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.

Bilateral bartholin's gland abscesses in a 4-year-old girl with vitamin a deficiency: a case report.

BACKGROUND: A Bartholin's gland abscess is one of the most common infections in women of reproductive age. Although Bartholin's gland abscesses have been reported in prepubertal children, they are rarer in prepubertal children than in adults. Herein, we report a case of bilateral Bartholin's gland abscesses in a 4-year-old girl with vitamin A deficiency. CASE PRESENTATION: A 4-year-old girl diagnosed with autism spectrum disorder was admitted to the hospital for close examination and treatment because of persistent fever and malaise. The child was a marked fussy eater and was diagnosed with corneal ulceration and night blindness secondary to vitamin A deficiency. Both of the patient's labia were swollen, and a diagnosis of a bilateral Bartholin's gland abscess was made using computed tomography. Incisional drainage was performed under general anesthesia. The patient's postoperative course was uneventful, and she was discharged from the hospital on day 8 after the surgery. During hospitalization, attempts were made to correct the vitamin deficiency by adding nutritional supplements to the diet. Three months after the surgery, no recurrence of abscesses was noted. CONCLUSIONS: Decreased immunocompetence and mucosal barrier function due to vitamin A deficiency is thought to be the underlying cause of Bartholin's gland abscesses. Although prepubertal Bartholin's gland abscesses have been reported, they are rare. To the best of our knowledge, no reports of bilateral Bartholin's gland abscesses potentially caused by vitamin A deficiency have been reported. When prepubertal girls present with Bartholin's gland abscesses, the presence of immunodeficiency due to vitamin or trace element deficiency should also be considered.

Type I Kounis syndrome in a young woman without chest pain: a case report.

BACKGROUND: Kounis syndrome is defined as the concurrence of acute coronary syndromes in the setting of allergic or anaphylactic reactions. It primarily affects men aged 40-70 years and is often associated with chest pain. This syndrome is often unrecognized and undiagnosed in clinical practice due to a low level of awareness. Herein, we present a case of type I Kounis syndrome in a young woman without chest pain. CASE PRESENTATION: A 28-year-old Japanese woman with a history of atopic dermatitis received a glycyrrhizin, glutathione, and neurotropin preparation (a preparation of inflamed skin extract from rabbits inoculated with vaccinia virus) at a dermatology clinic to treat pruritus caused by atopic dermatitis. Immediately after the administration, the patient developed abdominal pain and generalized body wheals. The patient was diagnosed with anaphylaxis and was transported to our hospital. She had no chest pain on arrival at our hospital; however, a 12-lead electrocardiogram showed ST elevation in leads I, aVL, V2, and V3, and an echocardiogram showed decreased wall motion in the anterior and lateral walls of the left ventricle. Sublingual nitroglycerin administration improved ST-segment elevation and left ventricular wall motion abnormalities. The patient underwent emergency coronary angiography, which revealed no significant stenosis, and was diagnosed with type I Kounis syndrome. CONCLUSION: Kounis syndrome without chest pain is rare in young women. Since it can be fatal in cases with severe allergic symptoms such as anaphylaxis, the possibility of concurrent acute coronary syndrome should be considered when treating systemic allergic reactions, regardless of age, sex, or the presence or absence of chest symptoms.

Cellulitis caused by Roseomonas mucosa in a child: a case report.

BACKGROUND: Roseomonas mucosa (R. mucosa) is a pink-pigmented, Gram-negative short rod bacterium. It is isolated from moist environments and skin, resistant to multiple drugs, including broad-spectrum cephalosporins, and a rare cause of infection with limited reports. R. mucosa mostly causes catheter-related bloodstream infections, with even fewer reports of skin and soft tissue infections. CASE PRESENTATION: A 10-year-old boy received topical steroid treatment for sebum-deficient eczema. A few days before the visit, he was bitten by an insect on the front of his right lower leg and scratched it due to itching. The day before the visit, redness, swelling, and mild pain in the same area were observed. Based on his symptoms, he was diagnosed with cellulitis. He was treated with sulfamethoxazole/trimethoprim, and his symptoms improved. Pus culture revealed R. mucosa. CONCLUSIONS: We report a rare case of cellulitis caused by R. mucosa. Infections caused by rare organisms that cause opportunistic infections, such as R. mucosa, should be considered in patients with compromised skin barrier function and regular topical steroid use. Gram stain detection of organisms other than Gram-positive cocci should be considered.

