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Lymphocyte hydrogen sulfide production predicts intravenous immunoglobulin resistance in children with Kawasaki disease: A preliminary, single-center, case-control study.

Vascularites - mer, 28/11/2018 - 23:58
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Lymphocyte hydrogen sulfide production predicts intravenous immunoglobulin resistance in children with Kawasaki disease: A preliminary, single-center, case-control study.

Medicine (Baltimore). 2018 Nov;97(47):e13069

Authors: Lin J, Zhao H, Jiao F, Ma L, Ma L

Abstract
The aim of the study was to identify whether lymphocyte hydrogen sulfide production is a potential biomarker to predict intravenous immunoglobulin (IVIG) resistance in children with Kawasaki disease (KD).This preliminary, single-center, case-control study conducted between June 2016 and March 2018 in Shaanxi Provincial People's Hospital, 85 children (50 with KD and 35 healthy controls) were included. Laboratory biomarkers were collected from the medical records. All patients with KD received 1 g/kg/d IVIG for 2 days and 30-50 mg/kg/d oral aspirin. The aspirin dose was reduced from 3 to 5 mg/kg/d after body temperature normalized. Plasma hydrogen sulfide levels were detected using sulfide electrode. Lymphocyte hydrogen sulfide levels were detected using the human hydrogen sulfide ELISA kits at the acute stage.Of 50 patients with KD, 31 and 19 were diagnosed with complete KD (cKD) and incomplete KD (iKD), respectively. Eleven patients with KD were resistant to IVIG treatment. The laboratory biomarker findings and levels of plasma and lymphocyte hydrogen sulfide were significantly different between the patients with KD and control group (P < .001). Moreover, lymphocyte hydrogen sulfide production was significantly greater in IVIG-resistant patients than in the IVIG-responsive patients, both in cKD and iKD (P = .018 and P < .001 respectively). Receiver operating characteristic curve indicated that when the lymphocyte hydrogen sulfide production was >15.285 nmol/min/10 lymphocytes, the sensitivity and specificity for predicting IVIG resistance were 90.9% and 76.9%, respectively.Lymphocyte hydrogen sulfide production could serve as a predictor of the therapeutic efficacy of IVIG in children with KD.

PMID: 30461609 [PubMed - indexed for MEDLINE]

Spontaneous Spinal Subarachnoid Hemorrhage: Presentation and Outcome.

Vascularites - mer, 28/11/2018 - 23:58
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Spontaneous Spinal Subarachnoid Hemorrhage: Presentation and Outcome.

J Stroke Cerebrovasc Dis. 2018 Oct;27(10):2792-2796

Authors: Yost MD, Rabinstein AA

Abstract
BACKGROUND: Spontaneous spinal subarachnoid hemorrhage (SSAH) is a rare but serious condition that can lead to a variety of medical complications.
METHODS: Using the Mayo Clinic Rochester database, consecutive patients admitted to the Mayo Clinic Rochester, Minnesota hospital with spontaneous SSAH (not iatrogenic or traumatic) between January 2000 and December 2015 were retrospectively reviewed. Demographic and clinical data and functional outcomes as categorized by the modified Rankin Scale (mRS) score at the time of discharge and at postadmission follow-up were assessed.
RESULTS: Eight patients (median age 70 years, range 51-87) were identified. Seven patients presented with acute back pain or headache. Seven patients had poorly controlled chronic hypertension. Two patients had vasculitis and 1 had an arteriovenous malformation. Complications included cord compression (n = 4), hyponatremia (n = 5), sulcal subarachnoid hemorrhage (n = 2), chronic arachnoiditis (n = 1), vasospasm (n = 1), ischemic infarct (n = 1), and late cord ischemia (n = 1). All patients were managed medically including blood pressure control and repeat imaging. One patient had an aneurysm, which was embolized, and 2 received steroids for vasculitis. One patient died during hospitalization and 1 died within a week of discharge. Five patients had improved and 1 had unchanged mRS score at posthospitalization follow-up.
CONCLUSIONS: SSAH should be considered in patients with sudden onset of severe back pain and headache, especially if as associated with lower extremity weakness or urinary retention. Our findings suggest that older patients with poorly controlled hypertension are at particular risk for spontaneous SSAH. In our study vascular anomalies were found in 25% of patients, vasculitis in 25%, hyponatremia in 63%, and cord compression in 50%. Cord compression may be managed conservatively. Mortality was 25% in the 3 months following the initial event, primarily due to other medical comorbidities.

PMID: 30064869 [PubMed - indexed for MEDLINE]

Peculiar histopathological features in minor salivary gland in lupus erythematosus.

Vascularites - mer, 28/11/2018 - 23:58
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Peculiar histopathological features in minor salivary gland in lupus erythematosus.

