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anti-Smith&#44; anti-Ro and anti-double-stranded DNA antibodies&#41; and had decreased complement levels&#46; Given the predominance of joint involvement&#44; at first&#44; he was treated with prednisone at 10<span class="elsevierStyleHsp" style=""></span>mg&#47;day and hydroxychloroquine at 400<span class="elsevierStyleHsp" style=""></span>mg&#47;day&#46; There was a partial response in the joints&#44; but none with respect to the skin&#46; He had a substantial deterioration of the cutaneous manifestations&#44; developing patches of alopecia areata&#44; palpebral edema and nonpruritic&#44; erythematous violaceous macular lesions on his face&#44; torso and proximal portion of extremities&#44; producing a serious physical and psychological distress for the patient&#46; In addition&#44; he had constitutional symptoms&#44; with fever and an elevation of serum markers of lupus activity&#46; A biopsy of the skin lesions revealed an interstitial lymphohistiocytic infiltrate in superficial and deep dermis&#44; with the presence of mucin deposits arranged around collagen fibers&#44; corresponding histopathologically to IGD &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; It was decided to treat the patient with high-dose corticosteroids &#40;1<span class="elsevierStyleHsp" style=""></span>mg&#47;kg body weight &#91;bw&#93;&#47;day of prednisone&#41;&#44; to which he responded only partially&#46; His response to the association of metronidazole &#40;250<span class="elsevierStyleHsp" style=""></span>mg&#47;8<span class="elsevierStyleHsp" style=""></span>h&#41; and mycophenolate mofetil &#40;1<span class="elsevierStyleHsp" style=""></span>g&#47;12<span class="elsevierStyleHsp" style=""></span>h&#41; was not satisfactory either&#44; and he did not tolerate methotrexate&#46; Given the high clinical activity and extension of the lesions&#44; we decided to start treatment with intravenous belimumab &#40;10<span class="elsevierStyleHsp" style=""></span>mg&#47;kg bw each month&#41;&#44; associated with hydroxychloroquine&#46; The patient showed an evident satisfactory response after 1 month of treatment&#44; with resolution of the skin lesions and alopecia&#44; as well as clinical remission and normalization of the biological parameters&#46; Four years after the start of this approach&#44; he remains in remission&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Interstitial granulomatous dermatitis is an uncommon skin condition&#44; that is occasionally associated with autoimmune diseases &#40;rheumatoid arthritis&#44; SLE&#44; vasculitis&#44; thyroiditis or diabetes&#41;&#44; lymphoproliferative disorders and even certain drugs like anti-tumor necrosis factor agents&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;2</span></a> The skin lesions usually develop in patients with SLE although&#44; in some cases&#44; they can also be the presenting sign of the disease&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> Its treatment has not been well established&#46; In the case of IGD associated with drugs&#44; it seems to be necessary to discontinue the causative agent and assess introducing treatment with corticosteroids&#46; In general lines&#44; these are the primary therapeutic options for IGD&#46; In cases of IGD associated with an autoimmune disease&#44; it may be necessary to combine corticosteroids with the treatment of the underlying disease&#46; For this&#44; we utilize immunosuppressive drugs&#44; like methotrexate&#44; mycophenolate&#44; hydroxychloroquine&#44; dapsone and even metronidazole &#40;the latter indicated in granulomatous diseases&#41;&#46; Our patient did not tolerate methotrexate or respond to other treatments&#46; With respect to biological therapy&#44; there are reports of cases of IGD associated with arthritis in which there was a good cutaneous response to infliximab&#44;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> etanercept&#44;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> tocilizumab&#44;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> ustekinumab<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> and immunoglobulins&#44;<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> treatments that were not indicated in our patient&#46; Belimumab has been seen to be effective in individuals with SLE and with cutaneous and joint involvement that is refractory to standard immunosuppressive agents&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> There is no evidence of its efficacy in IGD&#46; It is a fully humanized monoclonal antibody against B lymphocyte stimulator &#40;BlyS&#41;&#44; the action of which focuses on the indirect depletion of B lymphocytes and plasma cells&#44; thus reducing the production of antibodies&#46; Given the elevated clinical and serum activity&#44; considering the extension&#44; severity and refractoriness of the skin lesions&#44; we began treatment with belimumab&#44; which achieved complete resolution of IGD&#46; These results are surprising&#44; considering that the mechanism of action of this drug does not appear to be related to the pathophysiology of the granulomatous lesion&#46; The literature provides cases in which the development of IGD associated with autoimmune diseases was related to the presence of autoantibodies and immune complex deposition&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> This suggests that the depletion of these elements would impede the perpetuation of the inflammatory cascade causing the granuloma&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">In short&#44; the development of IGD during the course of SLE is an uncommon cutaneous condition&#44; with no established treatment&#44; and it may be resistant to different therapeutic strategies&#46; The case we report is the first in which belimumab was found to be effective in IGD associated with SLE&#46;</p></span>"
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Letter to the Editor
Interstitial Granulomatous Dermatitis Associated With Lupus: A Case Report
Dermatitis intersticial granulomatosa asociada al lupus: a propósito de un caso
Marta Arévalo
Corresponding author
marevalo@tauli.cat

Corresponding author.
