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High bleeding rates in δ-storage pool disease during surgeries and deliveries δ储存池病在手术和分娩中的高出血率
IF 3.4 3区 医学 Q2 HEMATOLOGY
Research and Practice in Thrombosis and Haemostasis
Pub Date : 2025-11-01 DOI: 10.1016/j.rpth.2025.103239
Elleke van Heerwaarde , Annick de Moor , Albert Huisman , Rolf Urbanus , Idske Kremer Hovinga , Roger Schutgens , Thrombocytopathy in the Netherlands (TiN) study group

Background

δ-storage pool disease (δ-SPD) involves various platelet disorders due to deficient or reduced secretion of dense granules, causing increased bleeding tendency, particularly during surgery and delivery. The optimal treatment to prevent bleeding in δ-SPD is unknown.

Objectives

To evaluate the efficacy of preventive treatment in a cohort of patients with δ-SPD.

Methods

In a single-center study, we retrospectively reviewed the medical files of δ-SPD patients diagnosed between 2016 and 2024. Data included invasive diagnostic procedures, surgeries (minor/major), and deliveries (vaginal/cesarean section) before and after diagnosis. Diagnosis was confirmed by platelet adenosine diphosphate levels (<1.4 μmol/1011 platelets). Bleeding complications were defined according to the International Society on Thrombosis and Haemostasis bleeding scale. Data on treatment and platelet transfusions were documented.

Results

Thirty-eight patients (mean age, 35.6 years; range, 0-67; 63.2% female) were included. The average adenosine diphosphate level was 0.82 μmol/1011 platelets (range, 0.2-1.4). A total of 161 interventions and deliveries were analyzed. The bleeding rate was dependent on the timing of diagnosis (51.5% before δ-SPD diagnosis and 16.8% after; P < .001) and on the use of preventive treatment (45.8% without and 17.9% with; P < .001). A total of 145 interventions were analyzed, including 63 minor and 82 major. Of these, 43/145 (29.7%) were complicated by bleeding (39 minor bleeds and 4 major bleeds). Sixteen deliveries were analyzed. Among 10 vaginal deliveries, 9 had postpartum hemorrhage, of whom 6 had no prophylaxis. All 6 cesarean sections received prophylactic platelet transfusion; no bleeding occurred.

Conclusion

We observed a high perioperative/peripartum bleeding rate in patients with δ-SPD. This decreased significantly after a correct diagnosis. In addition, bleeding rates were significantly lower among patients who received preventive treatment before the procedure.
δ-储存池病（δ-SPD）涉及由于致密颗粒分泌不足或减少而引起的各种血小板紊乱，导致出血倾向增加，特别是在手术和分娩期间。预防δ-SPD出血的最佳治疗方法尚不清楚。目的评价δ-SPD患者预防治疗的效果。方法采用单中心研究方法，回顾性分析2016年至2024年诊断为δ-SPD患者的医疗档案。数据包括诊断前后的侵入性诊断程序、手术（小/大）和分娩（阴道/剖宫产）。血小板二磷酸腺苷水平（1.4 μmol/1011个血小板）证实诊断。出血并发症的定义根据国际血栓和止血学会出血量表。记录了治疗和血小板输注的数据。结果共纳入38例患者，平均年龄35.6岁，年龄范围0 ~ 67岁，女性占63.2%。平均二磷酸腺苷水平为0.82 μmol/1011血小板（范围0.2 ~ 1.4）。总共分析了161例干预和分娩。出血率与诊断时间（δ-SPD诊断前为51.5%，诊断后为16.8%,P < 0.001）和预防治疗的使用（未治疗45.8%，治疗17.9%,P < 0.001）有关。共分析145项干预措施，其中次要干预63项，主要干预82项。其中43/145例（29.7%）合并出血，其中小出血39例，大出血4例。对16例分娩进行了分析。在10例阴道分娩中，9例发生产后出血，其中6例未采取预防措施。6例剖宫产均行预防性血小板输注；未发生出血。结论δ-SPD患者围手术期及围生期出血发生率较高。这在正确诊断后显著下降。此外，术前接受预防性治疗的患者出血率明显较低。
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