Venous malformations are congenital anomalies of the vascular system. The injection of bleomycin (a cytotoxic, antitumour drug) into the lesion is a safe and effective treatment for low-flow (venous and lymphatic) malformations, but its use systemically has been associated with pulmonary fibrosis. Intralesional injection of bleomycin is considered to have a lower risk, but caution should be used when planning treatment, with particular regard to respiratory function. Electroporation is the temporary application of an electrical field across a tissue to increase (briefly) the permeability of the cell membrane in that tissue. We successfully treated a venous malformation in a patient with severe respiratory compromise with a low dose of bleomycin into the lesion, which we augmented using electroporation. To the best of our knowledge, this is the first reported use of bleomycin electrosclerotherapy in the management of a venous malformation.
Vascular malformations are congenital anomalies of the vascular system and are a challenge both therapeutically and diagnostically. Current management mainly consists of techniques of intralesional sclerotherapy. Bleomycin is a cytotoxic, antitumour drug with sclerosant properties, and the use of it intralesionally is safe and effective for the treatment of venous malformations. There is an established link between its systemic use and pulmonary fibrosis, and a single case report shows this complication with intralesional injection of bleomycin also. We give our patients a specialist respiratory assessment, (which involves testing their pulmonary function) before treatment, to provide a baseline for comparison and diagnose any pre-existing respiratory conditions.
Intralesional injection of bleomycin is also used to manage various types of skin cancer, and is typically combined with the technique of electroporation (known as “electrochemotherapy”). Electroporation applies an electrical field across cells to increase the permeability of the membranes. We apply pulsed, short, high-intensity electrical fields to the tissues through electrodes placed into the lesions, and inject a drug (in this case, bleomycin), either systemically or intralesionally.
A 76-year-old man, who was a retired plant operator, presented to our clinic with a venous malformation. It had been noted in childhood, affected both cheeks, and extended into the mouth and tongue. Excision had been attempted twice in the past, and he had had a course of radiotherapy, neither of which had worked. He had lived with the malformation ever since, and sought referral to us only because of a steady increase in its size and symptoms. Ultrasound examination supported the physical findings of a venous malformation that affected the entire tongue and involved the cheeks bilaterally ( Fig. 1 ).
The malformation was accessible percutaneously and was therefore ideal for treatment with injection of intralesional bleomycin. The patient had poor respiratory function secondary to severe chronic obstructive pulmonary disease because of a history of smoking and exposure to coal dust.
We devised a management plan after discussion with him that involved a reduced dose of intralesional bleomycin, and we did serial pulmonary function tests between injections. We repeated treatment over a course of six sessions with six-week intervals, and continued it based on the response. Small improvements were seen after each session, but the overall response was minimal. We think that this was probably because of the reduced dose of bleomycin (a third of the standard) used.
After further discussion with him we did one more session of injections (still at a reduced dose) accompanied by electroporation. On review one month later, the malformation had reduced considerably in size, his symptoms had eased, and he had had no complications ( Fig. 2 ). No deterioration in respiratory function was seen throughout the duration of treatment.
Electrochemotherapy with bleomycin has been used extensively in the treatment of cutaneous malignancies without serious adverse events.
The use of intralesional bleomycin in the treatment of venous malformations is well established and associated with a favourable ratio of benefit:risk. A recent meta-analysis showed good to excellent rates of response in 87% of those treated.
In this instance, electrosclerotherapy enabled us to use a reduced dose of bleomycin. For units with access to electroporation devices, this may also be a simple way of improving response to intralesional bleomycin when treating venous malformations and the approach will be an exciting direction for future research.