Lupus. 2018 Sep;27(10):1706-1711

Authors: Bologna SB, Nico MMS, Florezi G, Cavalcante WS, Lourenço SV

Abstract
Salivary glands (SGs) can be affected by lupus erythematosus (LE). Many authors debate whether this condition is a secondary manifestation of Sjögren syndrome (SS) or a glandular aspect of LE. The present study investigated the histopathological aspects of biopsied minor salivary glands from LE patients to analyze their peculiar features that lead to xerostomia. Twenty-three minor labial salivary gland (MLSG) cases were included in the study; the diagnosis of LE was rendered according to the American College of Rheumatology criteria. Twenty-three healthy MLSGs were used as a control, for comparison. Regarding lupus MLSG, the presence of hyalinization and thickening of ductal basement membrane, perivascular inflammatory infiltrate, epithelial spongiosis with no ductal lymphocytic aggression, vacuolar degeneration of the ductal cells and acinar serous metaplasia were statistically significant compared to the control group. In the LE group, there was a statistically significant correlation between acinar atrophy and acinar fibrosis; acinar atrophy and ductal ectasia; acinar fibrosis and ductal ectasia; ductal atrophy and ductal spongiosis with no lymphocytic focus, interstitial inflammatory infiltrate intensity and vasculitis as well as vascular thrombi and vasculitis. There were no morphological differences between the three subtypes of lupus analyzed. Minor salivary glands from patients diagnosed with LE present peculiar histopathological changes and may be a multisystemic presentation.

PMID: 30060722 [PubMed - indexed for MEDLINE]

Clinical characteristics of lupus enteritis in Japanese patients: the large intestine-dominant type has features of intestinal pseudo-obstruction.

Vascularites - mer, 28/11/2018 - 23:58
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Clinical characteristics of lupus enteritis in Japanese patients: the large intestine-dominant type has features of intestinal pseudo-obstruction.

Lupus. 2018 Sep;27(10):1661-1669

Authors: Maruyama A, Nagashima T, Iwamoto M, Minota S

Abstract
This study was performed to investigate the clinical characteristics of lupus enteritis in Japanese patients with systemic lupus erythematosus (SLE). A total of 481 patients with SLE admitted to our hospital between 2001 and 2015 were retrospectively reviewed. Diagnosis of lupus enteritis was based on the following three criteria: (1) abdominal symptoms, (2) diffuse long-segment bowel thickening and (3) a requirement for glucocorticoid therapy. Lupus enteritis was identified in 17 patients (3.5%) and there were two distinct types: small intestine-dominant and large intestine-dominant. Significant differences between the two types were noted with respect to the age, frequency of biopsy-proven lupus nephritis, frequency of rectal involvement, maximum bowel wall thickness, and requirement for steroid pulse therapy. Among patients with large intestine-dominant lupus enteritis, 60% had extra-intestinal symptoms (hydroureter, bladder wall thickening, and bile duct dilatation) that are known complications of intestinal pseudo-obstruction. Two patients with large intestine-dominant lupus enteritis developed intestinal pseudo-obstruction either before or after diagnosis of lupus enteritis. Five patients (29%) developed recurrence during a median observation period of 7.2 years (1.4-14.4 years). In conclusion, large intestine-dominant lupus enteritis resembles intestinal pseudo-obstruction and these two diseases may have a common pathogenesis.

PMID: 30028259 [PubMed - indexed for MEDLINE]

Distinct regional brain atrophy pattern in multiple sclerosis and neuropsychiatric systemic lupus erythematosus patients.

Vascularites - mer, 28/11/2018 - 23:58
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Distinct regional brain atrophy pattern in multiple sclerosis and neuropsychiatric systemic lupus erythematosus patients.

Lupus. 2018 Sep;27(10):1624-1635

Authors: Kalinowska-Lyszczarz A, Pawlak MA, Pietrzak A, Pawlak-Bus K, Leszczynski P, Puszczewicz M, Paprzycki W, Kozubski W, Michalak S

