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Epithelioid Hemangioma of the Nasal Cavity: a Diagnostic Challenge

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Abstract

Epithelioid hemangioma also known as angio-lymphoid hyperplasia with eosinophilia is a rare benign vascular neoplasm of unknown etiology. It very rarely involves the nasal cavity. It always poses a diagnostic challenge for clinicians and is usually misdiagnosed as Kimura’s, IgG4-related disease, or malignant vascular tumors. The present case report describes an extremely rare presentation of epithelioid hemangioma inside the left nasal cavity causing complete obstruction and nasal septal deviation towards the right side in a young male. Hematoxylin and eosin-stained sections depicted a lobular proliferation of small capillary-sized vessels lined by plump epithelioid endothelial cells surrounding central vessels and scattered around them. These cells had abundant eosinophilic to amphophilic cytoplasm and enlarged nuclei with fine chromatin and central nucleoli. On immunohistochemistry, CD34 and CD31 highlighted the vascular proliferation and epithelioid endothelial cells. Erythroblast transformation-specific (ETS)-related gene (ERG) showed strong nuclear positivity in neoplastic plump epithelioid endothelial cells. EH is a benign vascular proliferation, but a high chance of recurrence is seen if complete resection is not done. As a result, the treatment of choice is complete surgical excision with clear margins. The case highlights a non-conventional presentation of epithelioid hemangioma and the importance of histomorphological features in diagnosing this entity.

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References

  1. Wells GC, Whimster IW (1969) Subcutaneous angiolymphoid hyperplasia with eosinophilia. Br J Dermatol 81:1–14

    Article  CAS  PubMed  Google Scholar 

  2. Googe PB, Harris NL, Mihm MC Jr (1987) Kimura’s disease and angiolymphoid hyperplasia with eosinophilia: two distinct histopathological entities. J Cutan Pathol 14:263–271

    Article  CAS  PubMed  Google Scholar 

  3. Ortins-Pina A, Llamas-Velasco M, Turpin S, Soares-de-Almeida L, Filipe P, Kutzner H (2018) FOSB immunoreactivity in endothelia of epithelioid hemangioma (angiolymphoid hyperplasia with eosinophilia). J Cutan Pathol 45(6):395–402

    Article  PubMed  Google Scholar 

  4. Guo R, Gavino ACP (2015) Angiolymphoid hyperplasia with eosinophilia. Arch Pathol Lab Med 139:683–686

    Article  PubMed  Google Scholar 

  5. Sedran L, Bonaso M, Mettus A, Roccia F (2018) Epithelioid hemangioma of the face. J Craniofac Surg 29:e736–e739

    Article  PubMed  Google Scholar 

  6. Fetsch JF, Sesterhenn IA, Miettinen M, Davis CJ Jr (2004) Epithelioid hemangioma of the penis: a clinicopathologic and immunohistochemical analysis of 19 cases, with special reference to exuberant examples often confused with epithelioid hemangioendothelioma and epithelioid angiosarcoma. Am J Surg Pathol 28:523–533

    Article  PubMed  Google Scholar 

  7. Mariatos G, Gorgoulis VG, Laskaris G, Kittas C (1999) Epithelioid hemangioma (angiolymphoid hyperplasia with eosinophilia) in the oral mucosa: a case report and review of the literature. Oral Oncol 35:435–438

    Article  CAS  PubMed  Google Scholar 

  8. Maheshwari V, Sharma R, Alam K, Khan AH (1999) Angiolymphoid hyperplasia with eosinophilia (alhe) versus kimura’s disease : changing concepts. Indian J Dermatol Venereol Leprol 65:186–188

    CAS  PubMed  Google Scholar 

  9. Chandrashekar BS, Madura C (2018) IADVL textbook of trichology, 1st edn. JP Medical Ltd., New Delhi, India

  10. Calonje E, Mackie RM, Burns T, Breathnach S, Cox N, Griffiths C (2010) Rook’s textbook of dermatology, 10th edn. Blackwell Publishing Ltd

  11. Rajendran R, Sivapathasundram B (2012) Shafer’s textbook of oral pathology, 6th edn. Elsevier, Amsterdam

    Google Scholar 

  12. Youssef A, Hasan AR, Youssef Y, Al-Soufi L, Elshimali Y, Alshehabi Z (2018) Angiolymphoid hyperplasia with eosinophilia: a case report. J Med Case Rep 12:89

    Article  PubMed  PubMed Central  Google Scholar 

  13. Van Ratingen AR, Linden V, Sillevis Smitt JH (2016) Case report: angiolymphoid hyperplasia with eosinophilia of the nose. Dermatol Case Rep 1:2

    Article  Google Scholar 

  14. Panduranga Kamath M, Bhojwani KM, Bhandarkar AM, Pai RR, Rent NH (2014) Angiolymphoid hyperplasia with eosinophilia of root of nose: a rare phenomenon. J Clin Diagn Res 8:144–145

    CAS  PubMed  PubMed Central  Google Scholar 

  15. Chiu SC (2013) An unusual case of angiolymphoid hyperplasia with eosinophilia of the nose. Ear Nose Throat J 92:E10–E11

    PubMed  Google Scholar 

  16. Baghestani S, Firooz A, Ghazisaidi MR (2011) A refractory case of angiolymphoid hyperplasia with eosinophilia successfully treated by surgery. J Dermatolog Treat 22:49–51

    Article  PubMed  Google Scholar 

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Authors and Affiliations

  1. Department of Pathology, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi, India

    Adil Aziz Khan, Sana Ahuja & Sufian Zaheer

  2. Department of Pathology, All India Institute of Medical Sciences, New Delhi, India

    Sumanta Das

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  1. Adil Aziz Khan
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  2. Sana Ahuja
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  3. Sufian Zaheer
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  4. Sumanta Das
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Correspondence to Sufian Zaheer.

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Khan, A.A., Ahuja, S., Zaheer, S. et al. Epithelioid Hemangioma of the Nasal Cavity: a Diagnostic Challenge. Indian J Surg Oncol 15, 181–184 (2024). https://doi.org/10.1007/s13193-024-01875-4

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  • DOI: https://doi.org/10.1007/s13193-024-01875-4

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