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cheilitis&#44; pseudomembranous conjunctivitis&#44; and ulcerative keratitis&#46; The patient also presented erythematous scaly lesions on the scalp&#44; maculopapular lesions and reddish-purple plaques on the torso and legs&#44; hyperkeratosis with fissures on the palms and soles&#44; and erosive lesions on the glans and scrotum &#40;<a class="elsevierStyleCrossRefs" href="#fig0005">Figs&#46; 1 and 2</a>&#41;&#59; all these signs appeared 2 months before the patient visited our department&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">A skin biopsy showed lichenoid interface dermatitis with necrotic keratinocytes &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; Direct immunofluorescence showed intercellular deposits of immunoglobulin &#40;Ig&#41; G and IgC3 in the epidermis&#46; Indirect immunofluorescence showed intercellular deposits when monkey esophagus was used as a substrate and was negative when rat bladder was used&#46; Immunoblotting identified envoplakin &#40;210<span class="elsevierStyleHsp" style=""></span>kDa&#41; and periplakin &#40;190<span class="elsevierStyleHsp" style=""></span>kDa&#41; antibodies&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">A study was performed to search for an occult neoplasm&#46; Computed tomography of the chest and abdomen revealed enlarged mesenteric&#44; mediastinal&#44; and axillary lymph nodes&#44; and a mass in the retroperitoneal soft tissue&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Lymph-node and bone-marrow biopsies were compatible with non-Hodgkin follicular lymphoma and a diagnosis of PAMS associated with non-Hodgkin follicular lymphoma was reached&#46; Treatment of the underlying neoplasm was instated with 8 cycles of chemotherapy using vincristine&#44; rituximab&#44; cyclophosphamide&#44; and prednisone&#59; 5<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day of ciclosporin and 1<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day of methylprednisone were also administered at gradually decreasing doses to treat the cutaneous manifestations&#46; Complete remission of the lymphoma was achieved with no recurrences to date&#44; and the skin lesions resolved completely in 2 weeks&#46; After 3 years of follow-up&#44; the patient presents only mild stomatitis&#44; which is being treated with 100<span class="elsevierStyleHsp" style=""></span>mg&#47;day of ciclosporin&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">PAMS is a heterogeneous autoimmune syndrome that involves several internal organs&#59; it is associated with a neoplasm and presents clearly defined clinical&#44; histologic&#44; and immunologic characteristics&#46; The clinical findings of paraneoplastic pemphigus are varied and may resemble pemphigus&#44; lichen planus&#44; erythema multiforme&#44; or graft-versus-host disease&#46; Stomatitis is usually present and often an early sign of the disease&#8212;so much so that its absence should call a diagnosis of paraneoplastic pemphigus into question&#46; It is characterized by very painful erosive lesions&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Anatomic pathology may show acantholysis but&#44; unlike in common pemphigus&#44; this is less marked and may be accompanied by an intense lichenoid mononuclear infiltrate in the dermal&#8211;epidermal junction&#44; with vacuolar degeneration&#44; suprabasal desquamation&#44; and keratinocyte necrosis &#40;lichenoid dermatitis&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Direct-immunofluorescence findings show intercellular immunoreactants &#40;IgG and complement&#41;&#44; as in pemphigus&#44; although IgG and&#47;or complement are also commonly found in the basement membrane&#59; this fact is useful in the differential diagnosis of common pemphigus with paraneoplastic pemphigus or PAMS&#46; It should be remembered that&#44; in some cases&#44; direct immunofluorescence tests may be negative&#44; possibly because of the predominance of lichenoid lesions or the presence of necrotic tissue in the biopsies&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Serum antibodies against plakins &#40;envoplakin&#44; periplakin&#44; desmoplakin 1 and 2&#44; plectin&#44; and an uncharacterized 170<span class="elsevierStyleHsp" style=""></span>kDa protein&#41; have been detected in all epithelia&#46; However&#44; in a particular subgroup of patients&#44; antidesmoplakin antibodies may be absent and antienvoplakin and antiperiplakin antibodies are