Subscribe to RSS
DOI: 10.1055/a-2129-5668
Intralesional Bevacizumab in Periorbital Recurrence of an Orbital Lymphaticovenous Malformation
Bevacizumab intraläsional bei einem periorbitalen Rezidiv einer orbitalen lymphatisch-venösen MalformationIntroduction
Lymphaticovenous malformations (LVMs) of the orbit, formerly known as orbital lymphangiomas, are vascular, venous, or lymphatic dominant malformations, accounting for 1 – 4% of all orbital lesions [1], [2], [3]. They are primarily seen within the first two decades of life [4] and can grow in size due to reaction of the lymphatic tissue during infection [5], thrombosis, or hemorrhage [6]. LVMs may involve the superficial parts of the ocular adnexa, including eyelids, conjunctiva, and the anterior orbit, leading to disfigurement, as well as deeper parts of the orbit, with the risk of optic nerve compression, proptosis, diplopia, reduced vision, or amblyopia [2], [7]. LVMs can be classified as macrocytic (> 1 cm3) and microcytic (< 1 cm3), referring to the size of the cysts forming the lesion [8]. Management of LVMs has always been challenging due to the diffuse growth of the lesions. Conservative observation of small and asymptomatic LVMs is preferred. There are different treatment modalities [6] if reduction of size is necessary, including tumor debulking, radiation, or injection of sclerosing agents, e.g., bleomycin [6], [9]. However, the recurrency rates as well as intra- and postoperative bleeding complications call for therapeutic alternatives. Understanding that vascular endothelial growth factor (VEGF) takes part in lymphangiogenesis brought up the idea to use it as a therapeutic target and to treat LVMs with an anti-VEGF drug like bevacizumab [6], [10]. We describe the case of a patient with periorbital recurrence of a periorbital LVM that was successfully treated with an intralesional injection of bevacizumab.
Publication History
Received: 22 December 2022
Accepted: 13 July 2023
Accepted Manuscript online:
14 July 2023
Article published online:
06 September 2023
© 2023. Thieme. All rights reserved.
Georg Thieme Verlag KG
Rüdigerstraße 14, 70469 Stuttgart, Germany
-
References
- 1 Katz SE, Rootman J, Vangveeravong S. et al. Combined venous lymphatic malformations of the orbit (so-called lymphangiomas). Association with noncontiguous intracranial vascular anomalies. Ophthalmology 1998; 105: 176-184 DOI: 10.1016/s0161-6420(98)92058-9.
- 2 Greene AK, Burrows PE, Smith L. et al. Periorbital lymphatic malformation: clinical course and management in 42 patients. Plast Reconstr Surg 2005; 115: 22-30
- 3 Bonavolonta G, Strianese D, Grassi P. et al. An analysis of 2,480 space-occupying lesions of the orbit from 1976 to 2011. Ophthalmic Plast Reconstr Surg 2013; 29: 79-86 DOI: 10.1097/IOP.0b013e31827a7622.
- 4 Wright JE, Sullivan TJ, Garner A. et al. Orbital venous anomalies. Ophthalmology 1997; 104: 905-913 DOI: 10.1016/s0161-6420(97)30208-5.
- 5 Jones IS. Lymphangiomas of the ocular adnexa. An analysis of sixty-two cases. Am J Ophthalmol 1961; 51: 481-509 DOI: 10.1016/0002-9394(61)92094-3.
- 6 Nassiri N, Rootman J, Rootman DB. et al. Orbital lymphaticovenous malformations: Current and future treatments. Surv Ophthalmol 2015; 60: 383-405 DOI: 10.1016/j.survophthal.2015.03.001.
- 7 Tunc M, Sadri E, Char DH. Orbital lymphangioma: an analysis of 26 patients. Br J Ophthalmol 1999; 83: 76-80 DOI: 10.1136/bjo.83.1.76.
- 8 Rootman J, Heran MK, Graeb DA. Vascular malformations of the orbit: classification and the role of imaging in diagnosis and treatment strategies*. Ophthalmic Plast Reconstr Surg 2014; 30: 91-104 DOI: 10.1097/IOP.0000000000000122.
- 9 Gooding C, Meyer D. Intralesional bleomycin: a potential treatment for refractory orbital lymphangiomas. Ophthalmic Plast Reconstr Surg 2014; 30: e65-e67 DOI: 10.1097/IOP.0b013e31829bb4a9.
- 10 Mustak H, Ugradar S, Goldberg R. et al. Bevacizumab and Bleomycin combination for treatment of orbital lymphatico-venous malformation recalcitrant to sclerosing therapy alone. Clin Exp Ophthalmol 2018; 46: 815-816 DOI: 10.1111/ceo.13159.
- 11 Tan JC, Mann S, Coroneo MT. Successful Treatment of Conjunctival Lymphangiectasia With Subconjunctival Injection of Bevacizumab. Cornea 2016; 35: 1375-1377 DOI: 10.1097/ICO.0000000000000899.
- 12 Liang WC, Wu X, Peale FV. et al. Cross-species vascular endothelial growth factor (VEGF)-blocking antibodies completely inhibit the growth of human tumor xenografts and measure the contribution of stromal VEGF. J Biol Chem 2006; 281: 951-961 DOI: 10.1074/jbc.M508199200.
- 13 Sidle DM, Maddalozzo J, Meier JD. et al. Altered pigment epithelium-derived factor and vascular endothelial growth factor levels in lymphangioma pathogenesis and clinical recurrence. Arch Otolaryngol Head Neck Surg 2005; 131: 990-995 DOI: 10.1001/archotol.131.11.990.
- 14 Abdelaziz O, Hassan F, Elessawy K. et al. Image-Guided Percutaneous Bleomycin and Bevacizumab Sclerotherapy of Orbital Lymphatic Malformations in Children. Cardiovasc Intervent Radiol 2019; 42: 433-440 DOI: 10.1007/s00270-018-2128-4.