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1. Evaluation of Sézary cell marker expression and cell death behaviour upon in vitro treatment by flow cytometry in Sézary syndrome patients.

Author

Melchers, S., Roemer, M., Albrecht, J. D., Assaf, C., von Gugelberg, C., Guenova, E., Klemke, C.‐D., Moritz, R. K. C., Schlaak, M., Stadler, R., Wehkamp, U., Wobser, M., Albrecht, T., Goerdt, S., Schneider, S., and Nicolay, J. P.

Subjects
MONONUCLEAR leukocytes, CELL death, DIMETHYL fumarate, CELL populations, CELL analysis
Abstract

The diagnosis of Sézary syndrome (SS) relies on the identification of blood Sézary cells (SC) by different markers via flow cytometry. Treatment of SS is challenging since its pathogenesis is characterized by cell death resistance rather than hyperproliferation. In this study, we establish an integrated approach that considers both the expression of SC markers and sensitivity to cell death both spontaneously and upon in vitro treatment. Peripheral blood mononuclear cells were isolated from 20 SS patients and analysed for the SC markers CD7 and CD26 loss as well as CD158k and PD1 gain. The cells were then treated with different established and experimental therapies in vitro and cell death was measured. Spontaneous and therapeutically induced cell death were measured and correlated to cellular marker profiles. In the marker‐positive cells, spontaneous cell death sensitivity was reduced. Different treatments in vitro managed to specifically induce cell death in the putative CTCL cell populations. Interestingly, a repeated analysis after 3 months of treatment revealed the CTCL cell death sensitivity to be restored by therapy. We propose this novel integrated approach comprising the evaluation of SC marker expression and analysis of cell death sensitivity upon treatment that can also enable a better therapy stratification. [ABSTRACT FROM AUTHOR]

Published
2024
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5. Update on the development of EORTC/CLTF questionnaires to assess HRQOL in primary cutaneous T-cell and B-cell lymphomas: phase I

Author

Molloy, K, primary, Jonak, C, additional, Pokert, S, additional, Lehner-Baumgartner, E, additional, Quaglino, P, additional, Borro, A, additional, Guenova, E, additional, Blanchard, G, additional, Fassnacht, C, additional, Wehkamp, U, additional, Cozzio, A, additional, Zeller, T, additional, Oerlemans, S, additional, and Scarisbrick, J, additional

Published
2022
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6. Immunophenotypic identification and characterization of CTCL tumor cells in blood using standardized flow cytometry: a European multicenter study

Author

Najidh, S, primary, Van der Sluijs Gelling, AJ, additional, Cozzio, A, additional, Dobos, G, additional, Bagot, M, additional, Beylot-Barry, M, additional, Guenova, E, additional, Nicolay, J, additional, Lima, M, additional, Ortiz-Romero, PL, additional, Papadavid, E, additional, Pujol, R, additional, Quaglino, P, additional, Stadler, R, additional, Wehkamp, U, additional, Whittaker, S, additional, Van Dongen, JJM, additional, Montero, J Flores, additional, Almeida, J, additional, and Vermeer, MH, additional

Published
2022
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8. Should we be imaging lymph nodes at initial diagnosis of early‐stage mycosis fungoides? Results from the PROspective Cutaneous Lymphoma International Prognostic Index (PROCLIPI) international study*

Author

Hodak, E., Sherman, S., Papadavid, E., Bagot, M., Querfeld, C., Quaglino, P., Prince, H.M., Ortiz-Romero, P.L., Stadler, R., Knobler, R., Guenova, E., Estrach, T., Patsatsi, A., Leshem, Y.A., Prague-Naveh, H., Berti, E., Alberti-Violetti, S., Cowan, R., Jonak, C., Nikolaou, V., Mitteldorf, C., Akilov, O., Geskin, L., Matin, R., Beylot-Barry, M., Vakeva, L., Sanches, J.A., Servitje, O., Weatherhead, S., Wobser, M., Yoo, J., Bayne, M., Bates, A., Dunnill, G., Marschalko, M., Buschots, A.M., Wehkamp, U., Evison, F., Hong, E., Amitay-Laish, I., Stranzenbach, R., Vermeer, M., Willemze, R., Kempf, W., Cerroni, L., Whittaker, S., Kim, Y.H., Scarisbrick, J.J., Cutaneous Lymphoma Int Consortium, HUS Inflammation Center, Clinicum, and Helsinki University Hospital Area

Subjects
medicine.medical_specialty, Skin Neoplasms, EUROPEAN-ORGANIZATION, SOCIETY, Physical examination, Dermatology, GUIDELINES, CLASSIFICATION, Cutaneous lymphoma, Cutaneous patch, 030207 dermatology & venereal diseases, 03 medical and health sciences, Mycosis Fungoides, 0302 clinical medicine, International Prognostic Index, Biopsy, SEZARY-SYNDROME, medicine, Humans, COMPUTED-TOMOGRAPHY, Prospective Studies, Stage (cooking), RESPONSE CRITERIA, Neoplasm Staging, Body surface area, Mycosis fungoides, medicine.diagnostic_test, business.industry, CONSORTIUM, Prognosis, medicine.disease, 3121 General medicine, internal medicine and other clinical medicine, SURVIVAL, Lymph Nodes, Radiology, business, TASK-FORCE
Abstract

