Radiotherapy in the Treatment of Lip localized Lymphangioma. Case Report and Literature Review
Year 2024,
Volume: 1 Issue: 1, 18 - 30, 20.03.2024
Sema Rakıcı
,
Yunus Çınar
Abstract
Abstract
Lymphangiomas are developmental malformations that occur as benign tumors of the lymphatic channels. They occur very early in life and mostly prefer the head and neck region. In this case, we present a 21-year-old male patient with predominantly lower lip hemangioma from birth. Despite repeated surgical procedures and cryotherapy, growth continued and could only be controlled with external radiotherapy.
However, the doses of radiotherapy described so far for the treatment of these lesions seem inadequate for the control large lesions. We had to beam 3 times to check this case. We found that an average doses of 54-56 Gy (50-60 Gy) recommended for the control of some benign vascular diseases can be used in these cases.
References
- 1. Miceli A, Stewart KM. Lymphangioma. [Updated 2023 Aug 8]. In: StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing; 2024 Jan-. Available from: https://www.ncbi.nlm.nih.gov/books/NBK470333/
- 2. Haidry N, Shivhare P, Vaidyanathan A, Mokhtar EA, Ghimire AC, Shivhare Sr P, et al. Lymphangioma of the Oral and Maxillofacial Region: A Report of Three Cases. Cureus. 2022;14(12).
- 3. Reinhardt MA, Nelson SC, Sencer SF, Bostrom BC, Kurachek SC, Nesbit ME. Treatment of childhood lymphangiomas with interferon-α. Journal of pediatric hematology/oncology. 1997;19(3):232-6.
- 4. Saijo M, Munro IR, Mancer K. Lymphangioma A Long-Term Follow-Up Study. Plastic and reconstructive surgery. 1975;56(6):642-51.
- 5. Chouchene F, Masmoudi F, Baaziz A, Maatouk F, Ghedira H. Oral manifestations and dental care management of a young patient with lymphangioma of the tongue: A case report. Clinical Case Reports. 2021;9(7):e04537.
- 6. Luzzatto C, Midrio P, Tchaprassian Z, Guglielmi M. Sclerosing treatment of lymphangiomas with OK-432. Archives of disease in childhood. 2000;82(4):316-8.
- 7. Hasan S, Ahmad SA, Kaur M, Panigrahi R, Panda S. Lymphangioma of the Lower Lip-A Diagnostic Dilemma: Report of a Rare Case with a Brief Literature Review. Case Rep Dent. 2022;2022:7890338.
- 8. Riechelmann H, Muehlfay G, Keck T, Mattfeldt T, Rettinger G. Total, subtotal, and partial surgical removal of cervicofacial lymphangiomas. Archives of Otolaryngology–Head & Neck Surgery. 1999;125(6):643-8.
- 9. Jian X-c. Surgical management of lymphangiomatous or lymphangiohemangiomatous macroglossia. Journal of oral and maxillofacial surgery. 2005;63(1):15-9.
- 10. Bhayya H, Pavani D, Tejasvi MA, Geetha P. Oral lymphangioma: A rare case report. Contemporary clinical dentistry. 2015;6(4):584.
- 11. Manzini M, Schweiger C, Manica D, Kuhl G. Response to OK-432 sclerotherapy in the treatment of cervical lymphangioma with submucosal extension to the airway. Brazilian Journal of Otorhinolaryngology. 2020;86:127-9.
- 12. AlGhamdi A, AlBaqami M. Lymphangioma of the tongue in a pediatric patient. Journal of Pediatric Surgery Case Reports. 2021;75:102053.
- 13. Kuriakose, M.A., Chakrabarti, S., Cheong, S.C., Kowalski, L.P., Novaes Pinheiro, T., Farah, C.S. (2019). Head and Neck Tumors. In: Farah, C., Balasubramaniam, R., McCullough, M. (eds) Contemporary Oral Medicine. Springer, Cham. https://doi.org/10.1007/978-3-319-72303-7_20.
- 14. Farah C, Balasubramaniam R, McCullough MJ. Contemporary Oral Medicine. Switzerland: Springer, 2019.
