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Ear lobule reduction using a sub-antitragal groove technique in patients with angiolymphoid hyperplasia with eosinophilia on the earlobe: a case report and literature review

  • Yijun Moon (Department of Plastic and Reconstructive Surgery, Korea University Anam Hospital, Korea University College of Medicine) ;
  • Haneul Kim (Department of Plastic and Reconstructive Surgery, Korea University Anam Hospital, Korea University College of Medicine) ;
  • Hojin Park (Department of Plastic and Reconstructive Surgery, Korea University Anam Hospital, Korea University College of Medicine)
  • Received : 2023.11.10
  • Accepted : 2024.04.26
  • Published : 2024.08.20

Abstract

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare condition characterized by nodular lesions predominantly in the head and neck region, often causing discomfort or pain. Treatment remains challenging because of its rarity and the lack of established guidelines. This report presents a case of ALHE affecting the earlobes that was successfully managed using ear lobule reduction surgery and subsequent intralesional steroid injections. A 31-year-old woman with a history of recurrent earlobe masses underwent a partial excision to avoid the loss of the earlobe. Histopathological examination confirmed Kimura disease, a variant of ALHE. Subsequent local methylprednisolone injections effectively controlled the remaining lesions, resulting in significant size reduction without notching. Various treatment modalities have been attempted for this condition; however, recurrence rates remain high. Surgical resection combined with intralesional corticosteroid injections is the preferred approach. In this case, a sub-antitragal groove technique for earlobe reduction was employed to preserve the lateral edge of the ear lobule, minimize the risk of deformity, and achieve a predictable outcome. The sub-antitragal groove technique offers an approach to reduce earlobe size without compromising aesthetics. Further research is required to elucidate the pathogenesis of ALHE and establish standardized treatment protocols for this rare condition.

