Outcome of bleomycin electrosclerotherapy of slow-flow malformations in adults and children

Abstract

Objectives: To evaluate the safety and clinical outcome of bleomycin electrosclerotherapy (BEST) for treating extracranial slow-flow malformations.

Methods: In this retrospective investigation of a multicenter cohort presenting symptomatic slow-flow malformations, patient records were analyzed with respect to procedural details and complications. A treatment-specific, patient-reported questionnaire was additionally evaluated, obtained 3-12 months after the last treatment, to assess the subjective outcomes, including mobility, aesthetic aspects, and pain, as well as the occurrence of postprocedural skin hyperpigmentation. All outcome parameters were compared according to patients' age.

Results: Overall, 325 BEST treatments were performed in 233 patients after intralesional and/or intravenous bleomycin injection. The total complication rate was 10.2% (33/325), including 29/352 (8.9%) major complications. Patient-reported mobility decreased in 10/133 (8.8%), was stable in 30/113 (26.5%), improved in 48/113 (42.5%), and was rated symptom-free in 25/113 (22.1%) patients. Aesthetic aspects were rated impaired compared to baseline in 19/113 (16.8%), stable in 21/133 (18.6%), improved in 62/113 (54.9%), and perfect in 11/133 (9.7%) patients. Postprocedural skin hyperpigmentation occurred in 78/113 (69%) patients, remaining unchanged in 24/78 (30.8%), reduced in 51/78 (65.5%), and completely resolved in 3/78 (3.8%) patients. The median VAS pain scale was 4.0 (0-10) preprocedural and 2.0 (0-9) postprocedural. Children/adolescents performed significantly better in all parameters compared to adults (≥ 16 years) (mobility, p = 0.011; aesthetic aspects, p < 0.001; pain, p < 0.001).

Conclusions: BEST is effective for treating slow-flow vascular malformations, with few but potentially significant major complications. Regarding patient-reported outcomes, children seem to benefit better compared to older patients, suggesting that BEST should not be restricted to adults.

Clinical relevance statement: Bleomycin electrosclerotherapy is a safe and effective approach and therapy should not be restricted to adults due to good clinical outcomes in children.

Keywords: BEST; Bleomycin; Electro-chemotherapy; Sclerotherapy; Slow-flow vascular malformations.

Fig. 1

27-year-old male patient with intramuscular venous malformation (VM) of the upper extremity undergoing two subsequent bleomycin electrosclerotherapies (BESTs). a, b T2-weighted (T2w) and fat-suppressed (fs) T1-weighted (T1w) axial magnetic resonance (MR) images prior to treatment with BEST showing the extent of a symptomatic VM (asterisks) predominantly located in the triceps brachii muscle. c, d Digital subtraction angiography (DSA) images during first BEST session. The asterisk shows the filling of the dysplastic venous structures of the lesion after KM insertion. The arrow mark shows the parallelly inserted needle electrodes during therapy. e, f T2w and fs T1w axial MR images three months after the first BEST session revealing significant regredience of the vascularized parts of the VM (arrow) with accompanying improvement in the patient’s pain symptoms. g, h T2w and fs T1w axial MR images three months after the second BEST revealing near-complete resolution of the malformation. The arrow mark shows the volume-reduced lesion

Fig. 2

Four-year-old male patient with subcutaneous lymphatic malformation (LM) of the left cheek undergoing one bleomycin electrosclerotherapy (BEST). a, b T2-weighted, axial magnetic resonance images prior to treatment showing a partially micro- and partially macrocystic LM (arrows). c Clinical presentation of the patient prior to treatment including noticeable swelling of the affected region with consecutive mouth asymmetry. d Clinical presentation of the patient five days after BEST with extended postinterventional swelling (arrows) with consecutive dyspnea, which was strictly monitored, as well as skin alterations such as hyperpigmentation, necroses, and blebs (arrowhead). As oral food intake was not possible, the patient was fed intravenously, and prednisolone was administered. e–h The decreasing swelling and good healing of necrosis and blebs over time (arrowheads) while the distinct hyperpigmentation on the treated skin was pronounced on the maximum. i, j Significant reduction of skin hyperpigmentation six weeks after BEST (arrow) compared to (h)