- 15. Greinwald Jr JH, Burke DK, Sato Y, Poust RI, Kimura K, Bauman NM, et al. Treatment of lymphangiomas in children: an update of Picibanil (OK-432) sclerotherapy. Otolaryngology--Head and Neck Surgery. 1999;121(4):381-7.
- 16. Jham BC, da Silva Freire AR. Oral complications of radiotherapy in the head and neck. Brazilian journal of otorhinolaryngology. 2006;72(5):704-8.
- 17. Sroussi HY, Epstein JB, Bensadoun RJ, Saunders DP, Lalla RV, Migliorati CA, et al. Common oral complications of head and neck cancer radiation therapy: mucositis, infections, saliva change, fibrosis, sensory dysfunctions, dental caries, periodontal disease, and osteoradionecrosis. Cancer medicine. 2017;6(12):2918-31.
- 18. Bennardo L, Passante M, Cameli N, Cristaudo A, Patruno C, Nisticò SP, et al. Skin manifestations after ionizing radiation exposure: a systematic review. Bioengineering. 2021;8(11):153.
- 19. Wege J, Anabtawi M, Blackwell MA, Patterson A. Lymphangioma Formation Following Hyaluronic Acid Injection for Lip Augmentation. Cureus. 2021;13(1).
- 20. Nagata M, Semha I, Ooya K, Urago A, Yonezawa S, Sakae K. Malignant endothelial neoplasm arising in the area of lymphangioma: immunohistochemical and ultrastructural observation. Journal of Oral Pathology & Medicine. 1984;13(6):560-72.
- 21. Fukuda M, Matsuda K, Itoh M, Shimoda H, Echigo S, Teshima T. A case of lymphangioma in the region of lower lip and buccal mucosa of infant with repetition of recurrences. Japanese Journal of Oral and Maxillofacial Surgery. 1989;35(3):673-9.
- 22. Balakrishnan A, Bailey C. Lymphagioma of the tongue. A review of pathogenesis, treatment and the use of surface laser photocoagulation. The Journal of Laryngology & Otology. 1991;105(11):924-9.
- 23. Park YW, Kim SM, Min BG, Park IW, Lee SK. Lymphangioma involving the mandible: immunohistochemical expressions for the lymphatic proliferation. Journal of oral pathology & medicine. 2002;31(5):280-3.
- 24. Gul A, Kucuker I, Can E, Niyaz L, Yucel OE. A propranolol nonresponsive mass: lymphangioma-mimicking hemangioma. Journal of Craniofacial Surgery. 2015;26(1):327-8.
- 25. Nagaoka H, Okada H, Yamamoto H, Kondoh T. Cavernous lymphangioma of the lower lip: clinicopathological and immunohistochemical study. International Journal of Oral-Medical Sciences. 2008;7(2):123-7.
- 26. Kolay SK, Jha PK, Sinha RK, Kumar P. Oral lymphangioma: a review. J Clin Den Res Edu. 2012;1(1):42-6.
- 27. Goud S, Swapna T, Bindu H, Shylaja SRG. Extra nasopharyngeal angiofibroma simulating a mucocele: a new location for the rare entity. Journal of Clinical and Diagnostic Research: JCDR. 2017;11(1):ZD28.
- 28. de Carvalho FK, Pinheiro TN, da Silva RAB, de Queiroz AM, da Silva LAB, Nelson-Filho P. Lymphangioma of the lower lip mimicking a mucocele in children. Journal of Dentistry for Children. 2015;82(2):116-9.
- 29. Fernandes AIB, Cunha RG, De Farias MML, Barbosa JB, Kimura MK, Câmara J, et al. Lymphangioma of the Lower Lip of a 68-Year-Old Woman: a Case Report. Oral surgery, oral medicine, oral pathology and oral radiology. 2018;126(3):e95.
- 30. Matharu J, Patel V. Cryotherapy for the management of a lip lymphangioma–a case report and literature review. Oral Surgery. 2019;12(3):230-7.
- 31. Díaz-Flores L, Gutiérrez R, Gayoso S, García M, González-Gómez M, Díaz-Flores Jr L, et al. Intussusceptive angiogenesis and its counterpart intussusceptive lymphangiogenesis. Histology and Histopathology. 2020;35(10):1083-103.
