Randall-type monoclonal immunoglobulin deposition disease: novel insights from a nationwide cohort study Article Swipe
Florent Joly
,
Camille Cohen
,
Vincent Javaugue
,
Sébastien Bender
,
Mohamed Belmouaz
,
Bertrand Arnulf
,
Bertrand Knebelmann
,
M. Nouvier
,
Vincent Audard
,
François Provôt
,
Viviane Gnemmi
,
Dominique Nochy
,
Jean Michel Goujon
,
Arnaud Jaccard
,
Guy Touchard
,
Jean Paul Fermand
,
Christophe Sirac
,
Frank Bridoux
·
YOU?
·
· 2018
· Open Access
·
· DOI: https://doi.org/10.1182/blood-2018-09-872028
YOU?
·
· 2018
· Open Access
·
· DOI: https://doi.org/10.1182/blood-2018-09-872028
Monoclonal immunoglobulin deposition disease (MIDD) is a rare complication of B-cell clonal disorders, defined by Congo red negative–deposits of monoclonal light chain (LCDD), heavy chain (HCDD), or both (LHCDD). MIDD is a systemic disorder with prominent renal involvement, but little attention has been paid to the description of extrarenal manifestations. Moreover, mechanisms of pathogenic immunoglobulin deposition and factors associated with renal and patient survival are ill defined. We retrospectively studied a nationwide cohort of 255 patients, with biopsy-proven LCDD (n = 212) (including pure LCDD [n = 154], LCDD with cast nephropathy (CN) [n = 58]), HCDD (n = 23), or LHCDD (n = 20). Hematological diagnosis was monoclonal gammopathy of renal significance in 64% and symptomatic myeloma in 34%. Renal presentation was acute kidney injury in patients with LCCD and CN, and chronic glomerular disease in the other types, 35% of whom had symptomatic extrarenal (mostly hepatic and cardiac) involvement. Sequencing of 18 pathogenic LC showed high isoelectric point values of variable domain complementarity determining regions, possibly accounting for tissue deposition. Among 169 patients who received chemotherapy (bortezomib-based in 58%), 67% achieved serum free light chain (FLC) response, including very good partial response (VGPR) or above in 52%. Renal response occurred in 62 patients (36%), all of whom had achieved hematological response. FLC response ≥ VGPR and absence of severe interstitial fibrosis were independent predictors of renal response. This study highlights an unexpected frequency of extrarenal manifestations in MIDD. Rapid diagnosis and achievement of deep FLC response are key factors of prognosis.
Related Topics
Concepts
Medicine
Internal medicine
Nephropathy
Multiple myeloma
Monoclonal gammopathy of undetermined significance
Gastroenterology
Renal biopsy
Monoclonal
Pathology
Immunology
Biopsy
Antibody
Monoclonal antibody
Endocrinology
Diabetes mellitus
Metadata
- Type
- article
- Language
- en
- Landing Page
- https://doi.org/10.1182/blood-2018-09-872028
- https://ashpublications.org/blood/article-pdf/133/6/576/1750586/blood872028.pdf
- OA Status
- bronze
- Cited By
- 113
- References
- 43
- Related Works
- 10
- OpenAlex ID
- https://openalex.org/W2905748745
All OpenAlex metadata
Raw OpenAlex JSON
- OpenAlex ID
-
https://openalex.org/W2905748745Canonical identifier for this work in OpenAlex
- DOI
-
https://doi.org/10.1182/blood-2018-09-872028Digital Object Identifier
- Title
-
Randall-type monoclonal immunoglobulin deposition disease: novel insights from a nationwide cohort studyWork title
- Type
-
articleOpenAlex work type
- Language
-
enPrimary language
- Publication year
-
2018Year of publication
- Publication date
-
2018-12-21Full publication date if available
- Authors
-
