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[PMID]: 29256478
[Au] Autor:Sokolova OV
[Ad] Address:Department of Pathological Anatomy with the course of forensic medicine, Saint-Petersburg State Pediatric Medical University, Ministry of Health of the Russia, Saint-Petersburg, Russia, 194100; Department of Forensic Medicine and Jurisprudence Science, I.P. Pavlov First Saint-Petersburg State Medical University, Saint-Petersburg, Russia, 197022.
[Ti] Title:Sudebno-meditsinskaia otsenka strukturnykh izmenenii tkani golovnogo mozga v sluchaiakh vnezapnoi smerti ot alkogol'noi kardiomiopatii. [The forensic medical evaluation of the changes in the cerebral tissue in the case of sudden death from alcoholic cardiomyopathy].
[So] Source:Sud Med Ekspert;60(6):9-12, 2017.
[Is] ISSN:0039-4521
[Cp] Country of publication:Russia (Federation)
[La] Language:rus
[Ab] Abstract:This article presents the results of the retrospective analysis of the protocols of forensic medical autopsies and histological studies of the cerebral tissues together with the data obtained by their statistical treatment. The objective of present work was to study and evaluate the structural changes in the cerebral tissues associated with sudden cardiac death from alcoholic cardiomyopathy. It was shown that the morphological changes in the endothelial lining of the microcirculatory bed of the cerebral tissue can be a consequence of both the direct cytotoxic action of ethanol or its metabolites and the influence of cellular modulators liberation of which results in enhanced vascular permeability associated with trophic disturbances in the tissue. These changes provide a substrate for the development of the dystrophic and necrobiotic processes in the main structural components of the organ in question. The clustering of glial cells around atrophic neutrons or instead of the dead ones in the brain as well as around the microcysts of the cerebral tissue as a compensatory response to the lesion can be in its turn considered as a pathognomonic sign of chronic alcoholic intoxication. The severe dystrophic and destructive processes proceeding in the main components of the histohematological barrier revealed in the cerebral tissue as a result of the present analysis are believed to reflect the toxic action of ethanol and/or its metabolites that play an important role in the development of dyscirculatory disorders responsibly for cerebral tissue hypoxia.
[Pt] Publication type:ENGLISH ABSTRACT; JOURNAL ARTICLE
[Em] Entry month:1712
[Cu] Class update date: 180227
[Lr] Last revision date:180227
[St] Status:In-Process
[do] DOI:10.17116/sudmed20176069-12

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[PMID]: 28871936
[Au] Autor:Nockowski P; Wozniak Z; Reich A; Maj J
[Ti] Title:Xanthoma-like Skin Changes in an Elderly Woman with a Normal Lipid Profile.
[So] Source:Acta Dermatovenerol Croat;25(2):167-169, 2017 Jul.
[Is] ISSN:1847-6538
[Cp] Country of publication:Croatia
[La] Language:eng
[Ab] Abstract:Dear Editor, An 83-year-old woman developed yellow-brownish infiltrates, nodules, and tumors mimicking xanthomas, mostly involving the periorbital and chest area within three months (Figure 1). She had no abnormalities in serum cholesterol or triglycerides levels. A detailed laboratory analysis revealed the presence of mild monoclonal gammopathy with a presence of immunoglobulin G (IgG) kappa light chains; however, according to hematologist consultation, it did not require medical intervention. Imaging assessment and ultrasound examination did not show any specific involvement of internal organs. The skin biopsy demonstrated necrobiotic areas alternated with foci of xanthogranulomatous infiltration throughout the reticular dermis with extension into subcutaneous tissue. The granulomatous infiltrate was composed of epithelioid, foamy histiocytes in addition to conspicuous giant cells of the Touton type and foreign body type, as well as variable numbers of lymphocytes, plasma cells, and neutrophiles. Lipid vacuoles were seen within the foci of necrobiosis and xanthogranulomatous infiltration (Figure 2). Two months after first admission to our department, the first signs of necrosis within the lesions were noted, and massive necrosis of skin lesions occurred after the following 5 months (Figure 1). Based on the clinical manifestation and histological and laboratory findings, the diagnosis of necrobiotic xanthogranuloma (NXG) was established. In our patient, the extremely late onset of the disease, its very aggressive course, and the absence of malignant hematological disorder were remarkable. The general condition improved after local treatment and a low dose of prednisone. However, patient anamnesis revealed myocardial infarction in the past, congestive heart failure, and atrial fibrillation. Eventually, the patient died due to acute heart failure before alkylating agents could be administered; we consider the patient's death to have been unrelated to NXG. NXG is a rare, chronic granulomatous disorder which was first described in 1980 by Kossard and Winkelmann (1). Currently, less than one hundred fifty cases of this syndrome have been reported in the literature