Background Early-stage mycosis fungoides (MF) includes involvement of dermatopathic lymph nodes (LNs) or early lymphomatous LNs. There is a lack of unanimity among current guidelines regarding the indications for initial staging imaging in early-stage presentation of MF in the absence of enlarged palpable LNs. Objectives To investigate how often imaging is performed in patients with early-stage presentation of MF, to assess the yield of LN imaging, and to determine what disease characteristics promoted imaging. Methods A review of clinicopathologically confirmed newly diagnosed patients with cutaneous patch/plaque (T1/T2) MF from PROspective Cutaneous Lymphoma International Prognostic Index (PROCLIPI) data. Results PROCLIPI enrolled 375 patients with stage T1/T2 MF: 304 with classical MF and 71 with folliculotropic MF. Imaging was performed in 169 patients (45%): 83 with computed tomography, 18 with positron emission tomography-computed tomography and 68 with ultrasound. Only nine of these (5%) had palpable enlarged (>= 15 mm) LNs, with an over-representation of plaques, irrespectively of the 10% body surface area cutoff that distinguishes T1 from T2. Folliculotropic MF was not more frequently imaged than classical MF. Radiologically enlarged LNs (>= 15 mm) were detected in 30 patients (18%); only seven had clinical lymphadenopathy. On multivariate analysis, plaque presentation was the sole parameter significantly associated with radiologically enlarged LNs. Imaging of only clinically enlarged LNs upstaged 4% of patients (seven of 169) to at least IIA, whereas nonselective imaging upstaged another 14% (24 of 169). LN biopsy, performed in eight of 30 patients, identified N3 (extensive lymphomatous involvement) in two and N1 (dermatopathic changes) in six. Conclusions Physical examination was a poor determinant of LN enlargement or involvement. Presence of plaques was associated with a significant increase in identification of enlarged or involved LNs in patients with early-stage presentation of MF, which may be important when deciding who to image. Imaging increases the detection rate of stage IIA MF, and identifies rare cases of extensive lymphomatous nodes, upstaging them to advanced-stage IVA2.

Published
2020
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10. Clinical, histopathological and prognostic features of primary cutaneous acral CD8+ T-cell lymphoma and other dermal CD8+ cutaneous lymphoproliferations: results of an EORTC Cutaneous Lymphoma Group workshop*

Author

Kempf, W., Petrella, T., Willemze, R., Jansen, P., Berti, E., Santucci, M., Geissinger, E., Cerroni, L., Maubec, E., Battistella, M., Goodlad, J., Guenova, E., Lappalainen, K., Ranki, A., Craig, P., Calonje, E., Martin, B., Whittaker, S., Oschlies, I., Wehkamp, U., Nicolay, J. P., Wobser, M., Scarisbruck, J., Pimpinelli, N., Stadler, R., Kerl French, K., Quaglino, P., Lin, J., Chen, L., Beer, M., Emanuel, P., Dalle, S., and Robson, A.

Subjects
Cutaneous, Skin Neoplasms, Lymphoma, CD8-Positive T-Lymphocytes, Humans, Prognosis, Retrospective Studies, Lymphoma, T-Cell, Cutaneous, T-Cell
Published
2022

14. Angiolymphoid hyperplasia with eosinophilia infiltrated by GATA3+ Th2 cells responding to dupilumab.

Author

Seidl, U., Bohne, A. S., Kaeding, M., Bauer, L., Weichenthal, M., Wehkamp, U., Hutloff, A., Schwarz, T., Gerdes, S., and Heine, G.

Subjects
TH2 cells, DUPILUMAB, PULMONARY eosinophilia, EOSINOPHILIA, HYPERPLASIA, CELL physiology
Abstract

Eosinophils are characteristic in ALHE with unknown function.[3] We depleted eosinophils with benralizumab, an antibody directed against the IL-5 receptor alpha chain.[4] However, eosinophils were not the driving force in ALHE in this patient. To further characterize the infiltrating lymphocytes, the ALHE skin biopsies were stained for the T cell marker CD4 (Figure 1c,e), the Th2-lineage marker GATA3[5] (Figure 1d,e) and the B cell lineage marker Pax5 (data not shown). [Extracted from the article]

Published
2023
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15. Should we be imaging lymph nodes at initial diagnosis of early-stage mycosis fungoides? Results from the PROspective Cutaneous Lymphoma International Prognostic Index (PROCLIPI) international study

Author

Hodak, E. Sherman, S. Papadavid, E. Bagot, M. Querfeld, C. Quaglino, P. Prince, H. M. Ortiz-Romero, P. L. and Stadler, R. Knobler, R. Guenova, E. Estrach, T. and Patsatsi, A. Leshem, Y. A. Prague-Naveh, H. Berti, E. and Alberti-Violetti, S. Cowan, R. Jonak, C. Nikolaou, V and Mitteldorf, C. Akilov, O. Geskin, L. Matin, R. and Beylot-Barry, M. Vakeva, L. Sanches, J. A. Servitje, O. and Weatherhead, S. Wobser, M. Yoo, J. Bayne, M. Bates, A. and Dunnill, G. Marschalko, M. Buschots, A. M. Wehkamp, U. and Evison, F. Hong, E. Amitay-Laish, I Stranzenbach, R. and Vermeer, M. Willemze, R. Kempf, W. Cerroni, L. and Whittaker, S. Kim, Y. H. Scarisbrick, J. J. Cutaneous Lymphoma Int Consortium

Abstract

Background Early-stage mycosis fungoides (MF) includes involvement of dermatopathic lymph nodes (LNs) or early lymphomatous LNs. There is a lack of unanimity among current guidelines regarding the indications for initial staging imaging in early-stage presentation of MF in the absence of enlarged palpable LNs. Objectives To investigate how often imaging is performed in patients with early-stage presentation of MF, to assess the yield of LN imaging, and to determine what disease characteristics promoted imaging. Methods A review of clinicopathologically confirmed newly diagnosed patients with cutaneous patch/plaque (T1/T2) MF from PROspective Cutaneous Lymphoma International Prognostic Index (PROCLIPI) data. Results PROCLIPI enrolled 375 patients with stage T1/T2 MF: 304 with classical MF and 71 with folliculotropic MF. Imaging was performed in 169 patients (45%): 83 with computed tomography, 18 with positron emission tomography-computed tomography and 68 with ultrasound. Only nine of these (5%) had palpable enlarged (>= 15 mm) LNs, with an over-representation of plaques, irrespectively of the 10% body surface area cutoff that distinguishes T1 from T2. Folliculotropic MF was not more frequently imaged than classical MF. Radiologically enlarged LNs (>= 15 mm) were detected in 30 patients (18%); only seven had clinical lymphadenopathy. On multivariate analysis, plaque presentation was the sole parameter significantly associated with radiologically enlarged LNs. Imaging of only clinically enlarged LNs upstaged 4% of patients (seven of 169) to at least IIA, whereas nonselective imaging upstaged another 14% (24 of 169). LN biopsy, performed in eight of 30 patients, identified N3 (extensive lymphomatous involvement) in two and N1 (dermatopathic changes) in six. Conclusions Physical examination was a poor determinant of LN enlargement or involvement. Presence of plaques was associated with a significant increase in identification of enlarged or involved LNs in patients with early-stage presentation of MF, which may be important when deciding who to image. Imaging increases the detection rate of stage IIA MF, and identifies rare cases of extensive lymphomatous nodes, upstaging them to advanced-stage IVA2.

