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The skin ulcer healed in 3 weeks&#46; After a remission induction therapy&#44; anthracycline was administered for 3 days and cytarabine for 7&#44; PSL was down-titrated to 25<span class="elsevierStyleHsp" style=""></span>mg&#44; and no recurrences of the cervical lesions were seen&#46; However&#44; new and painful subcutaneous indurations appeared on the extremities with high fever&#46; The blood test showed elevated C-reactive protein levels &#40;2&#46;51<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#41; and decreased platelet count &#40;1&#46;0<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">4</span>&#47;&#956;L&#41;&#46; The physical examination revealed the presence of multiple tender subcutaneous erythematous indurations &#40;about 0&#46;5&#8211;2<span class="elsevierStyleHsp" style=""></span>cm in diameter&#41;&#44; on the forearms and thighs&#44; as well as sacral ulcer &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>b&#44; c&#41;&#46; Local samples for bacteriological and mycological tests tested negative&#44; and the patient&#39;s response to antibiotics was poor&#46; Skin biopsy of the subcutaneous induration on the left forearm showed hemorrhage&#44; neutrophil and lymphocyte infiltration of the deep dermis&#44; and neutrophilic lobular panniculitis&#44; without any signs of vasculitis &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>d&#44; e&#41;&#46; The lesions on the extremities and sacral area improved without up-titration of the dose of PSL&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">In the current case&#44; deep ulcers at the central venous catheter insertion site on the right neck were considered a pathergy reaction&#44; a characteristic feature of PG&#46; There were no ulcerative lesions suggestive of PG on any part of the body other than the neck&#46; After 2 months&#44; painful subcutaneous erythematous nodules appeared on the extremities&#44; and a skin biopsy revealed the presence of neutrophilic lobular panniculitis&#46; Because neither septal panniculitis nor Miescher radial granuloma was seen&#44; erythema nodosum was excluded&#46; PG and neutrophilic panniculitis are both categorized as deep neutrophilic dermatoses&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> suggesting a shared pathogenesis&#46; Neutrophilic lobular panniculitis is observed in subcutaneous Sweet&#39;s syndrome&#46; PG and Sweet&#39;s syndrome are the representative diseases of neutrophilic dermatosis&#44; and both diseases often clinically present with similar features&#44; which are difficult to differentiate&#44; especially in patients with hematologic disorders&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">2&#8211;4</span></a> Such overlapped or atypical cases have been reported under the term &#8216;neutrophilic dermatosis of myeloproliferative disorders&#8217;&#44; suggesting a linking of entities of both disorders in a continuous pathologic spectrum&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a> Although the cervical ulcer was not biopsized in the present case&#44; ulcerative neutrophilic panniculitis cannot be ruled out&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">The VEXAS syndrome &#40;vacuoles&#44; E1 enzyme&#44; X-linked&#44; autoinflammatory&#44; somatic&#41; is an inflammatory disease due to somatic UBA1 variants presenting with myelodysplasia&#44; and autoinflammatory symptoms including chondritis&#44; vasculitis&#44; and neutrophilic dermatosis&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a> The VEXAS syndrome was listed as a differential disease because of the common complication of neutrophilic dermatosis and myelodysplastic syndrome&#46; However&#44; it was ruled out in this case because the granulocyte progenitor cells did not contain cytoplasmic vacuoles in the bone marrow biopsy&#46; The current case of myelodysplastic syndrome presented with different clinical phenotypes such as deep ulcer&#44; and painful erythematous subcutaneous nodules at different points in time&#44; both of which were due to activated neutrophils&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflict of interests</span><p id="par0025" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflict of interest&#46;</p></span></span>"
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Case and Research Letter
A Case of Pyoderma Gangrenosum-Like Ulcers Progressing into Neutrophilic Panniculitis in a Patient With Myelodysplastic Syndrome
Un caso de úlceras similares al pioderma gangrenoso que posteriormente desarrollaron paniculitis neutrofílica en un paciente con síndrome mielodisplásico
K. Irie
Autor para correspondencia
kinuko07@fmu.ac.jp

Corresponding author.