Abstract
Differentiation of systemic lupus erythematosus (SLE) from multiple sclerosis (MS) can be challenging, especially when neuropsychiatric (NP) symptoms are accompanied by white matter lesions in the brain. Given the lack of discriminative power of currently applied tools for their differentiation, there is an unmet need for other measures that can aid in distinguishing between the two autoimmune disorders. In this study we aimed at exploring whether brain atrophy measures could serve as markers differentiating MS and SLE. Thirty-seven relapsing-remitting MS and 38 SLE patients with nervous system manifestations, matched according to age and disease duration, underwent 1.5 Tesla magnetic resonance imaging (MRI), including volumetric sequences, and clinical assessment. Voxelwise analysis was performed using ANTS-SyN elastic registration protocol, FSL Randomise and Gamma methods. Cortical and subcortical segmentation was performed with Freesurfer 5.3 pipeline using T1-weighted MPRAGE sequence data. Using MRI volumetric markers of general and subcortical gray matter atrophy and clinical variables, we built a stepwise multivariable logistic diagnostic model to identify MRI parameters that best differentiate MS and SLE patients. We found that the best volumetric predictors to distinguish them were: fourth ventricle volume (sensitivity 0.86, specificity 0.57, area under the curve, AUC 0.77), posterior corpus callosum (sensitivity 0.81, specificity 0.57, AUC 0.68), and third ventricle to thalamus ratio (sensitivity 0.42, specificity 0.84, AUC 0.65). The same classifiers were identified in a subgroup analysis that included patients with a short disease duration. In MS brain atrophy and lesion load correlated with clinical disability, while in SLE age was the main determinant of brain volume. This study proposes new imaging parameters for differential diagnosis of MS and SLE with central nervous system involvement. We show there is a different pattern of atrophy in MS and SLE, and the key structural volumes that are differentially affected include fourth ventricle and posterior section of corpus callosum, followed by third ventricle to thalamus ratio. Different correlation patterns between volumetric and clinical data may suggest that while in MS atrophy is driven mainly by disease activity, in SLE it is mostly associated with age. However, these results need further replication in a larger cohort.

PMID: 29950159 [PubMed - indexed for MEDLINE]

Septic Pulmonary Emboli From Peripheral Suppurative Thrombophlebitis: A Case Report and Literature Review.

Vascularites - mer, 28/11/2018 - 23:58
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Septic Pulmonary Emboli From Peripheral Suppurative Thrombophlebitis: A Case Report and Literature Review.

Vasc Endovascular Surg. 2018 Nov;52(8):633-635

Authors: Ho VT, Rothenberg KA, McFarland G, Tran K, Aalami OO

Abstract
BACKGROUND:: We report the case of a 90-year old woman who presented with septic pulmonary emboli due to suppurative thrombophlebitis at an old peripheral intravenous site.
METHODS:: After unsuccessful treatment with antibiotics, the patient was taken to the operating room for excision and drainage of the purulent superficial vein.
RESULTS:: We review the literature and discuss the presentation, risk factors, treatment options, and complications of this often-overlooked disease entity.
CONCLUSIONS:: Suppurative thrombophlebitis is a rare but morbid disease that requires a high level of clinical suspicion to diagnose.

PMID: 29909751 [PubMed - indexed for MEDLINE]

Endovascular Thrombectomy of Septic Thrombophlebitis of the Inferior Vena Cava: Case Report and Review of the Literature.

Vascularites - mer, 28/11/2018 - 23:58
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Endovascular Thrombectomy of Septic Thrombophlebitis of the Inferior Vena Cava: Case Report and Review of the Literature.

Vasc Endovascular Surg. 2018 Nov;52(8):641-647

Authors: Talaie T, Drucker C, Aicher B, Khalifeh A, Lal B, Sarkar R, Toursavadkohi S

Abstract
We describe the cases of 2 patients who had septic thrombophlebitis and were successfully managed with endovascular thrombectomy. Patient A developed septic thrombophlebitis of the inferior vena cava after several retroperitoneal resections for metastatic renal cell carcinoma. The thrombus was successfully removed via endovascular mechanical balloon thrombectomy. Patient B was a patient with pancreatic adenocarcinoma involving the portal vein who developed a septic inferior vena cava thrombus extending from the level and beyond the renal veins, for which she underwent endovascular thrombectomy. We argue that this approach is safe and feasible. It should be considered as a supplemental treatment modality for select decompensating patients who require lifesaving interventions and have contraindications to traditional management of surgical thrombectomy or excision of the involved venous segment.

PMID: 29898647 [PubMed - indexed for MEDLINE]

Intractable Hemophagocytic Syndrome Associated with Systemic Lupus Erythematosus Resistant to Corticosteroids and Intravenous Cyclophosphamide That Was Successfully Treated with Cyclosporine A.

Vascularites - mer, 28/11/2018 - 23:58
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Intractable Hemophagocytic Syndrome Associated with Systemic Lupus Erythematosus Resistant to Corticosteroids and Intravenous Cyclophosphamide That Was Successfully Treated with Cyclosporine A.

