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Prevalence of Mycobacterium avium subspecies paratuberculosis IS 900 DNA in biopsy tissues from patients with Crohn's disease: histopathological and molecular comparison with Johne's disease in Fars province of Iran.

Vascularites - sam, 16/02/2019 - 16:14
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Prevalence of Mycobacterium avium subspecies paratuberculosis IS 900 DNA in biopsy tissues from patients with Crohn's disease: histopathological and molecular comparison with Johne's disease in Fars province of Iran.

BMC Infect Dis. 2019 Jan 07;19(1):23

Authors: Zarei-Kordshouli F, Geramizadeh B, Khodakaram-Tafti A

Abstract
BACKGROUND: Crohn's disease is a chronic enteritis of humans that affects the gastrointestinal tract, especially the terminal ileum, cecum and colon. The etiology of this disease is still unknown but seems to be multifactorial. There are reports about the potential link between Crohn's disease in humans and the causative agent of Johne's disease in ruminants. Because of the prevalence of Johne's disease in the Fars Province of Iran, the aim of this study was to investigate the prevalence of MAP in the biopsy tissues of patients affected by Crohn's disease in this area.
METHODS: The study was performed from April 2015 to June 2017 at Namazi Hospital, Shiraz University of Medical Sciences, and School of Veterinary Medicine, Shiraz University, Shiraz, Iran. Intestinal biopsies of 30 patients (12 male and 18 female; mean age, 34 years; range 4-77 years) with the confirmed diagnosis of Crohn's disease and 30 patients diagnosed as non-inflammatory bowel disease (19 male and 11 female; mean age, 38 years; range 13-68 years) were studied by molecular, histopathological and histochemical methods. Also, similar numbers of adult goats affected by Johne's disease were studied, comparatively. DNA extractions of tissue specimens were subjected to PCR to amplify a 413-bp sequence of the IS900 gene.
RESULTS: Using IS900-PCR, the overall prevalence of MAP in patients affected by Crohn's disease and non-inflammatory bowel disease were 47 and 13%, respectively. In addition, the prevalence of MAP in goats affected by Johne's disease was 70%. Using acid-fast histochemical staining, only 7% of Crohn's disease patients were weakly positive as paucibacillary and 43% of Johne's disease cases were moderate to strongly positive as multibacillary. Histopathologically, granulomatous enteritis (83 and 90%), lymphoplasmacytic enteritis (17 and 14%), edema and lymphangiectasia (67 and 96%), and vasculitis (20 and 73%) were common findings in Crohn's and Johne's diseases, respectively.
CONCLUSION: Our findings demonstrate a remarkable association between MAP and CD in this population, and support an etiologic relationship between MAP infection in humans and the development of CD. MAP infection in human tissue may display species-specific pathologic findings, as occurs with other zoonotic pathogens.

PMID: 30616527 [PubMed - indexed for MEDLINE]

Superior Mesenteric Artery Vasculitis in Behçet's Disease: A Case Report and Literature Review.

Vascularites - sam, 16/02/2019 - 16:14
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Superior Mesenteric Artery Vasculitis in Behçet's Disease: A Case Report and Literature Review.

Intern Med. 2019 Jan 01;58(1):127-133

Authors: Kakehi E, Adachi S, Fukuyasu Y, Hashimoto Y, Yoshida M, Osaka T, Hirotani A, Danbara H, Shimizu K, Fujita R, Kotani K, Matsumura M

Abstract
A 55-year-old male presented with abdominal pain that had begun about 5 days ago. Physical examination revealed oral aphtha, genital aphthosis, and pseudofolliculitis, and the patient was diagnosed with incomplete Behçet's disease (BD). Contrast-enhanced computed tomography (CECT) showed dilation of the superior mesenteric artery and mesenteric infiltration of inflammation, indicating vasculo-BD. The symptoms were improved by 3-day of intravenous methylprednisolone pulse therapy followed by oral prednisolone. A literature review suggested that vasculo-BD should be included as a differential diagnosis in cases with unexplained abdominal pain, arterial dilation, and mesenteric invasion, and CECT examination and steroid therapy should be considered.

PMID: 30146589 [PubMed - indexed for MEDLINE]

Serum uric acid could be served as an independent marker for increased risk and severity of ascending aortic dilatation in Behçet's disease patients.