therefore considered to be more specific in PAMS&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;6</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Paraneoplastic pemphigus is a mucocutaneous manifestation of a severe PAMS and precedes diagnosis of the neoplasm in approximately a third of patients&#46; Clinical suspicion of paraneoplastic pemphigus is essential for early diagnosis and treatment of both PAMS and the neoplasm&#44; and thus for preventing fatal complications&#44; such as bronchiolitis obliterans&#46;<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7&#44;8</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">Our patient presented with a case of PAMS associated with lymphoma&#46; Diagnosis was made early and&#44; unlike most cases associated with malignant tumors reported in the literature&#44; the outcome was excellent&#44; with the patient alive more than 2 years after diagnosis&#46;</p></span></span>"
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        "texto" => "<p id="par0070" class="elsevierStylePara elsevierViewall">The authors would like to thank Professor J&#46; Manuel Mascar&#243;-Galy&#44; Hospital Clinic Barcelona&#44; Spain&#44; and Josep Herrero&#44; Dermatology Department&#44; Hospital del Mar&#44; Barcelona&#44; Spain&#44; for performing the immunoblotting technique&#46;</p>"
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Cases and Research Letters
Follicular Lymphoma With Paraneoplastic Autoimmune Multiorgan Syndrome
Síndrome multiorgánico autoinmune paraneoplásico asociado a linfoma folicular
I. Hidalgoa,
Autor para correspondencia
ihidalgoparra@gmail.com

Corresponding author.
, F. Martinezb, C. Graua, I. Gila, A. Azóna
a Sección de Dermatología, Hospital Universitari Sant Joan, Reus, Spain
b Servicio de Oncologia, Hospital Universitari Sant Joan, Reus, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Paraneoplastic pemphigus is a rare entity first described by Anhalt et al&#46; in 1990&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It is an autoimmune disease of the skin and mucosa&#44; associated with neoplasm&#44; which is generally lymphoid in origin&#46; Because several organs are involved as well as the skin and because the physiopathologic mechanisms associated with the mucosal&#44; skin and internal-organ lesions are not limited to the presence of antibodies specific to adhesion molecules&#44; in 2001&#44; Nguyen et al&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> proposed the term paraneoplastic autoimmune multiorgan syndrome &#40;PAMS&#41;&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">We describe a patient with PAMS associated with non-Hodgkin lymphoma&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Case Description</span><p id="par0015" class="elsevierStylePara elsevierViewall">A 69-year-old man with a personal history of cerebrovascular accident and type 2 diabetes mellitus visited our department with oral erosions and ulcers that had appeared 6 months earlier&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Physical examination revealed erosive glossitis&#44; cheilitis&#44; pseudomembranous conjunctivitis&#44; and ulcerative keratitis&#46; The patient also presented erythematous scaly lesions on the scalp&#44; maculopapular lesions and reddish-purple plaques on the torso and legs&#44; hyperkeratosis with fissures on the palms and soles&#44; and erosive lesions on the glans and scrotum &#40;<a class="elsevierStyleCrossRefs" href="#fig0005">Figs&#46; 1 and 2</a>&#41;&#59; all these signs appeared 2 months before the patient visited our department&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">A skin biopsy showed lichenoid interface dermatitis with necrotic keratinocytes &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; Direct immunofluorescence showed intercellular deposits of immunoglobulin &#40;Ig&#41; G and IgC3 in the epidermis&#46; Indirect immunofluorescence showed intercellular deposits when monkey esophagus was used as a substrate and was negative when rat bladder was used&#46; Immunoblotting identified envoplakin &#40;210<span class="elsevierStyleHsp" style=""></span>kDa&#41; and periplakin &#40;190<span class="elsevierStyleHsp" style=""></span>kDa&#41; antibodies&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">A study was performed to search for an occult neoplasm&#46; Computed tomography of the chest and abdomen revealed enlarged mesenteric&#44; mediastinal&#44; and axillary lymph nodes&#44; and a mass in the retroperitoneal