Keywords

References

  1. Akdeniz N, Kosem M, Calka O, Bilgili SG, Metin A, Gelincik I. Intralesional bleomycin for angiolymphoid hyperplasia. Arch Dermatol 2007;143:841-4. 
  2. Busquets AC, Sanchez JL. Angiolymphoid hyperplasia with eosinophilia induced by trauma. Int J Dermatol 2006;45:1211-4. 
  3. Fink CM, Maggio KL. Angiolymphoid hyperplasia with eosinophilia revisited: lack of durable response to intralesional interferon alfa-2a. Arch Dermatol 2011;147:507-8. 
  4. Isohisa T, Masuda K, Nakai N, Takenaka H, Katoh N. Angiolymphoid hyperplasia with eosinophilia treated successfully with imiquimod. Int J Dermatol 2014;53:e43-4. 
  5. Park JS, Lee MJ. A case of angiolymphoid hyperplasia with eosinophilia (ALHE) in the genital area accompanied by varicocele. Int J Dermatol 2009;48:1264-6. 
  6. Adler BL, Krausz AE, Minuti A, Silverberg JI, Lev-Tov H. Epidemiology and treatment of angiolymphoid hyperplasia with eosinophilia (ALHE): a systematic review. J Am Acad Dermatol 2016;74:506-12. 
  7. Buder K, Ruppert S, Trautmann A, Brocker EB, Goebeler M, Kerstan A. Angiolymphoid hyperplasia with eosinophilia and Kimura's disease: a clinical and histopathological comparison. J Dtsch Dermatol Ges 2014;12:224-8. 
  8. Kempf W, Haeffner AC, Zepter K, Sander CA, Flaig MJ, Mueller B, et al. Angiolymphoid hyperplasia with eosinophilia: evidence for a T-cell lymphoproliferative origin. Hum Pathol 2002;33:1023-9. 
  9. Ito S, Oda T, Matsuo A, Takechi H, Uchida T, Watanabe A, et al. Observation of angiolymphoid hyperplasia with eosinophilia (ALHE) at three arterial sites and its association with membranous nephropathy. Intern Med 2015;54:1933-9. 
  10. Grimwood R, Swinehart JM, Aeling JL. Angiolymphoid hyperplasia with eosinophilia. Arch Dermatol 1979;115:205-7. 
  11. Barnes L, Koss W, Nieland ML. Angiolymphoid hyperplasia with eosinophilia: a disease that may be confused with malignancy. Head Neck Surg 1980;2:425-34. 
  12. Nelson DA, Jarratt M. Angiolymphoid hyperplasia with eosinophilia. Pediatr Dermatol 1984;1:210-4. 
  13. Zou A, Hu M, Niu B. Comparison between Kimura's disease and angiolymphoid hyperplasia with eosinophilia: case reports and literature review. J Int Med Res 2021;49:3000605211040976. 
  14. Wells GC, Whimster IW. Subcutaneous angiolymphoid hyperplasia with eosinophilia. Br J Dermatol 1969;81:1-14. 
  15. Chong WS, Thomas A, Goh CL. Kimura's disease and angiolymphoid hyperplasia with eosinophilia: two disease entities in the same patient: case report and review of the literature. Int J Dermatol 2006;45:139-45. 
  16. Bonfils P, Moya-Plana A, Badoual C, Naderi S, Malinvaud D, Laccourreye O. Intraparotid Kimura disease. Eur Ann Otorhinolaryngol Head Neck Dis 2013;130:87-9. 
  17. Su S, Chen X, Li J, Yu J, Zhang L. Kimura's disease with membranoproliferative glomerulonephritis: a case report with literature review. Ren Fail 2019;41:126-30. 
  18. Kim WJ, Kim HK. Current concepts of Kimura disease: pathophysiology and evolution of treatment. Arch Craniofac Surg 2022;23:249-55. 
  19. Rosai J, Gold J, Landy R. The histiocytoid hemangiomas: a unifying concept embracing several previously described entities of skin, soft tissue, large vessels, bone, and heart. Hum Pathol 1979;10:707-30. 
  20. Deutsch BC, Schwam ZG, Kaul VZ, Wanna GB. Angiolymphoid hyperplasia with eosinophilia of the external ear. Am J Otolaryngol 2019;40:453-5. 
  21. Abrahamson TG, Davis DA. Angiolymphoid hyperplasia with eosinophilia responsive to pulsed dye laser. J Am Acad Dermatol 2003;49(2 Suppl Case Reports):S195-6. 
  22. Kaur T, Sandhu K, Gupta S, Kanwar AJ, Kumar B. Treatment of angiolymphoid hyperplasia with eosinophilia with the carbon dioxide laser. J Dermatolog Treat 2004;15:328-30. 
  23. Kadurina MI, Dimitrov BG, Bojinova ST, Tonev SD. Angiolymphoid hyperplasia with eosinophilia: successful treatment with the Nd:YAG laser. J Cosmet Laser Ther 2007;9:107-11. 
  24. Wozniacka A, Omulecki A, Torzecka JD. Cryotherapy in the treatment of angiolymphoid hyperplasia with eosinophilia. Med Sci Monit 2003;9:CS1-4. 
  25. McClintic EA, Ting A, Yeatts RP. Oral propranolol as an alternative therapy for orbital angiolymphoid hyperplasia with eosinophilia. Ophthalmic Plast Reconstr Surg 2016;32:e139-40. 
  26. Miller CJ, Ioffreda MD, Ammirati CT. Mohs micrographic surgery for angiolymphoid hyperplasia with eosinophilia. Dermatol Surg 2004;30:1169-73. 
  27. Miller CC. Cosmetic surgery: the correction of featural imperfections. Oak Printing Company; 1908. 
  28. Kim JH, Yang YB, Oh DY. Correction of pixie ear: infraauricular skin redraping method. Arch Craniofac Surg 2022;23:187-9. 
  29. Constant E. Reduction of the hypertrophic earlobe. Plast Reconstr Surg 1979;64:264-7. 
  30. Van Putte L, Colpaert SD. Earlobe reduction with minimally visible scars: the sub-antitragal groove technique. Aesthetic Plast Surg 2017;41:335-8.