Co-occurrence of myositis and neuropathy after anti-CD30 therapy in a late-adolescent Hodgkin lymphoma patient
Abstract Objective Immune-related adverse events (irAEs) are recognized in oncology, particularly with immune checkpoint inhibitors and other targeted therapies. Brentuximab Vedotin (BV), is an anti-CD30 antibody–drug conjugate- its association with immune-mediated myositis remains unexplored. We re...
Saved in:
| Main Authors: | , , , , , , , , , , , , |
|---|---|
| Format: | Article |
| Language: | English |
| Published: |
BMC
2025-06-01
|
| Series: | Acta Neuropathologica Communications |
| Subjects: | |
| Online Access: | https://doi.org/10.1186/s40478-025-02056-2 |
| Tags: |
Add Tag
No Tags, Be the first to tag this record!
|
| _version_ | 1849420445658382336 |
|---|---|
| author | Adela Della Marina Lydia Rink Andreas Hentschel Michael M. Schündeln Christopher Nelke Heike Kölbel Calvin Tucht Vera Dobelmann Tobias Ruck Tim Hagenacker Teresinha Evangelista Ulrike Schara-Schmidt Andreas Roos |
| author_facet | Adela Della Marina Lydia Rink Andreas Hentschel Michael M. Schündeln Christopher Nelke Heike Kölbel Calvin Tucht Vera Dobelmann Tobias Ruck Tim Hagenacker Teresinha Evangelista Ulrike Schara-Schmidt Andreas Roos |
| author_sort | Adela Della Marina |
| collection | DOAJ |
| description | Abstract Objective Immune-related adverse events (irAEs) are recognized in oncology, particularly with immune checkpoint inhibitors and other targeted therapies. Brentuximab Vedotin (BV), is an anti-CD30 antibody–drug conjugate- its association with immune-mediated myositis remains unexplored. We report a case of an adolescent with Hodgkin lymphoma (HL) who developed neuropathy and myositis following BV therapy. Materials & methods The diagnostic work-up included MRI as well as microscopic analyses (histology, electron microscopy, and immunostainings including CD30 and MxA) of a gastrocnemius muscle biopsy. Proteomic analysis was also performed on the same biopsy, and paradigmatic protein dysregulations were validated through immunostaining. Serum NCAM1 levels were measured using ELISA. Results The patient, diagnosed with HL at 15 years, developed neuropathy after Vincristine treatment and was switched to BV. During BV therapy, she experienced progressive muscle weakness and foot drop, leading to discontinuation. MRI confirmed myositis, and biopsy revealed neurogenic and inflammatory changes with complement deposition and mitochondrial dysfunction. Proteomics showed upregulation of inflammatory relevant proteins, with HPRT1 (749.43-fold) being the most increased one. Intravenous immunoglobulin (IVIG) therapy improved muscle strength. Discussion Myositis following BV therapy has not been reported. Findings suggest an immune-mediated mechanism with B-cell involvement. Given the response to IVIG, B-cell-directed therapies may be beneficial. This case identifies BV-induced myositis as a novel irAE. |
| format | Article |
| id | doaj-art-723c5bf8fcf042df9c85c3831a14b83d |
| institution | Kabale University |
| issn | 2051-5960 |
| language | English |
| publishDate | 2025-06-01 |
| publisher | BMC |
| record_format | Article |
| series | Acta Neuropathologica Communications |
| spelling | doaj-art-723c5bf8fcf042df9c85c3831a14b83d2025-08-20T03:31:45ZengBMCActa Neuropathologica Communications2051-59602025-06-011311810.1186/s40478-025-02056-2Co-occurrence of myositis and neuropathy after anti-CD30 therapy in a late-adolescent Hodgkin lymphoma patientAdela Della Marina0Lydia Rink1Andreas Hentschel2Michael M. Schündeln3Christopher Nelke4Heike Kölbel5Calvin Tucht6Vera Dobelmann7Tobias Ruck8Tim Hagenacker9Teresinha Evangelista10Ulrike Schara-Schmidt11Andreas Roos12Department of Pediatric Neurology, Center for Neuromuscular Disorders in Children and Adolescents, University Hospital Essen, University of Duisburg-EssenDivision of Pediatric Hematology and Oncology, Department of Pediatrics III, University Hospital Essen, University of Duisburg-EssenLeibniz-Institut für Analytische Wissenschaften -ISAS- E.V.Division of Pediatric Hematology and Oncology, Department of Pediatrics III, University Hospital Essen, University of Duisburg-EssenDepartment of Neurology, Medical Faculty and University Hospital Düsseldorf, Heinrich-Heine-University DüsseldorfDepartment of Pediatric Neurology, Center for Neuromuscular Disorders in Children and Adolescents, University Hospital Essen, University of Duisburg-EssenDepartment of Pediatric Neurology, Center for Neuromuscular Disorders in Children and Adolescents, University Hospital Essen, University of Duisburg-EssenDepartment of Neurology, Medical Faculty and University Hospital Düsseldorf, Heinrich-Heine-University DüsseldorfDepartment of Neurology, Medical Faculty and University Hospital Düsseldorf, Heinrich-Heine-University DüsseldorfDepartment of Neurology, Center for Translational Neuro- and Behavioral Sciences (C-TNBS), University Hospital Essen, University of Duisburg-EssenInstitute of Myology, Neuromuscular