Sepsis following acute pyelonephritis caused by Trueperella bernardiae: a case report and literature review.

BACKGROUND: Trueperella bernardiae is a coryneform, gram-positive bacterium that is a commensal of the skin and upper respiratory tract. It is treated as a contaminant and rarely causes infections. Blood, urine, and abscesses have been previously reported as the most common sites of infection. Infections caused by T. bernardiae are rarely reported in bedridden very old patients with reduced activities of daily living (ADL). In this report, we describe a case of sepsis due to acute pyelonephritis caused by T. bernardiae in a very old patient with impaired ADL. CASE PRESENTATION: A 94-year-old woman had a home visit from her local physician. She was bedridden and used diapers. On the day of admission, she presented with fever and dyspnea and was admitted with a diagnosis of sepsis associated with acute pyelonephritis. T. bernardiae was detected in blood and urine cultures; furthermore, multiple bacteria were detected in a urine culture. She was treated with ampicillin/sulbactam 3 g every 12 h on the day of admission. The fever was controlled, and inhaled oxygen 1 L/min via a nasal cannula was administered for dyspnea until hospitalization day 2. On hospitalization day 2, her fever resolved to 36 °C. Antimicrobials were de-escalated and changed to cephazolin and then to cephalexin on hospitalization days 9 and 16, respectively, and were continued until day 22. On hospitalization day 28, the urinary tract infection flared up; however, her fever resolved by hospitalization day 38 after the re-administration of antimicrobial agents. She was discharged on hospitalization day 60. CONCLUSIONS: We encountered a rare case of sepsis following acute pyelonephritis caused by T. bernardiae infection. When bedridden, diaper-using, very old patients present with urinary tract infections caused by multiple bacteria, the presence of rare opportunistic organisms, such as T. bernardiae, should be considered.

Hyponatremia due to adrenal insufficiency after a Mamushi bite: a case report.

BACKGROUND: Mamushi bites are the most common venomous snake bites in Japan, with known complications including rhabdomyolysis and acute kidney injury; however, adrenal insufficiency as a result of snake bites has not been previously reported. We report a case of empty sella with transient adrenal insufficiency during hospitalization for a Mamushi bite. CASE PRESENTATION: An 84-year-old man was admitted to our hospital with a Mamushi bite on the right fifth finger. Serum sodium (Na) level remained in the normal range. On the ninth day of admission, he developed hyponatremia, with a serum Na level of 114 mEq/L and serum cortisol level of 4.0 µg/dL (reference value 4.5-21.1 µg/dL). His serum Na level was restored within the normal range after administration of corticosteroids with 3% NaCl solution. Both rapid adrenocorticotrophin and corticotropin-releasing hormone loading tests showed low cortisol response. Based on the results of the hormone loading tests, a diagnosis of pituitary adrenal insufficiency was made. Contrast-enhanced pituitary magnetic resonance imaging (MRI) showed primary empty sella. After discontinuation of corticosteroids, the hyponatremia did not recur, and the patient was discharged on the 24th day of hospitalization. After discharge, the patient visited an outpatient clinic, but hyponatremia recurrence was not observed. CONCLUSIONS: This is the first report of hyponatremia due to pituitary adrenal insufficiency during hospitalization for a Mamushi bite in a patient with empty sella. When hyponatremia occurs during hospitalization for a Mamushi bite, cortisol measurement, hormone loading test, and head MRI should be performed to search for pituitary lesions because of the possibility of adrenal insufficiency caused by snake venom.

Diffusion-weighted whole-body magnetic resonance imaging with background body signal suppression was useful in a patient with isolated myocardial abscess confined to the right atrial wall: a case report.

