Abstract
We present a challenging case of HIV-related lymphangioma-like Kaposi sarcoma (LLKS) affecting the oral cavity. A 54-year-old Brazilian male patient was referred to our center complaining of bleeding lesions affecting the oral cavity for 2 months. Interestingly, these oral lesions were the first clinical manifestation of HIV infection. Clinically, multiple erythematous nodular and patch lesions were observed. An incisional biopsy was performed, revealing numerous microscopic angled and irregular vascular channels lined with flattened endothelial cells. More cellularized and solid areas consisting of more fusiform cells with little pleomorphism and with slit-like vascular channels were noted. Based on immunoreactivity for CD31, CD34, D2-40, and HHV-8, the final diagnosis was oral LLKS. Highly active antiretroviral therapy (HAART) was initiated with dolutegravir, tenofovir, and lamivudine. During follow-up, the patient showed KS metastases to other sites and a chemotherapeutic protocol was initiated. Regression of the oral lesion was clearly noted by the clinicians 1 year after the KS diagnosis. Dentists should be able to recognize systemic diseases that affect the oral cavity such as KS in order to make an early diagnosis of its oral manifestations and to implement effective therapeutic measures to ensure a better prognosis.
Similar content being viewed by others
References
Agaimy A, Mueller SK, Harrer T, Bauer S, Thompson LDR (2018) Head and neck Kaposi sarcoma: clinicopathological analysis of 11 cases. Head Neck Pathol 12:511–516
Cesarman E, Damania B, Krown SE, Martin J, Bower M, Whitby D (2019) Kaposi sarcoma Nat Rev Dis Primers 5:9
Kaposi M (1872) Idiopathisches multiples pigmentsarkom der haut [German]. Arch Dermatol Syph 4:265–273
Dittmer DP, Damania B (2016) Kaposi sarcoma-associated herpesvirus: immunobiology, oncogenesis, and therapy. J Clin Invest 126:3165–3175
Mesri EA, Cesarman E, Boshoff C (2010) Kaposi’s sarcoma and its associated herpesvirus. Nat Rev Cancer 10:707–719
El-Naggar AK, Chan JKC, Grandis JR, Takata T, Slootweg PJ (2017) WHO classification of head and neck tumours, 4th edn. IARC Press, Lyon
Pantanowitz L, Khammissa RAG, Lemmer J, Feller L (2013) Oral HIV-associated Kaposi sarcoma. J Oral Pathol Med 42:201–207
Will BM, Scott EB, Scott MP (2019) Oral Kaposi sarcoma: a case report and literature update. N Y State Dent J; Hempstead Vol. 85, Ed. 6: 22–24
Wang J, Stebbing J, Bower M (2007) HIV-associated Kaposi sarcoma and gender 4(3):266–273
Patton LL (2013) Oral Lesions associated with human immunodeficiency virus disease. Dent Clin North Am 57:673–698
AIDSinfo [Internet]. UNAIDS. 2020. Available from: http://aidsinfo.unaids.org/. Accessed Dec 2020
Brasil. Ministério da Saúde. Secretaria de Vigilância em Saúde. Departamento de Doenças de Condições Crônicas e Infecções Sexualmente Transmissíveis (DCCI) (2019) Relatório de monitoramento clínico do HIV / Ministério da Saúde, Secretaria de Vigilância em Saúde, Departamento de Doenças de Condições Crônicas e Infecções Sexualmente Transmissíveis (DCCI). – Brasília: Ministério da Saúde
Mangal TD, Pascom ARP, Vesga JF, Meireles MV, Benzakenb AS, Hallett TB (2019) Estimating HIV incidence from surveillance data indicates a second wave of infections in Brazil. Epidemics 27:77–85
Benzaken AS, Pereira GFM, Costa L, Tanuri A, Santos AF, Soares MA (2019) Antiretroviral treatment, government policy and economy of HIV/AIDS in Brazil: is it time for HIV cure in the country? AIDS Res Ther 16:19
Ribeiro LCS, Freitas MIF, Tupinambás U, Lana FCF (2020) Late diagnosis of human immunodeficiency virus infection and associated factors. Rev Lat Am Enfermagem 28:e3342
Patil N, Chaurasia VR, Babaji P, Ramesh D, Jhamb K, Sharma AM (2015) The effect of highly active antiretroviral therapy on the prevalence of oral manifestation in human immunodeficiency virus-infected patients in Karnataka, India. Eur J Dent 9:47–52
Oliscovicz NF, Pomarico L, Castro GF, Souza IP (2015) Effect of highly active antiretroviral therapy use on oral manifestations in pediatric patients infected with HIV. Indian J Dent Res 26:200–204
Patton LL (2016) Current strategies for prevention of oral manifestations of human immunodeficiency virus. Oral Surg Oral Med Oral Pathol Oral Radiol 121:29–38
de Almeida VL, Lima IFP, Ziegelmann PK, Paranhos LR, de Matos FR (2017) Impact of highly active antiretroviral therapy on the prevalence of oral lesions in HIV-positive patients: a systematic review and meta-analysis. Int J Oral Maxillofac Surg 46:1497–1504
Lager I, Altini M, Coleman H, Ali H (2003) Oral Kaposi’s sarcoma: a clinicopathologic study from South Africa. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 96:701–710
Mohanna S, Bravo F, Ferrufino JC, Sanchez J, Gotuzzo E (2007) Classic Kaposi’s sarcoma presenting in the oral cavity of two HIV-negative Quechua patients. Med Oral Patol Oral Cir Bucal 12:E365–E368
Fatahzadeh M, Schwartz RA (2013) Oral Kaposi’s sarcoma: a review and update. Int J Dermatol 52(6):666–672
Martellotta F, Berretta M, Vaccher E, Schioppa O, Zanet E, Tirelli U (2009) AIDS-related Kaposi’s sarcoma: state of the art and therapeutic strategies. Curr HIV Res 7(6):634–638
Bunn BK, Carvalho MV, Louw M, Vargas PA, van Heerden WF (2013) Microscopic diversity in oral Kaposi sarcoma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 115:241–248
Pugalagiri P, Muller S, Cox DP, Kessler HP, Wright JM, Cheng YSL (2013) Lymphangioma-like Kaposi sarcoma of the oral mucosa. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 116:84–90
Kotzias G, Agunbiade M, Isaac L, Khaladj M (2017) Human immunodeficiency virus–negative–associated lymphangioma-like Kaposi’s Sarcoma with variable clinical presentations. J Am Podiatr Med Assoc 107:461–466
Acknowledgements
This study was supported by the Coordination of Improvement of Higher Education Personnel (CAPES, Finance Code 001), Brazil. L.F.S. is the recipient of fellowships. We would like to thank the Brazilian National Council for Scientific and Technological Development for financial support (CNPq). M.D.M. and P.A.V. are research fellows of CNPq. The authors are also grateful to the São Paulo State Research Foundation (FAPESP 2016/21785-4) for a student scholarship.
Author information
Authors and Affiliations
Corresponding author
Ethics declarations
Ethical approval
For this type of retrospective case report, formal consent is not required.
Informed consent
No identifier information is included in the case report, and the study meets the waiver criteria for the institutional review board.
Conflict of interest
The authors declare no competing interests.
Additional information
Publisher’s note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
About this article
Cite this article
Schuch, L.F., Kovalski, L.N.S., Leite, A.A. et al. Oral lymphangioma-like Kaposi sarcoma: a Brazilian case report in a scenario of a still high number of HIV infections. Oral Maxillofac Surg 26, 171–176 (2022). https://doi.org/10.1007/s10006-021-00974-8
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s10006-021-00974-8