Abstract：
BACKGROUND: Chronic histiocytic intervillositis (CHI) is a rare inflammatory placental disease characterized by diffuse infiltration of monocytes into the intervillous space and is associated with adverse pregnancy outcomes. No treatment is currently validated and although in some small reports, steroids with hydroxychloroquine have been described. There are no data for other therapies in refractory cases.
METHODS: We here report four cases of patients with a history of CHI treated with immunoglobulins during a subsequent pregnancy. The four patients with recurrent CHI had failed to previous immunomodulatory therapies with steroids and hydroxychloroquine. All patients had at least four pregnancy losses with histopathological confirmation of CHI for at least one pregnancy loss. The usual pregnancy-loss etiology screening and immunological screening were negative for all the patients.
RESULTS: For three patients, intravenous immunoglobulins were initiated at the βHCG positivity at 1 g/kg every 15 days until delivery. In one case with combined therapy since the beginning of the pregnancy, intravenous immunoglobulins were introduced at 20 WG because of severe growth restriction. Two patients had live births at 36 WG and one patient at 39 WG. One patient, who presented early first-trimester hypertension and severe placental lesions, failed to intravenous immunoglobulins and had a pregnancy loss at 15 WG.
CONCLUSIONS: This is the first report demonstrating the potential benefit of intravenous immunoglobulins in recurrent chronic intervillositis. Larger studies are needed to confirm this potential benefit for patients presenting severe cases of recurrent CHI.
METHODS: We here report four cases of patients with a history of CHI treated with immunoglobulins during a subsequent pregnancy. The four patients with recurrent CHI had failed to previous immunomodulatory therapies with steroids and hydroxychloroquine. All patients had at least four pregnancy losses with histopathological confirmation of CHI for at least one pregnancy loss. The usual pregnancy-loss etiology screening and immunological screening were negative for all the patients.
RESULTS: For three patients, intravenous immunoglobulins were initiated at the βHCG positivity at 1 g/kg every 15 days until delivery. In one case with combined therapy since the beginning of the pregnancy, intravenous immunoglobulins were introduced at 20 WG because of severe growth restriction. Two patients had live births at 36 WG and one patient at 39 WG. One patient, who presented early first-trimester hypertension and severe placental lesions, failed to intravenous immunoglobulins and had a pregnancy loss at 15 WG.
CONCLUSIONS: This is the first report demonstrating the potential benefit of intravenous immunoglobulins in recurrent chronic intervillositis. Larger studies are needed to confirm this potential benefit for patients presenting severe cases of recurrent CHI.
摘要：
背景：慢性组织细胞性颌间炎（CHI）是一种罕见的炎症性胎盘疾病，其特征是单核细胞弥漫性渗入绒毛间隙，并与不良妊娠结局有关。目前没有验证治疗方法，尽管在一些小报告中，类固醇与羟氯喹已经被描述。在难治性病例中没有其他疗法的数据。
方法：我们在此报告4例具有CHI病史的患者在随后的妊娠期间接受免疫球蛋白治疗。4例CHI复发患者先前未能接受类固醇和羟氯喹的免疫调节治疗。所有患者至少有4次妊娠损失，组织病理学证实CHI至少有1次妊娠损失。所有患者的常规妊娠丢失病因筛查和免疫筛查均为阴性。
结果：对于三名患者，在βHCG阳性时,每15天开始静脉注射免疫球蛋白,剂量为1g/kg,直至分娩.在一个病例中，从怀孕开始，联合治疗，由于严重的生长限制,在20WG时引入了静脉注射免疫球蛋白.两名患者在36WG活产，一名患者在39WG活产。一个病人,出现早孕期高血压和严重胎盘病变的患者，免疫球蛋白静脉注射失败，在15WG时妊娠失败。
结论：这是首次报告，证明静脉注射免疫球蛋白对复发性慢性颌间炎的潜在益处。需要更大规模的研究来证实这种对患有严重CHI复发病例的患者的潜在益处。
方法：我们在此报告4例具有CHI病史的患者在随后的妊娠期间接受免疫球蛋白治疗。4例CHI复发患者先前未能接受类固醇和羟氯喹的免疫调节治疗。所有患者至少有4次妊娠损失，组织病理学证实CHI至少有1次妊娠损失。所有患者的常规妊娠丢失病因筛查和免疫筛查均为阴性。
结果：对于三名患者，在βHCG阳性时,每15天开始静脉注射免疫球蛋白,剂量为1g/kg,直至分娩.在一个病例中，从怀孕开始，联合治疗，由于严重的生长限制,在20WG时引入了静脉注射免疫球蛋白.两名患者在36WG活产，一名患者在39WG活产。一个病人,出现早孕期高血压和严重胎盘病变的患者，免疫球蛋白静脉注射失败，在15WG时妊娠失败。
结论：这是首次报告，证明静脉注射免疫球蛋白对复发性慢性颌间炎的潜在益处。需要更大规模的研究来证实这种对患有严重CHI复发病例的患者的潜在益处。
