A Multicentric Castleman Disease Associated with Mixed Warm and Cold Antibody-Mediated AHA Responsive to Siltuximab
© 2023 S. Karger AG, Basel..
Castleman disease is non-clonal lymphoproliferative disorders defined by hypertrophy of lymph nodes. The multicentric form (MCD), in which multiple lymph node stations are involved, is not associated with HHV8 infection, but considered idiopathic, although IL-6 appears to play a central role in its pathogenesis. Here, we report the case of a patient who presented with mixed autoimmune hemolytic anemia (AIHA) and adenopathy that was very challenging to diagnose due to very low values of hemoglobin and refractoriness of obtaining any improvement of AIHA with standard first and second lines of therapy (steroids, rituximab, immunoglobulin, erythropoietin, and cyclosporine). When we safely proceeded to lymph node biopsy, a diagnosis of MCD was established. This permitted the treatment with siltuximab, an anti-IL-6 monoclonal antibody. After only 1 week, hemoglobin raised and he was discharged. After 1 year, he was still in remission. This case underlines the challenges in diagnosis of MCD, and the first case of response to siltuximab after the failure of rituximab to relieve mixed AIHA.
| Medienart: |
E-Artikel |
|---|
| Erscheinungsjahr: |
2024 |
|---|---|
| Erschienen: |
2024 |
| Enthalten in: |
Zur Gesamtaufnahme - volume:69 |
|---|---|
| Enthalten in: |
Chemotherapy - 69(2024), 1 vom: 27., Seite 35-39 |
| Sprache: |
Englisch |
|---|
| Beteiligte Personen: |
Plano, Federica [VerfasserIn] |
|---|
| Links: |
|---|
| Themen: |
4F4X42SYQ6 |
|---|
| Anmerkungen: |
Date Completed 28.02.2024 Date Revised 28.02.2024 published: Print-Electronic Citation Status MEDLINE |
|---|
| doi: |
10.1159/000533235 |
|---|
| funding: |
|
|---|---|
| Förderinstitution / Projekttitel: |
|
| PPN (Katalog-ID): |
NLM361326114 |
|---|
| LEADER | 01000caa a22002652 4500 | ||
|---|---|---|---|
| 001 | NLM361326114 | ||
| 003 | DE-627 | ||
| 005 | 20240229170235.0 | ||
| 007 | cr uuu---uuuuu | ||
| 008 | 231226s2024 xx |||||o 00| ||eng c | ||
| 024 | 7 | |a 10.1159/000533235 |2 doi | |
| 028 | 5 | 2 | |a pubmed24n1310.xml |
| 035 | |a (DE-627)NLM361326114 | ||
| 035 | |a (NLM)37634492 | ||
| 040 | |a DE-627 |b ger |c DE-627 |e rakwb | ||
| 041 | |a eng | ||
| 100 | 1 | |a Plano, Federica |e verfasserin |4 aut | |
| 245 | 1 | 2 | |a A Multicentric Castleman Disease Associated with Mixed Warm and Cold Antibody-Mediated AHA Responsive to Siltuximab |
| 264 | 1 | |c 2024 | |
| 336 | |a Text |b txt |2 rdacontent | ||
| 337 | |a ƒaComputermedien |b c |2 rdamedia | ||
| 338 | |a ƒa Online-Ressource |b cr |2 rdacarrier | ||
| 500 | |a Date Completed 28.02.2024 | ||
| 500 | |a Date Revised 28.02.2024 | ||
| 500 | |a published: Print-Electronic | ||
| 500 | |a Citation Status MEDLINE | ||
| 520 | |a © 2023 S. Karger AG, Basel. | ||
| 520 | |a Castleman disease is non-clonal lymphoproliferative disorders defined by hypertrophy of lymph nodes. The multicentric form (MCD), in which multiple lymph node stations are involved, is not associated with HHV8 infection, but considered idiopathic, although IL-6 appears to play a central role in its pathogenesis. Here, we report the case of a patient who presented with mixed autoimmune hemolytic anemia (AIHA) and adenopathy that was very challenging to diagnose due to very low values of hemoglobin and refractoriness of obtaining any improvement of AIHA with standard first and second lines of therapy (steroids, rituximab, immunoglobulin, erythropoietin, and cyclosporine). When we safely proceeded to lymph node biopsy, a diagnosis of MCD was established. This permitted the treatment with siltuximab, an anti-IL-6 monoclonal antibody. After only 1 week, hemoglobin raised and he was discharged. After 1 year, he was still in remission. This case underlines the challenges in diagnosis of MCD, and the first case of response to siltuximab after the failure of rituximab to relieve mixed AIHA | ||
| 650 | 4 | |a Case Reports | |
| 650 | 4 | |a Autoimmune hemolytic anemia | |
| 650 | 4 | |a Castleman disease | |
| 650 | 4 | |a Rituximab | |
| 650 | 4 | |a Siltuximab | |
| 650 | 7 | |a siltuximab |2 NLM | |
| 650 | 7 | |a T4H8FMA7IM |2 NLM | |
| 650 | 7 | |a Rituximab |2 NLM | |
| 650 | 7 | |a 4F4X42SYQ6 |2 NLM | |
| 650 | 7 | |a Antibodies, Monoclonal |2 NLM | |
| 650 | 7 | |a Hemoglobins |2 NLM | |
| 700 | 1 | |a Mancuso, Salvatrice |e verfasserin |4 aut | |
| 700 | 1 | |a Camarda, Giulia Maria |e verfasserin |4 aut | |
| 700 | 1 | |a Butera, Maria Giulia |e verfasserin |4 aut | |
| 700 | 1 | |a Sucato, Giuseppe |e verfasserin |4 aut | |
| 700 | 1 | |a Alecci, Giuseppe |e verfasserin |4 aut | |
| 700 | 1 | |a Florena, Ada Maria |e verfasserin |4 aut | |
| 700 | 1 | |a Perrone, Salvatore |e verfasserin |4 aut | |
| 700 | 1 | |a Siragusa, Sergio Mario |e verfasserin |4 aut | |
| 773 | 0 | 8 | |i Enthalten in |t Chemotherapy |d 1965 |g 69(2024), 1 vom: 27., Seite 35-39 |w (DE-627)NLM000137006 |x 1421-9794 |7 nnns |
| 773 | 1 | 8 | |g volume:69 |g year:2024 |g number:1 |g day:27 |g pages:35-39 |
| 856 | 4 | 0 | |u http://dx.doi.org/10.1159/000533235 |3 Volltext |
| 912 | |a GBV_USEFLAG_A | ||
| 912 | |a GBV_NLM | ||
| 951 | |a AR | ||
| 952 | |d 69 |j 2024 |e 1 |b 27 |h 35-39 | ||