Cerebral Scedosporium apiospermum infection presenting with intestinal manifestations

We present a case of cerebral Scedosporium apiospermum infection presenting with intestinal manifestations in a 64-year-old male patient on immunosuppression for orthotopic liver transplantation. At admission, the patient’s chief complaint was chronic watery diarrhea and he was found to have colonic ulcers on endoscopy. His hospital course was complicated by a tonic–clonic seizure caused by a left frontal brain abscess, with the causative agent being identified by culture. He was treated with lobectomy, high-dose intravenous voriconazole, and liposomal amphotericin with clinical, endoscopic, and histologic improvement. To our knowledge, S. apiospermum has not been previously described as a cause of colitis. The septate branching appearance of the Scedosporium species is similar to the more common Aspergillus species. This case of gastrointestinal Scedosporium brings into question previously reported cases of isolated gastrointestinal aspergillosis diagnosed by histopathology. Clinical suspicion for S. apiospermum must be maintained in immunosuppressed patients presenting with neurologic and gastrointestinal symptoms.     

Non-valvular Infective Endocarditis Caused by Sarocladium kiliense in an Immunocompromised Patient with Aplastic Anemia

Sarocladium kiliense is ubiquitous in the human environment and is an emerging opportunistic pathogen, especially among immunocompromised hosts. A 77-year-old man diagnosed with aplastic anemia suffered from non-valvular endocarditis. After he passed away, fungal hyphae were observed in several lesions on a postmortem examination. Polymerase chain reaction (PCR) and a DNA sequence analysis revealed S. kiliense as the causative organism. This is the first case report of non-valvular fungal endocarditis caused by S. kiliense identified by PCR and a DNA sequence analysis in an immunocompromised patient. Although rare, invasive fungal infection caused by S. kiliense should be considered in immunocompromised hosts.     

A Resuscitated Case of Acute Myocardial Infarction with both Familial Hypercholesterolemia Phenotype Caused by Possibly Oligogenic Variants of the PCSK9 and ABCG5 Genes and Type I CD36 Deficiency

A 56-year-old postmenopausal woman with out-of-hospital cardiac arrest caused by acute myocardial infraction was successfully resuscitated by intensive treatments and recovered without any neurological disability. She was diagnosed as having familial hypercholesterolemia (FH) based on a markedly elevated low-density lipoprotein cholesterol (LDL-C) level and family history of premature coronary artery disease. Genetic testing in her family members showed that a variant of the proprotein convertase subtilisin/kexin type 9 ( PCSK9 ) gene (c.2004C＞A, p.S668R), which had been previously reported as having uncertain significance, was associated with FH, indicating that the variant is a potential candidate for the FH phenotype. Next-generation sequencing analysis for the proband also showed that there was a heterozygous mutation of the ATP-binding cassette sub-family G member 5 ( ABCG5 ) gene (c.1166G＞A, R389H), which has been reported to increase LDL-C level and the risk of cardiovascular disease. She was also diagnosed as having type 1 CD36 deficiency based on a lack of myocardial uptake of ¹²³ I-labeled 15-(p-iodophenyl)-3-R,S-methyl-pentadecanoic acid in scintigraphy and the absence of CD36 antigen in both monocytes and platelets in flow cytometry. She had a homozygous mutation of the CD36 gene (c.1126-5_1127delTTTAGAT), which occurs in a canonical splice site (acceptor) and is predicted to disrupt or distort the normal gene product. To our knowledge, this is the first report of a heterozygous FH phenotype caused by possibly oligogenic variants of the PCSK9 and ABCG5 genes complicated with type I CD36 deficiency caused by a novel homozygous mutation. Both FH phenotype and CD36 deficiency might have caused extensive atherosclerosis, leading to acute myocardial infarction in the present case.     

Severe Plasmodium vivax cerebral malaria complicated by hemophagocytic lymphohistiocytosis treated with artesunate and doxycycline

Malaria-related hemophagocytic lymphohistiocytosis is a rare, potentially fatal, hyperinflammatory disease entity which can be challenging to diagnose and treat. It is usually associated with Plasmodium falciparum infection. It is less frequently associated with Plasmodium vivax. Here we report an unusual case of a 23-year-old healthy Nigerian man who presented with fever, microangiopathic hemolytic anemia, acute renal failure, and confusion, and was diagnosed as having cerebral malaria-related hemophagocytic lymphohistiocytosis caused by P. vivax infection. He was successfully treated with intravenous artesunate and doxycycline with dramatic clinical improvement.     