worldwide (2,3). The disease occurs during adulthood, slightly more frequently in women, and usually after the age of 60 years, although the youngest reported patient was 17 years old (3). The disease initially manifests as xanthoma-like eruptions of yellowish or red-orange papules and nodules that coalesce into indurated plaques (4). The size of the lesions typically increases over time or with the next recurrences. In comparison to hyperlipemic and normolipemic xanthomas, the lesions are firmer, more prominent, and more polymorphic (3) with superficial telangiectasias, sometimes erythematous and/or violaceous borders, and atrophy (5). Ulcerations of the lesions were observed in about 50% of patients and tended to be extensive and progressive (4). Skin lesions of NXG can occur anywhere on the body. However, about two-thirds of patients had periorbital involvement, particularly on the upper and/or lower eyelids or elsewhere on the face. The second most commonly affected site was the trunk, predominantly the chest (3-6). However, many skin lesions first appear on the trunk or extremities and subsequently involve the periorbital area (4). More than one body area was affected in about 90% of the published cases (3,4). In individual cases, the occurrence of NXG was noted within scars, after trauma, or in a previously X-ray irradiated area (5). Lesions may be asymptomatic; however, over half of patients asked reported various symptoms, predominantly itching but also burning, tenderness, and even pain (4,5). Periorbital skin lesions are often accompanied by ophthalmic manifestations, mainly scleritis, choroiditis, or conjunctivitis (3), and with complications such as blepharoptosis, restricted ocular motility, and proptosis (4,5). Extracutaneous lesions are most commonly seen in the respiratory tract, including the lungs and larynx, followed by the myocardium, oral cavity, skeletal muscles, kidneys, ovaries, intestine, and other sites (5,6). Extracutaneous involvement was reported in less than 20% of cases (3), but its frequency seems to have increased in recent years (5). Regarding laboratory abnormalities, the majority of patients with NXG (70% and up to 90% depending on the studied population) have a monoclonal gammopathy (more often IgG-kappa than IgG-lambda). Elevated erythrocyte sedimentation rate, anemia, leukopenia, low C1 and C4 levels, and cryoglobulinemia are also frequently present (3-6). Incisional biopsy is recommended to confirm the diagnosis of NXG, but correlations between the clinical presentation and specific histopathologic findings have been poorly characterized so far. The histopathology shows an inflammatory infiltrate composed of macrophages, foam cells, plasma cells, and other inflammatory cells as well as Touton and foreign body-type giant cells in the dermis and subcutaneous tissue. Necrobiosis is usually present, and nodular lymphoid aggregates are common. Cholesterol clefts or asteroid bodies are rare or absent. The epidermis may be atrophic or normal. Special stains are not helpful in establishing the diagnosis of NXG, but immunohistochemistry for CD68 is positive while it is always for CD1a and PS100 negative, like in non-X histiocytosis (4,5). In patients without a known myeloproliferative disorder, bone marrow biopsy may reveal atypical or increased plasma cells and, very rarely, true multiple myeloma (5). As mentioned above, NXG can be a manifestation of multiple myeloma. However, chronic lymphocyte leukemia, B-cell lymphoma, and other lymphoproliferative diseases have also been reported in patients with NXG (3). Remarkably, hematological disorders may emerge many years before or after the onset of skin lesions (even up to 11 years) (4). According to available literature data, the course of the disease is usually chronic and slowly progressive, and the prognosis is relatively good in the absence of co-occurrence of malignant hematological disorders ([5-7). Aside from hyperlipemic and normolipemic xanthomas, the differential diagnosis of NXG includes multifocal necrobiosis lipoidica, granuloma annulare, foreign-body granuloma, juvenile xanthogranuloma, rheumatoid nodules, and amyloidosis (4). In 5 cases from the literature, xanthoma and NXG were present at the same time (3). Despite several hypotheses, the etiopathogenesis of NXG remains unknown (3,4,8). For that reason and due to the rarity of the disease, the optimal therapy has not been not defined. Frequently, chlorambucil or melphalan have been used alone or in combination with prednisone (4). Treatment may result in remission of symptoms on the skin, but it does not provide a permanent cure (8). There are also single reports of the successful use of thalidomide, lenalidomide, cyclophosphamide, dexamethasone, interferon 2a and 2b, plasmapheresis and hydroxychloroquine, azathioprine, infliximab, and autologous bone marrow transplantation (3). Methotrexate seems to be ineffective (9). Local therapy, including local steroids, laser CO2, or radiotherapy, results in partial improvement (3,4). Skin lesions which relapsed or were unresponsive to treatment could be excised surgically and the defects resurfaced with skin grafts. [2].