Published
2021

16. Treatment of early-stage mycosis fungoides: results from the PROspective Cutaneous Lymphoma International Prognostic Index (PROCLIPI) study

Author

Quaglino, P. Prince, H. M. Cowan, R. Vermeer, M. and Papadavid, E. Bagot, M. Servitjie, O. Berti, E. Guenova, E. Stadler, R. Querfeld, C. Busschots, A. M. Hodak, E. and Patsatsi, A. Sanches, J. Maule, M. Yoo, J. Kevin, M. and Fava, P. Ribero, S. Zocchi, L. Rubatto, M. Fierro, M. T. Wehkamp, U. Marshalko, M. Mitteldorf, C. Akilov, O. Ortiz-Romero, P. Estrach, T. Vakeva, L. Enz, P. A. and Wobser, M. Bayne, M. Jonak, C. Rubeta, M. Forbes, A. and Bates, A. Battistella, M. Amel-Kashipaz, R. Vydianath, B. Combalia, A. Georgiou, E. Hauben, E. Hong, E. K. and Jost, M. Knobler, R. Amitay-Laish, I. Miyashiro, D. and Cury-Martins, J. Martinez, X. Muniesa, C. Prag-Naveh, H. and Stratigos, A. Nikolaou, V. Quint, K. Ram-Wolff, C. and Rieger, K. Stranzenbach, R. Szepesi, A. Alberti-Violetti, S. and Felicity, E. Cerroni, L. Kempf, W. Whittaker, S. and Willemze, R. Kim, Y. Scarisbrick, J. J.

Abstract

Background The PROspective Cutaneous Lymphoma International Prognostic Index (PROCLIPI) study is a prospective analysis of an international database. Here we examine front-line treatments and quality of life (QoL) in patients with newly diagnosed mycosis fungoides (MF). Objectives To identify (i) differences in first-line approaches according to tumour-nodes-metastasis-blood (TNMB) staging; (ii) parameters related to a first-line systemic approach and (iii) response rates and QoL measures. Methods In total, 395 newly diagnosed patients with early-stage MF (stage IA-IIA) were recruited from 41 centres in 17 countries between 1 January 2015 and 31 December 2018 following central clinicopathological review. Results The most common first-line therapy was skin-directed therapy (SDT) (322 cases, 81 center dot 5%), while a smaller percentage (44 cases, 11 center dot 1%) received systemic therapy. Expectant observation was used in 7 center dot 3%. In univariate analysis, the use of systemic therapy was significantly associated with higher clinical stage (IA, 6%; IB, 14%; IIA, 20%; IA-IB vs. IIA, P < 0 center dot 001), presence of plaques (T1a/T2a, 5%; T1b/T2b, 17%; P < 0 center dot 001), higher modified Severity Weighted Assessment Tool (> 10, 15%

Published
2021

19. Treatment of early-stage mycosis fungoides: results from the PROspective Cutaneous Lymphoma International Prognostic Index (PROCLIPI) study

Author

Quaglino, P, Prince, HM, Cowan, R, Vermeer, M, Papadavid, E, Bagot, M, Servitjie, O, Berti, E, Guenova, E, Stadler, R, Querfeld, C, Busschots, AM, Hodak, E, Patsatsi, A, Sanches, J, Maule, M, Yoo, J, Kevin, M, Fava, P, Ribero, S, Zocchi, L, Rubatto, M, Fierro, MT, Wehkamp, U, Marshalko, M, Mitteldorf, C, Akilov, O, Ortiz-Romero, P, Estrach, T, Vakeva, L, Enz, PA, Wobser, M, Bayne, M, Jonak, C, Rubeta, M, Forbes, A, Bates, A, Battistella, M, Amel-Kashipaz, R, Vydianath, B, Combalia, A, Georgiou, E, Hauben, E, Hong, EK, Jost, M, Knobler, R, Amitay-Laish, I, Miyashiro, D, Cury-Martins, J, Martinez, X, Muniesa, C, Prag-Naveh, H, Stratigos, A, Nikolaou, V, Quint, K, Ram-Wolff, C, Rieger, K, Stranzenbach, R, Szepesi, A, Alberti-Violetti, S, Felicity, E, Cerroni, L, Kempf, W, Whittaker, S, Willemze, R, Kim, Y, Scarisbrick, JJ, Quaglino, P, Prince, HM, Cowan, R, Vermeer, M, Papadavid, E, Bagot, M, Servitjie, O, Berti, E, Guenova, E, Stadler, R, Querfeld, C, Busschots, AM, Hodak, E, Patsatsi, A, Sanches, J, Maule, M, Yoo, J, Kevin, M, Fava, P, Ribero, S, Zocchi, L, Rubatto, M, Fierro, MT, Wehkamp, U, Marshalko, M, Mitteldorf, C, Akilov, O, Ortiz-Romero, P, Estrach, T, Vakeva, L, Enz, PA, Wobser, M, Bayne, M, Jonak, C, Rubeta, M, Forbes, A, Bates, A, Battistella, M, Amel-Kashipaz, R, Vydianath, B, Combalia, A, Georgiou, E, Hauben, E, Hong, EK, Jost, M, Knobler, R, Amitay-Laish, I, Miyashiro, D, Cury-Martins, J, Martinez, X, Muniesa, C, Prag-Naveh, H, Stratigos, A, Nikolaou, V, Quint, K, Ram-Wolff, C, Rieger, K, Stranzenbach, R, Szepesi, A, Alberti-Violetti, S, Felicity, E, Cerroni, L, Kempf, W, Whittaker, S, Willemze, R, Kim, Y, and Scarisbrick, JJ

