Diagnosis of Primary Mast Cell Disorders in Anaphylaxis: Value of KIT D816V in Peripheral Blood.

Anaphylaxis KIT D816V mutation Mast cell Mastocytosis Mastocytosis in the skin Monoclonal mast cell activation syndrome Primary mast cell disorder Tryptase

Journal

The journal of allergy and clinical immunology. In practice
ISSN: 2213-2201
Titre abrégé: J Allergy Clin Immunol Pract
Pays: United States
ID NLM: 101597220

Informations de publication

Date de publication:
08 2021
Historique:
received: 16 12 2020
revised: 30 03 2021
accepted: 13 04 2021
pubmed: 12 5 2021
medline: 28 10 2021
entrez: 11 5 2021
Statut: ppublish

Résumé

Anaphylaxis is frequent in patients suffering from primary mast cell disorders (PMCDs). In patients without mastocytosis in the skin (MIS) and a baseline serum tryptase (bST) less than 30 ng/mL, the diagnosis of PMCD is challenging. In these patients, detection of the KIT D816V mutation in peripheral blood (PB) has been suggested as screening tool for a PMCD. In this study, we investigated whether KIT D816V in PB can contribute to the decision to perform a bone marrow (BM) biopsy in patients with anaphylaxis without MIS and a bST less than 30 ng/mL. We selected 74 patients with severe anaphylaxis without MIS and a bST less than 30 ng/mL. All underwent a BM biopsy. KIT D816V mutation was quantified in both PB and BM using digital droplet polymerase chain reaction (ddPCR). Diagnosis of a PMCD was established in 40 patients (54%). Median bST for patients with and without PMCD was, respectively, 9.5 ng/mL (range 4.2-27 ng/mL) and 4.9 ng/mL (range 2.2-20.3 ng/mL) (P <.001). KIT D816V in PB was detected in 16 out of 40 (40%) patients with PMCD. KIT D816V in BM was detected in 22 out of 40 (55%) patients with PMCD. In patients without MIS and a bST less than < 30 ng/mL who experience anaphylaxis, determination of KIT D816V mutation in PB is of limited help in deciding when to proceed to a BM biopsy. Therefore, KIT D816V in PB mutation analysis should be interpreted together with scoring tools to make a better assessment in identifying patients who should undergo BM biopsy.

Sections du résumé

BACKGROUND
Anaphylaxis is frequent in patients suffering from primary mast cell disorders (PMCDs). In patients without mastocytosis in the skin (MIS) and a baseline serum tryptase (bST) less than 30 ng/mL, the diagnosis of PMCD is challenging. In these patients, detection of the KIT D816V mutation in peripheral blood (PB) has been suggested as screening tool for a PMCD.
OBJECTIVE
In this study, we investigated whether KIT D816V in PB can contribute to the decision to perform a bone marrow (BM) biopsy in patients with anaphylaxis without MIS and a bST less than 30 ng/mL.
METHODS
We selected 74 patients with severe anaphylaxis without MIS and a bST less than 30 ng/mL. All underwent a BM biopsy. KIT D816V mutation was quantified in both PB and BM using digital droplet polymerase chain reaction (ddPCR).
RESULTS
Diagnosis of a PMCD was established in 40 patients (54%). Median bST for patients with and without PMCD was, respectively, 9.5 ng/mL (range 4.2-27 ng/mL) and 4.9 ng/mL (range 2.2-20.3 ng/mL) (P <.001). KIT D816V in PB was detected in 16 out of 40 (40%) patients with PMCD. KIT D816V in BM was detected in 22 out of 40 (55%) patients with PMCD.
CONCLUSIONS
In patients without MIS and a bST less than < 30 ng/mL who experience anaphylaxis, determination of KIT D816V mutation in PB is of limited help in deciding when to proceed to a BM biopsy. Therefore, KIT D816V in PB mutation analysis should be interpreted together with scoring tools to make a better assessment in identifying patients who should undergo BM biopsy.

Identifiants

pubmed: 33975032
pii: S2213-2198(21)00569-9
doi: 10.1016/j.jaip.2021.04.062
pii:
doi:

Substances chimiques

Proto-Oncogene Proteins c-kit EC 2.7.10.1

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

3176-3187.e3

Informations de copyright

Copyright © 2021 American Academy of Allergy, Asthma & Immunology. Published by Elsevier Inc. All rights reserved.