, Mireia Moreno, Jordi Gratacós
Servicio de Reumatología, Hospital Universitari Parc Taulí de Sabadell, Sabadell, Barcelona, Spain
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Histological image of a skin biopsy&#46; The interstitial lymphohistiocytic infiltrate can be observed in superficial and deep dermis&#44; with the presence of mucin deposits surrounding the collagen fibers&#46; Histopathological view of interstitial granulomatous dermatitis&#46;</p>"
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Recently&#44; we treated a patient with interstitial granulomatous dermatitis &#40;IGD&#41; associated with systemic lupus erythematosus &#40;SLE&#41;&#46; Given the refractoriness of the condition to standard therapy&#44; treatment with belimumab was begun&#44; and the response was satisfactory&#46; In view of the absence of data in the literature on the efficacy of belimumab in IGD associated with SLE&#44; the report of this case was considered to be interesting&#44; as it would enable us to examine new therapeutic lines in refractory IGD&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">The patient was a 40-year-old white man who had been diagnosed with SLE at the age of 35 on the basis of leukopenia&#44; oral and nasal ulcers&#44; arthritis of the hands&#44; myositis and cutaneous involvement in the form of diffuse alopecia&#46; He was also positive for immunological markers &#40;antinuclear&#44; anti-Smith&#44; anti-Ro and anti-double-stranded DNA antibodies&#41; and had decreased complement levels&#46; Given the predominance of joint involvement&#44; at first&#44; he was treated with prednisone at 10<span class="elsevierStyleHsp" style=""></span>mg&#47;day and hydroxychloroquine at 400<span class="elsevierStyleHsp" style=""></span>mg&#47;day&#46; There was a partial response in the joints&#44; but none with respect to the skin&#46; He had a substantial deterioration of the cutaneous manifestations&#44; developing patches of alopecia areata&#44; palpebral edema and nonpruritic&#44; erythematous violaceous macular lesions on his face&#44; torso and proximal portion of extremities&#44; producing a serious physical and psychological distress for the patient&#46; In addition&#44; he had constitutional symptoms&#44; with fever and an elevation of serum markers of lupus activity&#46; A biopsy of the skin lesions revealed an interstitial lymphohistiocytic infiltrate in superficial and deep dermis&#44; with the presence of mucin deposits arranged around collagen fibers&#44; corresponding histopathologically to IGD &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; It was decided to treat the patient with high-dose corticosteroids &#40;1<span class="elsevierStyleHsp" style=""></span>mg&#47;kg body weight &#91;bw&#93;&#47;day of prednisone&#41;&#44; to which he responded only partially&#46; His response to the association of metronidazole &#40;250<span class="elsevierStyleHsp" style=""></span>mg&#47;8<span class="elsevierStyleHsp" style=""></span>h&#41; and mycophenolate mofetil &#40;1<span class="elsevierStyleHsp" style=""></span>g&#47;12<span class="elsevierStyleHsp" style=""></span>h&#41; was not satisfactory either&#44; and he did not tolerate methotrexate&#46; Given the high clinical activity and extension of the lesions&#44; we decided to start treatment with intravenous belimumab &#40;10<span class="elsevierStyleHsp" style=""></span>mg&#47;kg bw each month&#41;&#44; associated with hydroxychloroquine&#46; The patient showed an evident satisfactory response after 1 month of treatment&#44; with resolution of the skin lesions and alopecia&#44; as well as clinical remission and normalization of the biological parameters&#46; Four years after the start of this approach&#44; he remains in remission&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Interstitial granulomatous dermatitis is an uncommon skin condition&#44; that is occasionally associated with autoimmune diseases &#40;rheumatoid arthritis&#44; SLE&#44; vasculitis&#44; thyroiditis or diabetes&#41;&#44; lymphoproliferative disorders and even certain drugs like anti-tumor necrosis factor agents&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;2</span></a> The skin lesions usually develop in patients with SLE although&#44; in some cases&#44; they can also be the presenting sign of the disease&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> Its treatment has not been well established&#46; In the case of IGD associated with drugs&#44; it seems to be necessary to discontinue the causative agent and assess introducing treatment with corticosteroids&#46; In general lines&#44; these are the primary therapeutic options for IGD&#46; In cases of IGD associated with an autoimmune disease&#44; it may be necessary to combine corticosteroids with the treatment of the underlying disease&#46; For this&#44; we utilize immunosuppressive drugs&#44; like methotrexate&#44; mycophenolate&#44; hydroxychloroquine&#44; dapsone and even metronidazole &#40;the latter indicated in granulomatous diseases&#41;&#46; Our patient did not tolerate methotrexate or respond to other treatments&#46; With respect to biological therapy&#44; there are reports of cases of IGD associated with arthritis in which there was a good cutaneous response to infliximab&#44;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> etanercept&#44;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> tocilizumab&#44;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> ustekinumab<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> and immunoglobulins&#44;<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> treatments that were not indicated in our patient&#46; Belimumab has been seen to be effective in individuals with SLE and with cutaneous and joint involvement that is refractory to standard immunosuppressive agents&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> There is no evidence of its efficacy in IGD&#46; It is a fully humanized monoclonal antibody against B lymphocyte stimulator &#40;BlyS&#41;&#44; the action of which focuses on the indirect depletion of B lymphocytes and plasma cells&#44; thus reducing the production of antibodies&#46; Given the elevated clinical and serum activity&#44; considering the extension&#44; severity and refractoriness of the skin lesions&#44; we began treatment with belimumab&#44; which achieved complete resolution of IGD&#46; These results are surprising&#44; considering that the mechanism of action of this drug does not appear to be related to the pathophysiology of the granulomatous lesion&#46; The literature provides cases in which the development of IGD associated with autoimmune diseases was related to the presence of autoantibodies and immune complex deposition&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> This suggests that the depletion of these elements would impede the perpetuation of the inflammatory cascade causing the granuloma&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">In short&#44; the development of IGD during the course of SLE is an uncommon cutaneous condition&#44; with no established treatment&#44; and it may be resistant to different therapeutic strategies&#46; The case we report is the first in which belimumab was found to be effective in IGD associated with SLE&#46;</p></span>"
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Article information
ISSN: 21735743
Original language: English
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Idiomas
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