Safety and efficacy of brentuximab vedotin as a treatment for lymphoproliferative disorders in primary immunodeficiencies

Thomas Pincez¹, Julie Bruneau², Laureline Berteloot³, Eve Piekarski⁴, Caroline Thomas⁵, Ambroise Marçais⁶, Amélie Trinquand⁷, Martin Castelle¹, Nicolas Garcelon⁸, Dominique Plantaz⁹, Morgane Cheminant⁶, Despina Moshous¹⁰, Thierry Jo Molina¹¹, Olivier Hermine⁶, Elizabeth Macintyre¹², Alain Fischer¹³, Stéphane Blanche¹⁰, Felipe Suarez⁶, Bénédicte Neven¹⁴

Affiliations

¹ Pediatric Hematology-Immunology and Rheumatology Department, Hôpital Necker-Enfants Malades, AP-HP, Paris.
² Pathology Department, Hôpital Necker-Enfants Malades, AP-HP, Paris; Paris University, Paris; INSERM UMR 1163, Institut Imagine, Paris.
³ Medical Imaging Department, Hôpital Necker-Enfants Malades, APHP, Paris.
⁴ Nuclear Medicine Department, Hôpital Bichat-Claude Bernard, AP-HP, Paris.
⁵ Pediatric Oncology-Hematology Department, Hôpital Enfant-Adolescent, CHU Nantes, Nantes.
⁶ Paris University, Paris; INSERM UMR 1163, Institut Imagine, Paris; Department of Hematology, Hôpital Necker-Enfants Malades, AP-HP, Paris.
⁷ Laboratory of Onco-Hematology, Hôpital Necker-Enfants Malades, AP-HP, Paris.
⁸ Paris University, Paris; INSERM UMR 1163, Institut Imagine, Paris.
⁹ Pediatric Hematology-Oncology Department, Grenoble University Hospital, Grenoble.
¹⁰ Pediatric Hematology-Immunology and Rheumatology Department, Hôpital Necker-Enfants Malades, AP-HP, Paris; Paris University, Paris; INSERM UMR 1163, Institut Imagine, Paris.
¹¹ Pathology Department, Hôpital Necker-Enfants Malades, AP-HP, Paris; Paris University, Paris.
¹² Paris University, Paris; Laboratory of Onco-Hematology, Hôpital Necker-Enfants Malades, AP-HP, Paris; INSERM UMR 1151, Institut Necker-Enfants Malades, Paris.
¹³ Pediatric Hematology-Immunology and Rheumatology Department, Hôpital Necker-Enfants Malades, AP-HP, Paris; Paris University, Paris; INSERM UMR 1163, Institut Imagine, Paris; Collège de France, Paris, France.
¹⁴ Pediatric Hematology-Immunology and Rheumatology Department, Hôpital Necker-Enfants Malades, AP-HP, Paris; Paris University, Paris; INSERM UMR 1163, Institut Imagine, Paris. benedicte.neven@aphp.fr.

PMID: 33054064
PMCID: PMC7556515
DOI: 10.3324/haematol.2019.230276

Safety and efficacy of brentuximab vedotin as a treatment for lymphoproliferative disorders in primary immunodeficiencies

Thomas Pincez et al. Haematologica. 2020.

. 2020 Sep 1;105(9):e461-464.

doi: 10.3324/haematol.2019.230276.

Authors

Affiliations

¹ Pediatric Hematology-Immunology and Rheumatology Department, Hôpital Necker-Enfants Malades, AP-HP, Paris.
² Pathology Department, Hôpital Necker-Enfants Malades, AP-HP, Paris; Paris University, Paris; INSERM UMR 1163, Institut Imagine, Paris.
³ Medical Imaging Department, Hôpital Necker-Enfants Malades, APHP, Paris.
⁴ Nuclear Medicine Department, Hôpital Bichat-Claude Bernard, AP-HP, Paris.
⁵ Pediatric Oncology-Hematology Department, Hôpital Enfant-Adolescent, CHU Nantes, Nantes.
⁶ Paris University, Paris; INSERM UMR 1163, Institut Imagine, Paris; Department of Hematology, Hôpital Necker-Enfants Malades, AP-HP, Paris.
⁷ Laboratory of Onco-Hematology, Hôpital Necker-Enfants Malades, AP-HP, Paris.
⁸ Paris University, Paris; INSERM UMR 1163, Institut Imagine, Paris.
⁹ Pediatric Hematology-Oncology Department, Grenoble University Hospital, Grenoble.
¹⁰ Pediatric Hematology-Immunology and Rheumatology Department, Hôpital Necker-Enfants Malades, AP-HP, Paris; Paris University, Paris; INSERM UMR 1163, Institut Imagine, Paris.
¹¹ Pathology Department, Hôpital Necker-Enfants Malades, AP-HP, Paris; Paris University, Paris.
¹² Paris University, Paris; Laboratory of Onco-Hematology, Hôpital Necker-Enfants Malades, AP-HP, Paris; INSERM UMR 1151, Institut Necker-Enfants Malades, Paris.
¹³ Pediatric Hematology-Immunology and Rheumatology Department, Hôpital Necker-Enfants Malades, AP-HP, Paris; Paris University, Paris; INSERM UMR 1163, Institut Imagine, Paris; Collège de France, Paris, France.
¹⁴ Pediatric Hematology-Immunology and Rheumatology Department, Hôpital Necker-Enfants Malades, AP-HP, Paris; Paris University, Paris; INSERM UMR 1163, Institut Imagine, Paris. benedicte.neven@aphp.fr.

PMID: 33054064
PMCID: PMC7556515
DOI: 10.3324/haematol.2019.230276

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Figures

**Figure 1.**
**Efficacy of brentuximab vedotin.** (Right) Swimmer plot of individual outcome. On the left, summary of clinical response, radiological response of target lesions according to Response Evaluation Criteria in Lymphoma criteria (% change in sum of diameters of target lesions) and metabolic uptake on [¹⁸F]2-fluoro- 2-deoxy-D-glucose positron emission tomography-computed tomography and polymerase chain reaction (PCR) Epstein Barr virus (EBV) log change for EBV-related B-cell lymphoproliferative disorder (LPD). In three cases, brentuximab vedotin (BV) was started while the LPD was in a partial response after two courses of COP (cyclophosphamide, vincristine and prednisone) for the LPD1 of P1, one injection of rituximab (375mg/m) and ten days of steroids for P2, and five injections of rituximab (375 mg/m) for P4. In two cases (P3 and P7), the LPD was refractory to rituximab. P1 presented a second LPD, or an immunogenetically evolved relapse, which was not associated with a loss of CD30 expression. The median time to an objective response was 2.6 months (range, 0.56-3.91). However, a clinical improvement was apparent in the first weeks after treatment. aHSCT: allogeneic hematopoietic stem cell transplantation.

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References

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Safety and efficacy of brentuximab vedotin as a treatment for lymphoproliferative disorders in primary immunodeficiencies

Affiliations

Safety and efficacy of brentuximab vedotin as a treatment for lymphoproliferative disorders in primary immunodeficiencies

Authors

Affiliations

Figures

References

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