Abstract

BACKGROUND: The PROspective Cutaneous Lymphoma International Prognostic Index (PROCLIPI) study is a prospective analysis of an international database. Here we examine front-line treatments and quality of life (QoL) in patients with newly diagnosed mycosis fungoides (MF). OBJECTIVES: To identify (i) differences in first-line approaches according to tumour-nodes-metastasis-blood (TNMB) staging; (ii) parameters related to a first-line systemic approach and (iii) response rates and QoL measures. METHODS: In total, 395 newly diagnosed patients with early-stage MF (stage IA-IIA) were recruited from 41 centres in 17 countries between 1 January 2015 and 31 December 2018 following central clinicopathological review. RESULTS: The most common first-line therapy was skin-directed therapy (SDT) (322 cases, 81·5%), while a smaller percentage (44 cases, 11·1%) received systemic therapy. Expectant observation was used in 7·3%. In univariate analysis, the use of systemic therapy was significantly associated with higher clinical stage (IA, 6%; IB, 14%; IIA, 20%; IA-IB vs. IIA, P < 0·001), presence of plaques (T1a/T2a, 5%; T1b/T2b, 17%; P < 0·001), higher modified Severity Weighted Assessment Tool (> 10, 15%; ≤ 10, 7%; P = 0·01) and folliculotropic MF (FMF) (24% vs. 12%, P = 0·001). Multivariate analysis demonstrated significant associations with the presence of plaques (T1b/T2b vs. T1a/T2a, odds ratio 3·07) and FMF (odds ratio 2·83). The overall response rate (ORR) to first-line SDT was 73%, while the ORR to first-line systemic treatments was lower (57%) (P = 0·027). Health-related QoL improved significantly both in patients with responsive disease and in those with stable disease. CONCLUSIONS: Disease characteristics such as presence of plaques and FMF influence physician treatment choices, and SDT was superior to systemic therapy even in patients with such disease characteristics. Consequently, future treatment guidelines for early-stage MF need to address these issues.

Published
2021

20. Should we be imaging lymph nodes at initial diagnosis of early-stage mycosis fungoides? Results from the PROspective Cutaneous Lymphoma International Prognostic Index (PROCLIPI) international study

Author

Hodak, E, Sherman, S, Papadavid, E, Bagot, M, Querfeld, C, Quaglino, P, Prince, HM, Ortiz-Romero, PL, Stadler, R, Knobler, R, Guenova, E, Estrach, T, Patsatsi, A, Leshem, YA, Prague-Naveh, H, Berti, E, Alberti-Violetti, S, Cowan, R, Jonak, C, Nikolaou, V, Mitteldorf, C, Akilov, O, Geskin, L, Matin, R, Beylot-Barry, M, Vakeva, L, Sanches, JA, Servitje, O, Weatherhead, S, Wobser, M, Yoo, J, Bayne, M, Bates, A, Dunnill, G, Marschalko, M, Buschots, AM, Wehkamp, U, Evison, F, Hong, E, Amitay-Laish, I, Stranzenbach, R, Vermeer, M, Willemze, R, Kempf, W, Cerroni, L, Whittaker, S, Kim, YH, Scarisbrick, JJ, Hodak, E, Sherman, S, Papadavid, E, Bagot, M, Querfeld, C, Quaglino, P, Prince, HM, Ortiz-Romero, PL, Stadler, R, Knobler, R, Guenova, E, Estrach, T, Patsatsi, A, Leshem, YA, Prague-Naveh, H, Berti, E, Alberti-Violetti, S, Cowan, R, Jonak, C, Nikolaou, V, Mitteldorf, C, Akilov, O, Geskin, L, Matin, R, Beylot-Barry, M, Vakeva, L, Sanches, JA, Servitje, O, Weatherhead, S, Wobser, M, Yoo, J, Bayne, M, Bates, A, Dunnill, G, Marschalko, M, Buschots, AM, Wehkamp, U, Evison, F, Hong, E, Amitay-Laish, I, Stranzenbach, R, Vermeer, M, Willemze, R, Kempf, W, Cerroni, L, Whittaker, S, Kim, YH, and Scarisbrick, JJ

Abstract

BACKGROUND: Early-stage mycosis fungoides (MF) includes involvement of dermatopathic lymph nodes (LNs) or early lymphomatous LNs. There is a lack of unanimity among current guidelines regarding the indications for initial staging imaging in early-stage presentation of MF in the absence of enlarged palpable LNs. OBJECTIVES: To investigate how often imaging is performed in patients with early-stage presentation of MF, to assess the yield of LN imaging, and to determine what disease characteristics promoted imaging. METHODS: A review of clinicopathologically confirmed newly diagnosed patients with cutaneous patch/plaque (T1/T2) MF from PROspective Cutaneous Lymphoma International Prognostic Index (PROCLIPI) data. RESULTS: PROCLIPI enrolled 375 patients with stage T1/T2 MF: 304 with classical MF and 71 with folliculotropic MF. Imaging was performed in 169 patients (45%): 83 with computed tomography, 18 with positron emission tomography-computed tomography and 68 with ultrasound. Only nine of these (5%) had palpable enlarged (≥ 15 mm) LNs, with an over-representation of plaques, irrespectively of the 10% body surface area cutoff that distinguishes T1 from T2. Folliculotropic MF was not more frequently imaged than classical MF. Radiologically enlarged LNs (≥ 15 mm) were detected in 30 patients (18%); only seven had clinical lymphadenopathy. On multivariate analysis, plaque presentation was the sole parameter significantly associated with radiologically enlarged LNs. Imaging of only clinically enlarged LNs upstaged 4% of patients (seven of 169) to at least IIA, whereas nonselective imaging upstaged another 14% (24 of 169). LN biopsy, performed in eight of 30 patients, identified N3 (extensive lymphomatous involvement) in two and N1 (dermatopathic changes) in six. CONCLUSIONS: Physical examination was a poor determinant of LN enlargement or involvement. Presence of plaques was associated with a significant increase in identification of enlarged or involved LNs in patients w