BACKGROUND: Myocardial abscess is often associated with infective endocarditis (IE), and isolated myocardial abscess without IE is rare. Echocardiography and computed tomography (CT) are often used to diagnose myocardial abscess; however, to the best of our knowledge, diffusion-weighted whole-body magnetic resonance imaging with background body signal suppression (DWIBS) has not been used. Here, we present a case of myocardial abscess without IE that was diagnosed using DWIBS. CASE PRESENTATION: A 72-year-old Japanese man with a history of hypertension, dyslipidemia, and retinitis pigmentosa presented to our hospital with malaise and a fever lasting 10 days. Blood test results showed elevated inflammatory marker levels (white blood cell count 18,700/µL and C-reactive protein level 23.0 mg/dL). Infection was suspected; however, the source of the infection could not be identified. DWIBS, which was performed on day 7 of admission to determine the source of infection, showed a high signal surrounding the right wall, suggesting inflammation. Contrast-enhanced CT performed on day 1 of hospitalization revealed a low-density area in the same region; however, the pathological implications of this finding could not be determined. Based on DWIBS findings, we concluded that the condition presented as a myocardial abscess that was confined specifically to the right atrial wall. Three sets of blood cultures revealed negative findings, and echocardiography showed no vegetation or valve regurgitation. Therefore, the patient was diagnosed with an isolated myocardial abscess uncomplicated with IE. An electrocardiogram on admission showed no P waves, and the patient had a junctional rhythm. However, on day 20 of hospitalization, he developed a complete atrioventricular block. After complete myocardial abscess healing following antibiotic treatment was confirmed, the patient underwent pacemaker implantation. Ten months after surgery, the patient had no signs of infection recurrence. CONCLUSIONS: Based on history and physical examination alone, diagnosis of an isolated myocardial abscess can be challenging. In addition to CT and echocardiography, DWIBS might be helpful for the diagnosis of myocardial abscesses.

Computed tomography values of pericardial effusion may predict chylopericardium: a case report.

BACKGROUND: Idiopathic chylopericardium is a rare disease characterized by filling of the pericardial cavity with chylous fluid and has no evident cause. Secondary chylopericardium usually results from injury or damage to the thoracic duct. The most common causes of secondary chylopericardium are trauma, thoracic or cardiac surgery, and congenital lymphangiomatosis. Conservative or surgical treatment can be pursued; however, surgical treatment is required if conservative treatment is unsuccessful. Pericardiocentesis plays a crucial role in the definitive diagnosis of chylopericardium. However, although a serious complication, its occurrence is infrequent. Non-invasive methods, such as computed tomography (CT), could be useful in predicting the color or characteristics of pericardial effusion. CASE PRESENTATION: A 37-year-old Japanese woman presented to our hospital with a cough that persisted for 1 week. Echocardiography revealed pericardial effusion, which was diagnosed as acute pericarditis and treated with loxoprofen. However, pericardial effusion increased, and the patient presented to the emergency room with cardiac tamponade 1 month later. Pericardiocentesis was performed, which confirmed that the pericardial effusion was chylopericardium. Lymphatic scintigraphy did not show any connection between the thoracic duct and pericardial cavity, and the patient was diagnosed with idiopathic chylopericardium. The patient underwent continuous drainage for 11 days. After completion of cardiac drainage, the patient was discharged from the hospital without any exacerbation. The CT attenuation value of the pericardial fluid was 11.00 Hounsfield units (HU). Compared with the other causes of pericardial effusions encountered at our hospital, the HU on CT scan of pericardial effusion was low in our study and similar to the values on CT scan of chylous ascites reported in previous studies. CONCLUSIONS: Although idiopathic chylopericardium is rare, it should be considered an important cause of pericardial effusion. Pericardiocentesis is necessary for definitive diagnosis; however, the CT findings of pericardial effusion may help predict the presence of chylous fluid.

Pulmonary syphilis with a cicatricial variant of organizing pneumonia: a case report.

