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based on a suspected clinical diagnosis of systemic vasculitis&#44; gave the following results&#58; white blood cell count&#44; 12&#160;500&#47;&#956;L&#59; hemoglobin&#44; 9&#46;8<span class="elsevierStyleHsp" style=""></span>g&#47;dL&#59; reactive thrombocytosis&#44; 658&#160;000&#47;&#956;L&#59; hypoalbuminemia&#44; 2&#46;0<span class="elsevierStyleHsp" style=""></span>g&#47;dL&#59; alanine aminotransferase&#44; 68 U&#47;L&#59; aspartate aminotransferase&#44; 62 U&#47;L&#59; &#947;-glutamyltransferase&#44; 181 U&#47;L&#59; alkaline phosphatase&#44; 326 U&#47;L&#59; creatinine clearance&#44; 88&#46;3<span class="elsevierStyleHsp" style=""></span>mL&#47;min with renal function of 86&#46;6&#37;&#59; C-reactive protein&#44; 1&#46;40<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#59; a negative Venereal Disease Research Laboratory test&#44; negative rheumatoid factor&#59; antineutrophil cytoplasmic antibodies &#40;c-ANCA&#41;&#44; 1&#58;40&#59; perinuclear &#40;p&#41; ANCA&#44; 1&#58;2560 U&#47;mL&#59; p-ANCA antilactoferrin&#44; 48&#46;2 U&#47;mL&#59; culture of the skin exudate&#44; <span class="elsevierStyleItalic">Escherichia coli</span>&#59; positive total hepatitis A virus antibody&#59; negative anti-hepatitis B and anti-hepatitis C virus antibodies&#59; negative human immunodeficiency virus &#40;enzyme-linked immunosorbent assay&#41;&#46; Skin biopsy revealed leukocytoclastic vasculitis &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The otorhinolaryngology department reported perforation of the cartilaginous portion of the nasal septum&#44; confirmed by computed tomography&#44; but biopsy of the nasal mucosa only revealed chronic inflammation&#46; The psychiatry department diagnosed the patient as having a mixed personality disorder and treatment was initiated with amitriptyline&#44; perphenazine&#44; and diazepam&#46; The general surgery department then performed surgical debridement of the ulcers and escharectomy&#46; The patient was treated with antibiotics &#40;trimethoprim-sulfamethoxazole&#44; 160&#47;800<span class="elsevierStyleHsp" style=""></span>mg&#44; administered intravenously every 12<span class="elsevierStyleHsp" style=""></span>hours for 14 days&#41;&#44; analgesics&#44; and thalidomide &#40;100<span class="elsevierStyleHsp" style=""></span>mg&#47;d&#41;&#59; prednisone &#40;0&#46;5<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#41; was added after achieving control of the infection&#46; The response was satisfactory and the patient was discharged after 37 days&#44; with the presence of granulation tissue and partial reepithelialization of the ulcerated areas&#46; Diagnosis at discharge was cocaine-related vasculitis based on the clinical findings&#44; histopathological study&#44; the positivity of serum markers &#40;p-ANCA and lactoferrin&#41;&#44; a history of long-term consumption of the drug&#44; and the exclusion of other causes&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Follow-up confirmed the reepithelialization of most lesions&#44; which developed into hypertrophic scars and retractile keloids&#44; currently awaiting surgical treatment by the plastic surgery department &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">Cocaine consumption is increasing worldwide&#44; leading to a rise in related diseases&#46; The spectrum of skin lesions caused by cocaine is broad and has been related to digital vasospasm&#44; bullous diseases&#44; small- and medium-vessel vasculitis&#44; which present as purpura&#44; necrotizing vasculitis&#44; urticarial vasculitis&#44; ulcers&#44; livedo reticularis&#44; Buerger disease&#44; pyoderma gangrenosum&#44; and gangrene&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> Some authors hold levamisole&#44; an adulterant found in the end product&#44; responsible for the damage in cases involving microvascular thrombosis and neutropenia&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> There are reports of cocaine consumption in patients with Wegener granulomatosis&#44;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> and since both may cause midline destructive lesions of the face&#44; distinguishing between them can be difficult&#46; In our patient&#44; the absence of granulomatous vasculitis of the respiratory tract&#44; renal involvement&#44; and high titers of c-ANCA enabled us to rule out this possibility&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">The study protocol for cocaine-related vasculitis is identical to drug-related vasculitis and includes a blood count&#44; erythrocyte sedimentation rate&#44; routine biochemistry&#44; chest x-ray&#44; urinalysis&#44; liver function tests&#44; and fecal occult blood&#46; More specific studies should subsequently be considered&#44; such as histopathology with or without direct immunofluorescence&#44; anticardiolipin antibodies&#44; homocysteine levels&#44; proteins S and C&#44; and cryoglobulins&#46; Serum levels of ANCA&#44; antinuclear antibodies&#44; rheumatoid factor&#44; complement&#44; and hepatitis B and C virus antibodies should also be determined&#44; together with human