Sarcoidosis, Vasculitis, and Diffuse Lung Diseases最新文献

{"title":"A case of nail sarcoidosis with rich clinical findings.","authors":"Can Baykal, Zeynep Yılmaz, Tuğba Atcı","doi":"10.36141/svdld.v39i3.11525","DOIUrl":"https://doi.org/10.36141/svdld.v39i3.11525","url":null,"abstract":"To the editor, As skin lesions are observed in 10-35% of sarcoidosis patients, nail involvement is very rare which generally develops in the setting of chronic disease (14). Nail dystrophy is the most frequent nail change among several described findings (1,2,4-7). Herein, we report a case of sarcoidosis showing rich nail findings some of them rarely reported or not well-described. A 36-year-old man with pulmonary sarcoidosis (stage one) (Figure 1a,b) presented with multiple subcutaneous nodules on the face and extremities (Figure 2a). Histopathologic examination confirmed cutaneous sarcoidosis. Median splitting was noticed on one fingernail plate accompanied by a subungual reddish nodule and increased convexity on the proximal portion of the nail plate (Figure 2b). Magnetic resonance imaging of hand demonstrated soft tissue expansion at the location with increased convexity. In addition, bone cysts were detected in adjacent thumb and middle fingers radiologically (Figure 3a,b). Systemic methylprednisolone therapy resulted in significant improvement of the nail changes after four months of treatment (Figure 2c), along with the skin lesions that resolved completely. However, two months after cessation of the therapy, skin lesions recurred and distal notching associated with onycholysis and increased convexity of the nail plate appeared (Figure 2d). All lesions regressed after readministration of systemic corticosteroid (Figure 2e). Three years later, he was admitted with ulcerated plaques and subcutaneous nodules on arms (Figure 4a) and severe nail involvement (Figure 4b). Increased convexity, longitudinal ridging and subungual hyperkeratosis were observed on the previously affected third digit of the right hand (Figure 2f ), while a subungual reddish hyperkeratotic nodule on the thumbnail was detected which was associated with partial (median) nail plate loss (Figure 4c). Pitting, mild subungual hyperkeratosis, erythronychia and onycholysis were present on the third digit of the left hand (Figure 4d). Furthermore, total dystrophy (opaque, fragile, irregularly thickened nail plate) and subungual hyperkeratosis were observed on both big toenails accompanied by red-violaceous periungual discoloration (paronychial involvement) and tiny papules evaluated as lupus pernio of digit (Figure 4b,e). All lesions including periungual erythema, papules and nail findings regressed dramatically (Figure 4f ) after moderate-dose systemic corticosteroid. Letter to editor","PeriodicalId":21394,"journal":{"name":"Sarcoidosis, Vasculitis, and Diffuse Lung Diseases","volume":"39 3","pages":"e2022032"},"PeriodicalIF":1.6,"publicationDate":"2022-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/1e/0d/SVDLD-39-32.PMC9766855.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10739102","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Pulmonary sarcoidosis with lung injury induced by shin'iseihaito.","authors":"Suzuki Kentaro, Yoshiro Kai, Masayuki Matsuda, Kazuhide Horimoto, Kazonori Iwai, Eriko Hamada, Yoshifumi Yamamoto, Masato Takano, Shigeo Muro","doi":"10.36141/svdld.v39i3.12895","DOIUrl":"https://doi.org/10.36141/svdld.v39i3.12895","url":null,"abstract":"Suzuki Kentaro, Yoshiro Kai, Masayuki Matsuda, Kazuhide Horimoto, Kazonori Iwai, Eriko Hamada, Yoshifumi Yamamoto, Masato Takano and Shigeo Muro Department of Respiratory Medicine, Minami-Nara General Medical Center, 8-1 Fukugami, Oyodo-cho, Yoshino-gun, Nara 638-8551, Japan; Department of Internal Medicine, Yoshino Hospital, 130-1, Tanji, Yoshino-cho, Yoshino-gun, Nara 639-3114, Japan; Department of Respiratory Medicine, Nara Medical University, 840 Shijo-cho, Kashihara City, Nara 634-8522, Japan; Department of Diagnostic Pathology, Minami-Nara General Medical Center, 8-1 Fukugami, Oyodo-cho, Yoshino-gun, Nara 638-8551, Japan","PeriodicalId":21394,"journal":{"name":"Sarcoidosis, Vasculitis, and Diffuse Lung Diseases","volume":"39 3","pages":"e2022026"},"PeriodicalIF":1.6,"publicationDate":"2022-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/ec/90/SVDLD-39-26.PMC9766847.