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
Corresponding author
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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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 3 | 5 | 8 |
| 2024 October | 61 | 47 | 108 |
| 2024 September | 49 | 29 | 78 |
| 2024 August | 65 | 63 | 128 |
| 2024 July | 53 | 40 | 93 |
| 2024 June | 67 | 22 | 89 |
| 2024 May | 50 | 33 | 83 |
| 2024 April | 48 | 32 | 80 |
| 2024 March | 55 | 47 | 102 |
| 2024 February | 55 | 33 | 88 |
| 2024 January | 38 | 42 | 80 |
| 2023 December | 39 | 41 | 80 |
| 2023 November | 41 | 57 | 98 |
| 2023 October | 35 | 27 | 62 |
| 2023 September | 37 | 30 | 67 |
| 2023 August | 24 | 20 | 44 |
| 2023 July | 35 | 51 | 86 |
| 2023 June | 40 | 36 | 76 |
| 2023 May | 39 | 39 | 78 |
| 2023 April | 52 | 28 | 80 |
| 2023 March | 57 | 42 | 99 |
| 2023 February | 41 | 32 | 73 |
| 2023 January | 24 | 53 | 77 |
| 2022 December | 33 | 60 | 93 |
| 2022 November | 19 | 41 | 60 |
| 2022 October | 18 | 30 | 48 |
| 2022 September | 21 | 49 | 70 |
| 2022 August | 21 | 34 | 55 |
| 2022 July | 21 | 36 | 57 |
| 2022 June | 16 | 27 | 43 |
| 2022 May | 29 | 62 | 91 |
| 2022 April | 27 | 54 | 81 |
| 2022 March | 36 | 71 | 107 |
| 2022 February | 28 | 38 | 66 |
| 2022 January | 33 | 69 | 102 |
| 2021 December | 21 | 57 | 78 |
| 2021 November | 34 | 67 | 101 |
| 2021 October | 37 | 70 | 107 |
| 2021 September | 39 | 55 | 94 |
| 2021 August | 34 | 41 | 75 |
| 2021 July | 35 | 39 | 74 |
| 2021 June | 20 | 37 | 57 |
| 2021 May | 23 | 29 | 52 |
| 2021 April | 36 | 85 | 121 |
| 2021 March | 64 | 18 | 82 |
| 2021 February | 47 | 36 | 83 |
| 2021 January | 34 | 24 | 58 |
| 2020 December | 36 | 20 | 56 |
| 2020 November | 16 | 26 | 42 |
| 2020 October | 21 | 17 | 38 |
| 2020 September | 21 | 20 | 41 |
| 2020 August | 22 | 45 | 67 |
| 2020 July | 32 | 23 | 55 |
| 2020 June | 18 | 29 | 47 |
| 2020 May | 12 | 26 | 38 |
| 2020 April | 15 | 19 | 34 |
| 2020 March | 19 | 37 | 56 |
| 2020 February | 0 | 12 | 12 |
| 2020 January | 0 | 14 | 14 |
| 2019 December | 0 | 19 | 19 |
| 2019 November | 0 | 7 | 7 |
| 2019 October | 0 | 4 | 4 |
| 2019 September | 0 | 3 | 3 |
| 2019 August | 0 | 1 | 1 |
| 2019 July | 0 | 6 | 6 |
| 2019 June | 0 | 3 | 3 |
| 2019 May | 0 | 48 | 48 |
| 2019 April | 0 | 8 | 8 |
| 2019 March | 0 | 6 | 6 |
| 2018 February | 9 | 6 | 15 |
| 2018 January | 13 | 6 | 19 |
| 2017 December | 11 | 9 | 20 |
| 2017 November | 15 | 3 | 18 |
| 2017 October | 15 | 8 | 23 |
| 2017 September | 11 | 12 | 23 |
| 2017 August | 11 | 14 | 25 |
| 2017 July | 12 | 2 | 14 |
| 2017 June | 19 | 49 | 68 |
| 2017 May | 15 | 22 | 37 |
| 2017 April | 15 | 11 | 26 |
| 2017 March | 11 | 64 | 75 |
| 2017 February | 11 | 8 | 19 |
| 2017 January | 14 | 5 | 19 |
| 2016 December | 28 | 7 | 35 |
| 2016 November | 27 | 10 | 37 |
| 2016 October | 26 | 20 | 46 |
| 2016 September | 24 | 12 | 36 |
| 2016 August | 38 | 10 | 48 |
| 2016 July | 30 | 12 | 42 |
| 2016 June | 11 | 10 | 21 |
| 2016 May | 3 | 8 | 11 |
| 2016 April | 4 | 13 | 17 |
| 2016 March | 3 | 1 | 4 |
| 2016 February | 11 | 2 | 13 |
| 2016 January | 10 | 2 | 12 |
| 2015 December | 10 | 0 | 10 |
| 2015 November | 15 | 0 | 15 |
| 2015 October | 7 | 5 | 12 |
| 2015 September | 11 | 3 | 14 |
| 2015 August | 9 | 5 | 14 |
| 2015 July | 25 | 91 | 116 |
| 2015 June | 26 | 15 | 41 |
| 2015 May | 68 | 24 | 92 |
| 2015 April | 37 | 23 | 60 |
| 2015 March | 42 | 22 | 64 |
| 2015 February | 32 | 9 | 41 |
| 2015 January | 29 | 19 | 48 |
| 2014 December | 26 | 12 | 38 |
| 2014 November | 20 | 10 | 30 |
| 2014 October | 29 | 20 | 49 |
| 2014 September | 32 | 20 | 52 |
| 2014 August | 30 | 25 | 55 |
| 2014 July | 43 | 26 | 69 |
| 2014 June | 40 | 17 | 57 |
| 2014 May | 45 | 16 | 61 |
| 2014 April | 36 | 12 | 48 |
| 2014 March | 57 | 19 | 76 |
| 2014 February | 27 | 14 | 41 |
| 2014 January | 28 | 19 | 47 |
| 2013 December | 44 | 22 | 66 |
| 2013 November | 24 | 21 | 45 |
| 2013 October | 24 | 22 | 46 |
| 2013 September | 35 | 26 | 61 |
| 2013 August | 38 | 29 | 67 |
| 2013 July | 41 | 32 | 73 |
| 2013 June | 30 | 34 | 64 |
| 2013 May | 24 | 25 | 49 |
| 2013 April | 37 | 35 | 72 |
| 2013 March | 41 | 42 | 83 |
| 2013 February | 15 | 21 | 36 |
| 2013 January | 2 | 2 | 4 |
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