Published
2021

21. Treatment of early‐stage mycosis fungoides: results from the PROspective Cutaneous Lymphoma International Prognostic Index (PROCLIPI) study*

Author

Quaglino, P., primary, Prince, H.M., additional, Cowan, R., additional, Vermeer, M., additional, Papadavid, E., additional, Bagot, M., additional, Servitjie, O., additional, Berti, E., additional, Guenova, E., additional, Stadler, R., additional, Querfeld, C., additional, Busschots, A.M., additional, Hodak, E., additional, Patsatsi, A., additional, Sanches, J., additional, Maule, M., additional, Yoo, J., additional, Kevin, M., additional, Fava, P., additional, Ribero, S., additional, Zocchi, L., additional, Rubatto, M., additional, Fierro, M.T., additional, Wehkamp, U., additional, Marshalko, M., additional, Mitteldorf, C., additional, Akilov, O., additional, Ortiz‐Romero, P., additional, Estrach, T., additional, Vakeva, L., additional, Enz, P.A., additional, Wobser, M., additional, Bayne, M., additional, Jonak, C., additional, Rubeta, M., additional, Forbes, A., additional, Bates, A., additional, Battistella, M., additional, Amel‐Kashipaz, R., additional, Vydianath, B., additional, Combalia, A., additional, Georgiou, E., additional, Hauben, E., additional, Hong, E.K., additional, Jost, M., additional, Knobler, R., additional, Amitay‐Laish, I., additional, Miyashiro, D., additional, Cury‐Martins, J., additional, Martinez, X., additional, Muniesa, C., additional, Prag‐Naveh, H., additional, Stratigos, A., additional, Nikolaou, V., additional, Quint, K., additional, Ram‐Wolff, C., additional, Rieger, K., additional, Stranzenbach, R., additional, Szepesi, Á., additional, Alberti‐Violetti, S., additional, Felicity, E., additional, Cerroni, L., additional, Kempf, W., additional, Whittaker, S., additional, Willemze, R., additional, Kim, Y., additional, and Scarisbrick, J.J., additional

Published
2021
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22. Role of B-cells in Mycosis Fungoides

Author

Nielsen, P, primary, Eriksen, J, additional, Sørensen, M, additional, Wehkamp, U, additional, Lindahl, L, additional, Bzorek, M, additional, Iversen, L, additional, Woetman, A, additional, Ødum, N, additional, Litman, T, additional, and Gjerdrum, L, additional

Published
2021
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24. Clinical diversity and treatment approaches to blastic plasmacytoid dendritic cell neoplasm: a retrospective multicentre study

Author

Brüggen, M.‐C., primary, Valencak, J., additional, Stranzenbach, R., additional, Li, N., additional, Stadler, R., additional, Jonak, C., additional, Bauer, W., additional, Porkert, S., additional, Blaschke, A., additional, Meiss, F., additional, Nicolay, J. P., additional, Wehkamp, U., additional, Schlaak, M., additional, Nguyen, V.A., additional, Romani, N., additional, Cozzio, A., additional, Gayathri, N., additional, Dimitriou, F., additional, French, L. E., additional, Dummer, R., additional, and Guenova, E., additional

Published
2020
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25. 样肉芽肿和 Sézary 综合征患者生活质量研究

Author

Molloy, K., primary, Jonak, C., additional, Sherida, F.J., additional, Woei‐A‐Ji, H., additional, Guenova, E., additional, Busschots, A.M., additional, Bervoets, A., additional, Hauben, E., additional, Knobler, R., additional, Porkert, S., additional, Fassnacht, C., additional, Cowan, R., additional, Papadavid, E., additional, Beylot‐Barry, M., additional, Berti, E., additional, Alberti Violetti, S., additional, Estrach, T., additional, Matin, R., additional, Akilov, O., additional, Vakeva, L., additional, Prince, M., additional, Bates, A., additional, Bayne, M., additional, Wachsmuch, R., additional, Wehkamp, U., additional, Marschalko, M., additional, Servitje, O., additional, Turner, D., additional, Weatherhead, S., additional, Wobser, M., additional, Sanches, J.A., additional, McKay, P., additional, Klemke, D., additional, Peng, C., additional, Howles, A., additional, Yoo, J., additional, Evison, F., additional, and Scarisbrick, J., additional

Published
2020
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26. A study of quality of life in people with mycosis fungoides and Sézary syndrome

Author

Molloy, K., primary, Jonak, C., additional, Sherida, F.J., additional, Woei‐A‐Ji, H., additional, Guenova, E., additional, Busschots, A.M., additional, Bervoets, A., additional, Hauben, E., additional, Knobler, R., additional, Porkert, S., additional, Fassnacht, C., additional, Cowan, R., additional, Papadavid, E., additional, Beylot‐Barry, M., additional, Berti, E., additional, Alberti Violetti, S., additional, Estrach, T., additional, Matin, R., additional, Akilov, O., additional, Vakeva, L., additional, Prince, M., additional, Bates, A., additional, Bayne, M., additional, Wachsmuch, R., additional, Wehkamp, U., additional, Marschalko, M., additional, Servitje, O., additional, Turner, D., additional, Weatherhead, S., additional, Wobser, M., additional, Sanches, J.A., additional, McKay, P., additional, Klemke, D., additional, Peng, C., additional, Howles, A., additional, Yoo, J., additional, Evison, F., additional, and Scarisbrick, J., additional

Published
2020
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27. Clinical diversity and treatment approaches to blastic plasmacytoid dendritic cell neoplasm: a retrospective multicentre study

Author

Bruggen, M-C., Valencak, J., Stranzenbach, R., Li, N., Stadler, R., Jonak, C., Bauer, W., Porkert, S., Blaschke, A., Meiss, F., Nicolay, J. P., Wehkamp, U., Schlaak, M., Nguyen, V. A., Romani, N., Cozzio, A., Gayathri, N., Dimitriou, F., French, L. E., Dummer, R., Guenova, E., Bruggen, M-C., Valencak, J., Stranzenbach, R., Li, N., Stadler, R., Jonak, C., Bauer, W., Porkert, S., Blaschke, A., Meiss, F., Nicolay, J. P., Wehkamp, U., Schlaak, M., Nguyen, V. A., Romani, N., Cozzio, A., Gayathri, N., Dimitriou, F., French, L. E., Dummer, R., and Guenova, E.