Intern Med. 2018 Sep 15;57(18):2747-2752

Authors: Toko H, Tsuboi H, Umeda N, Honda F, Ohyama A, Takahashi H, Abe S, Yokosawa M, Asashima H, Hagiwara S, Hirota T, Kondo Y, Matsumoto I, Sumida T

Abstract
Hemophagocytic syndrome (HPS) associated with systemic lupus erythematosus (SLE), dubbed acute lupus hemophagocytic syndrome (ALHS), is an intractable complication of SLE. A 24-year-old man who had been diagnosed with SLE three months previously, presented with fever, rash, hallucination, and pancytopenia accompanied with hyperferritinemia and bone marrow hemophagocytosis. He was diagnosed with ALHS and neuropsychiatric (NP)-SLE. Although 4 courses of methylprednisolone pulse therapy and 1 course of intravenous cyclophosphamide (IVCY) improved his NP-SLE, his ALHS did not respond. However, the addition of cyclosporine A (CsA) led to a rapid remission from ALHS. This suggests the usefulness of CsA in the treatment of intractable, corticosteroid- and IVCY-resistant ALHS.

PMID: 29780126 [PubMed - indexed for MEDLINE]

Significantly higher frequencies of hemoglobin, iron, vitamin B12, and folic acid deficiencies and of hyperhomocysteinemia in patients with Behcet's disease.

Vascularites - mer, 28/11/2018 - 23:58
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Significantly higher frequencies of hemoglobin, iron, vitamin B12, and folic acid deficiencies and of hyperhomocysteinemia in patients with Behcet's disease.

J Formos Med Assoc. 2018 Oct;117(10):932-938

Authors: Kuo YS, Chang JY, Wang YP, Wu YC, Wu YH, Sun A

Abstract
BACKGROUND/PURPOSE: Behcet's disease (BD) patients may have one or more hematinic deficiencies. This study evaluated whether there were significantly higher frequencies of hemoglobin, iron, vitamin B12, and folic acid deficiencies and of hyperhomocysteinemia in BD patients.
METHODS: The blood hemoglobin, iron, vitamin B12, folic acid, and homocysteine concentrations in 63 BD patients were measured and compared with the corresponding levels in 126 age- and sex-matched healthy control subjects.
RESULTS: We found that 30.2%, 34.9%, 6.3%, 6.3%, and 14.3% of 63 BD patients had hemoglobin, iron, vitamin B12, and folic acid deficiencies and hyperhomocysteinemia, respectively. Both 63 BD and 19 major-typed RAS/BD patients had significantly higher frequencies of hemoglobin, iron, vitamin B12, and folic acid deficiencies and of hyperhomocysteinemia than 126 healthy control subjects (all P-values < 0.05). Moreover, 44 minor-typed RAS/BD patients had significantly higher frequencies of hemoglobin and iron deficiencies than 126 healthy control subjects (both P-values < 0.001). Of the 19 anemic BD patients, one had pernicious anemia, one had macrocytic anemia, 12 had normocytic anemia, four had iron deficiency anemia, and one had thalassemia trait-induced anemia.
CONCLUSION: There are significantly higher frequencies of hemoglobin, iron, vitamin B12, and folic acid deficiencies and of hyperhomocysteinemia in BD patients. The normocytic anemia is the most common type of anemia in our 63 BD patients. We suggest that supplementations of vitamin BC capsules plus deficient vitamin B12 and/or folic acid may reduce the abnormally high serum homocysteine level to a relatively lower level in BD patients with hyperhomocysteinemia.

PMID: 29716764 [PubMed - indexed for MEDLINE]

An Autopsy Case of Myeloperoxidase-anti-neutrophil Cytoplasmic Antibody (MPO-ANCA)-associated Vasculitis Accompanied by Cryoglobulinemic Vasculitis Affecting the Kidneys, Skin, and Gastrointestinal Tract.

Vascularites - mer, 28/11/2018 - 23:58
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An Autopsy Case of Myeloperoxidase-anti-neutrophil Cytoplasmic Antibody (MPO-ANCA)-associated Vasculitis Accompanied by Cryoglobulinemic Vasculitis Affecting the Kidneys, Skin, and Gastrointestinal Tract.

Intern Med. 2018 Sep 15;57(18):2739-2745

Authors: Hasegawa J, Wakai S, Kono M, Imaizumi Y, Masuda S, Ishizu A, Honda K

Abstract
Anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV) and cryoglobulinemic vasculitis (CV) rarely coexist. An 83-year-old woman was admitted with rapidly progressive renal failure, gastrointestinal hemorrhage and purpura with myeloperoxidase (MPO)-ANCA positivity and cryoglobulinemia. Despite intensive immunosuppressive treatment, she died of aspergillus pneumonia. Autopsy revealed necrotizing crescentic glomerulitis in the majority of the glomeruli, accompanied by partially membranoproliferative-like glomerular changes. Immunofluorescence staining revealed the presence of neutrophil extracellular trap (NET) formation in the glomeruli and cutaneous arteries. These pathological findings suggested that MPO-AAV and/or CV caused NET formation, leading to lethal systemic vasculitis.

