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Case of erythema elevatum diutinum associated with IgA paraproteinemia successfully controlled with thalidomide and plasma exchange
No full textErythema elevatum diutinum (EED) is a rare chronic inflammatory dermatosis often associated with monoclonal IgA gammopathy and myeloma [1]. An 83-year-old man presented with multiple, symmetric, fibrotic, hyperkeratotic/crusted/ulcerated, pruritic/painful, firm, purplish-red nodules on the extensor surfaces of the interphalangeal, metacarpophalangeal and metatarsophalangeal joints, elbows and knees (Fig. 1) that appeared five years earlier. Skin biopsy showed a leukocytoclastic vasculitis consistent with EED. The patient was treated for one year with Dapsone, with progressive worsening. At that time, the laboratory tests showed an IgA kappa monoclonal protein. The serum IgA level was 760 mg/dL. Neither monoclonal urinary components nor Bence Jones proteinuria were found. Paraproteinemia was thus consistent with monoclonal gammopathy of undetermined significance (MGUS). Three weekly sessions of plasma exchange (PLEX) were introduced during the first and second week, two exchanges/week from the 3rd to 8th week, one exchange/week from the 9th to 12th week. From the 13th week we added thalidomide 200 mg/day and 100 mg/day alternatively and we reduced PLEX to one exchange every 2 weeks. The association of the two drugs was decided owing to the sudden and marked increase in IgA (520 mg/dL). PLEX was then discontinued while thalidomide was continued as monotherapy. Thalidomide was discontinued and PLEX was restarted from the 22nd to the 35th month. The rationale of returning to PLEX treatment was to avoid the neurotoxicity of thalidomide. Following 18 plasma exchanges the IgA level decreased from 760 to 356 mg/dL and the severe (+++) skin lesions significantly improved (++). Following the association of thalidomide with PLEX, from the 4th to 9th month, the skin lesions further improved, without flares (+), and the IgA levels were slightly above the normal range (Fig. 1). Subsequently, from the 10th to 21st month, the treatment with thalidomide alone (100 mg e.o.d.) allowed the cutaneous lesions to significantly improve, and to reach an IgA level slightly above the normal value (418 g/L). From the 22nd to 35th month, i.e. at the end of the therapy, treatment with PLEX alone maintained the skin lesions as well controlled, and the IgA levels around the high normal values (Fig. 1). Co-administration of both treatments from the 4th to 9th month maintained the efficacy of plasmapheresis when its frequency was reduced. The time course of the serum IgA concentration and skin lesions can be seen in Figure 2.
figure
Figure 1. Nodules/plaques on the extensor surfaces of the interphalangeal and metacarpophalangeal joints, at baseline (a) and after 9 months since the beginning of treatment and 6 months of the association of thalidomide with plasma exchange (PLEX) (from the 4th to 9th month) (b).
figure
Figure 2. Time course of serum IgA concentration and skin lesions (+++, severe; ++, moderate; +, mild) during treatment: PLEX, plasma exchange; Thal, thalidomide. aSerum IgA value at baseline (immediately before the first PLEX); Skin lesions at baseline (immediately before the first PLEX): +++ (severe, highest severity degree). bSerum IgA value after 2 months (T2) of treatment with PLEX alone; Skin lesion: ++ (moderate). c1Serum IgA value after 3 months of PLEX plus thalidomide; Skin lesions, + (mild). c2Serum IgA value after 6 months of PLEX plus thalidomide; Skin lesions, + (mild). dSerum IgA value after 21 months (T21) of treatment and after 11 months of Thal alone; Skin lesions, + (mild). eSerum IgA value after 35 months (T35) of treatment and after 15 months of PLEX alone; Skin lesions, + (mild). fTreatment from 1st month (start of treatment) to 35th month (end of treatment): PLEX, Plex alone; PLEX + Thal, 100 mg/d alternated with 200 mg/d; Thal alone, 100 mg/eod. *400 mL/dL = IgA upper normal vallue.
Dapsone is the first-line treatment, but with no effects on paraproteinemia. Alternatively, other systemic drugs have been suggested for EED treatment that have antioxidant or antinflammatory and immunomodulant properties but do not influence the synthesis of paraproteins and therefore were not administered. Circulating immunocomplexes, monoclonal antibodies, cytokines and paraproteins are removed from the plasma through the PLEX that must be associated with immunosuppressive or cytotoxic therapy, in order to avoid the hyperproduction of pathological plasmatic factors. Thalidomide has an anti-angiogenic property that plays a significant role in the prevention of the progression of a more advanced form of biologic MGUS toward a multiple myeloma. Efficacy of the therapeutical modalities suggested for EED has not yet been defined owing to the limited number of cases studied [2, 3]. To our knowledge, this is a novel treatment because it is the first case of EED associated with IgA paraproteinemia treated with thalidomide. Therefore, this drug could be evaluated to control the disease, and could be useful in maintaining the results of PLEX.
References
1
Comfere NI, Gibson LE. Erythema elevatum diutinum. In: Wolff K, Goldsmith LA, Katz SI, Gilehrest BA, Paller AS, Leffell DJ, eds. Fitzpatrick's Dermatology in General Medicine, 7th edn. New York: McGraw-Hill, 2008; 1616–1619.
Universita di Pisa
2
Chow RK, Benny WB, Coupe RL, Dodd WA, Ongley RC. Erythema elevatum diutinum associated with IgA paraproteinemia successfully controlled with intermittent plasma exchange. Arch Dermatol 1996;132:1360–1364.