Vascularites - sam, 16/02/2019 - 16:14
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Serum uric acid could be served as an independent marker for increased risk and severity of ascending aortic dilatation in Behçet's disease patients.

J Clin Lab Anal. 2019 Jan;33(1):e22637

Authors: Cai J, Zhang Y, Zou J, Shen Y, Luo D, Bao H, Chen Y, Ye J, Guan JL

Abstract
OBJECTIVE: This study aimed to investigate the correlation of serum uric acid (SUA) with risk and dilatation diameter of ascending aortic dilatation (AAD) in Behçet's disease (BD) patients.
METHODS: Seventeen BD patients complicated with AAD and 20 BD patients without AAD were consecutively enrolled and categorized into AAD group and control group, respectively. Ascending aortic dilatation was determined by two-dimensional doppler echocardiographic examination, and AAD was defined as a diameter of ascending aorta ≥3.8 and <4.4 cm. SUA was detected by quantitative immunoassay method.
RESULTS: Ascending aortic dilatation patients presented with higher proportion of male patients (P = 0.003), hypertension occurrence (P = 0.036) and increased diameter of ascending aorta (P < 0.001) compared to controls. SUA was elevated in AAD patients compared to controls (P = 0.002), and receiver operating characteristic curve displayed that SUA presented with great predictive value for AAD risk in BD patients with area under curve (AUC) 0.821 (95% CI 0.675-0.966). Pearson's analysis also disclosed that SUA was positively correlated with ascending aortic diameter in total BD patients. However, no difference of CRP (P = 0.219) or ESR (P = 0.320) between AAD patients and controls was observed, and no correlation of CRP (R = -0.150, P = 0.377) or ESR (R = 0.067, P = 0.692) with ascending aortic diameter in total BD patients was discovered either. Further multivariate logistic regression illuminated that SUA was an independent factor predicting AAD risk in BD patients (P = 0.031).
CONCLUSIONS: Serum uric acid could be served as an independent marker for increased risk and severity of AAD in BD patients.

PMID: 30101545 [PubMed - indexed for MEDLINE]

Aging in Primary Systemic Vasculitis: Implications for Diagnosis, Clinical Manifestations, and Management.

Vascularites - mer, 13/02/2019 - 15:24
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Aging in Primary Systemic Vasculitis: Implications for Diagnosis, Clinical Manifestations, and Management.

Drugs Aging. 2019 01;36(1):53-63

Authors: Berti A, Caporali R, Montecucco C, Paolazzi G, Monti S

Abstract
Anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitides (AAV) usually affect adults and older populations. The well recognized clinical (i.e., granulomatosis with polyangiitis [GPA], microscopic polyangiitis, and eosinophilic GPA) and serological phenotypes (i.e., anti-MPO-ANCA, anti-PR3-ANCA and ANCA negative) within AAV differ substantially for clinical, demographic, and epidemiological features, including age at presentation. Whether and how aging could contribute to the clinical expression of these disease phenotypes is intriguing and still overlooked. In addition, despite being predominantly a disease of the elderly, most of the studies analyzing drug interventions and the clinical trials on AAV explicitly excluded older patients, limiting the understanding of the disease in this subset of patients. In elderly patients induced with cyclophosphamide, a lower dose of treatment for patients aged 60 years or older and with reduced renal function has been recommended. Giant cell arteritis (GCA) and Takayasu's arteritis (TAK) are two primary systemic vasculitides involving large vessels that differ in age at presentation, with GCA patients being at least 10 years older than TAK patients. Different treatment approaches are effective in these conditions. However, a few authors have suggested that they might be considered expressions of the same clinical syndrome, rather than two different diseases. Novel insights into the role of senescence-related immunological and vascular processes might help to interpret the link between these two conditions. Overall, the impact of aging on all these vasculitides is complex and not easy to analyze. So far, few studies focusing on this topic have been published. We reviewed data on the clinical presentation, epidemiology, therapy, and disease- and treatment-related complications in patients affected by these vasculitides, highlighting the differences in young versus elderly subjects.

PMID: 30484240 [PubMed - indexed for MEDLINE]

[Kaposi sarcoma in a patient with ANCA vasculitis requiring hemodialysis].

Vascularites - mer, 13/02/2019 - 15:24
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[Kaposi sarcoma in a patient with ANCA vasculitis requiring hemodialysis].