BACKGROUND: Syphilis is a chronic disease that progresses in the primary, secondary, latent, and tertiary stages. Pulmonary manifestations of syphilis are rare, and their histological features have not been well-described. CASE PRESENTATION: A 78-year-old man was referred to our hospital because of a solitary nodular shadow in the right middle lung field on a chest radiograph. Five years prior, a rash appeared on both legs. He was tested for syphilis at a public health center, and the non-treponemal test result was negative. When he was approximately 35 years old, he had unspecified sexual intercourse. Chest computed tomography showed a 13-mm nodule with a cavity in S6 of the right lower lobe of the lung. Robot-assisted resection of the right lower lobe was performed because of suspected localized right lower lobe lung cancer. A cicatricial variant of organizing pneumonia (CiOP) was observed, and immunohistochemistry identified Treponema pallidum inside the macrophages in the nodule cavity. The rapid plasma regain (RPR) value was negative, and the Treponema pallidum hemagglutination assay was positive. The patient was diagnosed as having secondary syphilis with pulmonary involvement. Insidious progression of secondary syphilis may result in CiOP and a negative RPR test result. CONCLUSIONS: We report the first case of pulmonary syphilis with a histological pattern of CiOP. It may be asymptomatic and difficult to diagnose because the RPR test may be negative for a long period of time. When either non-treponemal or treponemal test results are positive, the possibility of pulmonary syphilis should be considered along with appropriate medical treatment.

Venous Thrombosis Due to Duplication of the Inferior Vena Cava with Decreased Protein C Activity: A Case Report.

Introduction: Duplication of the inferior vena cava (IVC) is a congenital venous malformation that occurs in 0.2%-3% of the population as a result of persistent left and right supracardinal veins. The IVC duplication is prone to deep vein thrombosis due to endothelial dysfunction and associated venous stasis. This is a rare case of recurrent venous thrombosis due to IVC duplication and decreased protein C activity. Case: A 57-year-old male presented with swelling of the left lower limb that gradually developed over a one-week period preceding his visit. He reported a history of superior mesenteric vein thrombosis, approximately three years ago, for which he received anticoagulation therapy for three months. Thoracoabdominal contrast-enhanced computed tomography (CT) revealed thrombi in the locations of the bilateral main pulmonary arteries, IVC duplication, left common iliac vein, left IVC, and left renal vein. Blood work confirmed protein C activity of 21% (baseline 64%-146%), that could have contributed to the recurrent IVC thrombosis and formation of pulmonary artery thrombus. Subsequently, the patient was hospitalized and started on anticoagulation therapy. The swelling in the left lower extremity gradually improved, and the patient was instructed to continue anticoagulation therapy permanently. Conclusion: When investigating venous thrombosis of unknown or recurrent origin, it is necessary to include venous malformations and abnormal activity of blood coagulation factors in differential diagnosis.

Asymptomatic Idiopathic Renal Infarction Detected Incidentally on Contrast-Enhanced Computed Tomography: A Case Report.

Background: Renal infarction is an extremely rare disease. Although more than 95% of cases are symptomatic, there have been no previously reported asymptomatic cases, without any abnormal blood and urine test findings. Furthermore, the efficacy of long-term treatment of idiopathic renal infarction remains unknown. Case Presentation: A 63-year-old Japanese male underwent laparoscopy; a very low anterior resection of the rectum for lower rectal cancer (stage II) four years and five months prior to diagnosis with renal infarction. During the follow-up imaging studies, asymptomatic idiopathic renal infarction was found incidentally. The blood and urine test findings were normal. Contrast-enhanced computed tomography revealed a linearly bordered area of poor contrast in the dorsal region of the right kidney; however, no renal artery lesions, thromboembolic disease, or coagulation abnormalities were observed. Initial treatment with rivaroxaban 15 mg/day resulted in the remission of the infarcted lesion. The anticoagulation therapy was terminated after about 18 months without any incidences of re-infarction or bleeding events. Conclusions: We reported a very rare case of asymptomatic idiopathic renal infarction where blood and urine tests revealed no abnormal findings, and it was diagnosed incidentally during a post-treatment follow-up examination for lower rectal cancer. Long-term anticoagulant therapy for idiopathic renal infarction should be terminated at an appropriate time, taking the risk of bleeding into account.

A Case of Milk-Alkali Syndrome Caused by Diuretic-Induced Alkalosis and Polypharmacy.