[Pt] Publication type:JOURNAL ARTICLE
[Em] Entry month:1709
[Cu] Class update date: 170905
[Lr] Last revision date:170905
[St] Status:In-Process

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[PMID]: 28500693
[Au] Autor:Baykal C; Polat Ekinci A; Yazganoglu KD; Buyukbabani N
[Ad] Address:Dermatology and Venereology, Istanbul Medical Faculty, Istanbul University, Istanbul, Turkey.
[Ti] Title:The clinical spectrum of xanthomatous lesions of the eyelids.
[So] Source:Int J Dermatol;56(10):981-992, 2017 Oct.
[Is] ISSN:1365-4632
[Cp] Country of publication:England
[La] Language:eng
[Ab] Abstract:Yellowish papules, nodules, or plaques, namely "xanthomatous" lesions, may be seen on the eyelids in the course of various disorders. The prototype is "xanthelasma palpebrarum" (XP) that is localized only to the eyelids and may be associated with hyperlipidemia. On the other hand, different types of normolipemic disorders may also cause xanthomatous eyelid lesions. Among these, Langerhans cell histiocytosis, diffuse normolipemic xanthoma, and non-Langerhans cell histiocytoses (papular xanthoma, juvenile xanthogranuloma, xanthoma disseminatum, adult-onset xanthogranuloma, adult-onset asthma and periocular xanthogranuloma, necrobiotic xanthogranuloma, Erdheim-Chester disease, Rosai-Dorfman disease, and reticulohistiocytosis) can be listed. The eyelid findings of this heterogeneous group of disorders are challenging to differentiate from each other due to common clinical aspects that may even sometimes mimic XP. Nodularity, induration, ulceration, diffuse eyelid involvement, and extension from eyelids to the neighboring skin may represent the clinical features of xanthomatous lesions other than XP. It is necessary to obtain a thorough history and exclude XP and then perform detailed dermatological and systemic examination, biopsy for histopathologic confirmation, and appropriate specific imaging screens. As some of the conditions may be associated with other systemic disorders, especially malignancies, the differentiation of xanthomatous eyelid lesions has a critical importance, and clinical signs can be guiding.
[Pt] Publication type:JOURNAL ARTICLE; REVIEW
[Em] Entry month:1705
[Cu] Class update date: 170912
[Lr] Last revision date:170912
[St] Status:In-Process
[do] DOI:10.1111/ijd.13637

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[PMID]: 27915419
[Au] Autor:Konradt G; Bianchi MV; Leite-Filho RV; da Silva BZ; Soares RM; Pavarini SP; Driemeier D
[Ad] Address:Departamento de Patologia Clínica Veterinária, Setor de Patologia Veterinária, Faculdade de Veterinária, Universidade Federal do Rio Grande do Sul (UFRGS), Av. Bento Gonçalves 9090, Prédio 42505, Porto Alegre, RS, 91540-000, Brazil. guikonradt@yahoo.com.br.
[Ti] Title:Necrotizing meningoencephalitis caused by Sarcocystis falcatula in bare-faced ibis (Phimosus infuscatus).
[So] Source:Parasitol Res;116(2):809-812, 2017 Feb.