Rev Med Interne. 2018 Dec;39(12):942-945

Authors: Jebali H, Saihi M, Omrane M, Kheder R, Chelly I, Haouet S, Beji S, Raïs L, Mami I, Krid M, Smaoui W, Ben Hmida F, Ben Fatma L, Zouaghi MK

Abstract
INTRODUCTION: Iatrogenic Kaposi's sarcoma is widely reported after transplantation. Less commonly, it occurs in patients receiving immunosuppressive therapy for ANCA associated vasculitis. We report here the rare association of Kaposi's sarcoma, prurigo nodularis and ANCA associated vasculitis in a hemodialysis patient.
CASE REPORT: We describe a 58-year-old woman who presented granulomatosis with polyangeiitis with alveolar hemorrhage and renal failure requiring hemodialysis. She developed cutaneous Kaposi's sarcoma seven weeks after the beginning of immunosuppressive therapy. Biological tests showed negative HHV8 virus infection. Lesions of Kaposi's sarcoma responded to a discontinuation of immunosuppressive drugs and a decreasing dosage of corticosteroids.
CONCLUSION: Our case showed that the immunosuppressed state related to multiple factors such as underlying disease, immunosuppressive therapy and hemodialysis may all have contributed to the development of this neoplastic disorder in our patient.

PMID: 30316478 [PubMed - indexed for MEDLINE]

Cyclophosphamide or Rituximab Treatment of Scleritis and Uveitis for Patients with Granulomatosis with Polyangiitis.

Vascularites - mer, 13/02/2019 - 15:24
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Cyclophosphamide or Rituximab Treatment of Scleritis and Uveitis for Patients with Granulomatosis with Polyangiitis.

Ophthalmic Res. 2019;61(1):44-50

Authors: Ahmed A, Foster CS

Abstract
PURPOSE: Vision-threatening ocular inflammation can be a devastating complication of granulomatosis with polyangiitis (GPA). Here we performed a retrospective observational study to describe the safety and efficacy of treating scleritis and uveitis with either cyclophosphamide or rituximab in GPA.
METHODS: A chart review of patients diagnosed with GPA-associated scleritis or uveitis, treated with either cyclophosphamide or rituximab as the final therapy at our clinic, was conducted. A total of 1 year of follow-up visits was required for inclusion in the study.
RESULTS: Thirteen patients (19 eyes) suffering from GPA-associated scleritis and/or uveitis were identified. As the final therapy, rituximab was administered to 9 patients and cyclophosphamide to 4. Mean duration of follow-up was 55 months (range 16-23 months). Remission was observed in all patients. Three patients had a flare of scleritis after the completion of therapy, and they were restarted on their respective agents. One patient had a flare of retinal vasculitis during rituximab therapy. One patient on cyclophosphamide experienced transient leukopenia. No adverse side effects of rituximab were noted throughout the course of treatment.
CONCLUSIONS: Cyclophosphamide and rituximab are safe and effective agents for controlling scleritis and uveitis associated with GPA, with eventual progression towards steroid-sparing remission.

PMID: 29635229 [PubMed - indexed for MEDLINE]

Concomitant cryoglobulinemic vasculitis and cold agglutinin disease successfully treated with bortezomib: A case report.

Vascularites - dim, 10/02/2019 - 11:56
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Concomitant cryoglobulinemic vasculitis and cold agglutinin disease successfully treated with bortezomib: A case report.

Medicine (Baltimore). 2019 Jan;98(4):e14201

Authors: Liu XH, Liu MX, Jin F, Zhang M, Zhang L

Abstract
RATIONALE: Concomitant cryoglobulinemic vasculitis and cold agglutinin disease (CAD) is an extremely uncommon clinical scenario. The role of bortezomib in the treatment of cryoglobulinemic vasculitis needs further investigation.
PATIENT CONCERNS: A 72-year-old Chinese woman presented with a 25-year history of cyanosis of the extremities after cold exposure, which worsened and was accompanied with purpuric skin lesions and proteinuria in recent years. Laboratory data demonstrated hemolysis. Cold agglutinin and cryoglobulin tests were positive. There was no evidence for malignancies after blood, image, and pathologic tests.
DIAGNOSES: Concomitant cryoglobulinemic vasculitis and CAD.
INTERVENTIONS: The patient was treated with bortezomib-based regimen, including bortezomib, cyclophosphamide, and dexamethasone.
OUTCOMES: The patient responded well to the treatment. Both symptoms and laboratory tests significantly improved. The patient's condition was in a state of sustained remission in the 6-month follow-up.
LESSONS: This rare case promotes further understanding of these 2 diseases and suggests that bortezomib is a promising treatment in type I cryoglobulinemic vasculitis.