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10739104","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Unusual bone localization of sarcoidosis mimicking metastatic lesions: case report and review of literature.","authors":"Besma Hamdi,&nbsp;Emna Ben Jemia,&nbsp;Monia Attia,&nbsp;Ikbel Khalfallah,&nbsp;Hend Riahi,&nbsp;Anissa Berraies,&nbsp;Mohamed Faouzi Ladeb,&nbsp;Soumaya Rammeh,&nbsp;Agnes Hamzaoui","doi":"10.36141/svdld.v39i3.8744","DOIUrl":"https://doi.org/10.36141/svdld.v39i3.8744","url":null,"abstract":"<p><p>Sarcoidosis is a multisystem disease of unknown origin. Diagnosis remains challenging, based on organ site involvement, histological confirmation of non-caseating granuloma and an appropriate clinical syndrome. Granulomatous bone involvement is rare and may be ignored because it is usually asymptomatic. Vertebrae, ribs and skull localizations are rarely reported. We described an interesting case of a woman with chronic and multiorgan sarcoidosis with unusual bone localizations.</p>","PeriodicalId":21394,"journal":{"name":"Sarcoidosis, Vasculitis, and Diffuse Lung Diseases","volume":"39 3","pages":"e2022034"},"PeriodicalIF":1.6,"publicationDate":"2022-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/f4/8d/SVDLD-39-34.PMC9766854.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10739105","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Value of pulmonary function testing identifying progressive pulmonary disease in fibrotic sarcoidosis: results of a prospective feasibility study.","authors":"Robert P Baughman,&nbsp;Rohit Gupta,&nbsp;Marc A Judson,&nbsp;Elyse E Lower,&nbsp;Surinder S Birring,&nbsp;Jeffrey Stewart,&nbsp;Rebecca Reeves,&nbsp;Athol U Wells","doi":"10.36141/svdld.v39i2.12940","DOIUrl":"https://doi.org/10.36141/svdld.v39i2.12940","url":null,"abstract":"","PeriodicalId":21394,"journal":{"name":"Sarcoidosis, Vasculitis, and Diffuse Lung Diseases","volume":"39 2","pages":"e2022011"},"PeriodicalIF":1.6,"publicationDate":"2022-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9437762/pdf/SVDLD-39-011.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"40364484","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"A rare case of pulmonary lymphomatoid granulomatosis complicated with venous thrombosis.","authors":"Senem Maral,&nbsp;Murat Albayrak,&nbsp;Hacer Berna Afacan Ozturk,&nbsp;Funda Incekara,&nbsp;Abdulkerim Yıldız,&nbsp;Pınar Comert,&nbsp;Aynur Albayrak,&nbsp;Merih Reis Aras,&nbsp;Sadi Kaya","doi":"10.36141/svdld.v38i4.10140","DOIUrl":"https://doi.org/10.36141/svdld.v38i4.10140","url":null,"abstract":"<p><p>Lymphomatoid granulomatosis (LG) is Epstein-Barr virus associated and aggressive B cell lymphoproliferative disease. The most common sites of involvement are lungs, skin, kidneys, liver and central nervous system. The clinical presentation of pulmonary LG may mimic infectious diseases, malignancies or vasculitis. While treatment approach of low grade disease is watch and wait, patients with advanced stage require aggressive treatment with chemotherapy. Patients with hematological malignancy as well as solid tumors are at increased risk of venous thromboembolic events (VTE). We reported here in a case of pulmonary LG who was complicated with VTE during treatment with chemo-immunotherapy After 4 cycles of R-CHOP, she achieved complete remission for LG and was followed up without relapse for 2 years. She was anticoagulated with Low-Molecular-Weight Heparin (LMWH) during chemotherapy period, and the thrombus improved over the next several weeks. While on this paper written, patient completed her pregnancy successfully under anticoagulation prophylaxis.</p>","PeriodicalId":21394,"journal":{"name":"Sarcoidosis, Vasculitis, and Diffuse Lung Diseases","volume":"38 4","pages":"e2021038"},"PeriodicalIF":1.6,"publicationDate":"2022-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/e3/c3/SVDLD-38-38.PMC8787372.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39747944","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Design and rationale of ProSar, the first Danish sarcoidosis registry.","authors":"Janne Møller, Ole Hilberg, Elisabeth Bendstrup","doi":"10.36141/svdld.v38i4.11605","DOIUrl":"10.36141/svdld.v38i4.11605","url":null,"abstract":"","PeriodicalId":21394,"journal":{"name":"Sarcoidosis, Vasculitis, and Diffuse Lung Diseases","volume":"38 4","pages":"e2021044"},"PeriodicalIF":1.4,"publicationDate":"2022-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/b6/c1/SVDLD-38-44.PMC8787369.