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皮尔逊综合征的临床及形态学骨髓特征:三例连续病例报告及文献复习

Clinical and Morphological Bone Marrow Characteristics of Pearson Syndrome: About Three Consecutive Cases and Review of the Literature.

作者信息

Degroot Gaetan-Nagim, Empain Aurélie, Dedeken Laurence, Demulder Anne, Rozen Laurence

机构信息

Laboratory of Hematology, Brussels University Hospital Laboratory (LHUB), Université Libre de Bruxelles (ULB), Brussels, Belgium.

Laboratory of Hematology, CHU Brugmann University Hospital, Université Libre de Bruxelles (ULB), Brussels, Belgium.

出版信息

Case Rep Pediatr. 2025 May 18;2025:3076141. doi: 10.1155/crpe/3076141. eCollection 2025.

Abstract

Pearson syndrome (PS) is a rare and fatal multisystem disorder caused by a mitochondrial DNA (mtDNA) deletion. Most patients develop refractory anemia in early infancy, rapidly followed by multiple complications such as failure to thrive, muscle hypotonia, pancreatic insufficiency, and renal tubulopathy. Although the definitive diagnosis is established by mtDNA sequencing, bone marrow (BM) cytology is a cornerstone of diagnosis, typically revealing precursor vacuolization and ring sideroblasts. We report here three cases of patients with PS encountered in our institution and summarize the clinical and hematological features of PS through a systematic review of the literature. The first symptoms mostly appear during the first month of life and rarely after 18 months. Hyporegenerative anemia, a hallmark of the disease, is the most common initial symptom, followed at a distance by neutropenia and thrombocytopenia. Gastrointestinal and metabolic symptoms such as failure to thrive and lactic acidosis are the most frequent non-hematological symptoms, even in the rare cases without hyporegenerative anemia. Vacuolization of BM precursors, observed in the vast majority of PS patient BMAs, is not influenced by the patient's age at sampling. Ring sideroblasts, the other feature of PS BMAs, are less frequent than progenitor vacuolization but increase significantly after 6 months of age. These abnormalities are just as common in patients with or without hematological symptoms, suggesting that BMA should be performed in all suspected PS cases, despite the absence of anemia. PS is a multisystem disorder requiring early diagnosis and a coordinate multidisciplinary management, involving clinicians and clinical biologists.

摘要

皮尔逊综合征(PS)是一种由线粒体DNA(mtDNA)缺失引起的罕见且致命的多系统疾病。大多数患者在婴儿早期出现难治性贫血,随后迅速出现多种并发症,如生长发育迟缓、肌张力低下、胰腺功能不全和肾小管病变。虽然通过mtDNA测序可确诊,但骨髓(BM)细胞学检查是诊断的基石,通常显示前体细胞空泡化和环形铁粒幼细胞。我们在此报告在我院遇到的3例PS患者,并通过对文献的系统回顾总结PS的临床和血液学特征。首发症状大多出现在出生后的第一个月,18个月后很少出现。再生障碍性贫血是该病的一个标志,是最常见的初始症状,其次是中性粒细胞减少和血小板减少。生长发育迟缓、乳酸酸中毒等胃肠道和代谢症状是最常见的非血液学症状,即使在罕见的无再生障碍性贫血的病例中也是如此。在绝大多数PS患者的骨髓穿刺涂片中观察到的BM前体细胞空泡化不受取样时患者年龄的影响。环形铁粒幼细胞是PS骨髓穿刺涂片的另一个特征,比祖细胞空泡化少见,但在6个月龄后显著增加。这些异常在有或无血液学症状的患者中同样常见,这表明尽管没有贫血,所有疑似PS病例均应进行骨髓穿刺检查。PS是一种多系统疾病,需要早期诊断和协调的多学科管理,涉及临床医生和临床生物学家。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5f23/12103953/88c2ddf7dfd2/CRIPE2025-3076141.001.jpg

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