Rothia aeria endocarditis in a patient with a bicuspid aortic valve: case report

Rothia aeria is an uncommon pathogen mainly associated with endocarditis in case reports. In previous reports, endocarditis by R. aeria was complicated by central nervous system embolization. In the case we report herein, endocarditis by R. aeria was diagnosed after acute self-limited diarrhea. In addition to the common translocation of R. aeria from the oral cavity, we hypothesize the possibility of intestinal translocation. Matrix-assisted laser desorption ionization-time of flight mass spectrometry and genetic sequencing are important tools that can contribute to early and more accurate etiologic diagnosis of severe infections caused by Gram-positive rods.     

Pyogenic Spondylitis Caused by Staphylococcus schleiferi in a Patient with Crohn's Disease

Staphylococcus schleiferi has rarely been reported to cause pyogenic spondylitis. A 42-year-old man had been treated for Crohn''s disease with immunosuppressive agents and home parenteral nutrition via a central vein (CV) port. The patient was admitted to our hospital, presenting with neck pain and a fever. A neurological examination showed slight weakness in his left-hand muscles, and he was diagnosed with pyogenic spondylitis of C6 and C7 vertebral bodies due to catheter-related blood stream infection caused by S. schleiferi. An early diagnosis by magnetic resonance imaging, CV port removal and antibiotic therapy targeting S. schleiferi improved his symptoms.     

Long-term Clinical Follow-up of Patients with Familial Hypomagnesemia with Secondary Hypocalcemia

Objective:Familial hypomagnesemia with secondary hypocalcemia (HSH) is an autosomal recessive disease caused by a mutation in the transient receptor potential melastatin 6 (TRPM6) gene and is characterized by selective magnesium malabsorption. Affected cases are usually diagnosed during infancy and usually present with seizures due to hypocalcemia and hypomagnesemia. Irreversible neurological deficits and arrhythmias can be observed without appropriate treatment. The aim was to evaluate the long-term follow-up of patients with genetically confirmed HSH.Methods:A total of six patients with HSH, two of whom were siblings, were included. Age at diagnosis, clinical, laboratory and follow-up data on admission were recorded. All 39 exons of the TRPM6 gene and flanking exon-intron junctions from genomic DNA were amplified and sequenced in all cases.Results:The median (range) follow-up duration was 12.1 (7.6-21.7) years. All cases were diagnosed in infancy. Four different mutations, three of which had not been previously reported, were detected in the TRPM6 gene. Treatment compliance was good and there were no severe complications in the long-term follow-up of cases. However, mental retardation, specific learning difficulty and attention deficit/hyperactive disorder were observed as comorbidities.Conclusion:Of the four different TRPM6 mutations in this small cohort, three had not been previously reported. The long-term prognosis of HSH appears to be good, given early diagnosis and good treatment compliance. This long-term follow-up and prognostic data and the three novel mutations will contribute to the published evidence concerning this rare condition, HSH, and it is hoped will prevent negative outcomes.     

Clinicoetiologic investigations on superficial mycoses of Warangal (A.P) India

ObjectiveTo study the clinical manifestations and etiological agents of superficial mycosis and to investigate their relationship in superficial mycosis occurring in Warangal, A.P. India.MethodsThe present study was conducted on 400 clinically diagnosed patients with dermatophytoses. Three different types of samples viz. skin scrapings, hair samples and nail clippings were collected for examinations. The samples were processed for microscopic examination, histological study and culturing of etiological agent. The incidence of various dermatophytic species in relation to clinical types was recorded.ResultsAmong different clinical types, tinea corporis was most common. The most frequently isolated dermatophytes were Trichophyton species which were isolated from all the three samples i.e. skin, hair and nail. A majority of the diagnosed patients showed erythematous, scaly annular lesions with raised and inflammatory borders with itching. Mixed infections, which constituted about 3.5%, were a combination of tinea corporis and tinea cruris. Either individually or in mixed infections, these two infections are caused by Trichophyton species. Tinea capitis infections which include ectothrix and endothrix are caused by species of Microsporum and Trichophyton.ConclusionsThe investigations reveal that superficial mycoses are very common in this region. Tinea corporis is the most common clinical manifestation. Majority of the infections are caused by Trichophyton species. Mixed infections constituted a small proportion.     

