Autopsy and Case Reports
https://www.autopsyandcasereports.org/article/doi/10.4322/acr.2023.435
Autopsy and Case Reports
Clinical Case Report and Review

Unveiling an oral hemangiolymphangioma

Rívea Inês Ferreira-Santos; Kamilla Alves Santos; Alexandre Prado Scherma; Jorge Esquiche León; Estela Kaminagakura

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Abstract

Hemangiolymphangioma is a very rare vascular malformation that develops as a combination of dilated venous and lymphatic vessels. We describe an unusual case of hemangiolymphangioma of the tongue affecting an adult man who complained of an uncomfortable, slowly progressing exophytic irregular dark red-violaceous nodular mass on his tongue that impaired speech and swallowing for two weeks. The clinical differential diagnoses were Kaposi’s sarcoma and a COVID-19-related lesion. A complete blood count and serology for HIV-1 and 2 and RT-PCR for COVID-19 were requested and results were negative. An incisional biopsy was performed. Microscopically, the lesion exhibited several dilated vessels lined by normal-appearing endothelial cells, some filled with prominent intravascular erythrocytes and others containing proteinaceous eosinophilic material resembling lymphatic vessels, in close association with hyperkeratosis, papillomatosis, and acanthosis. From immunohistochemical analysis, most vessels were found to be CD34 positive, some highlighted by α-SMA, whereas D2-40 was focal. Positive staining for some lymphatic and blood vessel markers, i.e., D2-40 and CD34, respectively, indicates a mixed derivation of the lesion. HHV-8 was negative. Clinical features, the congested blood vessels with ectasia in intimate association with hyperplastic epithelium, and the immunohistochemical profile supported the final diagnosis of oral hemangiolymphangioma. The patient underwent minimally invasive surgical excision with no intercurrences. After 18 months of follow-up, there were no signs of relapse.

Keywords

Angiokeratoma, lymphangioma, COVID-19, tongue, case reports

References

1 Kulkarni CV, Nema P, Patidar H, Soni S, Tiwari NP. Hemangiolymphangioma of neck - a rare case with review report. J Med Sci Clin Res. 2014;2(8):1869-72.

2 Murphy T, Ramai D, Lai J, Sullivan K, Grimes C. Adult neck hemangiolymphangioma: a case and review of its etiology, diagnosis and management. J Surg Case Rep. 2017;2017(8):rjx168. http://dx.doi.org/10.1093/jscr/rjx168. PMid:28928923.

3 Manickam S, Sasikumar P, Kishore BN, Joy S. Hemangiolymphangioma of buccal mucosa: a rare case report. J Oral Maxillofac Pathol. 2017;21(2):282-5. http://dx.doi.org/10.4103/jomfp.JOMFP_28_17. PMid:28932041.

4 Khaunte DDN, Kumar PS, Dhupar V, Naik M. Hemangiolymphangioma of buccal cheek - a rare case report with review of literature. J Dent Health Oral Disord Ther. 2020;11(5):150-4. http://dx.doi.org/10.15406/jdhodt.2020.11.00534.

5 Shetty D, Rai H, Rastogi P, Panda A, Ahuja N. Vascular malformations of the oral cavity in children and young adolescents - insights into their pathogenesis. Internet J Ped Neonatol. 2009;12(2):1-5.

6 Merhi BA, Nous A, Rajab M. Hemangiolymphangioma of tongue, report of a rare case. Eur J Biomed Pharm Sci. 2016;3(5):88-91.

7 Sobhana CR, Beena VT, Soni A, Choudhary K, Sapru D. Hemangiolymphangioma of buccal mucosa: report of a rare case and review of literature on treatment aspect. Natl J Maxillofac Surg. 2012;3(2):190-4. http://dx.doi.org/10.4103/0975-5950.111379. PMid:23833496.

8 Vilalta J, Mascaro JM. Hemangiolymphangioma of the tongue treated by transfixion technique. J Dermatol Surg Oncol. 1985;11(2):168-70. http://dx.doi.org/10.1111/j.1524-4725.1985.tb02986.x. PMid:2981911.