Abstract

Background Blastic plasmacytoid dendritic cell neoplasm (BPDCN) is a rare, aggressive type of haematologic precursor malignancy primarily often manifesting in the skin. We sought to provide a thorough clinical characterization and report our experience on therapeutic approaches toBPDCN. Methods In the present multicentric retrospective study, we collected allBPDCNcases occurring between 05/1999 and 03/2018 in 10 secondary care centres of the German-Swiss-Austrian cutaneous lymphoma working group. Results A total of 37BPDCNcases were identified and included. Almost 90% of the patients had systemic manifestations (bone marrow, lymph nodes, peripheral blood) in addition to skin involvement. The latter presented with various types of cutaneous lesions: nodular (in more than 2/3) and bruise-like (in 1/3) skin lesions, but also maculopapular exanthema (in circa 1/6). Therapeutically, 22 patients received diverse combinations of chemotherapeutic regimens and/or radiotherapy. Despite initial responses, all of them ultimately relapsed and died from progressive disease. Eleven patients underwent haematopoietic stem cell transplantation (HSCT; autologousHSCTn = 3, allo-HSCTn = 8). The mortality rate amongHSCTpatients was only 33.33% with a median survival time of 60.5 months. Conclusion Our study demonstrates the clinical diversity of cutaneousBPDCNmanifestations and the positive development observed after the introduction ofHSCT.

Published
2020

31. An overall response in skin is associated with improved HRQoL in patients with MF/SS enrolled in the PROCLIPI study

Author

Molloy, K. Jonak, C. Sherida, F. J. Woei-A-Ji, H. and Guenova, E. Busschots, A. M. Bervoets, A. Hauben, E. and Knobler, R. Porkert, S. Fassnacht, C. Cowan, R. and Papadavid, E. Beylot-Barry, M. Berti, E. Violetti, S. Alberti Estrach, T. Matin, R. Akilov, O. Vakeva, L. and Prince, M. Bates, A. Bayne, M. Wachsmuch, R. Wehkamp, U. and Marschalko, M. Servitje, O. Turner, D. Weatherhead, S. and Wobser, M. Sanches, J. Antonio Mckay, P. Klemke, D. and Howles, A. Yoo, J. Evison, F. Scarisbrick, J.

Published
2019

32. T-cell receptor rearrangements in the skin and blood of patients in the PROCLIPI study: detection of clonal rearrangements in the skin (and blood) correlates with the B-class of MF and SS patients

Author

Wehkamp, U. Whittaker, S. Servitje, O. Berti, E. and Querfeld, C. Bagot, M. Stadler, R. Stranzenbach, R. and Marschalko, M. Busschots, A. -M. Jost, M. Sanches, J. and Ortiz-Romero, P. Estrach, T. Vakeva, L. Jonak, C. and Akilov, O. Hodak, E. Mitteldorf, C. Bates, A. and Beylot-Barry, M. Cowan, R. Pujol, R. Matin, R. and Papadavid, E. Quaglino, P. Vermeer, M. Kempf, W. Kim, Y. and Scarisbrick, J. PROCLIPI Investigators

Published
2019

35. Characteristics associated with significantly worse quality of life in mycosis fungoides/Sézary syndrome from the Prospective Cutaneous Lymphoma International Prognostic Index ( PROCLIPI ) study

Author

Molloy, K., primary, Jonak, C., additional, Woei‐A‐Jin, F.J.S.H., additional, Guenova, E., additional, Busschots, A.M., additional, Bervoets, A., additional, Hauben, E., additional, Knobler, R., additional, Porkert, S., additional, Fassnacht, C., additional, Cowan, R., additional, Papadavid, E., additional, Beylot‐Barry, M., additional, Berti, E., additional, Alberti Violetti, S., additional, Estrach, T., additional, Matin, R., additional, Akilov, O., additional, Vakeva, L., additional, Prince, M., additional, Bates, A., additional, Bayne, M., additional, Wachsmuch, R., additional, Wehkamp, U., additional, Marschalko, M., additional, Servitje, O., additional, Turner, D., additional, Weatherhead, S., additional, Wobser, M., additional, Sanches, J.A., additional, McKay, P., additional, Klemke, D., additional, Peng, C., additional, Howles, A., additional, Yoo, J., additional, Evison, F., additional, and Scarisbrick, J., additional

Published
2019
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36. The PROCLIPI international registry of early-stage mycosis fungoides identifies substantial diagnostic delay in most patients

