Refractory pemphigus vulgaris and high‐intensity extracorporeal photopheresis: A case report
A 41‐year‐old man with oral pemphigus vulgaris (PV) presented to our clinic with a history of no response to numerous immunosuppressant agents and was referred for extracorporeal photopheresis (ECP) therapy. Although the patient underwent a high‐intensity ECP regimen for 5 months, which included two...
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| Published in | Photodermatology, photoimmunology & photomedicine Vol. 39; no. 3; pp. 296 - 300 |
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| Main Authors | , , , , , , , , , , , , |
| Format | Journal Article |
| Language | English |
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01.05.2023
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| Abstract | A 41‐year‐old man with oral pemphigus vulgaris (PV) presented to our clinic with a history of no response to numerous immunosuppressant agents and was referred for extracorporeal photopheresis (ECP) therapy. Although the patient underwent a high‐intensity ECP regimen for 5 months, which included two different photopheresis systems, his oral dysesthesia continued to interfere with oral intake, leading to continued weight loss and other adverse events. The intervention was associated with changes in several immune cell subpopulations without modifying the anti‐epidermal antibody titers, aligned with his poor clinical outcome. To the best of the authors' knowledge, this is the first report to examine immunophenotyping of a PV patient who was refractory to previous immunosuppression and recalcitrant to high‐intensity ECP therapy. |
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| AbstractList | A 41‐year‐old man with oral pemphigus vulgaris (PV) presented to our clinic with a history of no response to numerous immunosuppressant agents and was referred for extracorporeal photopheresis (ECP) therapy. Although the patient underwent a high‐intensity ECP regimen for 5 months, which included two different photopheresis systems, his oral dysesthesia continued to interfere with oral intake, leading to continued weight loss and other adverse events. The intervention was associated with changes in several immune cell subpopulations without modifying the anti‐epidermal antibody titers, aligned with his poor clinical outcome. To the best of the authors' knowledge, this is the first report to examine immunophenotyping of a PV patient who was refractory to previous immunosuppression and recalcitrant to high‐intensity ECP therapy. A 41-year-old man with oral pemphigus vulgaris (PV) presented to our clinic with a history of no response to numerous immunosuppressant agents and was referred for extracorporeal photopheresis (ECP) therapy. Although the patient underwent a high-intensity ECP regimen for five months, which included two different photopheresis systems, his oral dysesthesia continued to interfere with oral intake, leading to continued weight loss and other adverse events. The intervention was associated with changes in several immune cell subpopulations without modifying the anti-epidermal antibody titers, aligned with his poor clinical outcome. To the best of the authors' knowledge, this is the first report to examine immunophenotyping of a PV patient who was refractory to previous immunosuppression and recalcitrant to high-intensity ECP therapy. Abstract A 41‐year‐old man with oral pemphigus vulgaris (PV) presented to our clinic with a history of no response to numerous immunosuppressant agents and was referred for extracorporeal photopheresis (ECP) therapy. Although the patient underwent a high‐intensity ECP regimen for 5 months, which included two different photopheresis systems, his oral dysesthesia continued to interfere with oral intake, leading to continued weight loss and other adverse events. The intervention was associated with changes in several immune cell subpopulations without modifying the anti‐epidermal antibody titers, aligned with his poor clinical outcome. To the best of the authors' knowledge, this is the first report to examine immunophenotyping of a PV patient who was refractory to previous immunosuppression and recalcitrant to high‐intensity ECP therapy. |
| Author | Othman, Jekhsi Martinez, May Ann Brown, Sadaf Castillo‐Aleman, Yandy Marx Cato, Marlene Ponce Al Amin, Mariam Thabet Kistan, Jerusha Villegas‐Valverde, Carlos Agustin Al‐Kaabi, Fatema Mohammed Bencomo‐Hernandez, Antonio Alfonso Ventura‐Carmenate, Yendry Rivero‐Jimenez, Rene Antonio Victorino Roque, Jay Mary Rose |
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| Cites_doi | 10.1016/j.jaci.2020.11.008 10.1007/s10875-021-00991-y 10.1002/jca.21705 10.1016/s0190-9622(08)80560-9 10.1016/S0140-6736(19)31778-7 10.1038/s41590-022-01189-z 10.1111/cei.13232 10.1371/journal.pone.0134518 10.1111/bjd.15930 10.4103/ajts.AJTS_87_16 10.1038/s41590-020-0760-z |
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| Contributor | Mary Rose Victorino Roque, Jay Mohammed Al-Kaabi, Fatema Kistan, Jerusha Othman, Jekhsi Ann Martinez, May Brown, Sadaf Ponce Cato, Marlene Thabet Al Amin, Mariam |