9 Shetty DC, Urs AB, Rai HC, Ahuja N, Manchanda A. Case series on vascular malformation and their review with regard to terminology and categorization. Contemp Clin Dent. 2010;1(4):259-62. http://dx.doi.org/10.4103/0976-237X.76397. PMid:22114434.

10 Katsoulas N, Tosios KI, Argyris P, Koutlas IG, Sklavounou A. Lymphangioma circumscriptum, angiokeratoma, or superficial vascular ectasia with epithelial hyperplasia? Oral Surg Oral Med Oral Pathol Oral Radiol. 2014;118(2):e53-7. http://dx.doi.org/10.1016/j.oooo.2013.12.003. PMid:24491964.

11 Yarmand F, Seyyedmajidi M, Shirzadc A, Foroughi R, Bakhshian A. Lymphangiohemangioma of buccal mucosa: report of a rare case. J Oral Maxillofac Surg Med Pathol. 2016;28(4):358-61. http://dx.doi.org/10.1016/j.ajoms.2015.11.002.

12 Huang AY, Lin CL, Chen GS, Hu SC. Clinical features of Kaposi’s sarcoma: experience from a Taiwanese medical center. Int J Dermatol. 2019;58(12):1388-97. http://dx.doi.org/10.1111/ijd.14476. PMid:31102268.

13 Iranmanesh B, Khalili M, Amiri R, Zartab H, Aflatoonian M. Oral manifestations of COVID-19 disease: a review article. Dermatol Ther. 2021;34(1):e14578. http://dx.doi.org/10.1111/dth.14578. PMid:33236823.

14 Deliverska E. Hemangiolymphangioma of the mandible: case report. J IMAB. 2019;25(4):2729-32. http://dx.doi.org/10.5272/jimab.2019254.2729.

15 Wang L, Yuan W, Geng S, et al. Expression of lymphatic markers in angiokeratomas. J Cutan Pathol. 2014;41(7):576-81. http://dx.doi.org/10.1111/cup.12349. PMid:24666194.

16 Jian XC. Surgical management of lymphangiomatous or lymphangiohemangiomatous macroglossia. J Oral Maxillofac Surg. 2005;63(1):15-9. http://dx.doi.org/10.1016/j.joms.2004.04.024. PMid:15635551.

17 Kim SS. Intraosseous hemangiolymphangioma of the mandible: a case report. J Korean Assoc Oral Maxillofac Surg. 2003;29(3):182-5.

18 Hunchaisri N. Hemangiolymphangioma of the floor of mouth: a case report and literature review. J Med Health Sci. 2013;20(3):4-9.

19 Duque CS, Londoño AF, Penagos AM, Urquijo DP, Dueñas JP. Hypoglossal nerve monitoring, a potential application of intraoperative nerve monitoring in head and neck surgery. World J Surg Oncol. 2013;11:225. http://dx.doi.org/10.1186/1477-7819-11-225. PMid:24028712.

20 Gautam D, Pantha T. Lymphamgiohemangioma of face: case report. Nepalese J ENT Head Neck Surg. 2020;11(2):28-30.

21 Sion-Vardy N, Manor E, Puterman M, Bodner L. Solitary angiokeratoma of the tongue. Med Oral Patol Oral Cir Bucal. 2008;13(1):E12-4. PMid:18167473.

22 Meirelles DP, do Couto AM, Silva LVO, et al. Oral lymphatic malformations: a multicenter study of 208 cases and literature review. Head Neck. 2021;43(11):3562-71. http://dx.doi.org/10.1002/hed.26854. PMid:34517432.

23 Brahmbhatt AN, Skalski KA, Bhatt AA. Vascular lesions of the head and neck: an update on classification and imaging review. Insights Imaging. 2020;11(1):19. http://dx.doi.org/10.1186/s13244-019-0818-3. PMid:32034537.
 


Submitted date:
10/21/2022

Accepted date:
04/29/2023

Publication date:
05/24/2023

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