[Is] ISSN:1432-1955
[Cp] Country of publication:Germany
[La] Language:eng
[Ab] Abstract:The infection by S. falcatula is commonly associated with respiratory disease in captive psittacine birds, with a few case reports of this protozoan causing encephalitis in wild birds. We describe the clinical, pathological, and molecular aspects of an infection by S. falcatula in a bare-faced ibis (Phimosus infuscatus). Clinically, wing paralysis and mild motor incoordination were observed. At necropsy, the telencephalic cortex showed multifocal to coalescing yellowish soft areas. Histologically, multifocal to coalescent nonsuppurative necrotizing meningoencephalitis of telencephalic cortex, cerebellum, and brainstem was observed. Necrotic areas showed multiple protozoan organism characteristics of Sarcocystis sp. schizonts in the cytoplasm of endothelial cells or lying free in the neuropil. Partial genetic sequences of the gene encoding cytochrome b (CYTB), the gene encoding the beta subunit of RNA polymerase (RPOB) and the first internal transcribed spacer (ITS-1) from Sarcocystis sp. schizonts revealed that the parasite had ITS-1 sequences that were 100% identical to the homologous alleles from Sarcocystis sp. shed by Didelphis albiventris in Brazil. RPOB and CYTB sequences were 100% identical to homologous of S. falcatula available in Genbank. Thus, this is the first report of necrotizing meningoencephalitis caused by S. falcatula in bare-faced ibis (P. infuscatus).
[Mh] MeSH terms primary: Bird Diseases/parasitology
Meningoencephalitis/veterinary
Sarcocystis/isolation & purification
Sarcocystosis/veterinary
[Mh] MeSH terms secundary: Alleles
Animals
Bird Diseases/diagnosis
Bird Diseases/pathology
Birds
Brain/parasitology
Brain/pathology
Brazil
Cytochromes b/genetics
Male
Meningoencephalitis/diagnosis
Meningoencephalitis/parasitology
Meningoencephalitis/pathology
Necrobiotic Disorders
Sarcocystis/genetics
Sarcocystosis/diagnosis
Sarcocystosis/parasitology
Sarcocystosis/pathology
Sequence Analysis, DNA/veterinary
[Pt] Publication type:CASE REPORTS; JOURNAL ARTICLE
[Nm] Name of substance:9035-37-4 (Cytochromes b)
[Em] Entry month:1706
[Cu] Class update date: 171007
[Lr] Last revision date:171007
[Js] Journal subset:IM
[Da] Date of entry for processing:161205
[St] Status:MEDLINE
[do] DOI:10.1007/s00436-016-5341-6

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[PMID]: 27886405
[Au] Autor:Sfeir JG; Zogala RJ; Popii VB
[Ad] Address:Department of Medicine and Division of Endocrinology, Diabetes, Metabolism, and Nutrition, Mayo Clinic, Rochester, MN, USA.
[Ti] Title:Hypercalcemia in Necrobiotic Xanthogranuloma: First Reported Case and Insight Into Treatment.
[So] Source:J Bone Miner Res;32(4):784-787, 2017 04.
[Is] ISSN:1523-4681
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:Necrobiotic xanthogranuloma (NXG) is a rare systemic and progressive granulomatous disease first described in 1980. Given no established first-line therapy, treatment focuses on the control of skin lesions and associated complications. Despite it being a granulomatous disease, NXG has not been associated with hypercalcemia. About 140 cases of NXG have been reported to date but, to our knowledge, this is the first case to be complicated by hypercalcemia. Our case confirms a granulomatous disease-mediated production of 1α-hydroxylase leading to increased synthesis of 1,25-dihydroxyvitamin D and subsequent hypercalcemia. Based on this pathophysiology, we elected to start systemic glucocorticoids, titrated to clinical and metabolic response. Steroid-sparing agents need to be considered to avoid long-term complications but continue controlling this granulomatous disease. © 2017 American Society for Bone and Mineral Research.
[Mh] MeSH terms primary: Hypercalcemia
Necrobiotic Xanthogranuloma
[Mh] MeSH terms secundary: Aged, 80 and over
Female
Humans
Hypercalcemia/blood
Hypercalcemia/complications
Hypercalcemia/diagnosis
Hypercalcemia/therapy
Necrobiotic Xanthogranuloma/blood
Necrobiotic Xanthogranuloma/complications
Necrobiotic Xanthogranuloma/diagnosis
Necrobiotic Xanthogranuloma/therapy
[Pt] Publication type:CASE REPORTS; JOURNAL ARTICLE
[Em] Entry month:1707
[Cu] Class update date: 171124
[Lr] Last revision date:171124
[Js] Journal subset:IM
[Da] Date of entry for processing:161126
[St] Status:MEDLINE
[do] DOI:10.1002/jbmr.3047

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[PMID]: 27172694
[Au] Autor:Peeters MA; Laffitte E
[Ti] Title:Granulomatoses cutanées. [Cutaneous granulomatosis].
