Lymphoscintigraphy in Angiomyomatous Hamartomas and Primary Lower Limb Lymphedema
Lymphoscintigraphy in Angiomyomatous Hamartomas and Primary Lower Limb Lymphedema
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DOI：
10.1097/rlu.0b013e3181a7d013
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发表时间：
2009-07-01
影响因子：
10.6
通讯作者：
De Valck, Christine
中科院分区：
文献类型：
--
作者：
Bourgeois, Pierre;Dargent, Jean-Louis;De Valck, Christine
目的：淋巴结血管平滑肌瘤性错构瘤是一种罕见的血管良性疾病，病因不明，易发生于腹股沟区淋巴结。到目前为止，文献中仅描述了18例病例，4例患者的疾病报告与同侧肢体水肿或肿胀有关。材料与方法：对5例因疑诊淋巴结炎而行腹股沟淋巴结活检诊断为血管平滑肌瘤性错构瘤的患者进行淋巴结造影检查（1名15岁的女孩; 1名首次活检时9岁和第二次活检时11岁的男孩; 3名30岁、50岁和57岁的男性）。3例术后肢体水肿，1例术后肢体水肿。第五例患者在第二次淋巴结活检后出现对侧水肿。结果：所有病例的肢体淋巴结造影检查均显示手术侧广泛的淋巴结异常，4例在对侧。结论：这些观察结果不仅支持了淋巴管紊乱参与这些肿瘤发病机制的假设，而且支持了淋巴管造影术应在不明原因的腹股沟淋巴结炎，以诊断潜在的水肿的基础情况。
Purpose: Angiomyomatous hamartorna (AH) of the lymph node is a rare vascular benign disease of unknown etiology with a predisposition for the lymph nodes of the inguinal area. Only 18 cases have been described up to now in the literature and the disorder was reported to be associated with lymphedema or swelling of the ipsilateral limb in 4 patients. However, scintigraphic investigation of the lymphatic system in these patients was reported in only 2 cases.Material and Methods: Five patients where the biopsy of inguinal nodes for suspected lymphadenitis led to the diagnosis of angiomyomatous hamartoma were investigated using lymphoscintigraphic techniques (1 girl aged 15; 1 boy aged 9 at the time of first biopsy and 11 at the time of the second one; and 3 men aged 30, 50, and 57). The operated limb was lymphedematous in 3 and 1 developed lymphedema after biopsy. The fifth patient developed a contralateral lymphedema after his second nodal biopsy.Results: In all cases, lymphoscintigraphic investigation of the limbs showed extensive lymph node abnormalities on the operated side and in 4 cases on the opposite side.Conclusions: These observations support not only the hypothesis that lymphatic disturbance was involved in the pathogenesis of these tumors but also the proposition that lymphoscintigraphy should be performed in cases of inguinal lymphadenitis of unknown origin to diagnose the underlying situation of latent lymphedema.