Diagnosis of autoimmune lymphoproliferative syndrome caused by FAS deficiency in adults

A diagnosis of autoimmune lymphoproliferative syndrome caused by FAS deficiency during adulthood is unusual. We analyzed 17 cases of autoimmune lymphoproliferative syndrome caused by FAS deficiency diagnosed during adulthood in French reference centers for hereditary immunodeficiencies and for immune cytopenias. Twelve of the 17 patients had developed their first symptoms during childhood. The diagnosis of autoimmune lymphopro-liferative syndrome had been delayed for a variety of reasons, including unusual clinical manifestations, late referral to a reference center, and the occurrence of somatic FAS mutations. The 5 other patients presented their first symptoms after the age of 16 years. In these patients, three germline heterozygous FAS mutations were predicted to be associated with haploinsufficiency and a somatic event on the second FAS allele was observed in 2 cases. Autoimmune lymphoproliferative syndrome may well be diagnosed in adulthood. The occurrence of additional genetic events may account for the delayed disease onset.     

Ecthyma gangrenosum caused by Staphylococcus aureus in hematological malignancies: Case reports and literature review

Rationale:Ecthyma gangrenosum (EG) is a potentially life-threatening, systemic infection generally caused by Pseudomonas aeruginosa. Data on EG caused by Staphylococcus aureus in patients with hematological malignancies are scarce. The present case report aimed to describe the clinical features of EG caused by S. aureus in patients with hematological malignancies and to provide a comprehensive review of previous studies on the topic.Patient concerns:The first patient was a 61-year-old man with acute myeloid leukemia who presented fever and multiple lesions during chemotherapy. The second patient was a 47-year-old man with myelodysplastic syndrome who developed progressive erythematous necrotic plaques on his extremities and face.Diagnosis:Both cases were diagnosed as EG caused by S. aureus. While the first patient had concurrent methicillin-resistant S. aureus (MRSA) bacteremia, the second patient had positive results only for tissue culture of the skin lesion isolated methicillin-sensitive S. aureus.Interventions:Vancomycin was initiated with critical care to the first patient. Cefazolin was administered to the second patient for 3 weeks, followed by cephalexin for 1 week.Outcomes:The first patient died of a brain hemorrhage and multiple organ failure. The second patient was cured without relapse.Lessons:Of 18 patients in the previous and current studies with EG caused by S. aureus, 6 (33%) had an underlying hematological malignancy, and 10 (56%) had EG caused by MRSA. While 28% of the patients had positive blood cultures, all tissue cultures were positive. All 3 fatalities had concurrent bacteremia (MRSA caused two). EG caused by MRSA with concurrent bacteremia can be fatal, especially in patients with hematological malignancies. Although S. aureus-associated EG in patients with hematological malignancies is relatively uncommon, tissue cultures with an initial gram stain smear are essential for selecting appropriate empirical antimicrobials, including the coverage of S. aureus.     

Exercise capacity of cirrhotic patients with hepatopulmonary syndrome

Introduction. Hepatopulmonary syndrome (HPS) is characterized by a clinical triad of liver disease and/or portal hypertension, intrapulmonary vascular dilatation and abnormal arterial oxygenation. These conditions can worsen muscle strength, exercise capacity and functionality in the affected population.Objective. The objective of this study was to compare exercise capacity, functional condition and respiratory muscle strength in cirrhotic patients diagnosed with HPS and cirrhotic patients without this diagnosis.Material and methods. This cross-sectional study used a convenience sample consisting of 178 patients (92 patients with HPS and 86 patients without HPS) with a diagnosis of liver cirrhosis caused by either alcohol consumption or the hepatitis C virus (HCV). Peak oxygen consumption (VO₂ peak) was used to verify exercise capacity, the six-minute walk test (6MWT) was used to test functionality, and manovacuometry was used to evaluate the strength of the respiratory muscles. The Kolmogorov-Smirnov test and Student’s t-test were used for the statistical analysis. The data were analyzed using SPSS 16.00, and p < 0.05 was considered significant.Results. The group of patients with the diagnosis of HPS exhibited a lower VO₂ peak (14.2 ± 2.3 vs. 17.6 ± 2.6, p < 0.001), shorter distance walked in the 6MWT (340.8 ± 50.9 vs. 416.5 ± 91.4, p < 0.001), lower maximal inspiratory pressure (-49.1 ± 9.8 vs. -74.2 ± 13.9, p = 0.001) and lower maximum expiratory pressure (60.1 ± 12.2 vs. 76.8 ± 14.7, p = 0.001).Conclusion. The group of cirrhotic patients diagnosed with HPS exhibited lower values for VO₂ peak, distance walked in the 6MWT and respiratory muscle strength than the cirrhotic patients not diagnosed with HPS.     