PMID: 29709950 [PubMed - indexed for MEDLINE]

Dilated Cardiomyopathy with Eosinophilic Granulomatosis with Polyangiitis in Which Active Myocardial Inflammation Was Only Detected by Endomyocardial Biopsy.

Vascularites - mer, 28/11/2018 - 23:58
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Dilated Cardiomyopathy with Eosinophilic Granulomatosis with Polyangiitis in Which Active Myocardial Inflammation Was Only Detected by Endomyocardial Biopsy.

Intern Med. 2018 Sep 15;57(18):2675-2679

Authors: Nakayama T, Murai S, Ohte N

Abstract
Eosinophilic granulomatosis with polyangiitis (EGPA) is a rare type of systemic vasculitis. Cardiac involvement is the main cause of death in patients with this disease. We herein report a case of congestive heart failure in a patient with EGPA. Neither 67Ga scintigraphy nor cardiac magnetic resonance imaging detected inflammation of the myocardium; however, myocardial biopsy revealed numerous infiltrating inflammatory cells, thereby fulfilling the criteria of inflammatory dilated cardiomyopathy. We improved the left ventricular systolic function by increasing the patient's prednisolone dosage. This case shows that in some cases the detection myocardial inflammation - which allows for appropriate therapy - may only be achieved by myocardial biopsy.

PMID: 29709930 [PubMed - indexed for MEDLINE]

Role of Direct Immunofluorescence in Cutaneous Small-Vessel Vasculitis: Experience From a Tertiary Center.

Vascularites - mer, 28/11/2018 - 23:58
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Role of Direct Immunofluorescence in Cutaneous Small-Vessel Vasculitis: Experience From a Tertiary Center.

Am J Dermatopathol. 2018 Sep;40(9):661-666

Authors: Lath K, Chatterjee D, Saikia UN, Saikia B, Minz R, De D, Handa S, Radotra B

Abstract
Skin is commonly affected by vasculitic process and often subjected to biopsy. Cutaneous vasculitis can be either primary or part of a systemic vasculitic process. This study was conducted to evaluate the diagnostic utility of direct immunofluorescence (DIF) in determination of etiology of cutaneous vasculitis. All histologically proven cases of cutaneous vasculitis over the past two and half years were retrospectively analyzed along with their clinical and DIF findings (IgG, IgA, IgM, and C3). Within this study period, a total of 198 cases of small-vessel vasculitis were diagnosed based on skin biopsy and DIF findings. The mean age of patients was 31.2 years (range 1-84 years) with slight male dominance (M:F ratio 1.06:1). Henoch-Schonlein purpura/IgA vasculitis was the commonest clinical diagnosis (31%), followed by urticarial vasculitis (11%) and others. Idiopathic vasculitis was suspected in 33% cases. Overall, DIF was positive in 60% (119/198) cases, with vascular deposition of IgA being commonest, followed by C3. The clinical diagnosis of Henoch-Schonlein purpura could be confirmed in 61.5% (40/65) cases by DIF, whereas another 20 unsuspected cases were picked up as IgA vasculitis based on DIF findings. DIF findings confirmed lupus vasculitis in 50% cases. Other cases showed variable nonspecific deposition of C3 and IgM in 42% cases. DIF can be highly useful to classify cutaneous vasculitis, with maximum efficacy for diagnosis of IgA vasculitis and lupus vasculitis. It can aid in the accurate diagnosis even when the histological changes are minimal. All cases of suspected cutaneous vasculitis should be subjected to DIF.

PMID: 29672364 [PubMed - indexed for MEDLINE]

Routine CT scanning of patients retrieved to a tertiary centre on veno-venous extracorporeal membrane oxygenation: a retrospective risk benefit analysis.

Vascularites - mer, 28/11/2018 - 23:58
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Routine CT scanning of patients retrieved to a tertiary centre on veno-venous extracorporeal membrane oxygenation: a retrospective risk benefit analysis.

