Case report
Paraneoplastic Pemphigus or Paraneoplastic Autoimmune Multiorgan Syndrome. Report of 2 Cases in Children and a Review of the Literature
Pénfigo paraneoplásico/síndrome multiorgánico autoinmune paraneoplásico. Presentación de dos casos en la edad infantil. Revisión de la literatura
A.B. Cervinia, V. Tosib,
, S.H. Kimb, M. Bociana, G. Chantadac, C. Nousarid, O.G. Carballoe, A.M. Pierinia
Autor para correspondencia
a Servicio de Dermatología, Hospital de Pediatría Dr. Prof. Juan P. Garrahan, Buenos Aires, Argentina
b Dermatología Pediátrica U.B.A., Servicio de Dermatología, Hospital de Pediatría Dr. Prof. Juan P. Garrahan, Buenos Aires, Argentina
c Servicio de Hemato-Oncología, Hospital de Pediatría Dr. Prof. Juan P. Garrahan, Buenos Aires, Argentina
d Institute for Immunofluorescence Dermpath Diagnostic, Pompano Beach, Florida, USA
e Unidad Inmunología, Hospital Carlos G. Durand, Buenos Aires, Argentina
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Garrahan, Buenos Aires, Argentina" "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Dermatología Pediátrica U.B.A., Servicio de Dermatología, Hospital de Pediatría Dr. Prof. Juan P. Garrahan, Buenos Aires, Argentina" "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Servicio de Hemato-Oncología, Hospital de Pediatría Dr. Prof. Juan P. Garrahan, Buenos Aires, Argentina" "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] 3 => array:3 [ "entidad" => "Institute for Immunofluorescence Dermpath Diagnostic, Pompano Beach, Florida, USA" "etiqueta" => "<span class="elsevierStyleSup">d</span>" "identificador" => "aff0020" ] 4 => array:3 [ "entidad" => "Unidad Inmunología, Hospital Carlos G. Durand, Buenos Aires, Argentina" "etiqueta" => "<span class="elsevierStyleSup">e</span>" "identificador" => "aff0025" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Pénfigo paraneoplásico/síndrome multiorgánico autoinmune paraneoplásico. Presentación de dos casos en la edad infantil. Revisión de la literatura" ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2010-01-26" "fechaAceptado" => "2010-06-28" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Key words" "identificador" => "xpalclavsec84824" "palabras" => array:5 [ 0 => "Paraneoplastic pemphigus" 1 => "Castleman disease" 2 => "Hodgkin lymphoma" 3 => "Bronchiolitis obliterans" 4 => "Children" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec84825" "palabras" => array:5 [ 0 => "Pénfigo paraneoplásico" 1 => "Enfermedad de Castleman" 2 => "Linfoma de Hodgkin" 3 => "Bronquiolitis obliterante" 4 => "Edad infantil" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Paraneoplastic pemphigus is an autoimmune blistering disease associated with an occult or previously diagnosed tumor. Its clinical, histological, and immunological features have been clearly defined. It is characterized by the presence of polymorphic skin lesions and by erosions of the oral and genital mucosas that are refractory to conventional treatments. The histology can be variable and includes acantholysis or lichenoid dermatitis. Circulating autoantibodies are a constant feature and confirm the diagnosis. We describe 2 girls with paraneoplastic pemphigus associated with Hodgkin lymphoma in one and Castleman disease in the other. Both children had