Author

Scarisbrick, JJ, Quaglino, P, Prince, HM, Papadavid, E, Hodak, E, Bagot, M, Servitje, O, Berti, E, Ortiz-Romero, P, Stadler, R, Patsatsi, A, Knobler, R, Guenova, E, Child, F, Whittaker, S, Nikolaou, V, Tomasini, C, Amitay, I, Prag Naveh, H, Ram-Wolff, C, Battistella, M, Alberti-Violetti, S, Stranzenbach, R, Gargallo, V, Muniesa, C, Koletsa, T, Jonak, C, Porkert, S, Mitteldorf, C, Estrach, T, Combalia, A, Marschalko, M, Csomor, J, Szepesi, A, Cozzio, A, Dummer, R, Pimpinelli, N, Grandi, V, Beylot-Barry, M, Pham-Ledard, A, Wobser, M, Geissinger, E, Wehkamp, U, Weichenthal, M, Cowan, R, Parry, E, Harris, J, Wachsmuth, R, Turner, D, Bates, A, Healy, E, Trautinger, F, Latzka, J, Yoo, J, Vydianath, B, Amel-Kashipaz, R, Marinos, L, Oikonomidi, A, Stratigos, A, Vignon-Pennamen, M-D, Climent, F, Gonzalez-Barca, E, Georgiou, E, Senetta, R, Zinzani, P, Vakeva, L, Ranki, A, Busschots, A-M, Hauben, E, Bervoets, A, Woei-A-Jin, FJSH, Matin, R, Collins, G, Weatherhead, S, Frew, J, Bayne, M, Dunnill, G, McKay, P, Arumainathan, A, Azurdia, R, Benstead, K, Twigger, R, Rieger, K, Brown, R, Sanches, JA, Miyashiro, D, Akilov, O, McCann, S, Sahi, H, Damasco, FM, Querfeld, C, Folkes, A, Bur, C, Klemke, C-D, Enz, P, Pujol, R, Quint, K, Geskin, L, Hong, E, Evison, F, Vermeer, M, Cerroni, L, Kempf, W, Kim, Y, Willemze, R, Scarisbrick, JJ, Quaglino, P, Prince, HM, Papadavid, E, Hodak, E, Bagot, M, Servitje, O, Berti, E, Ortiz-Romero, P, Stadler, R, Patsatsi, A, Knobler, R, Guenova, E, Child, F, Whittaker, S, Nikolaou, V, Tomasini, C, Amitay, I, Prag Naveh, H, Ram-Wolff, C, Battistella, M, Alberti-Violetti, S, Stranzenbach, R, Gargallo, V, Muniesa, C, Koletsa, T, Jonak, C, Porkert, S, Mitteldorf, C, Estrach, T, Combalia, A, Marschalko, M, Csomor, J, Szepesi, A, Cozzio, A, Dummer, R, Pimpinelli, N, Grandi, V, Beylot-Barry, M, Pham-Ledard, A, Wobser, M, Geissinger, E, Wehkamp, U, Weichenthal, M, Cowan, R, Parry, E, Harris, J, Wachsmuth, R, Turner, D, Bates, A, Healy, E, Trautinger, F, Latzka, J, Yoo, J, Vydianath, B, Amel-Kashipaz, R, Marinos, L, Oikonomidi, A, Stratigos, A, Vignon-Pennamen, M-D, Climent, F, Gonzalez-Barca, E, Georgiou, E, Senetta, R, Zinzani, P, Vakeva, L, Ranki, A, Busschots, A-M, Hauben, E, Bervoets, A, Woei-A-Jin, FJSH, Matin, R, Collins, G, Weatherhead, S, Frew, J, Bayne, M, Dunnill, G, McKay, P, Arumainathan, A, Azurdia, R, Benstead, K, Twigger, R, Rieger, K, Brown, R, Sanches, JA, Miyashiro, D, Akilov, O, McCann, S, Sahi, H, Damasco, FM, Querfeld, C, Folkes, A, Bur, C, Klemke, C-D, Enz, P, Pujol, R, Quint, K, Geskin, L, Hong, E, Evison, F, Vermeer, M, Cerroni, L, Kempf, W, Kim, Y, and Willemze, R

Abstract

BACKGROUND: Survival in mycosis fungoides (MF) is varied and may be poor. The PROCLIPI (PROspective Cutaneous Lymphoma International Prognostic Index) study is a web-based data collection system for early-stage MF with legal data-sharing agreements permitting international collaboration in a rare cancer with complex pathology. Clinicopathological data must be 100% complete and in-built intelligence in the database system ensures accurate staging. OBJECTIVES: To develop a prognostic index for MF. METHODS: Predefined datasets for clinical, haematological, radiological, immunohistochemical, genotypic, treatment and quality of life are collected at first diagnosis of MF and annually to test against survival. Biobanked tissue samples are recorded within a Federated Biobank for translational studies. RESULTS: In total, 430 patients were enrolled from 29 centres in 15 countries spanning five continents. Altogether, 348 were confirmed as having early-stage MF at central review. The majority had classical MF (81·6%) with a CD4 phenotype (88·2%). Folliculotropic MF was diagnosed in 17·8%. Most presented with stage I (IA: 49·4%; IB: 42·8%), but 7·8% presented with enlarged lymph nodes (stage IIA). A diagnostic delay between first symptom development and initial diagnosis was frequent [85·6%; median delay 36 months (interquartile range 12-90)]. This highlights the difficulties in accurate diagnosis, which includes lack of a singular diagnostic test for MF. CONCLUSIONS: This confirmed early-stage MF cohort is being followed-up to identify prognostic factors, which may allow better management and improve survival by identifying patients at risk of disease progression. This study design is a useful model for collaboration in other rare diseases, especially where pathological diagnosis can be complex.