Milk-alkali syndrome, which is characterized by hypercalcemia, metabolic alkalosis, and renal dysfunction, typically results from the ingestion of large amounts of calcium and absorbable alkaline products. However, these symptoms can also manifest when alkalosis and calcium loading occur simultaneously, owing to other factors. We report a case of milk-alkali syndrome caused by loop-diuretic-induced alkaline load and polypharmacy in an 85-year-old Japanese woman with multiple comorbidities, including osteoporosis, hypertension, type 2 diabetes, dyslipidemia, and Parkinson's disease. The patient regularly took 14 drugs, including calcium L-aspartate, eldecalcitol, celecoxib, and a fixed-dose combination of losartan and hydrochlorothiazide. Immediately before admission, furosemide was administered for the treatment of edema. The patient presented with chest discomfort, general malaise, and clinical signs of dehydration, hypercalcemia, hypophosphatemia, hypokalemia, and hypomagnesemia, accompanied by electrocardiogram abnormalities, renal dysfunction, and chloride-resistant metabolic alkalosis. The hypercalcemia was specifically induced by calcium L-aspartate and eldecalcitol. The hypomagnesaemia and hypophosphatemia were caused by diuretics and hypercalcemia. Thus, all the oral medications were discontinued, and rehydration and electrolyte correction therapy were administered. The final diagnosis was milk-alkali syndrome caused by the concomitant use of loop diuretics and other medications, without absorbable alkaline preparation use. This case underscores the importance of considering drug-related factors, checking concomitant medications, and being aware of the benefits, harmful effects, and side effects of polypharmacy in older adults with multimorbidity.

Hereditary Congenital Methemoglobinemia Diagnosed at the Age of 79 Years: A Case Report.

Background: Cardiopulmonary disorders are the most common cause of central cyanosis, and methemoglobinemia is often overlooked in the differential diagnosis of patients with central cyanosis. In most cases, methemoglobinemia is acquired and hereditary congenital methemoglobinemia is rare. Only a few case reports of congenital methemoglobinemia can be found in PubMed. To date, only four cases of congenital methemoglobinemia diagnosed after the age of 50 years have been reported. Case Presentation: A 79-year-old Japanese woman presented at our hospital with the chief complaints of dyspnea and cyanosis. She exhibited cyanosis of the lips and extremities, and her SpO2 was 80%, with oxygen administration at 5 L/min. Blood gas analysis revealed a PaO2 of 325.4 mmHg and methemoglobin level of 36.9%. The SpO2 and PaO2 values were dissociated, and methemoglobin levels were markedly elevated. Genetic analysis revealed a nonsynonymous variant in the gene encoding nicotinamide adenine dinucleotide cytochrome (NADH) B5 reductase 3 (CYB5R3), and the patient was diagnosed with congenital methemoglobinemia. Conclusions: It is important to consider methemoglobinemia in the differential diagnosis of patients with central cyanosis. At 79 years of age, our patient represents the oldest patient with this diagnosis. This report indicates that it is crucial to consider the possibility of methemoglobinemia regardless of the patient's age.

Congenital Isolated Unilateral Agenesis of Pulmonary Arteries with 3-Year Follow-Up after Initiation of Treatment.

Background and objective: Unilateral agenesis of pulmonary arteries (UAPA) is a rare disease, with approximately 400 cases reported to date. UAPA is often associated with congenital heart disease, and the uncomplicated form is isolated UAPA, which accounts for approximately 30% of all cases of UAPA. The incidence of pulmonary hypertension due to UAPA has been reported to range from 19 to 44%. There is no consensus treatment for pulmonary hypertension associated with UAPA. We present the first reported case in which a three-drug combination, comprising of iloprost inhalation, riociguat, and ambrisentan, was administered to a patient with UAPA, and was followed-up for 3 years post-diagnosis. Case presentation: A 68-year-old Japanese woman presented to our hospital with dyspnea and chest discomfort. She underwent chest radiography, blood tests, and echocardiography; however, the cause of the patient's symptoms could not be identified. During regular follow-up, an echocardiography 21 months after the initial visit revealed elevated right ventricular pressure (peak tricuspid regurgitation velocity: 5.2 m/s and right ventricular systolic pressure: 120 mmHg) and a diagnosis of pulmonary hypertension was made. Contrast-enhanced computed tomography (CT) of the chest and a pulmonary blood flow scintigram were performed to investigate the cause of pulmonary hypertension, and isolated UAPA was diagnosed. The patient was treated with a three-drug combination of iloprost inhalation, riociguat, and ambrisentan and followed up for 3 years with good therapeutic outcomes. Conclusions: We present a case of pulmonary hypertension caused by isolated UAPA. Although rare, this disease can lead to pulmonary hypertension and should be treated cautiously. While there is no consensus regarding the treatment of this disease, a three-drug combination of iloprost inhalation, riociguat, and oral ambrisentan proved effective.