Dilated papilla with mucin extrusion is a potential predictor of acute pancreatitis associated with intraductal papillary mucinous neoplasms of pancreas

Background/objectivesAs intraductal papillary mucinous neoplasm (IPMN) of the pancreas is associated with acute pancreatitis (AP) in some cases, predicting the risk of pancreatitis is as important as predicting the risk of malignancy in IPMN cases. In this study, we attempted to clarify the characteristics of IPMN associated with AP, compared to those of IPMN not associated with AP.MethodsFrom January 2006 to March 2013, data from 88 patients who underwent surgery for IPMN were retrospectively investigated and analyzed. We evaluated clinical and pathological variables of each patient and compared patients with IPMN with AP to those without AP. Furthermore, we presented representative cases of mild and severe pancreatitis caused by IPMN.ResultsOverall, 12 of 88 patients with IPMN (13.6%) had AP. Seven of the 12 patients had a single episode of AP, whereas the remaining 5 patients were diagnosed with IPMN with repeated AP. Ten of 12 patients with AP were diagnosed with mild AP and the remaining 2 with severe AP. Regarding clinical findings, the proportion of dilated papilla with mucin extrusion was significantly higher in patients with IPMN with AP than in those without AP (p = 0.035). Histological findings indicated that the proportion of intestinal-subtype IPMN was significantly higher in patients with IPMN with AP (p = 0.013).ConclusionsAP caused by IPMN derives mostly from intestinal IPMN. Dilated papilla with mucin extrusion can be a potential predictor of AP.     

The First Case Report of Mediastinal Abscess Caused by Gemella bergeri

Most cases of mediastinal abscess occur as a postoperative complication of a thoracic surgical procedure or following trauma. The most common causative microorganism is Staphylococcus aureus, but it can be rarely caused by unusual microorganisms, such as Gemella species. These are relatively difficult-to-identify commensal microorganisms of the upper respiratory and gastrointestinal tracts and may cause several infections. A 66-year-old man was diagnosed with Gemella bergeri mediastinal abscess by the molecular detection of bacterial genes. He was successfully treated with penicillin antibiotic for eight weeks. To our knowledge, this is the first case report of mediastinal abscess caused by G. bergeri.     

Infective endocarditis with atypical clinical feature and relapse by Abiotrophia defectiva

A case of infective endocarditis caused by an uncommon agent Abiotrophia defectiva with atypical manifestations is presented. A 42-year-old woman previously had rheumatic heart disease, presented with the symptoms of fever and chills that resolved within 3 days under antibiotherapy. She was diagnosed with endocarditis due to A. defectiva. Despite culture-directed antibiotics being administered in the first admission, her symptoms and also blood culture growth relapsed 3 weeks later. She was successfully treated with antimicrobial therapy and surgical intervention including aorta and mitral valve replacement. This case demonstrates that A. defectiva should be considered as a causative organism of endocarditis particularly in the presence of atypical symptoms and should be followed up carefully in terms of relapses and complications.     

Biopsy-proven Streptococcus suis-associated Infectious Glomerulonephritis

A 64-year-old Japanese man who worked at a butcher shop was hospitalized for a fever, headache, and deafness. We diagnosed him with sepsis and meningitis caused by Streptococcus suis infection. The patient''s renal function declined rapidly, and hemodialysis was performed temporarily. A renal biopsy was performed, and the renal function tended to improve with antimicrobial therapy. This case seemed rather similar to one of staphylococcal-associated nephritis in that it showed mesangial proliferative nephritis with immunoglobulin A deposition, even though the nephritis was caused by streptococci. Similarly, intramembranous electron-dense deposits were characteristic findings. We present new findings of an in vivo renal biopsy in a case of S. suis-associated glomerulonephritis.     

Chest injuries and the 2011 Great East Japan Earthquake

BackgroundChest injuries caused during a major earthquake remain unclear. We have described profiles of patients with chest injuries who were diagnosed and treated at the area that was most affected by the Great East Japan Earthquake in 2011.MethodsWe retrospectively reviewed medical records of 3938 patients who were transferred to the Japanese Red Cross Ishinomaki Hospital during the first week after the earthquake (March 11–17). In total, 77 patients were declared dead on arrival at the hospital. Of the remaining 3861 patients, 42 (1.1%) sustained chest injuries. Diagnosis of the chest injury was based on results of physical examination, chest radiography, and computed tomography.ResultsChest injury was diagnosed in 42 patients, including 22 men and 20 women (age range, 21–99 years). The most common cause of injury was tsunami (n=21), followed by falls (n=9), and traffic accidents (n=1), although this information was missing in 11 cases. The most common type of chest injury was superficial trauma such as laceration and contusion (n=37). Only 5 patients had rib fractures with intrathoracic damages such as pneumothorax (n=3), hemothorax (n=1), and aspiration (n=1).ConclusionsThe number of patients with chest injury was surprisingly small. Most patients did not require hospitalization. The small number of survivors with serious chest injuries can most likely be explained by the tsunami caused by the earthquake.     