immunodeficiency virus serology&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">The case presented here was a drug-dependent patient whose clinical lesions were compatible with necrotizing vasculitis confirmed by histopathology&#44; with a diagnosis of an ANCA-positive cutaneous vasculitis &#40;p-ANCA&#44; 1&#58;2560 U&#47;mL&#44; antilactoferrin antibodies&#44; 48&#46;2 U&#47;mL&#41;&#46; The differential diagnosis included other forms of ANCA-positive small-vessel vasculitis&#46; The role of ANCA in the pathogenesis of vasculitis is still unclear&#46; One hypothesis suggests that ANCA stimulate neutrophil degranulation&#44; activation&#44; and apoptosis&#44; leading to direct and indirect endothelial damage&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Drug-induced ANCA show a perinuclear fluorescence pattern &#40;p-ANCA&#41; and positivity to several antigens &#40;myeloperoxidase&#44; cathepsin G&#44; proteinase 3&#44; azurocidin&#44; bactericidal&#47;permeability-increasing protein&#44; lactoferrin&#44; and human neutrophil elastase&#41;&#46; Reports in the literature suggest that the presence of lactoferrin and human neutrophil elastase supports the diagnosis of a cocaine-related syndrome&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;7</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The management of these patients is still a matter of debate&#44; but good outcomes have been reported with the use of systemic steroids&#44; nonsteroidal immunosuppressants&#44; antihistamines&#44; dapsone&#44; pentoxifylline&#44; and intravenous immunoglobulin&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> Our patient stopped taking cocaine and was treated with thalidomide and systemic steroids&#46; Although the outcome was excellent&#44; significant scarring could not be avoided&#46;</p></span>"
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Case and research letters
Leukocytoclastic Vasculitis Related to Cocaine Use
Vasculitis leucocitoclástica asociada a consumo de cocaína
Y. Salas-Espíndolaa, A. Peniche-Castellanosa,
Corresponding author
amelia_peniche@yahoo.com.mx

Corresponding author.
, I. López-Gehrkea, P. Mercadillo-Pérezb
a Servicio de Dermatología, Hospital General de México, México, D.F
b Servicio de Dermatopatología, Hospital General de México, México, D.F
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Vasculitis is a rare complication of cocaine abuse and poses significant diagnostic and management problems&#46; We report a case of this type of vasculitis&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 45-year-old woman with a history of cocaine abuse from age 23 years consulted because of a 20-day history of a widespread skin condition that affected practically the whole body surface&#44; was accompanied by fever and intense pain&#44; and prevented her from walking&#46; At the time of admission&#44; the patient was underweight&#44; had facies dolorosa&#44; normal vital signs&#44; and dry mucosas&#46; Physical examination revealed multiple ulcers of varying shape and size&#44; with a purulent&#44; foul-smelling exudate&#59; several were covered by necrotic slough &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Additional tests&#44; based on a suspected clinical diagnosis of systemic vasculitis&#44; gave the following results&#58; white blood cell count&#44; 12&#160;500&#47;&#956;L&#59; hemoglobin&#44; 9&#46;8<span class="elsevierStyleHsp" style=""></span>g&#47;dL&#59; reactive thrombocytosis&#44; 658&#160;000&#47;&#956;L&#59; hypoalbuminemia&#44; 2&#46;0<span class="elsevierStyleHsp" style=""></span>g&#47;dL&#59; alanine aminotransferase&#44; 68 U&#47;L&#59; aspartate aminotransferase&#44; 62 U&#47;L&#59; &#947;-glutamyltransferase&#44; 181 U&#47;L&#59; alkaline phosphatase&#44; 326 U&#47;L&#59; creatinine clearance&#44; 88&#46;3<span class="elsevierStyleHsp" style=""></span>mL&#47;min with renal function of 86&#46;6&#37;&#59; C-reactive protein&#44; 1&#46;40<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#59; a negative Venereal Disease Research Laboratory test&#44; negative rheumatoid factor&#59; antineutrophil cytoplasmic antibodies &#40;c-ANCA&#41;&#44; 1&#58;40&#59; perinuclear &#40;p&#41; ANCA&#44; 1&#58;2560 U&#47;mL&#59; p-ANCA antilactoferrin&#44; 48&#46;2 U&#47;mL&#59; culture of the skin exudate&#44; <span class="elsevierStyleItalic">Escherichia coli</span>&#59; positive total hepatitis A virus antibody&#59; negative anti-hepatitis B and anti-hepatitis C virus antibodies&#59; negative human immunodeficiency virus &#40;enzyme-linked immunosorbent assay&#41;&#46; Skin biopsy revealed leukocytoclastic vasculitis &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The otorhinolaryngology department reported perforation of the cartilaginous portion of the nasal septum&#44; confirmed by computed tomography&#44; but biopsy of the nasal mucosa only revealed chronic inflammation&#46; The psychiatry department diagnosed the patient as having a mixed personality disorder and treatment was initiated with amitriptyline&#44; perphenazine&#44; and diazepam&#46; The general surgery department then performed surgical debridement of the