Shwachman-Diamond syndrome and solid tumors: Three new patients from the French Registry for Severe Chronic Neutropenia and literature review.


Journal

Pediatric blood & cancer
ISSN: 1545-5017
Titre abrégé: Pediatr Blood Cancer
Pays: United States
ID NLM: 101186624

Informations de publication

Date de publication:
07 2021
Historique:
revised: 17 03 2021
received: 14 10 2020
accepted: 05 04 2021
pubmed: 20 4 2021
medline: 7 1 2022
entrez: 19 4 2021
Statut: ppublish

Résumé

Shwachman-Diamond syndrome with Shwachman-Bodian-Diamond syndrome (SBDS) biallelic variants is a rare disorder that predisposes the carrier to malignant hemopathies but solid-cancer predisposition is poorly known. Among 155 cases entered in the French Registry for Severe Chronic Neutropenia, three were identified with malignant solid tumors (ovary, breast, and esophagus). All cancers occurred during the fifth decade and, despite being localized at diagnosis, were rapidly fatal thereafter. No cancer was observed post transplantation in the 14 HSCT survivors. Based on the literature and our patient data, we can merely advance that this complication is predominantly diagnosed in adults.

Identifiants

pubmed: 33871916
doi: 10.1002/pbc.29071
doi:

Types de publication

Case Reports Journal Article Research Support, Non-U.S. Gov't Review

Langues

eng

Sous-ensembles de citation

IM

Pagination

e29071

Informations de copyright

© 2021 Wiley Periodicals LLC.

Références

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Donadieu J, Fenneteau O, Beaupain B, et al. Classification of and risk factors for hematologic complications in a French national cohort of 102 patients with Shwachman-Diamond syndrome. Haematologica. 2012;97:1312-1319.
Myers KC, Furutani E, Weller E, et al. Clinical features and outcomes of patients with Shwachman-Diamond syndrome and myelodysplastic syndrome or acute myeloid leukaemia: a multicentre, retrospective, cohort study. Lancet Haematol. 2020;7:e238-e246.
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Auteurs

Fares Bou Mitri (F)

Centre de Recherche Saint-Antoine, APHP, Armand-Trousseau Hospital, French Registry for Severe Chronic Neutropenia, CRMR of Chronic Neutropenia, Department of Pediatrics Hematology and Oncology, Sorbonne Université, Inserm, Paris, France.

Blandine Beaupain (B)

Centre de Recherche Saint-Antoine, APHP, Armand-Trousseau Hospital, French Registry for Severe Chronic Neutropenia, CRMR of Chronic Neutropenia, Department of Pediatrics Hematology and Oncology, Sorbonne Université, Inserm, Paris, France.

Jean-François Flejou (JF)

Department of Pathology, Saint-Antoine Hospital, APHP, Paris, France.

Matthieu Patient (M)

Department of Hematology and Oncology, Hôpital d'Instruction des Armées (HIA) Sainte-Anne, Toulon, France.

Ilona Okhremchuck (I)

Department of Pathology, HIA Sainte-Anne, Toulon, France.

Didier Blaise (D)

Department of Hematology, Paoli-Calmettes Institut, Marseille, France.

Faézeh Izadifar-Legrand (F)

Department of Hematology, Paoli-Calmettes Institut, Marseille, France.

Jean Alain Martignoles (JA)

Inserm, Centre de Recherche Saint-Antoine, Hématologie Biologique, Hôpital Saint-Antoine, APHP, Sorbonne Université, Paris, France.

François Delhommeau (F)

Inserm, Centre de Recherche Saint-Antoine, Hématologie Biologique, Hôpital Saint-Antoine, APHP, Sorbonne Université, Paris, France.

Christine Bellanne-Chantelot (C)

Department of Genetics, Pitié-Salpêtrière Hospital, APHP, Sorbonne University, Paris, France.

Jean-François Emile (JF)

Department of Pathology, Ambroise-Paré Hospital, APHP, Boulogne, France.

Jean Donadieu (J)

Centre de Recherche Saint-Antoine, APHP, Armand-Trousseau Hospital, French Registry for Severe Chronic Neutropenia, CRMR of Chronic Neutropenia, Department of Pediatrics Hematology and Oncology, Sorbonne Université, Inserm, Paris, France.

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