Peritonitis caused by Paracoccidioides spp. in a patient on continuous ambulatory peritoneal dialysis – A case report

Paracoccidioidomycosis is a systemic mycosis found mainly in South America and is the most prevalent endemic and systemic mycosis in Brazil. The purpose of this paper was to report the case of a male patient who developed peritonitis caused by Paracoccidioides spp. Forty-eight-year-old, male patient, with type I Diabetes mellitus and chronic kidney disease who was undergoing a Continuous Ambulatory Peritoneal Dialysis (CAPD) program. After eighteen months of peritoneal dialysis, the patient developed turbidity of the peritoneal fluid and was diagnosed with peritonitis. Direct mycological examination of the peritoneal fluid revealed yeasts with morphology suggestive of Paracoccidioides spp. The patient was treated with sulfamethoxazole-trimethoprim (1,600 mg/320 mg dose/day) for 61 days, but he died because a bacterial septic shock. The diagnosis of opportunistic PCM peritonitis was later confirmed by autopsy and Paracoccidioides spp. isolation. This is the first reported case of a patient on CAPD who experienced complications due peritonitis caused by opportunistic PCM.     

Infective Endocarditis Due to Treponema pallidum: A Case Diagnosed Using Polymerase Chain Reaction Analysis of Aortic Valve

Syphilis is a sexually transmitted disease caused by Treponema pallidum. Syphilitic aortitis might coexist in a dysfunctional aortic valve, but the etiology remains unclear, because microbiological diagnosis is difficult. A 62-year-old man with low-grade fever was diagnosed with aortitis and infective endocarditis, due to Treponema pallidum infection, using polymerase chain reaction analysis. This case suggests that syphilis might cause infective endocarditis.     

Serum FGF21 levels are increased in newly diagnosed type 2 diabetes with nonalcoholic fatty liver disease and associated with hsCRP levels independently

Objective

Fibroblast growth factor (FGF21) has beneficial effects on lipolysis. Highly sensitive C-reactive protein (hs-CRP) is a predictor of type 2 diabetes and nonalcoholic fatty liver disease (NAFLD). This study aimed to determine the levels of serum FGF21 and hs-CRP in newly diagnosed type 2 diabetes patients with and without NAFLD, and further explored the correlation between FGF21 with hs-CRP in newly diagnosed type 2 DM.

Research design and methods

69 patients with newly diagnosed type 2 DM and 30 normal subjects were included in the study. FGF21 and hs-CRP were measured by ELISA kits. The severity of NAFLD was measured by ultrasound.

Results

Serum FGF21 in newly diagnosed type 2 DM with NAFLD group were significantly increased (p < 0.01). There was no difference for the FGF21 level in normal control group and newly diagnosed type 2 DM without NFALD group. In type 2 DM group, the FGF21 level was positively correlated with hsCRP (r = 0.417, p < 0.001). In multiple stepwise regression models, only hsCRP was a significantly independent determinant for serum FGF21.

Conclusions

Serum levels of FGF21 are closely related to liver steatosis in newly diagnosed type 2 DM patients.     

Misdiagnosis of tuberculosis and the clinical relevance of non—tuberculous mycobacteria in Zambia

ObjectiveTo determine the accuracy of TB diagnosis of TB in Zambia in the era of increasing HIV prevalence.MethodsSputum of the clinically diagnosed TB cases was additionally subjected to liquid culture and molecular identification. This study distinguished between TB cases confirmed by positive Mycobacterium tuberculosis (M. tuberculosis) cultures and mycobacterial disease caused by non-tuberculous mycobacteria (NTM).ResultsOnly 49% of the 173 presumptively diagnosed TB cases was M. tuberculosis cultured, while in 13% (22) cases, a combination of M. tuberculosis and NTM was found. In 18% of the patients only NTM were cultured. In 28%, no mycobacteria was cultivable. HIV positive status was correlated with the isolation of NTM (P <0.05).ConclusionsThe diagnosis of tuberculosis based on symptoms, sputum smear and/or chest X-ray leads to significant numbers of false-positive TB cases in Zambia, most likely due to the increased prevalence of HIV. The role of NTM in tuberculosis-like disease also seems relevant to the false diagnosis of TB in Zambia.