oral and genital lesions that did not respond to conventional treatments. Biopsy revealed acantholysis in one and a lichenoid reaction in the other, and immunoassays confirmed the diagnosis. Chemotherapeutic treatment of the underlying disease was performed in both cases, together with high-dose corticosteroids for the skin and mucosal lesions. Both patients died due to respiratory failure. We suggest that paraneoplastic pemphigus, although rare in childhood and adolescence, should be included in the differential diagnosis of periorificial erosive dermatitis; this may assist in the detection of an occult neoplasm.</p>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">El pénfigo paraneoplásico es una enfermedad ampollosa autoinmune asociada a una neoplasia oculta o previamente diagnosticada, con manifestaciones clínicas, histológicas e inmunológicas bien definidas. Se caracteriza por erosiones de la mucosa orogenital refractarias a los tratamientos convencionales y la presencia de lesiones cutáneas polimorfas. La histología puede ser variada, con presencia de acantólisis o dermatitis liquenoide. La presencia de anticuerpos circulantes es un hallazgo constante que confirma el diagnóstico. Presentamos dos niñas con pénfigo paraneoplásico asociado a linfoma de Hodgkin y enfermedad de Castleman respectivamente. Presentaban compromiso orogenital refractario a tratamientos convencionales. En la histopatología se observó acantólisis y reacción liquenoide respectivamente. Los estudios inmunológicos confirmaron el diagnóstico. Ambas realizaron tratamiento quimioterápico para su enfermedad de base conjuntamente con altas dosis de corticosteroides para sus lesiones cutáneo-mucosas falleciendo por fallo respiratorio. Es de nuestro interés destacar que si bien es infrecuente la presencia de pénfigo paraneoplásico en la edad infantil y en la adolescencia, debe ser tenido en cuenta entre los diagnósticos diferenciales de las dermatosis erosivas periorificiales facilitando, de esta manera, el hallazgo de una neoplasia oculta.</p>" ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:15 [ 0 => array:3 [ "identificador" => "bib1" "etiqueta" => "1." "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Paraneoplastic pemphigus in children and adolescents" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "D. Mimouni" 1 => "G.J. Anhalt" 2 => "Z. Lazarova" 3 => "S. Aho" 4 => "S. Kazerounian" 5 => "D.J. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 4 | 5 | 9 |
| 2024 Octubre | 61 | 47 | 108 |
| 2024 Septiembre | 49 | 29 | 78 |
| 2024 Agosto | 65 | 63 | 128 |
| 2024 Julio | 53 | 40 | 93 |
| 2024 Junio | 67 | 22 | 89 |
| 2024 Mayo | 50 | 33 | 83 |
| 2024 Abril | 48 | 32 | 80 |
| 2024 Marzo | 55 | 47 | 102 |
| 2024 Febrero | 55 | 33 | 88 |
| 2024 Enero | 38 | 42 | 80 |
| 2023 Diciembre | 39 | 41 | 80 |
| 2023 Noviembre | 41 | 57 | 98 |
| 2023 Octubre | 35 | 27 | 62 |
| 2023 Septiembre | 37 | 30 | 67 |
| 2023 Agosto | 24 | 20 | 44 |
| 2023 Julio | 35 | 51 | 86 |
| 2023 Junio | 40 | 36 | 76 |
| 2023 Mayo | 39 | 39 | 78 |
| 2023 Abril | 52 | 28 | 80 |
| 2023 Marzo | 57 | 42 | 99 |
| 2023 Febrero | 41 | 32 | 73 |
| 2023 Enero | 24 | 53 | 77 |
| 2022 Diciembre | 33 | 60 | 93 |
| 2022 Noviembre | 19 | 41 | 60 |
| 2022 Octubre | 18 | 30 | 48 |
