Main Article Content

Abstract

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare reactive angio-proliferative lesion. ALHE was initially classified as the late stage of Kimura’s disease (KD), although studies later showed that they were two separate clinical entities. Diagnosing ALHE remains a clinical challenge. Here, we report a case of ALHE in a young man and review current literature with an emphasis on how to distinguish ALHE particularly from KD. A 26-year-old man presented with a subcutaneous nodule in right infra-auricular area. Recurrence had occurred after three surgical excisions. Neither enlargement of salivary glands nor lymph nodes were found. Hematological examinations and renal function were normal. The mass was removed surgically. Microscopic examination showed proliferation of vascular channels with accompanying mixed inflammatory infiltrate consisting of lymphocytes, plasma cells, and eosinophils. Based on clinical data and histopathological examination, the patient was diagnosed with ALHE. Several clinical features differ between ALHE and KD, such as gender predilection, hypereosinophilia, IgE levels, and renal involvement. However, clinical features can overlap, so definitive diagnosis relies on histopathological examination. The most important hallmark of ALHE is vascular proliferation with epithelioid endothelial cells. Distinguishing ALHE from KD is important due to the lack of systemic manifestations in ALHE. However, ALHE can be easily mistaken for other diseases due to its rarity. Careful microscopic examination is very important to distinguish ALHE from KD and other mimicking lesions.

Keywords

Angiolymphoid hyperplasia eosinophilia epithelioid hemangioma Kimura’s disease skin tumor

Article Details

How to Cite
Wahyuningsih, L., Pudjohartono, M. F., & Rinonce, H. T. (2021). Angiolymphoid hyperplasia with eosinophilia: A potential mimic of kimura’s disease. JKKI : Jurnal Kedokteran Dan Kesehatan Indonesia, 12(2), 198–203. https://doi.org/10.20885/JKKI.Vol12.Iss2.art14

References

  1. Adler BL, Krausz AE, Minuti A, Silverberg JI, Lev-Tov H. Epidemiology and treatment of angiolymphoid hyperplasia with eosinophilia (ALHE): A systematic review. Journal of the American Academy of Dermatology. 2016;74(3):506-512.e11.
  2. Guo R, Gavino ACP. Angiolymphoid hyperplasia with eosinophilia. Archives of Pathology and Laboratory Medicine. 2015;139(5):683–6.
  3. de Bastos JT, e Silva PMC, Cassia F de F, da Rocha CRM, de Freitas BMP, Avelleira JCR. Angiolymphoid hyperplasia with eosinophilia versus Kimura’s disease: A case report and a clinical and histopathological comparison. Anais Brasileiros de Dermatologia. 2017;92(3):392–4.
  4. Santosa C, Wardhana M, Saputra H. Angiolymphoid hyperplasia with eosinophilia with clinical pictures of keratoacanthoma: A rare case report. Clinical Case Reports. 2019;7(1):189–92.
  5. Wells GC, Whimster IW. Subcutaneous angiolymphoid hyperplasia with eosinophilia. British Journal of Dermatology. 1969;81(1):1–15.
  6. Mukherjee B, Kadaskar J, Priyadarshini O, Krishnakumar S, Biswas J. Angiolymphoid hyperplasia with eosinophilia of the orbit and adnexa. Ocular Oncology and Pathology. 2016;2(1):40–7.
  7. Guinovart RM, Bassas-Vila J, Morell L, Ferrándiz C. Angiolymphoid hyperplasia with eosinophilia: A clinicopathologic study of 9 cases. Actas Dermo-Sifiliograficas. 2014;105(2).
  8. Dhingra H, Nagpal R, Baliyan A, Alva SR. Kimura disease: Case report and brief review of literature. Medicine and Pharmacy Reports. 2019;92(2):195–9.
  9. Zhang G, Qi W, Li X, Sun G, Cao Y, Gao N. Clinical analysis of Kimura’s disease in 24 cases from China. BMC Surgery:2020;20:1.
  10. Buder K, Ruppert S, Trautmann A, Bröcker E-B, Goebeler M, Kerstan A. Angiolymphoid hyperplasia with eosinophilia and Kimura’s disease-a clinical and histopathological comparison. JDDG: Journal der Deutschen Dermatologischen Gesellschaft. 2014;12(3):224–8.
  11. Reddy PKS, Prasad ALS, Sumathy TK, Shivaswamy KN, Ranganathan C. An overlap of angiolymphoid hyperplasia with eosinophilia and Kimura’s disease: Successful treatment of skin lesions with cryotherapy. Indian Journal of Dermatology. 2015;60(2):216.
  12. Sardaro A, Bardoscia L, Petruzzelli MF, Portaluri M. Epithelioid hemangioendothelioma: An overview and update on a rare vascular tumor. Oncology Reviews. 2014;8
  13. Tenório J da R, Gonzaga AKG, Gonçalves PGP, de Oliveira DHIP, Queiroz LMG. Angiolymphoid hyperplasia with eosinophilia: A rare case in the oral cavity. Jornal Vascular Brasileiro. 2016;15(4):317–21.
  14. Youssef A, Hasan AR, Youssef Y, Al-Soufi L, Elshimali Y, Alshehabi Z. Angiolymphoid hyperplasia with eosinophilia: A case report. Journal of Medical Case Reports. 2018;12(1).
  15. Singh P, Singh A. A rare case of angiolymphoid hyperplasia with eosinophilia in the submental region. Journal of Oral and Maxillofacial Pathology. 2013;17(2):311–4.
  16. Krausz AE, Lev-Tov H, Adler BL, Silverberg JI, Friedman A EA. Treatment of angiolymphoid hyperplasia with eosinophilia: Systematic review. Journal of the American Academy of Dermatology. 2015;72(5):AB66.
  17. Tambe SA, Nayak CS. Successful management of angiolymphoid hyperplasia with eosinophilia by radiofrequency. Journal of Cutaneous and Aesthetic Surgery. 2017;10(2):116–8.