Morphology Unit & Neuromuscular Diseases Reference Center Nord/Est/Ile-de-France, Sorbonne Université, INSERM, GHU Pitié-SalpêtrièreDepartment of Pediatric Neurology, Center for Neuromuscular Disorders in Children and Adolescents, University Hospital Essen, University of Duisburg-EssenDepartment of Pediatric Neurology, Center for Neuromuscular Disorders in Children and Adolescents, University Hospital Essen, University of Duisburg-EssenAbstract Objective Immune-related adverse events (irAEs) are recognized in oncology, particularly with immune checkpoint inhibitors and other targeted therapies. Brentuximab Vedotin (BV), is an anti-CD30 antibody–drug conjugate- its association with immune-mediated myositis remains unexplored. We report a case of an adolescent with Hodgkin lymphoma (HL) who developed neuropathy and myositis following BV therapy. Materials & methods The diagnostic work-up included MRI as well as microscopic analyses (histology, electron microscopy, and immunostainings including CD30 and MxA) of a gastrocnemius muscle biopsy. Proteomic analysis was also performed on the same biopsy, and paradigmatic protein dysregulations were validated through immunostaining. Serum NCAM1 levels were measured using ELISA. Results The patient, diagnosed with HL at 15 years, developed neuropathy after Vincristine treatment and was switched to BV. During BV therapy, she experienced progressive muscle weakness and foot drop, leading to discontinuation. MRI confirmed myositis, and biopsy revealed neurogenic and inflammatory changes with complement deposition and mitochondrial dysfunction. Proteomics showed upregulation of inflammatory relevant proteins, with HPRT1 (749.43-fold) being the most increased one. Intravenous immunoglobulin (IVIG) therapy improved muscle strength. Discussion Myositis following BV therapy has not been reported. Findings suggest an immune-mediated mechanism with B-cell involvement. Given the response to IVIG, B-cell-directed therapies may be beneficial. This case identifies BV-induced myositis as a novel irAE.https://doi.org/10.1186/s40478-025-02056-2MyositisAnti-CD30Brentuximab vedotinChemotherapyHodgkin lymphoma |
| spellingShingle | Adela Della Marina Lydia Rink Andreas Hentschel Michael M. Schündeln Christopher Nelke Heike Kölbel Calvin Tucht Vera Dobelmann Tobias Ruck Tim Hagenacker Teresinha Evangelista Ulrike Schara-Schmidt Andreas Roos Co-occurrence of myositis and neuropathy after anti-CD30 therapy in a late-adolescent Hodgkin lymphoma patient Acta Neuropathologica Communications Myositis Anti-CD30 Brentuximab vedotin Chemotherapy Hodgkin lymphoma |
| title | Co-occurrence of myositis and neuropathy after anti-CD30 therapy in a late-adolescent Hodgkin lymphoma patient |
| title_full | Co-occurrence of myositis and neuropathy after anti-CD30 therapy in a late-adolescent Hodgkin lymphoma patient |
| title_fullStr | Co-occurrence of myositis and neuropathy after anti-CD30 therapy in a late-adolescent Hodgkin lymphoma patient |
| title_full_unstemmed | Co-occurrence of myositis and neuropathy after anti-CD30 therapy in a late-adolescent Hodgkin lymphoma patient |
| title_short | Co-occurrence of myositis and neuropathy after anti-CD30 therapy in a late-adolescent Hodgkin lymphoma patient |
| title_sort | co occurrence of myositis and neuropathy after anti cd30 therapy in a late adolescent hodgkin lymphoma patient |
| topic | Myositis Anti-CD30 Brentuximab vedotin Chemotherapy Hodgkin lymphoma |
| url | https://doi.org/10.1186/s40478-025-02056-2 |
| work_keys_str_mv | AT adeladellamarina cooccurrenceofmyositisandneuropathyafteranticd30therapyinalateadolescenthodgkinlymphomapatient AT lydiarink cooccurrenceofmyositisandneuropathyafteranticd30therapyinalateadolescenthodgkinlymphomapatient AT andreashentschel cooccurrenceofmyositisandneuropathyafteranticd30therapyinalateadolescenthodgkinlymphomapatient AT michaelmschundeln cooccurrenceofmyositisandneuropathyafteranticd30therapyinalateadolescenthodgkinlymphomapatient AT christophernelke cooccurrenceofmyositisandneuropathyafteranticd30therapyinalateadolescenthodgkinlymphomapatient AT heikekolbel cooccurrenceofmyositisandneuropathyafteranticd30therapyinalateadolescenthodgkinlymphomapatient AT calvintucht cooccurrenceofmyositisandneuropathyafteranticd30therapyinalateadolescenthodgkinlymphomapatient AT veradobelmann cooccurrenceofmyositisandneuropathyafteranticd30therapyinalateadolescenthodgkinlymphomapatient AT tobiasruck cooccurrenceofmyositisandneuropathyafteranticd30therapyinalateadolescenthodgkinlymphomapatient AT timhagenacker cooccurrenceofmyositisandneuropathyafteranticd30therapyinalateadolescenthodgkinlymphomapatient AT teresinhaevangelista cooccurrenceofmyositisandneuropathyafteranticd30therapyinalateadolescenthodgkinlymphomapatient AT ulrikescharaschmidt cooccurrenceofmyositisandneuropathyafteranticd30therapyinalateadolescenthodgkinlymphomapatient AT andreasroos cooccurrenceofmyositisandneuropathyafteranticd30therapyinalateadolescenthodgkinlymphomapatient |