PMID: 30681592 [PubMed - indexed for MEDLINE]

Tumor necrosis factor-alpha -308G/A gene polymorphism and novel biomarker profiles in patients with Takayasu arteritis.

Vascularites - dim, 10/02/2019 - 11:56
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Tumor necrosis factor-alpha -308G/A gene polymorphism and novel biomarker profiles in patients with Takayasu arteritis.

Indian Heart J. 2018 Dec;70 Suppl 3:S167-S172

Authors: Sarkar PG, Gupta MD, Girish MP, Bansal A, Kohli S, Saijpaul R, Tyagi S, Pasha Q

Abstract
BACKGROUND: Takayasu arteritis (TA) is an idiopathic chronic inflammatory disease of the aorta and its branches, leading to stenosis, occlusion, and aneurysmal dilatation. Tumor necrosis factor-alpha (TNF-α) is a cytokine with pleomorphic actions and plays a pivotal role in inflammation; the serum level of TNF-α is genetically determined. However, the literature lacks adequate information on the association of TNF-α polymorphisms with TA. Hence, the present study investigates the contribution of TNF-α polymorphism toward the complex etiology of TA.
METHODS: A cross-sectional study was performed in 87 patients with TA and 90 controls. A promoter region polymorphism of TNF-α, rs1800629 G/A, or -308G/A was genotyped in all the study subjects followed by a case-control association study. Furthermore, to understand the biomarker profile, levels of specific markers such as erythrocyte sedimentation rate, serum high-sensitivity C-reactive protein, interleukin-18, interleukin-6, and TNF-α were measured in all the study subjects.
RESULTS: All the inflammatory markers were significantly higher in the TA patients than in the controls. The genetic study (available for 57 TA patients and 36 controls) revealed that the TNF-α -308A allele was overrepresented in the TA patients (12% vs 7%). The TNF-α -308A allele correlated with the increased TNF-α levels, but it could not attain significance because of a small sample size.
CONCLUSION: The TNF-α -308G/A polymorphism is associated with TNF-α levels in Indian population, which might have implications for clinical risk stratification and treatment. The different TNF-α gene promoter polymorphism might contribute to the molecular pathogenesis of TA. However, further study of the underlying mechanism is warranted.

PMID: 30595251 [PubMed - indexed for MEDLINE]

Simultaneous Multi-Supra-Aortic Artery Bypass Successfully Implemented in 17 Patients with Type I Takayasu Arteritis.

Vascularites - dim, 10/02/2019 - 11:56
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Simultaneous Multi-Supra-Aortic Artery Bypass Successfully Implemented in 17 Patients with Type I Takayasu Arteritis.

Eur J Vasc Endovasc Surg. 2018 12;56(6):903-909

Authors: Chen ZG, Chen YX, Diao YP, Wu ZY, Yan S, Ma L, Liu CW, Li YJ

Abstract
OBJECTIVE: Type I Takayasu arteritis is common in Chinese patients and usually leads to cerebral ischaemia. There are limited data regarding simultaneous multi-supra-aortic artery (SMSAA) bypass in patients with type I Takayasu arteritis. The present study shares experience of using this method.
METHODS: Seventeen patients with Takayasu arteritis who received SMSAA bypass from June 2012 to March 2016 were included in this analysis. Demographic features and basic information were recorded. Follow up results, including major complications, patency rates, and survival rates, were also obtained. The EQ-5D questionnaire was also administered to evaluate the quality of life of patients.
RESULTS: Among these 17 patients, there were 77 supra-aortic arteries affected by TA and 37 arteries were reconstructed, 10 with great saphenous vein and 27 with artificial blood vessels. All the operations were technical successes and the mean operation time was 396.6 ± 93.4 min. The patients were followed for a mean of 25.2 ± 12.3 months. Two graft restenoses were found, and both were successfully recanalised by balloon dilatation. One patient suffered a minor stroke but was free from obvious sequelae. No major complications, recurrent symptoms, or deaths occurred during the follow up period. The EQ-5D questionnaire scores were significantly improved after the operation compared with the pre-operative scores (87.14 ± 8.25 vs. 58.93 ± 14.4, p < .01), and all the patients enjoyed a satisfactory quality of life.
CONCLUSION: SMSAA bypass in type I Takayasu arteritis can be effectively and safely performed with rigorous peri-operative management. Maintaining the patency of the bypass graft relieves the symptoms of cerebral ischaemia and helps sustain patients' quality of life.

