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Shwachman-Diamond 综合征与实体肿瘤:来自法国严重慢性中性粒细胞减少症登记处的 3 例新患者及文献复习。

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

机构信息

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.

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

出版信息

Pediatr Blood Cancer. 2021 Jul;68(7):e29071. doi: 10.1002/pbc.29071. Epub 2021 Apr 19.

DOI:10.1002/pbc.29071
PMID:33871916
Abstract

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.

摘要

Shwachman-Diamond 综合征伴 Shwachman-Bodian-Diamond 综合征(SBDS)双等位基因突变是一种罕见疾病,使携带者易患恶性血液病,但实体瘤易感性知之甚少。在法国严重慢性中性粒细胞减少症登记处登记的 155 例病例中,有 3 例被诊断为恶性实体瘤(卵巢、乳腺和食管)。所有癌症均发生在第五十年,尽管在诊断时局限于局部,但此后迅速致命。在 14 例 HSCT 幸存者中,未观察到移植后癌症。根据文献和我们的患者数据,我们只能提出这种并发症主要在成人中诊断。

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引用本文的文献

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Transl Breast Cancer Res. 2024 Jul 22;5:26. doi: 10.21037/tbcr-24-13. eCollection 2024.
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Posttransplant complications in patients with marrow failure syndromes: are we improving long-term outcomes?骨髓衰竭综合征患者移植后的并发症:我们是否改善了长期预后?
Hematology Am Soc Hematol Educ Program. 2023 Dec 8;2023(1):141-148. doi: 10.1182/hematology.2023000471.
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Shwachman-Diamond syndromes: clinical, genetic, and biochemical insights from the rare variants.
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