, T. Yamamoto
Department of Dermatology, Fukushima Medical University, Fukushima, Japan
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The skin ulcer healed in 3 weeks&#46; After a remission induction therapy&#44; anthracycline was administered for 3 days and cytarabine for 7&#44; PSL was down-titrated to 25<span class="elsevierStyleHsp" style=""></span>mg&#44; and no recurrences of the cervical lesions were seen&#46; However&#44; new and painful subcutaneous indurations appeared on the extremities with high fever&#46; The blood test showed elevated C-reactive protein levels &#40;2&#46;51<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#41; and decreased platelet count &#40;1&#46;0<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">4</span>&#47;&#956;L&#41;&#46; The physical examination revealed the presence of multiple tender subcutaneous erythematous indurations &#40;about 0&#46;5&#8211;2<span class="elsevierStyleHsp" style=""></span>cm in diameter&#41;&#44; on the forearms and thighs&#44; as well as sacral ulcer &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>b&#44; c&#41;&#46; Local samples for bacteriological and mycological tests tested negative&#44; and the patient&#39;s response to antibiotics was poor&#46; Skin biopsy of the subcutaneous induration on the left forearm showed hemorrhage&#44; neutrophil and lymphocyte infiltration of the deep dermis&#44; and neutrophilic lobular panniculitis&#44; without any signs of vasculitis &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>d&#44; e&#41;&#46; The lesions on the extremities and sacral area improved without up-titration of the dose of PSL&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">In the current case&#44; deep ulcers at the central venous catheter insertion site on the right neck were considered a pathergy reaction&#44; a characteristic feature of PG&#46; There were no ulcerative lesions suggestive of PG on any part of the body other than the neck&#46; After 2 months&#44; painful subcutaneous erythematous nodules appeared on the extremities&#44; and a skin biopsy revealed the presence of neutrophilic lobular panniculitis&#46; Because neither septal panniculitis nor Miescher radial granuloma was seen&#44; erythema nodosum was excluded&#46; PG and neutrophilic panniculitis are both categorized as deep neutrophilic dermatoses&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> suggesting a shared pathogenesis&#46; Neutrophilic lobular panniculitis is observed in subcutaneous Sweet&#39;s syndrome&#46; PG and Sweet&#39;s syndrome are the representative diseases of neutrophilic dermatosis&#44; and both diseases often clinically present with similar features&#44; which are difficult to differentiate&#44; especially in patients with hematologic disorders&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">2&#8211;4</span></a> Such overlapped or atypical cases have been reported under the term &#8216;neutrophilic dermatosis of myeloproliferative disorders&#8217;&#44; suggesting a linking of entities of both disorders in a continuous pathologic spectrum&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a> Although the cervical ulcer was not biopsized in the present case&#44; ulcerative neutrophilic panniculitis cannot be ruled out&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">The VEXAS syndrome &#40;vacuoles&#44; E1 enzyme&#44; X-linked&#44; autoinflammatory&#44; somatic&#41; is an inflammatory disease due to somatic UBA1 variants presenting with myelodysplasia&#44; and autoinflammatory symptoms including chondritis&#44; vasculitis&#44; and neutrophilic dermatosis&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a> The VEXAS syndrome was listed as a differential disease because of the common complication of neutrophilic dermatosis and myelodysplastic syndrome&#46; However&#44; it was ruled out in this case because the granulocyte progenitor cells did not contain cytoplasmic vacuoles in the bone marrow biopsy&#46; The current case of myelodysplastic syndrome presented with different clinical phenotypes such as deep ulcer&#44; and painful erythematous subcutaneous nodules at different points in time&#44; both of which were due to activated neutrophils&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflict of interests</span><p id="par0025" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflict of interest&#46;</p></span></span>"
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