[So] Source:Rev Med Suisse;12(512):640-5, 2016 Mar 30.
[Is] ISSN:1660-9379
[Cp] Country of publication:Switzerland
[La] Language:fre
[Ab] Abstract:Granulomatous skin diseases represent a large group of reactive dermatoses occurring in response to different stimuli and whose skin biopsy is characterized by a granulomatous inflammatory infiltrate. By their very heterogeneous clinical presentation, it seems difficult to distinguish them and make their diagnosis. Clinically, it may be useful to separate localized forms of disseminated forms of cutaneous granulomatosis, although this distinction is often artificial. We will discuss here the main forms of localized and disseminated cutaneous granulomatosis, and, in light of recent literature data, mention different therapeutic options in each case.
[Mh] MeSH terms primary: Granuloma/pathology
Skin Diseases/pathology
[Mh] MeSH terms secundary: Granuloma/therapy
Granuloma, Foreign-Body/pathology
Humans
Necrobiotic Disorders/pathology
Sarcoidosis/pathology
Skin Diseases/therapy
Skin Diseases, Infectious/complications
Skin Diseases, Infectious/pathology
[Pt] Publication type:ENGLISH ABSTRACT; JOURNAL ARTICLE; REVIEW
[Em] Entry month:1606
[Cu] Class update date: 160513
[Lr] Last revision date:160513
[Js] Journal subset:IM
[Da] Date of entry for processing:160514
[St] Status:MEDLINE

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[PMID]: 26227781
[Au] Autor:Frew JW; Murrell DF; Haber RM
[Ad] Address:Department of Dermatology, Liverpool Hospital, Sydney, New South Wales, Australia.
[Ti] Title:Fifty shades of yellow: a review of the xanthodermatoses.
[So] Source:Int J Dermatol;54(10):1109-23, 2015 Oct.
[Is] ISSN:1365-4632
[Cp] Country of publication:England
[La] Language:eng
[Ab] Abstract:The xanthodermatoses consist of a heterogeneous group of cutaneous disorders characterized by the macroscopic yellow hue seen on examination. This hue is attributable to the chemical structure of the accumulating substances within the skin or surrounding tissues. The most common culprits are lipids (cholesterol and triglycerides), elastin, and bilirubin. Exogenous sources of yellow pigment include yellow dyes (including hennas) and metal salts. This article will focus on recognition of these entities, classified in terms of morphology and the site of initial eruption, in order to support the recognition and diagnosis of these widely variable conditions.
[Mh] MeSH terms primary: Elastic Tissue/physiopathology
Lipid Metabolism
Skin Diseases/pathology
Skin Diseases/physiopathology
Skin Pigmentation
[Mh] MeSH terms secundary: Amyloidosis/pathology
Ecchymosis/diagnosis
Extremities
Facial Dermatoses/pathology
Humans
Jaundice/etiology
Necrobiosis Lipoidica/pathology
Necrobiotic Xanthogranuloma/pathology
Nevus, Sebaceous of Jadassohn/pathology
Pseudoxanthoma Elasticum/pathology
Skin Diseases/etiology
Torso
Xanthogranuloma, Juvenile/pathology
Yellow Nail Syndrome/pathology
[Pt] Publication type:JOURNAL ARTICLE; REVIEW
[Em] Entry month:1606
[Cu] Class update date: 150923
[Lr] Last revision date:150923
[Js] Journal subset:IM
[Da] Date of entry for processing:150801
[St] Status:MEDLINE
[do] DOI:10.1111/ijd.12945

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[PMID]: 26143425
[Au] Autor:Kerstetter J; Wang J
[Ad] Address:Department of Pathology and Laboratory Medicine, Loma Linda University Medical Center, 11234 Anderson Street, Room #2151, Loma Linda, CA 92354, USA.