Florent Joly, Camille Cohen, Vincent Javaugue, Sébastien Bender, Mohamed Belmouaz, Bertrand Arnulf, Bertrand Knebelmann, M. Nouvier, Vincent Audard, François Provôt, Viviane Gnemmi, Dominique Nochy, Jean Michel Goujon, Arnaud Jaccard, Guy Touchard, Jean Paul Fermand, Christophe Sirac, Frank BridouxList of authors in order
- Landing page
-
https://doi.org/10.1182/blood-2018-09-872028Publisher landing page
- PDF URL
-
https://ashpublications.org/blood/article-pdf/133/6/576/1750586/blood872028.pdfDirect link to full text PDF
- Open access
-
YesWhether a free full text is available
- OA status
-
bronzeOpen access status per OpenAlex
- OA URL
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https://ashpublications.org/blood/article-pdf/133/6/576/1750586/blood872028.pdfDirect OA link when available
- Concepts
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Medicine, Internal medicine, Nephropathy, Multiple myeloma, Monoclonal gammopathy of undetermined significance, Gastroenterology, Renal biopsy, Monoclonal, Pathology, Immunology, Biopsy, Antibody, Monoclonal antibody, Endocrinology, Diabetes mellitusTop concepts (fields/topics) attached by OpenAlex
- Cited by
-
113Total citation count in OpenAlex
- Citations by year (recent)
-
2025: 14, 2024: 24, 2023: 12, 2022: 20, 2021: 20Per-year citation counts (last 5 years)
- References (count)
-
43Number of works referenced by this work
- Related works (count)
-
10Other works algorithmically related by OpenAlex
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| abstract_inverted_index.58%), | 181 |
| abstract_inverted_index.58]), | 96 |
| abstract_inverted_index.Among | 173 |
| abstract_inverted_index.Congo | 16 |
| abstract_inverted_index.LHCDD | 102 |
| abstract_inverted_index.MIDD. | 240 |
| abstract_inverted_index.Rapid | 241 |
| abstract_inverted_index.Renal | 121, 200 |
| abstract_inverted_index.above | 197 |
| abstract_inverted_index.acute | 124 |
| abstract_inverted_index.chain | 22, 25, 187 |
| abstract_inverted_index.heavy | 24 |
| abstract_inverted_index.light | 21, 186 |
| abstract_inverted_index.other | 139 |
| abstract_inverted_index.point | 160 |
| abstract_inverted_index.renal | 37, 61, 112, 228 |
| abstract_inverted_index.serum | 184 |
| abstract_inverted_index.study | 231 |
| abstract_inverted_index.(36%), | 206 |
| abstract_inverted_index.(MIDD) | 5 |
| abstract_inverted_index.(VGPR) | 195 |
| abstract_inverted_index.B-cell | 11 |
| abstract_inverted_index.clonal | 12 |
| abstract_inverted_index.cohort | 73 |
| abstract_inverted_index.domain | 164 |
| abstract_inverted_index.injury | 126 |
| abstract_inverted_index.kidney | 125 |
| abstract_inverted_index.little | 40 |
| abstract_inverted_index.severe | 221 |
| abstract_inverted_index.showed | 157 |
| abstract_inverted_index.tissue | 171 |
| abstract_inverted_index.types, | 140 |
| abstract_inverted_index.values | 161 |
| abstract_inverted_index.(HCDD), | 26 |
| abstract_inverted_index.(LCDD), | 23 |
| abstract_inverted_index.(mostly | 147 |
| abstract_inverted_index.absence | 219 |
| abstract_inverted_index.chronic | 134 |
| abstract_inverted_index.defined | 14 |
| abstract_inverted_index.disease | 4, 136 |
| abstract_inverted_index.factors | 58, 251 |
| abstract_inverted_index.hepatic | 148 |
| abstract_inverted_index.myeloma | 118 |
| abstract_inverted_index.partial | 193 |
| abstract_inverted_index.patient | 63 |
| abstract_inverted_index.studied | 70 |
| abstract_inverted_index.(LHCDD). | 29 |
| abstract_inverted_index.Abstract | 0 |
| abstract_inverted_index.achieved | 183, 211 |
| abstract_inverted_index.cardiac) | 150 |
| abstract_inverted_index.defined. | 67 |
| abstract_inverted_index.disorder | 34 |
| abstract_inverted_index.fibrosis | 223 |