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| Copyright | 2022 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd. This article is protected by copyright. All rights reserved. |
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| Keywords | case report Extracorporeal photopheresis mass cytometry autoimmune blistering disorders immunophenotyping pemphigus vulgaris |
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| References | 1989; 62 2022; 23 1992; 26 2020; 21 2017; 177 2019; 195 2019; 394 2021; 41 2021; 147 2017; 11 2019; 34 2015; 10 e_1_2_10_12_1 e_1_2_10_9_1 e_1_2_10_13_1 e_1_2_10_10_1 e_1_2_10_11_1 Rook AH (e_1_2_10_5_1) 1989; 62 e_1_2_10_2_1 e_1_2_10_4_1 e_1_2_10_3_1 e_1_2_10_6_1 e_1_2_10_8_1 e_1_2_10_7_1 |
| References_xml | – volume: 394 start-page: 882 issue: 10201 year: 2019 end-page: 894 article-title: Pemphigus publication-title: Lancet – volume: 34 start-page: 171 issue: 3 year: 2019 end-page: 354 article-title: Guidelines on the use of therapeutic apheresis in clinical practice – evidence‐based approach from the writing committee of the American Society for Apheresis: The eighth special issue publication-title: J Clin Apher – volume: 21 start-page: 1256 issue: 10 year: 2020 end-page: 1266 article-title: Early precursor T cells establish and propagate T cell exhaustion in chronic infection publication-title: Nat Immunol – volume: 11 start-page: 81 issue: 2 year: 2017 end-page: 86 article-title: Extracorporeal photopheresis: Review of technical aspects publication-title: Asian J Transfus Sci – volume: 23 start-page: 679 issue: 5 year: 2022 end-page: 691 article-title: CD66b‐CD64dimCD115‐cells in the human bone marrow represent neutrophil‐committed progenitors publication-title: Nat Immunol – volume: 10 start-page: e0134518 issue: 8 year: 2015 article-title: Modulation and apoptosis of neutrophil granulocytes by extracorporeal photopheresis in the treatment of chronic graft‐versus‐host disease publication-title: PLoS One – volume: 62 start-page: 647 issue: 6 year: 1989 end-page: 652 article-title: Treatment of autoimmune disease with extracorporeal photochemotherapy: pemphigus vulgaris – preliminary report publication-title: Yale J Biol Med – volume: 26 start-page: 779 issue: 5 Pt 1 year: 1992 end-page: 780 article-title: Pemphigus vulgaris treated with photopheresis publication-title: J Am Acad Dermatol – volume: 147 start-page: 2358 issue: 6 year: 2021 end-page: 2369 article-title: Immunophenotyping in pemphigus reveals a TH17/TFH17 cell‐dominated immune response promoting desmoglein1/3‐specific autoantibody production publication-title: J Allergy Clin Immunol – volume: 195 start-page: 369 issue: 3 year: 2019 end-page: 380 article-title: Monocytes show immunoregulatory capacity on CD4+ T cells in a human model of extracorporeal photopheresis publication-title: Clin Exp Immunol – volume: 41 start-page: 1016 issue: 5 year: 2021 end-page: 1030 article-title: Thymopoiesis, alterations in dendritic cells and tregs, and reduced T cell activation in successful extracorporeal photopheresis treatment of GVHD publication-title: J Clin Immunol – volume: 177 start-page: 1170 issue: 5 year: 2017 end-page: 1201 article-title: British Association of Dermatologists' guidelines for the management of pemphigus vulgaris 2017 publication-title: Br J Dermatol – ident: e_1_2_10_8_1 doi: 10.1016/j.jaci.2020.11.008 – ident: e_1_2_10_10_1 doi: 10.1007/s10875-021-00991-y – ident: e_1_2_10_3_1 doi: 10.1002/jca.21705 – ident: e_1_2_10_6_1 doi: 10.1016/s0190-9622(08)80560-9 – ident: e_1_2_10_2_1 doi: 10.1016/S0140-6736(19)31778-7 – ident: e_1_2_10_13_1 doi: 10.1038/s41590-022-01189-z – ident: e_1_2_10_11_1 doi: 10.1111/cei.13232 – volume: 62 start-page: 647 issue: 6 year: 1989 ident: e_1_2_10_5_1 article-title: Treatment of autoimmune disease with extracorporeal photochemotherapy: pemphigus vulgaris – preliminary report publication-title: Yale J Biol Med contributor: fullname: Rook AH – ident: e_1_2_10_12_1 doi: 10.1371/journal.pone.0134518 – ident: e_1_2_10_4_1 doi: 10.1111/bjd.15930 – ident: e_1_2_10_7_1 doi: 10.4103/ajts.AJTS_87_16 – ident: e_1_2_10_9_1 doi: 10.1038/s41590-020-0760-z |
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| Snippet | A 41‐year‐old man with oral pemphigus vulgaris (PV) presented to our clinic with a history of no response to numerous immunosuppressant agents and was referred... A 41-year-old man with oral pemphigus vulgaris (PV) presented to our clinic with a history of no response to numerous immunosuppressant agents and was referred... Abstract A 41‐year‐old man with oral pemphigus vulgaris (PV) presented to our clinic with a history of no response to numerous immunosuppressant agents and was... |
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| SubjectTerms | autoimmune blistering disorders case report extracorporeal photopheresis immunophenotyping mass cytometry pemphigus vulgaris |
| Title | Refractory pemphigus vulgaris and high‐intensity extracorporeal photopheresis: A case report |
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