- 32. dos Santos ALM, dos Santos Oliveira P, de Amorim Campos V, de Abreu Miranda RP, de Carvalho E. Giant mesenteric lymphangioma. Journal of Pediatric Surgery Case Reports. 2020;59:101491.
- 33. Kurude AA, Phiske MM, Kolekar KK, Nayak CS. Lymphangiomas: rare presentations in oral cavity and scrotum in pediatric age group. Indian Journal of Dermatology, Venereology and Leprology. 2020;86:230.
- 34. Goyal R, Jaiswal P, Akhter J, Dubey C, Akshay B, Gautam G. Lymphangioma circumscriptum of lower lip–A Rare Case Report.
- 35. Mäkinen T, Boon LM, Vikkula M, Alitalo K. Lymphatic Malformations: Genetics, Mechanisms and Therapeutic Strategies. Circ Res. 2021 Jun 25;129(1):136-154.
- 36. Sakhiya J, Sakhiya D, Kaklotar J, Hirapara B, Purohit M, Bhalala K, et al. Intralesional agents in dermatology: pros and cons. Journal of Cutaneous and Aesthetic Surgery. 2021;14(3):285.
- 37. Rakici SY, Tumkaya L, Edirvanli OC, Yazici U, Dursun E, Arpa M, Mercantepe T. Radioprotective effect of endogenous melatonin secretion associated with the circadian rhythm in irradiated rats. Int J Radiat Biol. 2019;95(9):1236-1241.
- 38. Rakici SY, Koksal V, Bedir R, Goksel S. An urban legend: Malignant transformation caused by radiotherapy in patients with presacral ganglioneuroma. The necessity and first-time administration of radiotherapy. Case report and literature review. J Cancer Res Ther. 2021;17(1):248-254.
- 39. Rakici, SY, Cinar , Eren M. Total scalp irradiation: the comparison of five different plans using volumetric modulated arc therapy-simultaneous integrated boost (VMAT-SIB) technique. Turk J Oncol. 2017;32(3):106–115.
Radiotherapy in the Treatment of Lip localized Lymphangioma. Case Report and Literature Review
Year 2024,
Volume: 1 Issue: 1, 18 - 30, 20.03.2024
Sema Rakıcı
,
Yunus Çınar
Abstract
Abstract
Lymphangiomas are developmental malformations that occur as benign tumors of the lymphatic channels. They occur very early in life and mostly prefer the head and neck region. In this case, we present a 21-year-old male patient with predominantly lower lip hemangioma from birth. Despite repeated surgical procedures and cryotherapy, growth continued and could only be controlled with external radiotherapy. However, the doses of radiotherapy described so far for the treatment of these lesions seem inadequate for the control large lesions. We had to beam 3 times to check this case. We found that an average doses of 54-56 Gy (50-60 Gy) recommended for the control of some benign vascular diseases can be used in these cases.
References
- 1. Miceli A, Stewart KM. Lymphangioma. [Updated 2023 Aug 8]. In: StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing; 2024 Jan-. Available from: https://www.ncbi.nlm.nih.gov/books/NBK470333/
- 2. Haidry N, Shivhare P, Vaidyanathan A, Mokhtar EA, Ghimire AC, Shivhare Sr P, et al. Lymphangioma of the Oral and Maxillofacial Region: A Report of Three Cases. Cureus. 2022;14(12).
- 3. Reinhardt MA, Nelson SC, Sencer SF, Bostrom BC, Kurachek SC, Nesbit ME. Treatment of childhood lymphangiomas with interferon-α. Journal of pediatric hematology/oncology. 1997;19(3):232-6.
- 4. Saijo M, Munro IR, Mancer K. Lymphangioma A Long-Term Follow-Up Study. Plastic and reconstructive surgery. 1975;56(6):642-51.
- 5. Chouchene F, Masmoudi F, Baaziz A, Maatouk F, Ghedira H. Oral manifestations and dental care management of a young patient with lymphangioma of the tongue: A case report. Clinical Case Reports. 2021;9(7):e04537.
