bacteremia caused by Desulfovibrio desulfuricans with the intestinal tract as the portal of entry: two case reports and a literature review.

BACKGROUND: Desulfovibrio desulfuricans (D. desulfuricans), a commensal anaerobic gram-negative rod endemic to the soil environment and human gastrointestinal tract, rarely causes bloodstream infections. We report two rare cases of bacteremia caused by D. desulfuricans in which the intestinal tract was the portal of entry. In addition, we summarize findings on D. desulfuricans. CASE PRESENTATION: Case 1: A 51-year-old man presented to the emergency department with the chief complaints of fever and right lower abdominal pain. He was admitted to the hospital with ascending colonic diverticulitis and received empirical antibacterial therapy with piperacillin/tazobactam. Blood culture revealed D. desulfuricans. The patient was discharged after 2 weeks of antimicrobial therapy. Case 2: A 95-year-old woman presented to our hospital with a chief complaint of fever. Owing to an elevated inflammatory response and pyuria, the patient was diagnosed with pyelonephritis and treated with ceftriaxone. Klebsiella pneumoniae was detected in her urine culture, while D. desulfuricans was detected in her blood culture. The patient was then treated with ampicillin/sulbactam for 14 days. The fecal occult blood test result was positive, suggesting a colonic mucosal lesion, such as a malignant tumor, may have been the portal of entry for D. desulfuricans bacteremia. Previous literature reviews indicate that D. desulfuricans bacteremia often results from liver or renal abscesses, intestinal lesions, among others, serving as the portal of entry. Although no specific underlying disease has been reported, it is more common in the older population. We encountered two cases of D. desulfuricans bacteremia and combined them with 15 cases from previous studies to explore the characteristics of the disease. The proportion of patients aged [Formula: see text]60 years was 73.7%; overall, 73.7% had gastrointestinal complications, and 63.2% had abdominal symptoms at the time of presentation. CONCLUSIONS: We encountered two rare cases of D. desulfurican bacteremia. This type of bacteremia is more common in elderly people over 60 years of age and is often associated with hepatobiliary and gastrointestinal diseases.

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.

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.

Heparin-induced thrombocytopenia during IgA vasculitis: a case report.

Immunoglobulin A (IgA) vasculitis is characterized by small vessel vasculitis involving immune complexes and IgA deposition. The development of heparin-induced thrombocytopenia (HIT) during IgA vasculitis is extremely rare. An 87-year-old man presented with general fatigue, leg edema, purpura, arthritis and renal disease. He was diagnosed with IgA vasculitis and was admitted to our hospital. Hemodialysis with heparin was initiated thrice a week on post-admission Day 11. On Day 21, during hemodialysis, the pressure in the dialysis circuit increased and the dialysis was interrupted. On Day 24, the platelet count rapidly decreased to 18 000/µl. The patient was diagnosed with HIT after testing positive for HIT antibodies; heparin was discontinued at this time. Precautions must be taken against the onset of HIT when initiating hemodialysis in patients with IgA vasculitis.