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39762874","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Routine Laboratory Biomarkers As Prognostic Indicators of Cardiac Sarcoidosis Outcomes.","authors":"Nikhil Kolluri,&nbsp;Tyler J Schmidt,&nbsp;Mohamed Y Elwazir,&nbsp;Suraj Kapa,&nbsp;Omar F Abou Ezzeddine,&nbsp;John P Bois,&nbsp;John A Schirger,&nbsp;Andrew N Rosenbaum,&nbsp;Leslie T Cooper","doi":"10.36141/svdld.v39i2.11136","DOIUrl":"https://doi.org/10.36141/svdld.v39i2.11136","url":null,"abstract":"<p><strong>Background: </strong>Biomarkers to monitor disease activity and predict major adverse cardiac events (MACE) in CS have not been described previously. We aimed to identify biomarkers to predict MACE in cardiac sarcoidosis (CS).</p><p><strong>Methods: </strong>Patients (N=232) diagnosed with CS were retrospectively enrolled. Biomarkers including angiotensin-converting enzyme (ACE), N-terminal brain natriuretic peptide (NT-proBNP), troponin T, and creatinine levels were evaluated against a primary end point of left ventricular assist device implantation, heart transplantation, or death, and a secondary end point of cardiac hospitalization-free survival.</p><p><strong>Results: </strong>Troponin T (hazard ratio [HR], 1.06 per 0.01 ng/mL; <i>P</i>=.006), NT-proBNP (HR, 1.31 per 1,000 pg/mL; <i>P</i><.001), and creatinine (HR, 4.02 per mg/dL; <i>P</i>=.01) were associated with the primary end point, even after adjusting for ejection fraction. NT-proBNP, B-type natriuretic peptide (BNP), creatinine, albumin, and calcium were associated with the secondary end point (<i>P</i><.05). ACE levels were associated with presence of late gadolinium enhancement (LGE) on cardiac magnetic resonance (CMR) imaging (mean difference, 14.7; <i>P</i>=.03); 1,25 dihydroxyvitamin D (1,25-OHVit-D) was associated with uptake on cardiac ¹⁸F-flurodeoxyglucose position emission tomography (FDG-PET, <i>P</i>=.03).</p><p><strong>Conclusions: </strong>Troponin T, NT-proBNP, and creatinine predict clinically significant outcomes in CS. ACE levels correlated with LGE on CMR, and 1,25-OHVit-D levels correlated with FDG-PET activity.</p>","PeriodicalId":21394,"journal":{"name":"Sarcoidosis, Vasculitis, and Diffuse Lung Diseases","volume":"39 3","pages":"e2022023"},"PeriodicalIF":1.6,"publicationDate":"2022-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/73/cc/SVDLD-39-23.PMC9766851.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10727473","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Patient profile and comparison of three diagnostic criteria for cardiac sarcoidosis in a tuberculosis endemic population.","authors":"Bijay Pattnaik, Sryma Pb, Mansi Verma, Sanjeev Kumar, Saurabh Mittal, Sudheer Arava, Pavan Tiwari, Vijay Hadda, Anant Mohan, Randeep Guleria, Karan Madan","doi":"10.36141/svdld.v38i4.10977","DOIUrl":"10.36141/svdld.v38i4.10977","url":null,"abstract":"<p><strong>Background: </strong>Cardiac sarcoidosis (CS) is an underdiagnosed and life-threatening condition. Histopathological diagnosis is difficult due to the risks and variable diagnostic yield of endomyocardial biopsy.</p><p><strong>Objectives: </strong>To study the clinical profile and compare the diagnostic criteria of CS in a cohort of sarcoidosis.</p><p><strong>Methods: </strong>A retrospective review of the Sarcoidosis database (375 patients) was performed to identify patients with CS. Demographic and clinical details were retrieved. We applied the available diagnostic criteria for the diagnosis of CS: The World Association of Sarcoidosis and Other Granulomatous Diseases (WASOG), Heart Rhythm Society (HRS), and Japanese Ministry of Health and Welfare (JMHW) criteria.</p><p><strong>Results: </strong>Out of the 375 patients, 15 (4%) were identified with CS. The median age was 41 years, and 53% were female. The most common symptoms were breathlessness, palpitation, and fatigue in 80%, 53.3%, and 46.6% of patients, respectively. Tuberculin positivity (≥ 10mm induration) was seen in 26.6%. 80% and 53.3% of the patients had abnormal ECG and 2D echocardiography findings, respectively. Six patients had a history of Ventricular tachycardia (40%). LV Ejection fraction was reduced in 12 subjects (80%). Cardiac-MRI showed late gadolinium enhancement in 53.3%. A definitive histopathological diagnosis for sarcoidosis was established in 86.6% (13/15) patients. Of the 15, all satisfied JMHW criteria and WASOG criteria (12 (80%) at least probable category, 3 (20%) possible CS), and 13 (86.6%) met HRS criteria for a diagnosis of CS.