- 6. Luzzatto C, Midrio P, Tchaprassian Z, Guglielmi M. Sclerosing treatment of lymphangiomas with OK-432. Archives of disease in childhood. 2000;82(4):316-8.
- 7. Hasan S, Ahmad SA, Kaur M, Panigrahi R, Panda S. Lymphangioma of the Lower Lip-A Diagnostic Dilemma: Report of a Rare Case with a Brief Literature Review. Case Rep Dent. 2022;2022:7890338.
- 8. Riechelmann H, Muehlfay G, Keck T, Mattfeldt T, Rettinger G. Total, subtotal, and partial surgical removal of cervicofacial lymphangiomas. Archives of Otolaryngology–Head & Neck Surgery. 1999;125(6):643-8.
- 9. Jian X-c. Surgical management of lymphangiomatous or lymphangiohemangiomatous macroglossia. Journal of oral and maxillofacial surgery. 2005;63(1):15-9.
- 10. Bhayya H, Pavani D, Tejasvi MA, Geetha P. Oral lymphangioma: A rare case report. Contemporary clinical dentistry. 2015;6(4):584.
- 11. Manzini M, Schweiger C, Manica D, Kuhl G. Response to OK-432 sclerotherapy in the treatment of cervical lymphangioma with submucosal extension to the airway. Brazilian Journal of Otorhinolaryngology. 2020;86:127-9.
- 12. AlGhamdi A, AlBaqami M. Lymphangioma of the tongue in a pediatric patient. Journal of Pediatric Surgery Case Reports. 2021;75:102053.
- 13. Kuriakose, M.A., Chakrabarti, S., Cheong, S.C., Kowalski, L.P., Novaes Pinheiro, T., Farah, C.S. (2019). Head and Neck Tumors. In: Farah, C., Balasubramaniam, R., McCullough, M. (eds) Contemporary Oral Medicine. Springer, Cham. https://doi.org/10.1007/978-3-319-72303-7_20.
- 14. Farah C, Balasubramaniam R, McCullough MJ. Contemporary Oral Medicine. Switzerland: Springer, 2019.
- 15. Greinwald Jr JH, Burke DK, Sato Y, Poust RI, Kimura K, Bauman NM, et al. Treatment of lymphangiomas in children: an update of Picibanil (OK-432) sclerotherapy. Otolaryngology--Head and Neck Surgery. 1999;121(4):381-7.
- 16. Jham BC, da Silva Freire AR. Oral complications of radiotherapy in the head and neck. Brazilian journal of otorhinolaryngology. 2006;72(5):704-8.
- 17. Sroussi HY, Epstein JB, Bensadoun RJ, Saunders DP, Lalla RV, Migliorati CA, et al. Common oral complications of head and neck cancer radiation therapy: mucositis, infections, saliva change, fibrosis, sensory dysfunctions, dental caries, periodontal disease, and osteoradionecrosis. Cancer medicine. 2017;6(12):2918-31.
- 18. Bennardo L, Passante M, Cameli N, Cristaudo A, Patruno C, Nisticò SP, et al. Skin manifestations after ionizing radiation exposure: a systematic review. Bioengineering. 2021;8(11):153.
- 19. Wege J, Anabtawi M, Blackwell MA, Patterson A. Lymphangioma Formation Following Hyaluronic Acid Injection for Lip Augmentation. Cureus. 2021;13(1).
- 20. Nagata M, Semha I, Ooya K, Urago A, Yonezawa S, Sakae K. Malignant endothelial neoplasm arising in the area of lymphangioma: immunohistochemical and ultrastructural observation. Journal of Oral Pathology & Medicine. 1984;13(6):560-72.
- 21. Fukuda M, Matsuda K, Itoh M, Shimoda H, Echigo S, Teshima T. A case of lymphangioma in the region of lower lip and buccal mucosa of infant with repetition of recurrences. Japanese Journal of Oral and Maxillofacial Surgery. 1989;35(3):673-9.
- 22. Balakrishnan A, Bailey C. Lymphagioma of the tongue. A review of pathogenesis, treatment and the use of surface laser photocoagulation. The Journal of Laryngology & Otology. 1991;105(11):924-9.
