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3 results on '"Rudolph Maarten Egeler"'

1. The international dataset on the association between Langerhans Cell Histiocytosis and other malignancies

Author

Francesca Bagnasco, Stefanie-Yvonne Zimmermann, Rudolph Maarten Egeler, Vasanta Rao Nanduri, Bruna Cammarata, Jean Donadieu, Thomas Lehrnbecher, and Riccardo Haupt

Subjects
Cancer predisposition, Cancer genetics, Second neoplasms, Langerhans Cell Histiocytosis, Computer applications to medicine. Medical informatics, R858-859.7, Science (General), Q1-390
Abstract

This article presents the international dataset of cases in which the association of Langerhans cell Histiocytosis (LCH) with other malignancies (AM) was documented occurring at any age before, concurrently or after LCH. These data are mostly derived from previously published manuscripts or from completed case report forms (CRFs) by Histiocyte Society (HS) members or colleagues. In particular, for each case of LCH-AM, the database reports all the available data about clinical and biologic characteristics of the two tumors, as well about treatment and status at follow-up. The AM were categorized as: i) leukemias [acute lymphoblastic or myeloid leukemia (ALL and AML, respectively), other leukemias] and myeloproliferative disorders; ii) lymphomas [Hodgkin lymphoma (HL) and non-Hodgkin lymphomas (NHL)] and iii) solid tumors.A total of 270 LCH-AM cases were documented, of which 116 (43%) occurred among children. After stratification by age at LCH diagnosis, using 18 years as cut-off between children and adults, we here provide details on the clinical characteristics in terms of LCH system involvement and affected organs, as well on the temporal relationship between the LCH and AM diagnoses, including details on the AM malignancy types. In 19 cases the LCH and the corresponding AM occurred in a different age group.The data set is available for future studies in view of new insights of the genetic or environmental determinants of LCH and/or of treatment related subsequent neoplasms.

Published
2022
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2. Langerhans cell histiocytosis and associated malignancies: A retrospective analysis of 270 patients

Author

Francesca Bagnasco, Stefanie Yvonne Zimmermann, Rudolph Maarten Egeler, Vasanta Rao Nanduri, Bruna Cammarata, Jean Donadieu, Thomas Lehrnbecher, and Riccardo Haupt

Subjects
Adult, Histiocytosis, Langerhans-Cell, Leukemia, Myeloid, Acute, Cancer Research, Lymphoma, Oncology, Humans, Prospective Studies, Child, Retrospective Studies
Abstract

The frequency of Langerhans cell histiocytosis (LCH) and associated malignancies (AM) is greater than statistically expected. Here, we analyze LCH-AM co-occurrence in both children and adults.Between 1991 and 2015, data were collected by regular questionnaires to members of the Histiocyte Society and searches in PubMed and Abstract Books. Patients were grouped by age at LCH diagnosis (≤ andgt;18 years), and types and timing of AM occurrence were plotted with respect to the LCH diagnosis. For the statistical analysis, only the first AM were considered.A total of 285 LCH-AM in 270 patients were identified, 116 (43%) ≤ 18 years, and 154 (57%)gt;18 years. In childhood LCH-AM pairs, leukemias and myeloproliferative disorders (n = 58; 50.0%) prevailed over solid tumors (n = 43; 37.1%) and lymphoma (n = 15; 12.9%). In adults, solid tumors were reported in 61 patients (39.6%), lymphoma, and leukemias and myeloproliferative disorders in 56 (36.4%) and 37 (24.0%) patients, respectively. In most children, AM followed LCH (n = 69, 59.5%), whereas in adults, LCH and AM occurred concurrently in 69 patients (44.8%). In children, T-lineage acute lymphoblastic leukemia (ALL) and promyelocytic acute myeloid leukemia (AML) and retinoblastoma were over-represented and thyroid carcinoma in adults.The largest collection of data on LCH-AM to date clearly indicates inherent relationships between specific types of AM and LCH, which may be due to therapy effects, clonal evolution, and germ-line predisposition, respectively. Prospective thorough genetic analysis is warranted and will hopefully shed light on the association of LCH and second neoplasms.

Published
2022
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3. Multiple infusions of mesenchymal stromal cells induce sustained remission in children with steroid-refractory, grade III-IV acute graft-versus-host disease

Author

Rudolph Maarten Egeler, Helene Roelofs, Marco Zecca, Benedetta Contoli, Jaap Jan Zwaginga, Alice Bertaina, Giovanna Giorgiani, Katarina Le Blanc, Maria Ester Bernardo, Francesco Frassoni, M.A. Avanzini, Arjan C. Lankester, Lynne M. Ball, Cornelia M. Jol-van der Zijde, Willem E. Fibbe, Franco Locatelli, Antonella Conforti, Maarten J. D. van Tol, Ball, L. M., Bernardo, M. E., Roelofs, H., van Tol, M. J. D., Contoli, B., Zwaginga, J. J., Avanzini, M. A., Conforti, A., Bertaina, A., Giorgiani, G., Jol-van der Zijde, C. M., Zecca, M., Le Blanc, K., Frassoni, F., Egeler, R. M., Fibbe, W. E., Lankester, A. C., and Locatelli, F.

Subjects
Male, medicine.medical_specialty, Stromal cell, Adolescent, Mesenchymal stromal cells, Graft vs Host Disease, steroid-refractory acute graft-versus-host disease, Mesenchymal Stem Cell Transplantation, Gastroenterology, Cohort Studies, haematopoietic stem cell transplantation in children, Internal medicine, Acute graft versus host disease, medicine, Humans, Haematopoietic stem cell transplantation in children, Cumulative incidence, In patient, Child, Steroid-refractory acute graft-versus-host disease, business.industry, Transplantation-related mortality, Mesenchymal stem cell, Remission Induction, Infant, Mesenchymal Stem Cells, Hematology, Surgery, Settore MED/38 - PEDIATRIA GENERALE E SPECIALISTICA, Child, Preschool, Hematologic Neoplasms, Cohort, Acute Disease, Female, Steroids, Sustained remission, Neoplasm Grading, Steroid refractory, business, mesenchymal stromal cells, transplantation-related mortality
Abstract

Mesenchymal stromal cell (MSC) infusions have been reported to be effective in patients with steroid-refractory, acute graft-versus-host disease (aGvHD) but comprehensive data on paediatric patients are limited. We retrospectively analysed a cohort of 37 children (aged 3months-17years) treated with MSCs for steroid-refractory grade III-IV aGvHD. All patients but three received multiple MSC infusions. Complete response (CR) was observed in 24 children (65%), while 13 children had either partial (n=8) or no response (n=5). Cumulative incidence of transplantation-related mortality (TRM) in patients who did or did not achieve CR was 17% and 69%, respectively (P=0·001). After a median follow-up of 2·9years, overall survival (OS) was 37%; it was 65% vs. 0% in patients who did or did not achieve CR, respectively (P=0·001). The median time from starting steroids for GvHD treatment to first MSC infusion was 13d (range 5-85). Children treated between 5 and 12d after steroid initiation showed a trend for better OS (56%) and lower TRM (17%) as compared with patients receiving MSCs 13-85d after steroids (25% and 53%, respectively; P=0·22 and 0·06, respectively). Multiple MSC infusions are safe and effective for children with steroid-refractory aGvHD, especially when employed early in the disease course. © 2013 John Wiley & Sons Ltd.

Published
2013

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