</p><p><strong>Conclusion: </strong>In a cohort of 375 patients with sarcoidosis in a tuberculosis endemic setting, 4% were diagnosed with cardiac sarcoidosis. Histopathological diagnosis may be obtained by sampling from extracardiac sites. JMHW and WASOG criteria perform equally well in TB endemic settings.</p>","PeriodicalId":21394,"journal":{"name":"Sarcoidosis, Vasculitis, and Diffuse Lung Diseases","volume":"38 4","pages":"e2021040"},"PeriodicalIF":1.4,"publicationDate":"2022-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/cf/78/SVDLD-38-40.PMC8787371.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39747946","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Co-existence of metastatic uterine sarcoma and orbital sarcoidosis: A case report and literature review.","authors":"Surabhi Shalini,&nbsp;Faye Mellington","doi":"10.36141/svdld.v38i4.11570","DOIUrl":"https://doi.org/10.36141/svdld.v38i4.11570","url":null,"abstract":"<p><p>A 77-year old lady with previously treated endometrial sarcoma presented with progressive left upper lid mechanical ptosis secondary to superior orbital mass. An orbital biopsy confirmed the diagnosis of orbital sarcoidosis. Further systemic work up revealed suspicious pulmonary nodules which were found to be endometrial sarcoma metastases rather than systemic sarcoidosis on image guided biopsy. She was treated with 20 months of chemotherapy for metastatic sarcoma. The ptosis completely resolved, however, pulmonary metastases progressed despite chemotherapy. The co- existence of malignancy with sarcoidosis should be considered in all cases of new onset sarcoidosis. Biopsy of suspicious lesions, close observation and multidisciplinary team management is advocated for these patients.</p>","PeriodicalId":21394,"journal":{"name":"Sarcoidosis, Vasculitis, and Diffuse Lung Diseases","volume":"38 4","pages":"e2021043"},"PeriodicalIF":1.6,"publicationDate":"2022-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/db/a8/SVDLD-38-43.PMC8787375.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39762873","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"DNA sequencing validation by PCR-RFLP for evaluating butyrophilin-like 2 rs2076530 polymorphism in Iranian patients with sarcoidosis.","authors":"Alireza Javadi,&nbsp;Masoud Zarei,&nbsp;Mihan Pourabdollah,&nbsp;Makan Sadr,&nbsp;Arda Kiani,&nbsp;Atefeh Abedini,&nbsp;Masoud Shamaei","doi":"10.36141/svdld.v38i4.11152","DOIUrl":"https://doi.org/10.36141/svdld.v38i4.11152","url":null,"abstract":"<p><strong>Background: </strong>Sarcoidosis is a multifactorial immune disorder with an uncertain origin. A single nucleotide polymorphism (G→A, rs2076530) in the butyrophilin-like 2 (BTNL2) gene results in the formation of truncating protein. This study aimed to genotype the predisposition of the BTNL2 rs2076530 polymorphism in Iranian patients with sarcoidosis using the RFLP technique.</p><p><strong>Materials and methods: </strong>In this study, 80 patients with sarcoidosis and 80 healthy individuals were included. The rs2076530 polymorphism of the BTNL2 gene was genotyped using the PCR-RFLP method by AvrII restriction enzyme and confirmed by DNA sequencing (Capillary electrophoresis 3130, ABI).</p><p><strong>Results: </strong>There was a statistically significant difference between proportions of patients with AA (47,5%) and controls (27.5%) (OR=2.38, 95%CI:1.23-4.61, P=0.009). In addition, a significant difference was observed in the frequency of the A allele (62.5%) in sarcoidosis (OR=2.14, 95%CI:1.37-3.35, <i>P</i>=0.001). A Bonferroni correction with <i>P</i><0.0038 indicates a statistical difference for genotype AA (<i>P</i>=0.009). In an effective model, binary logistic regression analysis indicates a statistical association between AA genotype and sarcoidosis (<i>P</i>=0.018 with 60% prediction). Based on the gene analysis study using DNA sequencing, all of the mentioned mutations were seen via RFLP.</p><p><strong>Conclusion: </strong>According to our findings, the BTNL2 rs2076530 A allele in the Iranian population is associated with susceptibility to sarcoidosis. This designed PCR-RFLP method for detecting SNPs is effective as DNA sequencing.</p>","PeriodicalId":21394,"journal":{"name":"Sarcoidosis, Vasculitis, and Diffuse Lung Diseases","volume":"38 4","pages":"e2021050"},"PeriodicalIF":1.6,"publicationDate":"2022-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/a3/56/SVDLD-38-50.PMC8787370.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39762879","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}