Abstract
Background: The link between cystic lymphatic malformation (cLM) and normal lymphatic system has become the focus of research. This study aimed to assess the outcomes of indocyanine green (ICG) lymphography-guided inflow occlusion combined with bleomycin sclerotherapy for the management of macro or mixed cLM in children.Methods: Between June 2018 and October 2020, inflow occlusion combined with bleomycin sclerotherapy was performed in 81 cLM patients (age range from 6 months to 8 years). All cases were evaluated by the following parameters: cLM location, histological typing, number of afferent lymph vessels, dermal backflow, curative effects, treatment frequency, and postoperative complications. The duration of postoperative follow-up was from 10 to 16 months.Results: All cLM cases could be found with at least one lymphatic inflow. Excellent outcomes were observed in 68 cases (84.0%), 11 cases (13.6%) experienced good outcomes, and two (2.5%) cases had fair outcome. No case experienced repeated treatment for more than three times. Wound infection, fever, and scar hyperplasia were the independent adverse events, which were managed by symptomatic treatment.Conclusion: Inflow occlusion combined with bleomycin sclerotherapy renders a safe and efficient approach for the management of macro or mixed cLM.
Highlights
Cystic lymphatic malformations are nonmalignant vascular malformations of the lymphatic system, characterized by the dilated cysts lined by lymphatic endothelial cells [1, 2]
It may change in size resulting from local infection, intracapsular hemorrhage or trauma, and even resolve spontaneously in rare cases [3,4,5]. Cystic lymphatic malformations (cLM) has been managed by multidisciplinary teams worldwide, each according to their own preferences regarding therapeutic intervention approaches, including surgical resection, sclerotherapy, oral sirolimus, radiofrequency ablation, and laser therapy [6,7,8,9,10,11]
Given that some of cLMs with inflow pattern are connected to lymphatic system, posttreatment accumulation of lymph in the lesion could always be observed, which may eventually lead to local recurrence and repeated sclerotherapy [12, 13]
Summary
Cystic lymphatic malformations (cLM) are nonmalignant vascular malformations of the lymphatic system, characterized by the dilated cysts lined by lymphatic endothelial cells [1, 2]. According to the classification system of the International Society for the Study of Vascular Anomalies (ISSVA), cLM can be histologically categorized into three subtypes, including macrocystic, microcystic, and mixed cystic. It may change in size resulting from local infection, intracapsular hemorrhage or trauma, and even resolve spontaneously in rare cases [3,4,5]. The link between cLM and normal lymphatic system has gradually become the focus of research nowadays. This study aimed to assess the outcomes of indocyanine green (ICG) lymphography-guided inflow occlusion combined with bleomycin sclerotherapy for the management of macro or mixed cLM in children
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