PMID: 30318396 [PubMed - indexed for MEDLINE]

Use of Apremilast for Aphthous Ulcers in a Patient With Behçet’s Syndrome

Vascularites - jeu, 07/02/2019 - 09:34
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Use of Apremilast for Aphthous Ulcers in a Patient With Behçet’s Syndrome

J Drugs Dermatol. 2018 Dec 01;17(12):1328-1329

Authors: Saini A, Ferguson C, Salkey K

Abstract
Behçet’s syndrome is an inflammatory disease characterized by aphthous oral ulcers and several systemic manifestations, which include genital ulcers, ocular disease, skin lesions, arthritis, and vascular disease

PMID: 30586267 [PubMed - indexed for MEDLINE]

Rare and life-threatening complication after an attempted lower third molar extraction: Lemierre syndrome.

Vascularites - jeu, 07/02/2019 - 09:34
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Rare and life-threatening complication after an attempted lower third molar extraction: Lemierre syndrome.

Ann R Coll Surg Engl. 2019 Feb;101(2):e52-e54

Authors: Varvara G, Bernardi S, Piattelli M, Cutilli T

Abstract
Lemierre's syndrome is also known as the forgotten disease, and is a rare but life-threatening complication that can arise after surgical extractions of infected mandibular third molars. Owing to its rarity, oral and maxillofacial surgeons might not immediately recognise or can underestimate the pathological signs, and consequently do not apply the appropriate therapy to treat the syndrome. Here, we report on the occurrence and management of a case of Lemierre's syndrome, where the complications affected the right sigmoid sinus. Since the condition appear to be underreported and not properly highlighted, eventual systematic review and meta-analysis of the occurrence of the Lemierre's syndrome are highly recommended.

PMID: 30372118 [PubMed - indexed for MEDLINE]

Henoch-Schönlein Purpura After Living Donor Liver Transplantation: Report of the First Case.

Vascularites - lun, 04/02/2019 - 07:13
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Henoch-Schönlein Purpura After Living Donor Liver Transplantation: Report of the First Case.

Transplant Proc. 2018 Dec;50(10):4050-4052

Authors: Aliyev V, Yagi S, Hammad A, Badawy A, Taura K, Okajima H, Takaori K, Kaido T, Uemoto S

Abstract
Henoch-Schönlein purpura (HSP) is a systemic vasculitis affecting the small vessels that mainly presents in children and young adults. It is characterized by tissue deposition of immunoglobulin A (IgA) immune complexes with the classic manifestations of purpura, arthritis, arthralgia, and gastrointestinal and renal involvements. We report a case of HSP nephritis that occurred 2 years after living-donor liver transplantation (LDLT). After pulse steroid administration, the patient's symptoms disappeared and blood markers normalized. To the best of our knowledge, this is the first HSP case to be reported in a liver transplant recipient.

PMID: 30522857 [PubMed - indexed for MEDLINE]

Successful Hybrid Zone 0 Landing Thoracic Endovascular Aortic Repair for Ascending Aortic Pseudoaneurysm after Bentall Procedure and Coronary Artery Bypass Grafting in Takayasu Arteritis.

Vascularites - lun, 04/02/2019 - 07:13
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Successful Hybrid Zone 0 Landing Thoracic Endovascular Aortic Repair for Ascending Aortic Pseudoaneurysm after Bentall Procedure and Coronary Artery Bypass Grafting in Takayasu Arteritis.

Ann Vasc Surg. 2019 Jan;54:335.e7-335.e10

Authors: Goto T, Shimaura K, Kuratani T, Kin K, Sakamoto T, Watanabe K, Watanabe Y, Kudo T, Sawa Y

Abstract
Pseudoaneurysm at the suture line is one of the most common complications in aortic surgery for Takayasu arteritis (TA) and is associated with a high mortality rate. A 52-year-old man with TA, who previously underwent the Bentall procedure and 2 redo surgeries for coronary artery obstruction and a pseudoaneurysm of a coronary button, was diagnosed with an anastomotic pseudoaneurysm in the ascending aorta. Hybrid zone 0 debranching thoracic endovascular aortic repair was performed, and the patient was discharged uneventfully on postoperative day 8.