ulcers and escharectomy&#46; The patient was treated with antibiotics &#40;trimethoprim-sulfamethoxazole&#44; 160&#47;800<span class="elsevierStyleHsp" style=""></span>mg&#44; administered intravenously every 12<span class="elsevierStyleHsp" style=""></span>hours for 14 days&#41;&#44; analgesics&#44; and thalidomide &#40;100<span class="elsevierStyleHsp" style=""></span>mg&#47;d&#41;&#59; prednisone &#40;0&#46;5<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#41; was added after achieving control of the infection&#46; The response was satisfactory and the patient was discharged after 37 days&#44; with the presence of granulation tissue and partial reepithelialization of the ulcerated areas&#46; Diagnosis at discharge was cocaine-related vasculitis based on the clinical findings&#44; histopathological study&#44; the positivity of serum markers &#40;p-ANCA and lactoferrin&#41;&#44; a history of long-term consumption of the drug&#44; and the exclusion of other causes&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Follow-up confirmed the reepithelialization of most lesions&#44; which developed into hypertrophic scars and retractile keloids&#44; currently awaiting surgical treatment by the plastic surgery department &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">Cocaine consumption is increasing worldwide&#44; leading to a rise in related diseases&#46; The spectrum of skin lesions caused by cocaine is broad and has been related to digital vasospasm&#44; bullous diseases&#44; small- and medium-vessel vasculitis&#44; which present as purpura&#44; necrotizing vasculitis&#44; urticarial vasculitis&#44; ulcers&#44; livedo reticularis&#44; Buerger disease&#44; pyoderma gangrenosum&#44; and gangrene&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> Some authors hold levamisole&#44; an adulterant found in the end product&#44; responsible for the damage in cases involving microvascular thrombosis and neutropenia&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> There are reports of cocaine consumption in patients with Wegener granulomatosis&#44;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> and since both may cause midline destructive lesions of the face&#44; distinguishing between them can be difficult&#46; In our patient&#44; the absence of granulomatous vasculitis of the respiratory tract&#44; renal involvement&#44; and high titers of c-ANCA enabled us to rule out this possibility&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">The study protocol for cocaine-related vasculitis is identical to drug-related vasculitis and includes a blood count&#44; erythrocyte sedimentation rate&#44; routine biochemistry&#44; chest x-ray&#44; urinalysis&#44; liver function tests&#44; and fecal occult blood&#46; More specific studies should subsequently be considered&#44; such as histopathology with or without direct immunofluorescence&#44; anticardiolipin antibodies&#44; homocysteine levels&#44; proteins S and C&#44; and cryoglobulins&#46; Serum levels of ANCA&#44; antinuclear antibodies&#44; rheumatoid factor&#44; complement&#44; and hepatitis B and C virus antibodies should also be determined&#44; together with human immunodeficiency virus serology&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">The case presented here was a drug-dependent patient whose clinical lesions were compatible with necrotizing vasculitis confirmed by histopathology&#44; with a diagnosis of an ANCA-positive cutaneous vasculitis &#40;p-ANCA&#44; 1&#58;2560 U&#47;mL&#44; antilactoferrin antibodies&#44; 48&#46;2 U&#47;mL&#41;&#46; The differential diagnosis included other forms of ANCA-positive small-vessel vasculitis&#46; The role of ANCA in the pathogenesis of vasculitis is still unclear&#46; One hypothesis suggests that ANCA stimulate neutrophil degranulation&#44; activation&#44; and apoptosis&#44; leading to direct and indirect endothelial damage&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Drug-induced ANCA show a perinuclear fluorescence pattern &#40;p-ANCA&#41; and positivity to several antigens &#40;myeloperoxidase&#44; cathepsin G&#44; proteinase 3&#44; azurocidin&#44; bactericidal&#47;permeability-increasing protein&#44; lactoferrin&#44; and human neutrophil elastase&#41;&#46; Reports in the literature suggest that the presence of lactoferrin and human neutrophil elastase supports the diagnosis of a cocaine-related syndrome&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;7</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The management of these patients is still a matter of debate&#44; but good outcomes have been reported with the use of systemic steroids&#44; nonsteroidal immunosuppressants&#44; antihistamines&#44; dapsone&#44; pentoxifylline&#44; and intravenous immunoglobulin&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> Our patient stopped taking cocaine and was treated with thalidomide and systemic steroids&#46; Although the outcome was excellent&#44; significant scarring could not be avoided&#46;</p></span>"
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ISSN: 15782190
Original language: English
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