Epstein-Barr virus-associated infectious mononucleosis with acute epididymitis: a case report.

BACKGROUND: Infectious mononucleosis due to the Epstein-Barr virus is an infectious disease that causes the appearance of atypical lymphocytes in the peripheral blood; it mainly presents with fever, tonsillar pharyngitis, and lymphadenopathy. In addition to hepatitis, splenomegaly, and rashes, it can involve different organs. Here, a case of epididymitis as a rare complication in a patient with Epstein-Barr virus-associated infectious mononucleosis was reported. CASE PRESENTATION: A healthy 23-year-old man visited an outpatient clinic with fever and pharyngitis. Tonsillar pharyngitis, lymphadenopathy, atypical lymphocytes in the peripheral blood, liver dysfunction, and splenomegaly were observed. The patient was diagnosed with infectious mononucleosis based on clinical signs. The next day, the patient developed left testicular pain and was immediately transferred to the emergency outpatient ward. Pain, redness, and swelling were observed in the left scrotum. Ultrasonography revealed swelling of the epididymis and increased blood flow, and the patient was hospitalized with a diagnosis of left epididymitis. The patient's symptoms improved with symptomatic treatment and was discharged on day 16 after admission. Changes in antibody titers established a definitive diagnosis of infectious mononucleosis caused by the Epstein-Barr virus. Based on the disease course, the patient was also diagnosed with infectious mononucleosis associated with unilateral epididymitis. CONCLUSIONS: This is the first case report of Epstein-Barr virus-associated infectious mononucleosis complicated with acute epididymitis. Infectious mononucleosis can cause numerous organ-related complications; thus, physicians and healthcare workers should remain cognizant of Epstein-Barr virus-associated complications throughout the body and not just in the primary organs affected by infectious mononucleosis.

Guillain-Barré syndrome with transition from hashimoto's to graves' disease: a case report.

BACKGROUND: On rare occasions, there can be a transition from Hashimoto's to Graves' disease. However, there are no reported cases of transition from Hashimoto's to Graves' disease triggered by the onset of Guillain-Barré syndrome. CASE PRESENTATION: Sixteen years prior, a 55-year-old woman was diagnosed with Hashimoto's disease and followed up without medication. One week after the appearance of signs of intestinal inflammation, weakness in the extremities was observed, and a cerebrospinal fluid test was positive for anti-GM1 IgG antibody, leading to the diagnosis of Guillain-Barré syndrome. In addition, hyperthyroidism was observed at the time of admission, and Graves' disease was diagnosed based on autoantibodies and thyroid echoes. Numbness in the extremities was relieved by high-dose intravenous gamma globulin. CONCLUSION: With the onset of Guillain-Barré syndrome, helper T cells became predominantly type 1, effector B cells increased in number, and thyroid-stimulating antibodies were produced, leading to the conclusion that Hashimoto's disease progressed to Graves' disease. Therefore, it is necessary to pay attention to the transition of thyroid function during Guillain-Barré syndrome.

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.

Meningitis retention syndrome caused by varicella zoster virus in a patient without a rash: a case report.

BACKGROUND: Meningitis retention syndrome (MRS) is a rare condition that presents with acute urinary retention as a complication of aseptic meningitis. Cases of MRS due to varicella zoster virus (VZV) infection without a rash are rare. We report the case of a patient who had no signs of meningitis or VZV infection, including a rash. CASE PRESENTATION: A 58-year-old man presented with dysesthesia of the lower limbs and acute urinary retention. He had fever but no rash and no signs of meningitis. He was diagnosed to have VZV infection based on the detection of VZV DNA in the cerebrospinal fluid. He responded satisfactorily to a course of intravenous acyclovir and experienced no sequelae during a 2-year follow-up period. CONCLUSION: MRS due to aseptic meningitis of viral origin should be considered in the differential diagnosis of acute urinary retention even in the absence of specific signs and symptoms of meningitis or a suggestive rash.

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.

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.