PMID: 30114506 [PubMed - indexed for MEDLINE]

Relapsing granulomatosis with polyangiitis provoked by trauma: A case report.

Vascularites - ven, 01/02/2019 - 05:51
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Relapsing granulomatosis with polyangiitis provoked by trauma: A case report.

Medicine (Baltimore). 2019 Jan;98(3):e14189

Authors: Deng S, Xing Y, Wang H, Kong X

Abstract
RATIONALE: The precise cause of granulomatosis with polyangiitis (GPA) remains unclear. Herein we present a case of refractory GPA occurring after trauma.
PATIENT CONCERNS: A 43-year-old man suffered fractures of the left orbital in an accident, and then received surgical repair. At a postoperative follow-up, he developed maxillary sinusitis. Seven months later, he was hospitalized with symptoms of cough, high-grade fever, and loss of weight.
DIAGNOSIS: He was diagnosed with GPA based on the symptoms of upper and lower respiratory tract involvement, granulomas in a lung biopsy specimen, and presence of PR3-ANCA.
INTERVENTIONS: Initially the patient responded well to the treatment of glucocorticoids and cyclophosphamide (CYC). Forty days later, he was hospitalized again with symptoms of fever, cephalgia, and recurrent ulcerated subcutaneous nodules, due to the vasculitic manifestations of GPA. This time he was treated with cyclophosphamide and glucocorticoids with no improvement, and then switched to rituximab.
OUTCOMES: After 4 doses of rituximab, the symptom of cephalgia disappeared and subcutaneous ulcerations and conjunctival congestion diminished.
LESSONS: Trauma may act as an inflammatory stimulus to affect the course of disease in GPA. The combination of glucocorticoids and cyclophosphamide is the standard therapy for GPA. Nevertheless, patients who have no response to CYC, especially with predominantly vasculitic manifestations, could resort to rituximab.

PMID: 30653170 [PubMed - indexed for MEDLINE]

The change of Th17/Treg cells and IL-10/IL-17 in Chinese children with Henoch-Schonlein purpura: A PRISMA-compliant meta-analysis.

Vascularites - ven, 01/02/2019 - 05:51
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The change of Th17/Treg cells and IL-10/IL-17 in Chinese children with Henoch-Schonlein purpura: A PRISMA-compliant meta-analysis.

Medicine (Baltimore). 2019 Jan;98(3):e13991

Authors: Li B, Ren Q, Ling J, Tao Z, Yang X, Li Y

Abstract
BACKGROUND: To date, the relationship of Th17 and Treg cells to Henoch-Schonlein purpura (HSP) in children remains controversial. Therefore, a systematic review and meta-analysis was conducted to reveal the potential role of the Th17 and Treg cells in children in acute stage of HSP.
METHODS: PubMed, Embase, Web of Science and China National Knowledge Internet (CNKI) were systematically searched for eligible studies up to November 03, 2017. Quality assessment was carried out according to the modification of the Newcastle-Ottawa Scale (NOS). The data were analyzed by Stata SE12.0 (StataCorp, College Station, TX). Standard mean difference (SMD) with 95% confidence intervals (CI) was calculated continuous data.
RESULTS: A total of 25 eligible studies were identified after a thorough literature search. The pooled results of the meta-analysis showed that values of Th17 frequency (SMD = 2.60; 95% CI: 1.98 to 3.23; P < .0001; I = 90.3%, P < .0001) and IL-17 level (SMD = 3.53; 95% CI: 2.71 to 4.35; P < .0001; I = 95.6%, P < .001) were significantly higher in children with HSP as compared to healthy children. In contrast, our analysis showed significant lower values of Treg frequency (SMD = -2.86; 95% CI: -3.53 to -2.19; P < .001; I = 92.4%, P < .001). However, no significance of IL-10 level was observed between children with HSP and healthy children (SMD = -1.22; 95% CI: -2.78 to 0.33; P < .01; I = 95.9%, P < .001).
CONCLUSION: In conclusion, our meta-analysis indicated that increased frequency of Th17 cells and level of IL-17, but lower frequency of Treg cells are associated with HSP in childhood. Considering the limitations of this meta-analysis, large-scaled studies need to be conducted to validate the current results.