soft tissue&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Lymph-node and bone-marrow biopsies were compatible with non-Hodgkin follicular lymphoma and a diagnosis of PAMS associated with non-Hodgkin follicular lymphoma was reached&#46; Treatment of the underlying neoplasm was instated with 8 cycles of chemotherapy using vincristine&#44; rituximab&#44; cyclophosphamide&#44; and prednisone&#59; 5<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day of ciclosporin and 1<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day of methylprednisone were also administered at gradually decreasing doses to treat the cutaneous manifestations&#46; Complete remission of the lymphoma was achieved with no recurrences to date&#44; and the skin lesions resolved completely in 2 weeks&#46; After 3 years of follow-up&#44; the patient presents only mild stomatitis&#44; which is being treated with 100<span class="elsevierStyleHsp" style=""></span>mg&#47;day of ciclosporin&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">PAMS is a heterogeneous autoimmune syndrome that involves several internal organs&#59; it is associated with a neoplasm and presents clearly defined clinical&#44; histologic&#44; and immunologic characteristics&#46; The clinical findings of paraneoplastic pemphigus are varied and may resemble pemphigus&#44; lichen planus&#44; erythema multiforme&#44; or graft-versus-host disease&#46; Stomatitis is usually present and often an early sign of the disease&#8212;so much so that its absence should call a diagnosis of paraneoplastic pemphigus into question&#46; It is characterized by very painful erosive lesions&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Anatomic pathology may show acantholysis but&#44; unlike in common pemphigus&#44; this is less marked and may be accompanied by an intense lichenoid mononuclear infiltrate in the dermal&#8211;epidermal junction&#44; with vacuolar degeneration&#44; suprabasal desquamation&#44; and keratinocyte necrosis &#40;lichenoid dermatitis&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Direct-immunofluorescence findings show intercellular immunoreactants &#40;IgG and complement&#41;&#44; as in pemphigus&#44; although IgG and&#47;or complement are also commonly found in the basement membrane&#59; this fact is useful in the differential diagnosis of common pemphigus with paraneoplastic pemphigus or PAMS&#46; It should be remembered that&#44; in some cases&#44; direct immunofluorescence tests may be negative&#44; possibly because of the predominance of lichenoid lesions or the presence of necrotic tissue in the biopsies&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Serum antibodies against plakins &#40;envoplakin&#44; periplakin&#44; desmoplakin 1 and 2&#44; plectin&#44; and an uncharacterized 170<span class="elsevierStyleHsp" style=""></span>kDa protein&#41; have been detected in all epithelia&#46; However&#44; in a particular subgroup of patients&#44; antidesmoplakin antibodies may be absent and antienvoplakin and antiperiplakin antibodies are therefore considered to be more specific in PAMS&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;6</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Paraneoplastic pemphigus is a mucocutaneous manifestation of a severe PAMS and precedes diagnosis of the neoplasm in approximately a third of patients&#46; Clinical suspicion of paraneoplastic pemphigus is essential for early diagnosis and treatment of both PAMS and the neoplasm&#44; and thus for preventing fatal complications&#44; such as bronchiolitis obliterans&#46;<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7&#44;8</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">Our patient presented with a case of PAMS associated with lymphoma&#46; Diagnosis was made early and&#44; unlike most cases associated with malignant tumors reported in the literature&#44; the outcome was excellent&#44; with the patient alive more than 2 years after diagnosis&#46;</p></span></span>"
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        "texto" => "<p id="par0070" class="elsevierStylePara elsevierViewall">The authors would like to thank Professor J&#46; Manuel Mascar&#243;-Galy&#44; Hospital Clinic Barcelona&#44; Spain&#44; and Josep Herrero&#44; Dermatology Department&#44; Hospital del Mar&#44; Barcelona&#44; Spain&#44; for performing the immunoblotting technique&#46;</p>"
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ISSN: 15782190
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