| abstract_inverted_index.occurred | 202 |
| abstract_inverted_index.patients | 128, 175, 205 |
| abstract_inverted_index.possibly | 168 |
| abstract_inverted_index.received | 177 |
| abstract_inverted_index.regions, | 167 |
| abstract_inverted_index.response | 194, 201, 215, 248 |
| abstract_inverted_index.survival | 64 |
| abstract_inverted_index.systemic | 33 |
| abstract_inverted_index.variable | 163 |
| abstract_inverted_index.Moreover, | 51 |
| abstract_inverted_index.attention | 41 |
| abstract_inverted_index.diagnosis | 107, 242 |
| abstract_inverted_index.frequency | 235 |
| abstract_inverted_index.including | 190 |
| abstract_inverted_index.patients, | 76 |
| abstract_inverted_index.prominent | 36 |
| abstract_inverted_index.response, | 189 |
| abstract_inverted_index.response. | 213, 229 |
| abstract_inverted_index.(including | 83 |
| abstract_inverted_index.Monoclonal | 1 |
| abstract_inverted_index.Sequencing | 152 |
| abstract_inverted_index.accounting | 169 |
| abstract_inverted_index.associated | 59 |
| abstract_inverted_index.deposition | 3, 56 |
| abstract_inverted_index.disorders, | 13 |
| abstract_inverted_index.extrarenal | 49, 146, 237 |
| abstract_inverted_index.gammopathy | 110 |
| abstract_inverted_index.glomerular | 135 |
| abstract_inverted_index.highlights | 232 |
| abstract_inverted_index.mechanisms | 52 |
| abstract_inverted_index.monoclonal | 20, 109 |
| abstract_inverted_index.nationwide | 72 |
| abstract_inverted_index.pathogenic | 54, 155 |
| abstract_inverted_index.predictors | 226 |
| abstract_inverted_index.prognosis. | 253 |
| abstract_inverted_index.unexpected | 234 |
| abstract_inverted_index.achievement | 244 |
| abstract_inverted_index.deposition. | 172 |
| abstract_inverted_index.description | 47 |
| abstract_inverted_index.determining | 166 |
| abstract_inverted_index.independent | 225 |
| abstract_inverted_index.isoelectric | 159 |
| abstract_inverted_index.nephropathy | 92 |
| abstract_inverted_index.symptomatic | 117, 145 |
| abstract_inverted_index.chemotherapy | 178 |
| abstract_inverted_index.complication | 9 |
| abstract_inverted_index.interstitial | 222 |
| abstract_inverted_index.involvement, | 38 |
| abstract_inverted_index.involvement. | 151 |
| abstract_inverted_index.presentation | 122 |
| abstract_inverted_index.significance | 113 |
| abstract_inverted_index.Hematological | 106 |
| abstract_inverted_index.biopsy-proven | 78 |
| abstract_inverted_index.hematological | 212 |
| abstract_inverted_index.immunoglobulin | 2, 55 |
| abstract_inverted_index.manifestations | 238 |
| abstract_inverted_index.complementarity | 165 |
| abstract_inverted_index.manifestations. | 50 |
| abstract_inverted_index.retrospectively | 69 |
| abstract_inverted_index.(bortezomib-based | 179 |
| abstract_inverted_index.negative–deposits | 18 |
| cited_by_percentile_year.max | 100 |
| cited_by_percentile_year.min | 99 |
| corresponding_author_ids | https://openalex.org/A5010587107, https://openalex.org/A5091901169 |
| countries_distinct_count | 1 |
| institutions_distinct_count | 18 |
| corresponding_institution_ids | https://openalex.org/I154526488, https://openalex.org/I4210099442, https://openalex.org/I4210111187 |
| sustainable_development_goals[0].id | https://metadata.un.org/sdg/3 |
| sustainable_development_goals[0].score | 0.7900000214576721 |
| sustainable_development_goals[0].display_name | Good health and well-being |
| citation_normalized_percentile.value | 0.94930203 |
| citation_normalized_percentile.is_in_top_1_percent | False |
| citation_normalized_percentile.is_in_top_10_percent | True |