- 23. Park YW, Kim SM, Min BG, Park IW, Lee SK. Lymphangioma involving the mandible: immunohistochemical expressions for the lymphatic proliferation. Journal of oral pathology & medicine. 2002;31(5):280-3.
- 24. Gul A, Kucuker I, Can E, Niyaz L, Yucel OE. A propranolol nonresponsive mass: lymphangioma-mimicking hemangioma. Journal of Craniofacial Surgery. 2015;26(1):327-8.
- 25. Nagaoka H, Okada H, Yamamoto H, Kondoh T. Cavernous lymphangioma of the lower lip: clinicopathological and immunohistochemical study. International Journal of Oral-Medical Sciences. 2008;7(2):123-7.
- 26. Kolay SK, Jha PK, Sinha RK, Kumar P. Oral lymphangioma: a review. J Clin Den Res Edu. 2012;1(1):42-6.
- 27. Goud S, Swapna T, Bindu H, Shylaja SRG. Extra nasopharyngeal angiofibroma simulating a mucocele: a new location for the rare entity. Journal of Clinical and Diagnostic Research: JCDR. 2017;11(1):ZD28.
- 28. de Carvalho FK, Pinheiro TN, da Silva RAB, de Queiroz AM, da Silva LAB, Nelson-Filho P. Lymphangioma of the lower lip mimicking a mucocele in children. Journal of Dentistry for Children. 2015;82(2):116-9.
- 29. Fernandes AIB, Cunha RG, De Farias MML, Barbosa JB, Kimura MK, Câmara J, et al. Lymphangioma of the Lower Lip of a 68-Year-Old Woman: a Case Report. Oral surgery, oral medicine, oral pathology and oral radiology. 2018;126(3):e95.
- 30. Matharu J, Patel V. Cryotherapy for the management of a lip lymphangioma–a case report and literature review. Oral Surgery. 2019;12(3):230-7.
- 31. Díaz-Flores L, Gutiérrez R, Gayoso S, García M, González-Gómez M, Díaz-Flores Jr L, et al. Intussusceptive angiogenesis and its counterpart intussusceptive lymphangiogenesis. Histology and Histopathology. 2020;35(10):1083-103.
- 32. dos Santos ALM, dos Santos Oliveira P, de Amorim Campos V, de Abreu Miranda RP, de Carvalho E. Giant mesenteric lymphangioma. Journal of Pediatric Surgery Case Reports. 2020;59:101491.
- 33. Kurude AA, Phiske MM, Kolekar KK, Nayak CS. Lymphangiomas: rare presentations in oral cavity and scrotum in pediatric age group. Indian Journal of Dermatology, Venereology and Leprology. 2020;86:230.
- 34. Goyal R, Jaiswal P, Akhter J, Dubey C, Akshay B, Gautam G. Lymphangioma circumscriptum of lower lip–A Rare Case Report.
- 35. Mäkinen T, Boon LM, Vikkula M, Alitalo K. Lymphatic Malformations: Genetics, Mechanisms and Therapeutic Strategies. Circ Res. 2021 Jun 25;129(1):136-154.
- 36. Sakhiya J, Sakhiya D, Kaklotar J, Hirapara B, Purohit M, Bhalala K, et al. Intralesional agents in dermatology: pros and cons. Journal of Cutaneous and Aesthetic Surgery. 2021;14(3):285.
- 37. Rakici SY, Tumkaya L, Edirvanli OC, Yazici U, Dursun E, Arpa M, Mercantepe T. Radioprotective effect of endogenous melatonin secretion associated with the circadian rhythm in irradiated rats. Int J Radiat Biol. 2019;95(9):1236-1241.
- 38. Rakici SY, Koksal V, Bedir R, Goksel S. An urban legend: Malignant transformation caused by radiotherapy in patients with presacral ganglioneuroma. The necessity and first-time administration of radiotherapy. Case report and literature review. J Cancer Res Ther. 2021;17(1):248-254.
- 39. Rakici, SY, Cinar , Eren M. Total scalp irradiation: the comparison of five different plans using volumetric modulated arc therapy-simultaneous integrated boost (VMAT-SIB) technique. Turk J Oncol. 2017;32(3):106–115.