| 2022 Septiembre | 21 | 49 | 70 |
| 2022 Agosto | 21 | 34 | 55 |
| 2022 Julio | 21 | 36 | 57 |
| 2022 Junio | 16 | 27 | 43 |
| 2022 Mayo | 29 | 62 | 91 |
| 2022 Abril | 27 | 54 | 81 |
| 2022 Marzo | 36 | 71 | 107 |
| 2022 Febrero | 28 | 38 | 66 |
| 2022 Enero | 33 | 69 | 102 |
| 2021 Diciembre | 21 | 57 | 78 |
| 2021 Noviembre | 34 | 67 | 101 |
| 2021 Octubre | 37 | 70 | 107 |
| 2021 Septiembre | 39 | 55 | 94 |
| 2021 Agosto | 34 | 41 | 75 |
| 2021 Julio | 35 | 39 | 74 |
| 2021 Junio | 20 | 37 | 57 |
| 2021 Mayo | 23 | 29 | 52 |
| 2021 Abril | 36 | 85 | 121 |
| 2021 Marzo | 64 | 18 | 82 |
| 2021 Febrero | 47 | 36 | 83 |
| 2021 Enero | 34 | 24 | 58 |
| 2020 Diciembre | 36 | 20 | 56 |
| 2020 Noviembre | 16 | 26 | 42 |
| 2020 Octubre | 21 | 17 | 38 |
| 2020 Septiembre | 21 | 20 | 41 |
| 2020 Agosto | 22 | 45 | 67 |
| 2020 Julio | 32 | 23 | 55 |
| 2020 Junio | 18 | 29 | 47 |
| 2020 Mayo | 12 | 26 | 38 |
| 2020 Abril | 15 | 19 | 34 |
| 2020 Marzo | 19 | 37 | 56 |
| 2020 Febrero | 0 | 12 | 12 |
| 2020 Enero | 0 | 14 | 14 |
| 2019 Diciembre | 0 | 19 | 19 |
| 2019 Noviembre | 0 | 7 | 7 |
| 2019 Octubre | 0 | 4 | 4 |
| 2019 Septiembre | 0 | 3 | 3 |
| 2019 Agosto | 0 | 1 | 1 |
| 2019 Julio | 0 | 6 | 6 |
| 2019 Junio | 0 | 3 | 3 |
| 2019 Mayo | 0 | 48 | 48 |
| 2019 Abril | 0 | 8 | 8 |
| 2019 Marzo | 0 | 6 | 6 |
| 2018 Febrero | 9 | 6 | 15 |
| 2018 Enero | 13 | 6 | 19 |
| 2017 Diciembre | 11 | 9 | 20 |
| 2017 Noviembre | 15 | 3 | 18 |
| 2017 Octubre | 15 | 8 | 23 |
| 2017 Septiembre | 11 | 12 | 23 |
| 2017 Agosto | 11 | 14 | 25 |
| 2017 Julio | 12 | 2 | 14 |
| 2017 Junio | 19 | 49 | 68 |
| 2017 Mayo | 15 | 22 | 37 |
| 2017 Abril | 15 | 11 | 26 |
| 2017 Marzo | 11 | 64 | 75 |
| 2017 Febrero | 11 | 8 | 19 |
| 2017 Enero | 14 | 5 | 19 |
| 2016 Diciembre | 28 | 7 | 35 |
| 2016 Noviembre | 27 | 10 | 37 |
| 2016 Octubre | 26 | 20 | 46 |
| 2016 Septiembre | 24 | 12 | 36 |
| 2016 Agosto | 38 | 10 | 48 |
| 2016 Julio | 30 | 12 | 42 |
| 2016 Junio | 11 | 10 | 21 |
| 2016 Mayo | 3 | 8 | 11 |
| 2016 Abril | 4 | 13 | 17 |
| 2016 Marzo | 3 | 1 | 4 |
| 2016 Febrero | 11 | 2 | 13 |
| 2016 Enero | 10 | 2 | 12 |
| 2015 Diciembre | 10 | 0 | 10 |
| 2015 Noviembre | 15 | 0 | 15 |
| 2015 Octubre | 7 | 5 | 12 |
| 2015 Septiembre | 11 | 3 | 14 |
| 2015 Agosto | 9 | 5 | 14 |
| 2015 Julio | 25 | 91 | 116 |
| 2015 Junio | 26 | 15 | 41 |
| 2015 Mayo | 68 | 24 | 92 |
| 2015 Abril | 37 | 23 | 60 |
| 2015 Marzo | 42 | 22 | 64 |
| 2015 Febrero | 32 | 9 | 41 |
| 2015 Enero | 29 | 19 | 48 |
| 2014 Diciembre | 26 | 12 | 38 |
| 2014 Noviembre | 20 | 10 | 30 |
| 2014 Octubre | 29 | 20 | 49 |
| 2014 Septiembre | 32 | 20 | 52 |
| 2014 Agosto | 30 | 25 | 55 |
| 2014 Julio | 43 | 26 | 69 |
| 2014 Junio | 40 | 17 | 57 |
| 2014 Mayo | 45 | 16 | 61 |
| 2014 Abril | 36 | 12 | 48 |
| 2014 Marzo | 57 | 19 | 76 |
| 2014 Febrero | 27 | 14 | 41 |
| 2014 Enero | 28 | 19 | 47 |
| 2013 Diciembre | 44 | 22 | 66 |
| 2013 Noviembre | 24 | 21 | 45 |
| 2013 Octubre | 24 | 22 | 46 |
| 2013 Septiembre | 35 | 26 | 61 |
| 2013 Agosto | 38 | 29 | 67 |
| 2013 Julio | 41 | 32 | 73 |
| 2013 Junio | 30 | 34 | 64 |
| 2013 Mayo | 24 | 25 | 49 |
| 2013 Abril | 37 | 35 | 72 |
| 2013 Marzo | 41 | 42 | 83 |
| 2013 Febrero | 15 | 21 | 36 |
| 2013 Enero | 2 | 2 | 4 |
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