Scleroterapia con bleomicina per malformazioni linfatiche dopo fallimento dell’escissione chirurgica: caso clinico

Vlahovic, Aleksandar M.; Gazikalovic, Ana; Adjic, Oto

doi:10.14639/0392-100x-105513

Cited by 15 publications

(8 citation statements)

References 12 publications

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“…The skin fibrosis model was prepared by subcutaneous injection of bleomycin (20 μg/mice) into the mice (Figure A). Prior reports confirmed that this dose is safe for mice, as no severe toxicity or side effects were observed during the preparation of the animal model. , NAVI treatments were started on day 14. The treatment groups were as follows: untreated, COA (50%), NAVI (50 μM), and COA (50%)/NAVI (50 μM).…”

Section: Resultsmentioning

confidence: 99%

Topical Administration of an Apoptosis Inducer Mitigates Bleomycin-Induced Skin Fibrosis

Huda

Peurta

Guerena

et al. 2023

ACS Pharmacol. Transl. Sci.

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Pathological fibrosis is distinguished from physiological wound healing by persistent myofibroblast activation, suggesting that therapies that induce myofibroblast apoptosis selectively could prevent progression and potentially reverse the established fibrosis, such as for scleroderma (a heterogeneous autoimmune disease characterized by multiorgan fibrosis). Navitoclax (NAVI) is a BCL-2/BCL-xL inhibitor with antifibrotic properties and has been investigated as a potential therapeutic for fibrosis. NAVI makes myofibroblasts particularly vulnerable to apoptosis. However, despite NAVI's significant potency, clinical translation of BCL-2 inhibitors, NAVI in this case, is hindered due to the risk of thrombocytopenia. Therefore, in this work, we utilized a newly developed ionic liquid formulation of NAVI for direct topical application to the skin, thereby avoiding systemic circulation and off-targetmediated side effects. The ionic liquid composed of choline and octanoic acid (COA) at a 1:2 molar ionic ratio increases skin diffusion and transportation of NAVI and maintains their retention within the dermis for a prolonged duration. Topical administration of NAVI-mediated BCL-xL and BCL-2 inhibition results in the transition of myofibroblast to fibroblast and ameliorates pre-existing fibrosis, as demonstrated in a scleroderma mouse model. We have observed a significant reduction of α-SMA and collagen, which are known as fibrosis marker proteins, as a result of the inhibition of antiapoptotic proteins BCL-2/BCL-xL. Overall, our findings show that COA-assisted topical delivery of NAVI upregulates apoptosis specific to myofibroblasts, with minimal presence of the drug in the systemic circulation, resulting in an accelerated therapeutic effect with no discernible drug-associated toxicity.

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Section: Resultsmentioning

confidence: 99%

Topical Administration of an Apoptosis Inducer Mitigates Bleomycin-Induced Skin Fibrosis

Huda

Peurta

Guerena

et al. 2023

ACS Pharmacol. Transl. Sci.

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“…The potential risks associated with bleomycin include pulmonary fibrosis, anaphylaxis and hyperpigmentation of the skin. 24 In comparison, doxycycline is relatively inexpensive and can be easily prepared; it is widely available in many ophthalmic centres. 16 Doxycycline may be considered as a safe, effective and cheaper alternative for bleomycin in the treatment of periorbital LMs.…”

Section: Discussionmentioning

confidence: 99%

Doxycycline sclerotherapy in patients with periorbital lymphatic malformation: A case series and literature review

Lai

Kwok

Lam

et al. 2023

European Journal of Ophthalmology

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Purpose To describe and report the outcomes of doxycycline sclerotherapy in patients with periorbital lymphatic malformations(LMs). Basic procedures A retrospective review of consecutive patients diagnosed with periorbital LMs and who received doxycycline sclerotherapy at Hong Kong Eye Hospital and Queen Elizabeth Hospital, Hong Kong between January 2016 and June 2022. Doxycycline was prepared with a concentration of 100 mg diluted in 10 mL water for injection. A 23-gauge needle aiming at the center of the macrocyst was used to aspirate fluid from the lesion; this was then followed by an intralesional injection of 0.5 to 2 ml of doxycycline depending on the size of the cavity. Main findings A total of eight patients(six females) were included in this study. All of them received doxycycline sclerotherapy for periorbital LMs(five extraconal, three intraconal). The median age for receiving sclerotherapy was 29 years old. Seven patients had macrocystic LMs, and one had mixed macro- and microcystic LM. Two of the LMs had venous components radiologically. The average number of sclerotherapy treatment in one patient was 1.4 ± 0.7times. Seven of the eight patients had excellent response radiologically or clinically. One patient showed a satisfactory response after three cycles of sclerotherapy. No recurrence was experienced at median follow-up of 14 months. None of the patients experienced visual threatening or systemic complication. Principle conclusions Our preliminary experience with doxycycline sclerotherapy has shown encouraging results for the treatment of macrocystic or mixed-type periorbital LMs, with a favourable safety profile. Further clinical trials with longer follow-ups are warranted on this topic.

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“…Given a localized lesion, surgery resection and sclerotherapy are always the main choices for the management of both macro and mixed cLM (3,(16)(17)(18)(19). However, a series of cLM cases treated by surgery or sclerotherapy suffer from limited efficacy or local recurrence, especially for the cLM at the cervicofacial region, axilla, and joints.…”

Section: Discussionmentioning

confidence: 99%

Inflow Occlusion Combined With Bleomycin Sclerotherapy for Management of Macro/Mixed Cystic Lymphatic Malformation in Children

Han

Cui

et al. 2021

Front. Pediatr.

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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.

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Scleroterapia con bleomicina per malformazioni linfatiche dopo fallimento dell’escissione chirurgica: caso clinico

Cited by 15 publications

References 12 publications

Topical Administration of an Apoptosis Inducer Mitigates Bleomycin-Induced Skin Fibrosis

Topical Administration of an Apoptosis Inducer Mitigates Bleomycin-Induced Skin Fibrosis

Doxycycline sclerotherapy in patients with periorbital lymphatic malformation: A case series and literature review

Inflow Occlusion Combined With Bleomycin Sclerotherapy for Management of Macro/Mixed Cystic Lymphatic Malformation in Children

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