PMID: 30653104 [PubMed - indexed for MEDLINE]

Bilateral Optic Perineuritis as Initial Presentation of Giant Cell Arteritis.

Vascularites - ven, 01/02/2019 - 05:51
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Bilateral Optic Perineuritis as Initial Presentation of Giant Cell Arteritis.

Neurologist. 2019 Jan;24(1):26-28

Authors: Pappolla A, Silveira F, Norscini J, Miquelini L, Patrucco L

Abstract
Giant cell arteritis is a large vessel vasculitis with neurological manifestations that range from visual disturbances to ischemic stroke. Among the former, visual acuity and visual field defects are prevalent, with anterior ischemic optic neuropathy being their most frequent cause. We present a clinical case in which the presenting ocular feature of the disease was a peripheral monocular visual field defect secondary to optic perineuritis.

PMID: 30586031 [PubMed - indexed for MEDLINE]

Transcranial Doppler Evaluation in Takayasu Arteritis With Oculo-Cerebrovascular Complications.

Vascularites - ven, 01/02/2019 - 05:51
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Transcranial Doppler Evaluation in Takayasu Arteritis With Oculo-Cerebrovascular Complications.

Neurologist. 2019 Jan;24(1):17-21

Authors: Christiansen ME, O'Carroll CB, Kumar G, Larsen BT, Dumitrascu OM

Abstract
INTRODUCTION: Takayasu arteritis is a large-vessel vasculitis that may cause oculo-cerebral ischemia. We report a patient with visual loss as initial manifestation, and discuss transcranial Doppler (TCD) findings before and after surgical revascularization.
CASE DESCRIPTION: A 19-year-old female developed episodes of transient positional right vision loss, progressing to permanent right vision loss and bright light-induced left amaurosis. Examination demonstrated subclavian bruits, palpable epigastrium aortic pulsation, faint radial pulses, right retinal pallor, arteriolar narrowing, and bilateral boxcarring. Head and neck computed tomography angiogram demonstrated left subclavian origin and right common carotid artery occlusion, and severe innominate and left common carotid artery stenosis. TCD demonstrated right ophthalmic artery flow reversal and nonpulsatile waveforms with dampened spectra in the right anterior circulation. Corticosteroids, methotrexate, infliximab, and dual-antiplatelet therapy were initiated. Eleven weeks later, the patient underwent ascending aortic aneurysm repair and bilateral carotid artery bypass with an aortic graft. Pathology was consistent with chronic active Takayasu arteritis. Two weeks postoperatively, left eye visual symptoms resolved; right visual loss persisted. Postoperative TCD showed marked improvement in cerebral perfusion.
CONCLUSIONS: Retinal ischemia in young women should prompt emergent inflammatory and systemic vascular evaluation. In our subject, prolonged right retinal ischemia had dismal prognosis despite carotid-aortic revascularization, whereas left retinal boxcarring reversed. Surgical revascularization is recommended for severely symptomatic oculo-cerebrovascular disease, once inflammation is better controlled with immunosuppressive therapy. TCD documented the presence and monitored the subsequent resolution of blood flow changes causing retinal and brain hypoxia.

PMID: 30586029 [PubMed - indexed for MEDLINE]

Pleuroparenchymal fibroelastosis as a histological background of autoimmune diseases.

Vascularites - ven, 01/02/2019 - 05:51
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Pleuroparenchymal fibroelastosis as a histological background of autoimmune diseases.