Perfusion. 2018 09;33(6):438-444

Authors: Richmond KM, Warburton KG, Finney SJ, Shah S, Reddi BAJ

Abstract
INTRODUCTION: Comprehensive clinical examination can be compromised in patients on veno-venous extracorporeal membrane oxygenation (VV-ECMO). Adjunctive diagnostic imaging strategies range from bedside imaging only to routine computed tomography (CT). The risk-benefit of either approach remains to be evaluated. Patients retrieved to the Royal Brompton Hospital (RBH) on VV-ECMO routinely undergo admission CT imaging of head, chest, abdomen and pelvis. This study aimed to identify how frequently changes in therapy or adverse events could be attributed to routine CT scanning.
METHODS: Demographic and clinical data were gathered retrospectively from patients retrieved to RBH on VV-ECMO (January 2014-2016). Scans were categorized as 'routine' or requested to clarify a specific clinical uncertainty. Clinical records were reviewed to identify attributable management changes and CT- related adverse events. Seventy-two patients were retrieved on VV-ECMO (median age 44 years) and 65 scanned on admission (mean radiation dose 2344mGy-cm). Routine head CT head yielded novel clinical information in 11 patients, 10 of whom had unexpected intracranial haemorrhage and, subsequently, had their anticoagulation withheld. Routine thoracic CT identified unexpected positive findings in three patients (early fibrosis, pulmonary vasculitis, pneumomediastinum), eliciting management variation in one (steroid administration). Routine abdomen/pelvis CT identified new information in three patients (adrenal haemorrhage, hepatosteatosis, splenic infarction), changing the management in one (withholding anticoagulation).
RESULTS: CT scanning was not associated with consequential adverse events (e.g. accidental decannulation, gas entrainment into the circuit, hypoxia, hypotension). Median transfer/scan time was 78 minutes, requiring five ITU staff-members. In our cohort, a policy of routine head CT changed the management in 17% of patients; the yield from routine chest, abdomen and pelvis CT was modest. CT transfer was safe, but resource intensive.
CONCLUSION: Prospective studies should evaluate whether routine CT impacts outcome.

PMID: 29529977 [PubMed - indexed for MEDLINE]

Utility of a blood culture time to positivity-incorporated scoring model in predicting vascular infections in adults with nontyphoid Salmonella bacteremia.

Vascularites - mer, 28/11/2018 - 23:58
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Utility of a blood culture time to positivity-incorporated scoring model in predicting vascular infections in adults with nontyphoid Salmonella bacteremia.

J Microbiol Immunol Infect. 2018 Oct;51(5):652-658

Authors: Lin JJ, Weng TH, Tseng WP, Chen SY, Fu CM, Lin HW, Liao CH, Lee TF, Hsueh PR, Chen SY

Abstract
BACKGROUND: Vascular infections (VI) are potentially catastrophic complications of nontyphoid Salmonella (NTS). We aimed to develop a scoring model incorporating information from blood culture time to positivity (TTP-NTSVI) and compared the prediction capability for VI among adults with NTS bacteremia between TTP-NTSVI and a previously published score (Chen-NTSVI).
METHODS: This retrospective cohort study enrolled 217 adults with NTS bacteremia ≧ 50 years old. We developed a TTP-NTSVI score by multiple logistic regression modeling to identify independent predictors for imaging-confirmed VI and assigned a point value weighting by the corresponding natural logarithm of the odds ratio for each model predictor. Chen-NTSVI score includes hypertension, male sex, serogroup C1, coronary arterial disease (CAD) as positive predictors, and malignancy and immunosuppressive therapy as negative predictors. The prediction capability of the two scores was compared by area under the receiver operating characteristic curve (AUC).
RESULTS: The mean age was 68.3 ± 11.2 years-old. Serogroup D was the predominant isolate (155/217, 71.4%). Seventeen (7.8%) patients had VI. Four independent predictors for VI were identified: male sex (24.9 [2.59-239.60]; 6) (odds ratio [95% confidence interval]; assigned score point), peripheral arterial occlusive disease (9.41 [2.21-40.02]; 4), CAD (4.0 [1.16-13.86]; 3), and TTP <10 h (4.67 [1.42-15.39]; 3). Youden's index showed best cutoff value of ≧7 with 70.6% sensitivity and 82.5% specificity. TTP-NTSVI score had higher AUC than Chen-NTSVI (0.851 vs 0.741, P = 0.039).
CONCLUSION: While the previously reported scoring model performed well, a TTP-incorporated scoring model was associated with improved capability in predicting NTSVI.

PMID: 29501434 [PubMed - indexed for MEDLINE]

High-resolution Intracranial Vessel Wall Imaging in Monitoring Treatment Response in Primary CNS Angiitis.

Vascularites - dim, 25/11/2018 - 22:16
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High-resolution Intracranial Vessel Wall Imaging in Monitoring Treatment Response in Primary CNS Angiitis.

Neurologist. 2018 Nov;23(6):188-190

Authors: Tsivgoulis G, Papadimitropoulos GN, Lachanis S, Palaiodimou L, Zompola C, Antonellou R, Voumvourakis K