Auteurs

Leander P De Puysseleyr (LP)

Faculty of Medicine and Health Sciences, Department of Immunology, Allergology, and Rheumatology and the Infla-Med Centre of Excellence University of Antwerp; and Department of Immunology, Allergology, and Rheumatology, Antwerp University Hospital, Antwerp, Belgium.

Didier G Ebo (DG)

Faculty of Medicine and Health Sciences, Department of Immunology, Allergology, and Rheumatology and the Infla-Med Centre of Excellence University of Antwerp; and Department of Immunology, Allergology, and Rheumatology, Antwerp University Hospital, Antwerp, Belgium; Department of Immunology and Allergology, AZ Jan Palfijn Gent, Ghent, Belgium. Electronic address: immuno@uantwerpen.be.

Jessy Elst (J)

Faculty of Medicine and Health Sciences, Department of Immunology, Allergology, and Rheumatology and the Infla-Med Centre of Excellence University of Antwerp; and Department of Immunology, Allergology, and Rheumatology, Antwerp University Hospital, Antwerp, Belgium.

Margaretha A Faber (MA)

Faculty of Medicine and Health Sciences, Department of Immunology, Allergology, and Rheumatology and the Infla-Med Centre of Excellence University of Antwerp; and Department of Immunology, Allergology, and Rheumatology, Antwerp University Hospital, Antwerp, Belgium.

Marie-Line van der Poorten (MV)

Faculty of Medicine and Health Sciences, Department of Immunology, Allergology, and Rheumatology and the Infla-Med Centre of Excellence University of Antwerp; and Department of Immunology, Allergology, and Rheumatology, Antwerp University Hospital, Antwerp, Belgium; Faculty of Medicine and Health Sciences, Department of Paediatrics and the Infla-Med Centre of Excellence, University of Antwerp and Department of Paediatrics, Antwerp University Hospital, Antwerp, Belgium.

Athina L Van Gasse (AL)

Faculty of Medicine and Health Sciences, Department of Immunology, Allergology, and Rheumatology and the Infla-Med Centre of Excellence University of Antwerp; and Department of Immunology, Allergology, and Rheumatology, Antwerp University Hospital, Antwerp, Belgium; Faculty of Medicine and Health Sciences, Department of Paediatrics and the Infla-Med Centre of Excellence, University of Antwerp and Department of Paediatrics, Antwerp University Hospital, Antwerp, Belgium.

Chris H Bridts (CH)

Faculty of Medicine and Health Sciences, Department of Immunology, Allergology, and Rheumatology and the Infla-Med Centre of Excellence University of Antwerp; and Department of Immunology, Allergology, and Rheumatology, Antwerp University Hospital, Antwerp, Belgium.

Christel Mertens (C)

Faculty of Medicine and Health Sciences, Department of Immunology, Allergology, and Rheumatology and the Infla-Med Centre of Excellence University of Antwerp; and Department of Immunology, Allergology, and Rheumatology, Antwerp University Hospital, Antwerp, Belgium.

Michel Van Houdt (M)

Faculty of Medicine and Health Sciences, Department of Immunology, Allergology, and Rheumatology and the Infla-Med Centre of Excellence University of Antwerp; and Department of Immunology, Allergology, and Rheumatology, Antwerp University Hospital, Antwerp, Belgium.

Margo M Hagendorens (MM)

Faculty of Medicine and Health Sciences, Department of Immunology, Allergology, and Rheumatology and the Infla-Med Centre of Excellence University of Antwerp; and Department of Immunology, Allergology, and Rheumatology, Antwerp University Hospital, Antwerp, Belgium; Faculty of Medicine and Health Sciences, Department of Paediatrics and the Infla-Med Centre of Excellence, University of Antwerp and Department of Paediatrics, Antwerp University Hospital, Antwerp, Belgium.

Anke Verlinden (A)

Department of Haematology, Antwerp University Hospital, Edegem, Belgium.

Katrien Vermeulen (K)

Department of Clinical Biology, Antwerp University Hospital, Edegem, Belgium.

Marie-Berthe Maes (MB)

Department of Clinical Biology, Antwerp University Hospital, Edegem, Belgium.

Zwi N Berneman (ZN)

Department of Haematology, Antwerp University Hospital, Edegem, Belgium.

Vito Sabato (V)

Faculty of Medicine and Health Sciences, Department of Immunology, Allergology, and Rheumatology and the Infla-Med Centre of Excellence University of Antwerp; and Department of Immunology, Allergology, and Rheumatology, Antwerp University Hospital, Antwerp, Belgium; Department of Immunology and Allergology, AZ Jan Palfijn Gent, Ghent, Belgium.

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