[Ti] Title:Adult Orbital Xanthogranulomatous Disease: A Review with Emphasis on Etiology, Systemic Associations, Diagnostic Tools, and Treatment.
[So] Source:Dermatol Clin;33(3):457-63, 2015 Jul.
[Is] ISSN:1558-0520
[Cp] Country of publication:United States
[La] Language:eng
[Ab] Abstract:Adult orbital xanthogranulomatous diseases are rare entities and encompass a group of disorders with varying manifestations that are poorly understood. Taken as a group, there are non-Langerhans histiocytic disorders (type II) that are diagnosed histologically by the presence of foamy histiocytes, Touton giant cells, and varying degrees of fibrosis. Based on the accompanying systemic associations, there are 4 main categories of adult xanthogranulomatous disease: adult-onset xanthogranuloma, adult-onset asthma and periocular xanthogranuloma, necrobiotic xanthogranuloma, and Erdheim-Chester disease. Herein, we discuss the etiopathogenesis, systemic associations, methods of diagnosis, and treatment options for these disorders.
[Mh] MeSH terms primary: Cytoreduction Surgical Procedures
Erdheim-Chester Disease/therapy
Granuloma/therapy
Immunosuppressive Agents/therapeutic use
Necrobiotic Xanthogranuloma/therapy
Orbital Diseases/therapy
Skin/pathology
Xanthomatosis/therapy
[Mh] MeSH terms secundary: Adult
Asthma/epidemiology
Erdheim-Chester Disease/diagnosis
Erdheim-Chester Disease/epidemiology
Granuloma/diagnosis
Granuloma/epidemiology
Humans
Lymphatic Diseases/epidemiology
Lymphoproliferative Disorders/epidemiology
Necrobiotic Xanthogranuloma/diagnosis
Necrobiotic Xanthogranuloma/epidemiology
Orbital Diseases/diagnosis
Orbital Diseases/epidemiology
Paraproteinemias/epidemiology
Xanthomatosis/diagnosis
Xanthomatosis/epidemiology
[Pt] Publication type:JOURNAL ARTICLE; REVIEW
[Nm] Name of substance:0 (Immunosuppressive Agents)
[Em] Entry month:1603
[Cu] Class update date: 150706
[Lr] Last revision date:150706
[Js] Journal subset:IM
[Da] Date of entry for processing:150706
[St] Status:MEDLINE

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[PMID]: 25806933
[Au] Autor:van de Veerdonk FL; Netea MG; van der Meer JW
[Ti] Title:Case 39-2014: A girl with Crohn's disease and pulmonary nodules.
[So] Source:N Engl J Med;372(13):1272-3, 2015 Mar 26.
[Is] ISSN:1533-4406
[Cp] Country of publication:United States
[La] Language:eng
[Mh] MeSH terms primary: Crohn Disease/complications
Granulomatosis with Polyangiitis/diagnosis
Lung/pathology
Necrobiotic Disorders/diagnosis
[Mh] MeSH terms secundary: Female
Humans
[Pt] Publication type:COMMENT; LETTER
[Em] Entry month:1504
[Cu] Class update date: 150326
[Lr] Last revision date:150326
[Js] Journal subset:AIM; IM
[Da] Date of entry for processing:150326
[St] Status:MEDLINE
[do] DOI:10.1056/NEJMc1500453#SA1

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[PMID]: 25806932
[Au] Autor:El Saleeby CM; Kaplan JL; Nelson BA
[Ti] Title:Case 39-2014: a girl with Crohn's disease and pulmonary nodules.
[So] Source:N Engl J Med;372(13):1273, 2015 Mar 26.
[Is] ISSN:1533-4406
[Cp] Country of publication:United States
[La] Language:eng
[Mh] MeSH terms primary: Crohn Disease/complications
Granulomatosis with Polyangiitis/diagnosis
Lung/pathology
Necrobiotic Disorders/diagnosis
[Mh] MeSH terms secundary: Female
Humans
[Pt] Publication type:COMMENT; LETTER
[Em] Entry month:1504
[Cu] Class update date: 150326
[Lr] Last revision date:150326
[Js] Journal subset:AIM; IM
[Da] Date of entry for processing:150326
[St] Status:MEDLINE
[do] DOI:10.1056/NEJMc1500453


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