Published
2019

40. T-cell receptor rearrangements in the skin and blood of patients in the PROCLIPI study: detection of clonal rearrangements in the skin (and blood) correlates with the B-class of MF and SS patients

Author

Wehkamp, U., primary, Whittaker, S., additional, Servitje, O., additional, Berti, E., additional, Querfeld, C., additional, Bagot, M., additional, Stadler, R., additional, Stranzenbach, R., additional, Marschalko, M., additional, Busschots, A.-M., additional, Jost, M., additional, Sanches, J., additional, Ortiz-Romero, P., additional, Estrach, T., additional, Vakeva, L., additional, Jonak, C., additional, Akilov, O., additional, Hodak, E., additional, Mitteldorf, C., additional, Bates, A., additional, Beylot-Barry, M., additional, Cowan, R., additional, Pujol, R., additional, Matin, R., additional, Papadavid, E., additional, Quaglino, P., additional, Vermeer, M., additional, Kempf, W., additional, Kim, Y., additional, and Scarisbrick, J., additional

Published
2019
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41. An overall response in skin is associated with improved HRQoL in patients with MF/SS enrolled in the PROCLIPI study

Author

Molloy, K., primary, Jonak, C., additional, Sherida, F.J., additional, Woei-A-Ji, H., additional, Guenova, E., additional, Busschots, A.M., additional, Bervoets, A., additional, Hauben, E., additional, Knobler, R., additional, Porkert, S., additional, Fassnacht, C., additional, Cowan, R., additional, Papadavid, E., additional, Beylot-Barry, M., additional, Berti, E., additional, Alberti Violetti, S., additional, Estrach, T., additional, Matin, R., additional, Akilov, O., additional, Vakeva, L., additional, Prince, M., additional, Bates, A., additional, Bayne, M., additional, Wachsmuch, R., additional, Wehkamp, U., additional, Marschalko, M., additional, Servitje, O., additional, Turner, D., additional, Weatherhead, S., additional, Wobser, M., additional, Antonio Sanches, J., additional, McKay, P., additional, Klemke, D., additional, Howles, A., additional, Yoo, J., additional, Evison, F., additional, and Scarisbrick, J., additional

Published
2019
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44. The PROCLIPI international registry of early‐stage mycosis fungoides identifies substantial diagnostic delay in most patients

Author

Scarisbrick, J.J., primary, Quaglino, P., additional, Prince, H.M., additional, Papadavid, E., additional, Hodak, E., additional, Bagot, M., additional, Servitje, O., additional, Berti, E., additional, Ortiz‐Romero, P., additional, Stadler, R., additional, Patsatsi, A., additional, Knobler, R., additional, Guenova, E., additional, Child, F., additional, Whittaker, S., additional, Nikolaou, V., additional, Tomasini, C., additional, Amitay, I., additional, Prag Naveh, H., additional, Ram‐Wolff, C., additional, Battistella, M, additional, Alberti‐Violetti, S., additional, Stranzenbach, R., additional, Gargallo, V., additional, Muniesa, C., additional, Koletsa, T., additional, Jonak, C., additional, Porkert, S., additional, Mitteldorf, C., additional, Estrach, T., additional, Combalia, A., additional, Marschalko, M., additional, Csomor, J., additional, Szepesi, A., additional, Cozzio, A., additional, Dummer, R., additional, Pimpinelli, N., additional, Grandi, V., additional, Beylot‐Barry, M., additional, Pham‐Ledard, A., additional, Wobser, M., additional, Geissinger, E., additional, Wehkamp, U., additional, Weichenthal, M., additional, Cowan, R., additional, Parry, E., additional, Harris, J., additional, Wachsmuth, R., additional, Turner, D., additional, Bates, A., additional, Healy, E., additional, Trautinger, F., additional, Latzka, J., additional, Yoo, J., additional, Vydianath, B., additional, Amel‐Kashipaz, R., additional, Marinos, L., additional, Oikonomidi, A., additional, Stratigos, A., additional, Vignon‐Pennamen, M.‐D., additional, Battistella, M., additional, Climent, F., additional, Gonzalez‐Barca, E., additional, Georgiou, E., additional, Senetta, R., additional, Zinzani, P., additional, Vakeva, L., additional, Ranki, A., additional, Busschots, A.‐M., additional, Hauben, E., additional, Bervoets, A., additional, Woei‐A‐Jin, F.J.S.H., additional, Matin, R., additional, Collins, G., additional, Weatherhead, S., additional, Frew, J., additional, Bayne, M., additional, Dunnill, G., additional, McKay, P., additional, Arumainathan, A., additional, Azurdia, R., additional, Benstead, K., additional, Twigger, R., additional, Rieger, K., additional, Brown, R., additional, Sanches, J.A., additional, Miyashiro, D., additional, Akilov, O., additional, McCann, S., additional, Sahi, H., additional, Damasco, F.M., additional, Querfeld, C., additional, Folkes, A., additional, Bur, C., additional, Klemke, C.‐D., additional, Enz, P., additional, Pujol, R., additional, Quint, K., additional, Geskin, L., additional, Hong, E., additional, Evison, F., additional, Vermeer, M., additional, Cerroni, L., additional, Kempf, W., additional, Kim, Y., additional, and Willemze, R., additional

Published
2018
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47. 480 Atopic dermatitis is an IL-13/IL10 driven disease with high molecular heterogeneity compared to psoriasis

Author

Tsoi, L.C., primary, Rodrigues, E., additional, Degenhardt, F., additional, Baurecht, H., additional, Wehkamp, U., additional, Volks, N., additional, Szymczak, S., additional, Swindell, W., additional, Sarkar, M., additional, Raja, K., additional, Patrick, M.T., additional, Gao, Y., additional, Uppala, R., additional, Perez White, B.E., additional, Getsios, S., additional, Harms, P., additional, Maverakis, E., additional, Elder, J.T., additional, Franke, A., additional, Gudjonsson, J.E., additional, and Weidinger, S., additional

Published
2018
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50. 107 - An overall response in skin is associated with improved HRQoL in patients with MF/SS enrolled in the PROCLIPI study

Author

Molloy, K., Jonak, C., Sherida, F.J., Woei-A-Ji, H., Guenova, E., Busschots, A.M., Bervoets, A., Hauben, E., Knobler, R., Porkert, S., Fassnacht, C., Cowan, R., Papadavid, E., Beylot-Barry, M., Berti, E., Alberti Violetti, S., Estrach, T., Matin, R., Akilov, O., Vakeva, L., Prince, M., Bates, A., Bayne, M., Wachsmuch, R., Wehkamp, U., Marschalko, M., Servitje, O., Turner, D., Weatherhead, S., Wobser, M., Antonio Sanches, J., McKay, P., Klemke, D., Howles, A., Yoo, J., Evison, F., and Scarisbrick, J.

Published
2019
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