Virchows Arch. 2019 Jan;474(1):97-104

Authors: Kinoshita Y, Watanabe K, Ishii H, Kushima H, Hamasaki M, Fujita M, Nabeshima K

Abstract
Patients with autoimmune disease-related interstitial lung disease (AID-ILD) occasionally develop radiologic pleuroparenchymal fibroelastosis (PPFE)-like lesions. However, the significance of AID as an etiology of PPFE has not been fully elucidated. The aim of this study is to verify the increase of elastic fibers in AID-ILD patients and evaluate the prevalence of histological PPFE in patients with AID-ILD. We selected cases of clinically diagnosed AID-ILD and idiopathic pulmonary fibrosis (IPF), in which an autopsy had been performed or in which the patient had undergone pneumonectomy for lung transplantation. We quantified the collagen fibers and elastic fibers in each lobe as the percentage of the non-aerated lung area (collagen fiber score and elastic fiber score, respectively) in histological specimens from a total of 73 patients (AID-ILD, n = 24; IPF, n = 49). There were no significant differences in the collagen fiber scores of the AID-ILD and IPF groups. Meanwhile, the elastic fiber scores of the AID-ILD group were significantly greater than those of the IPF group in the whole lung (17.3 ± 7.70 vs 11.6 ± 4.55), and the upper (16.6 ± 8.11 vs 11.2 ± 5.18), and lower (18.0 ± 9.68 vs 12.0 ± 5.55) lobes (all p < 0.01). Histological PPFE pattern was found in 12 of 24 AID-ILD patients (50%), and histological PPFE pattern as a dominant pattern of fibrosis was found in 2 of the 24 patients (8%). Thus, PPFE can be a manifestation of AID-ILD.

PMID: 30327882 [PubMed - indexed for MEDLINE]

Cutaneous vasculitis in a patient with ankylosing spondylitis: A case report.

Vascularites - mar, 29/01/2019 - 05:45
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Cutaneous vasculitis in a patient with ankylosing spondylitis: A case report.

Medicine (Baltimore). 2019 Jan;98(3):e14121

Authors: Ye C, Li W

Abstract
RATIONALE: Cutaneous vasculitis is usually found in patients with rheumatoid arthritis (RA) as an extra-articular manifestation, but rare in patients with ankylosing spondylitis (AS). In this case, we describe an AS patient who developed large skin lesions, of which the histological findings were consistent with cutaneous vasculitis.
PATIENT CONCERNS: A 22-year-old male who was diagnosed as HLA-27 positive AS for 5 years. However, in the last year, he suffered a recurrent skin lesion in both lower extremities especially the dorsum of feet, accompanying with intense pruritus.
DIAGNOSIS: The originally diagnosis of the skin lessons was dermatitis, and then a skin biopsy pathology showed it was consistent with cutaneous vasculitis.
INTERVENTION: At first he was treated with gentamycin and ethacridine for nearly 2 weeks, the symptoms were relieved. But then he suffered severe cutaneous lesions with swell at the ulcerous margin in his dorsum of right foot. The patient was treated with dexamethasone 10 mg intravenous drip daily for 2 weeks, and followed by methylprednisolone 160 mg oral administration daily after discharge. The skin lesions were gradually healed in several months OUTCOMES:: This patient was followed up at 1-month, 5-month, and 10-month after discharged, the skin lessons was gradually healed and never occurred.
LESSONS: This case indicated that cutaneous vasculitis might be an extra-articular manifestation of AS in which IgA may plays a pathogenic part. All this may be associated with the damage of cytokines and autoantibodies to vascular endothelial wall caused by active inflammatory phase.

PMID: 30653140 [PubMed - indexed for MEDLINE]

Update on the epidemiology, risk factors, and outcomes of systemic vasculitides.

Vascularites - mar, 29/01/2019 - 05:45
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Update on the epidemiology, risk factors, and outcomes of systemic vasculitides.

Best Pract Res Clin Rheumatol. 2018 04;32(2):271-294

Authors: Berti A, Dejaco C

Abstract
Antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV) and giant cell arteritis (GCA) are the most common primary systemic vasculitides of the adult population, while polymyalgia rheumatica (PMR) is a clinical syndrome often associated with GCA. Incidence and prevalence rates of AAV have been increasing in the last decades, whereas those of GCA and PMR have remained stable. The mutual interplay between environmental and genetic risk factors leading to the development of these diseases has been further analyzed in the last years. The role of infectious agents has repeatedly been studied with regard to Staphylococcus aureus, associated with relapse in granulomatosis with polyangiitis, and Herpes zoster, potentially contributing to GCA development. Remission of disease and prevention of disease-related complications are the most important outcomes for all systemic vasculitides. Although these goals are achieved in the majority of patients receiving modern therapies, the prevention of treatment-related complications, especially glucocorticoid side effects, is still an unmet need that is common to AAV, GCA, and PMR.

PMID: 30527432 [PubMed - indexed for MEDLINE]

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