CrossRef,PubMed,CAS,Web of Science® Times Cited: 20
Universita di Pisa
3
Galeone M, Arunachalam M, Bassi A, Scarfì F, Difonzo EM. Erythema elevatum diutinum. QJM 2014;107:227–228.
CrossRef,PubMed,Web of Science® Times Cited:
Going Beyond Counting First Authors in Author Co-citation Analysis
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counts are defined. Co-citation counting provides the data on which all subsequent statistical analyses and mappings
are based, and we compare ACA results based on two different types of co-citation counting - the traditional type that
only counts the first one among a cited work's authors on the one hand and a non-traditional type that takes into
account the first 5 authors of a cited work on the other hand. Results indicate that the picture produced through this non-traditional author co-citation counting contains more coherent author groups and is therefore considerably clearer. However, this picture represents fewer specialties in the research field being studied than that produced through the traditional first-author co-citation counting when the same number of top-ranked authors is selected and analyzed. Reasons for these effects are discussed
Variations on the Author
Full text link“Variations on the Author” discusses two of Eduardo Coutinho’s recent films (Um Dia na Vida, from 2010, and Últimas Conversas, posthumously released in 2015) and their contribution to the general question of documentary authorship. The director’s filmography is characterized by a consistent yet self-effacing form of authorial self-inscription: Coutinho often features as an interviewer that rather than express opinions propels discourses; an interviewer that is good at listening. This mode of self-inscription characterizes him as an author who is not expressive but who is nonetheless markedly present on the screen. In Um Dia na Vida, however, Coutinho is completely absent form the image, while Últimas Conversas, on the contrary, includes a confessional prologue that moves the director from the margins to the center of his films. This article examines the ways in which these works stand out in the filmography of a director who offers new insights into the notion of cinematic authorship
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Full text linkWe conducted a full-scale evaluative citation analysis study of scholars in the XML research field to explore just how different from each other author rankings resulting from different citation counting methods actually are, and to demonstrate the capability of emerging data and tools on the Web in supporting more realistic citation counting methods. Our results contest some common arguments for the continued
use of first-author citation counts in the evaluation of scholars, such as high correlations between author rankings by first-author citation counts and other citation
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koamabayili/VECTRON-author-checklist: VECTRON author checklist
No full textWe have done our best to complete the author checklist relating to the use of animals in the hut study. Note that the objective for the hut study was to evaluate the IRS treatment applications for residual efficacy against Anopheles mosquitoes, including the local An. coluzzii mosquito population. Cows were only used to attract mosquitoes into the huts and no tests were carried out directly on the cows. The author checklist is intended for use with studies where experiments are carried out on animals, which is why we have had such difficulty in completing this for the hut study, as many of the questions do not relate to how the cows were used
Author-wise bibliometric analysis based on entropy.
No full textAuthor-wise bibliometric analysis based on entropy.</p
Author Under Sail The Imagination of Jack London, 1893-1902
No full textIn Author Under Sail, Jay Williams offers the first complete literary biography of Jack London as a professional writer engaged in the labor of writing. It examines the authorial imagination in London's work, the use of imagination in both his fiction and nonfiction, and the ways he defined imagination in the creative process in his business dealings with his publishers, editors, and agents. In this first volume of a two-volume biography, Williams traverses the years 1893 to 1902, from London's "Story of a Typhoon" to The People of the Abyss. The Jack London who emerges in the pages of Author Under Sail is a writer whose partnership with publishers, most notably his productive alliance with George Brett of Macmillan, was one of the most formative in American literary history. London pioneered many author models during the heyday of realism and naturalism, blurring the boundaries of these popular genres by focusing on absorption and theatricality and the representation of the seen and unseen. London created an impassioned, sincere, and extremely personal realism unlike that of other American writers of the time. Author Under Sail is a literary tour de force that reveals the full range of London as writer, creative citizen, and entrepreneur at the same time it sheds light on the maverick side of machine-age literature.Intro -- Title Page -- Copyright Page -- Dedication -- Contents -- Acknowledgments -- Introduction -- 1. Spirit Truth -- 2. From Absorption to Theatricality and Back Again -- 3. "I Will Build a New Present" -- 4. Sons as Authors -- 5. Fathers as Publishers -- 6. The Daughter as Author -- 7. Lovers as Authors -- 8. At Sea with the Family -- 9. Yellow News, Yellow Stories -- 10. The Return Home -- Notes -- Bibliography -- Index -- About Jay WilliamsIn Author Under Sail, Jay Williams offers the first complete literary biography of Jack London as a professional writer engaged in the labor of writing. It examines the authorial imagination in London's work, the use of imagination in both his fiction and nonfiction, and the ways he defined imagination in the creative process in his business dealings with his publishers, editors, and agents. In this first volume of a two-volume biography, Williams traverses the years 1893 to 1902, from London's "Story of a Typhoon" to The People of the Abyss. The Jack London who emerges in the pages of Author Under Sail is a writer whose partnership with publishers, most notably his productive alliance with George Brett of Macmillan, was one of the most formative in American literary history. London pioneered many author models during the heyday of realism and naturalism, blurring the boundaries of these popular genres by focusing on absorption and theatricality and the representation of the seen and unseen. London created an impassioned, sincere, and extremely personal realism unlike that of other American writers of the time. Author Under Sail is a literary tour de force that reveals the full range of London as writer, creative citizen, and entrepreneur at the same time it sheds light on the maverick side of machine-age literature.Description based on publisher supplied metadata and other sources.Electronic reproduction. Ann Arbor, Michigan : ProQuest Ebook Central, YYYY. Available via World Wide Web. Access may be limited to ProQuest Ebook Central affiliated libraries
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