Abstract
INTRODUCTION: High-resolution vessel wall imaging (HR-VWI) is emerging as a tool of notable utility in the diagnosis of intracranial vessel pathology. Its role in monitoring vessel wall disease response to treatment, however, is less well-established.
CASE REPORT: We report the case of a 45-year-old man with left middle and anterior cerebral artery infarcts and an National Institute of Health Stroke Scale (NIHSS) score of 2. Time-of-flight magnetic resonance angiography and digital subtraction angiography showed multifocal intracranial vessel pathology without extracranial vessel involvement. Comprehensive investigation with echocardiography and 24 hours Holter electrocardiography was unrevealing and the coagulation and routine autoimmune panel results were within normal limits. Cerebrospinal fluid showed mildly elevated protein and a diagnosis of probable primary central nervous system (PCNS) angiitis was made. The diagnosis was corroborated by intracranial HR-VWI, which showed homogenous, concentric enhancement of the left supraclinoid internal carotid artery (ICA) wall. The patient received high-dose IV methylprednisolone and cyclophosphamide. Repeat brain magnetic resonance imaging with HR-VWI at 3 and 9 months showed reduction and final resolution of vessel wall enhancement without recurrent infarcts. He has since remained clinically stable with an NIHSS score of 0 on low-dose oral glucocorticoids.
CONCLUSIONS: Our report illustrates the utility of HR-VWI in diagnosing a case of PCNS angiitis through the demonstration of a vasculitic pattern of mural enhancement. Furthermore, it has provided evidence of disease response to treatment, assisting us in modifying treatment accordingly. Tracking disease activity and response to treatment in cases of central nervous system vasculitis can be another important use of HR-VWI in clinical practice besides assisting in establishing the diagnosis.

PMID: 30379741 [PubMed - indexed for MEDLINE]

Hughes-Stovin syndrome: the diagnostic and therapeutic challenges of peripheral pulmonary artery aneurysms.

Vascularites - dim, 25/11/2018 - 22:16
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Hughes-Stovin syndrome: the diagnostic and therapeutic challenges of peripheral pulmonary artery aneurysms.

QJM. 2018 Oct 01;111(10):729-730

Authors: Robinson C, Miller D, Will M, Dhaun N, Walker W

PMID: 29860510 [PubMed - indexed for MEDLINE]

Performance of noninvasive laser Doppler flowmetry and laser speckle contrast imaging methods in diagnosis of Buerger disease: A case report.

Vascularites - jeu, 22/11/2018 - 20:41
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Performance of noninvasive laser Doppler flowmetry and laser speckle contrast imaging methods in diagnosis of Buerger disease: A case report.

Medicine (Baltimore). 2018 Oct;97(43):e12979

Authors: Omarjee L, Larralde A, Jaquinandi V, Stivalet O, Mahe G

Abstract
RATIONALE: Buerger disease (BD) is a nonatherosclerotic, inflammatory, segmental vascular occlusive disease, which affects small and medium-sized arteries and veins and is triggered by substantial tobacco exposure. Angiographic findings consistent with BD are required for diagnosis. Laser Doppler flowmetry (LDF) and laser speckle contrast imaging (LSCI) could represent potential noninvasive alternative techniques to angiography.
PATIENT CONCERNS: We report the case of a 49-year-old smoker who developed an ischemic ulcer in the distal segment of the second finger of the left hand. He had no medical history.
DIAGNOSES: In our vascular center, LDF and LSCI are conducted routinely for digital artery disease diagnosis. LDF was indicative of digital obstructive artery disease (DOAD). Postocclusive reactive hyperemia, assessed by LCSI, demonstrated no skin blood flow (SBF) perfusion in the distal phalanx of the thumb, index, middle, and auricular fingers. Angiography confirmed BD, showing distally located multisegmental vessel occlusion and corkscrew collaterals in this patient's hands.
INTERVENTIONS: Ilomedine treatment was initiated and smoking cessation was definitive.
OUTCOMES: Recently, the patient had an improvement in clinical condition despite the persistence of a small zone of necrosis of the left index finger 28 days post-treatment.
LESSONS: Our observation suggests that where suspicion of BD is based on clinical criteria, combining LDF and LSCI could represent a noninvasive, safe means of reaching BD diagnosis. Further clinical trials are necessary to confirm this novel observation.

PMID: 30412126 [PubMed - indexed for MEDLINE]

A case report of granulomatous polyangiitis complicated by tuberculous lymphadenitis.

Vascularites - jeu, 22/11/2018 - 20:41
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A case report of granulomatous polyangiitis complicated by tuberculous lymphadenitis.

Medicine (Baltimore). 2018 Oct;97(43):e12430

Authors: Iijima Y, Kobayashi Y, Uchida Y, Tsutsui T, Kakizaki Y, Naganuma T, Tsukamoto K, Oyama T, Miyashita Y

Abstract
RATIONAL: Granulomatous polyangiitis (GPA) is a type of vasculitis involving medium and small arteries, typically affecting the upper and lower respiratory tract with coexisting glomerulonephritis. GPA is also characterized by necrotizing granulomatous inflammation and the presence of antineutrophil cytoplasm antibodies (ANCA). So far, various infections have lead to elevation of titers of serum ANCA, making it difficult to diagnose.
PATIENT CONCERNS: We report a 50-year-old woman who was diagnosed as tuberculous lymphadenitis. During the treatment by anti-tuberculosis (TB) drugs, rapidly progressive renal failure and pleurisy had appeared with elevated titer of PR3-ANCA. Renal biopsy revealed crescentic glomerulonephritis.
DIAGNOSIS: Renal biopsy revealed crescentic glomerulonephritis and diagnosis of GPA was made.
INTERVENTIONS: Steroid therapy had been started with continuation of anti-TB drugs.
OUTCOMES: Renal dysfunction had gradually recovered and pleurisy had disappeared with decreasing titer of PR3-ANCA.
LESSONS: This is the first report of GPA complicated by TB infection. When we encounter a case with rapidly progressive renal failure during the TB infection, complication of GPA should be suspected as 1 of the different diagnosis.

PMID: 30412059 [PubMed - indexed for MEDLINE]

Beneficial use of serum ferritin and heme oxygenase-1 as biomarkers in adult-onset Still's disease: A multicenter retrospective study.

Vascularites - jeu, 22/11/2018 - 20:41
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Beneficial use of serum ferritin and heme oxygenase-1 as biomarkers in adult-onset Still's disease: A multicenter retrospective study.

Mod Rheumatol. 2018 Sep;28(5):858-864

Authors: Kirino Y, Kawaguchi Y, Tada Y, Tsukamoto H, Ota T, Iwamoto M, Takahashi H, Nagasawa K, Takei S, Horiuchi T, Ichida H, Minota S, Ueda A, Ohta A, Ishigatsubo Y

Abstract
BACKGROUND: Heme oxygenase (HO)-1 is a heme-degrading enzyme highly expressed in monocyte/macrophage, serum levels of which may be promising biomarker for adult-onset Still's disease (AOSD). We here report data on the use of serum ferritin and HO-1 levels in AOSD.
METHODS: Under the Hypercytokinemia Study Group collaboration, we collected sera from a total of 145 AOSD patients. Three independent experts judged whether the patients were definite AOSD depending on the clinical information. These 91 'definite AOSD' patients were further divided into active, remission, and relapse groups. Forty-six cases of systemic vasculitis, sepsis, etc. were included as disease controls. Serum ferritin and HO-1 levels were measured using ELISA. Associations between clinical symptoms, serum ferritin, and HO-1 were explored. Multivariate regression analysis was performed to identify independent variables associated with definite AOSD diagnosis.
RESULTS: Serum ferritin and HO-1 levels were significantly higher in active and relapsed AOSD cases compared to disease controls, and were reduced by the treatment. Although a significant correlation was found between serum ferritin and HO-1 levels, a discrepancy was found in some cases such as iron-deficiency anemia. Receiver operating characteristic analysis identified optimal levels of serum ferritin (>819 ng/ml; sensitivity 76.1% and specificity 73.8%), and serum HO-1 (>30.2 ng/ml; sensitivity 84.8% and specificity 83.3%) that differentiated AOSD from controls. Interestingly, 88.9% of patients with AOSD who relapsed exceeded the cut-off value of serum HO-1 > 30.2 ng/ml, but only 50.0% exceeded serum ferritin >819 ng/ml (p = .013), suggesting that serum HO-1 levels may be a convenient indicator of AOSD disease status. Multivariate analysis identified neutrophilia, RF/ANA negativity, sore throat, and elevated serum HO-1 as independent variables associated with AOSD diagnosis.
CONCLUSION: We confirmed that serum ferritin and HO-1 serve as highly specific and sensitive biomarkers for AOSD. A future prospective study with large sample size is necessary to determine whether these biomarkers could be included in Yamaguchi's Criteria.

PMID: 29278009 [PubMed - indexed for MEDLINE]

Intestinal Behçet's disease with pyoderma gangrenosum successfully treated with the combination therapy of adalimumab and glucocorticoids.

Vascularites - jeu, 22/11/2018 - 20:41
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Intestinal Behçet's disease with pyoderma gangrenosum successfully treated with the combination therapy of adalimumab and glucocorticoids.

Mod Rheumatol. 2018 Sep;28(5):901-905

Authors: Kashiwado Y, Uchino A, Ota T, Nagano S

Abstract
A 24-year-old male presented with orogenital ulcers, folliculitis, and progressive painful skin ulcers with a raised inflammatory border. Colonoscopy revealed volcano-shaped intestinal ulcers in the ascending colon, and hence, he was diagnosed as intestinal Behçets disease (BD) with pyoderma gangrenosum (PG). Treatment with systemic glucocorticoids and adalimumab dramatically improved the patient's symptoms. Our case demonstrates that early induction of adalimumab may contribute to the successful treatment of such difficult-to-treat conditions as intestinal BD with PG.

PMID: 27142127 [PubMed - indexed for MEDLINE]

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