IP Archives of Cytology and Histopathology Research

Print ISSN: 2581-5725

Online ISSN: 2456-9267

CODEN : IACHCL

IP Archives of Cytology and Histopathology Research (ACHR) open access, peer-reviewed quarterly journal publishing since 2016 and is published under the Khyati Education and Research Foundation (KERF), is registered as a non-profit society (under the society registration act, 1860), Government of India with the vision of various accredited vocational courses in healthcare, education, paramedical, yoga, publication, teaching and research activity, with the aim of faster and better dissemination more...


  • Article highlights
  • Article tables
  • Article images

Article statistics

Viewed: 71

PDF Downloaded: 48

Priyadharsini J, Barman, and Pari: Angiolymphoid hyperplasia with eosinophilia – A case report


Introduction

Angiolymphoid hyperplasia with eosinophilia represent the benign end of the spectrum of vascular tumors. This entity is known by various names which includes Epithelioid hemangioma, Inflammatory angiomatous nodule, Papular angioplasia and intravenous atypical vascular proliferation.1 In 1969, Wells & Whimster first described this rare vascular tumor.2 The etiology of epithelioid hemangioma remains controversial, whether it is a true neoplasm or reaction to trauma, however former is mostly favoured.3 It present as a single or multiple nodules in head and neck areas especially around the ear with predilection for middle aged women.4

Case Report

A 47 year old female patient presented with multiple nodular swelling in the temporal region of the scalp. She complained of itching and occasional bleeding. She had a positive history of trauma 2 months ago and the swelling slowly progressed in size during this time course. Chest X-Ray and Skull X-Ray was normal. Serum IgE levels were normal. Peripheral blood smear examination revealed eosinophilia with absolute eosinophil count of 750 cells per microliter.

Dermatological examination revealed multiple erythematous nodular lesion with focal ulceration. These nodules range in size from 5cm in greatest dimension to 1 cm in greatest dimension located in left sided temporal region. A biopsy was carried out in one of the lesions and histopathological findings showed proliferation of small sized blood vessels lined by plump endothelial cells with occasional hobnailing in the background of dense inflammatory infiltrate composed of eosinophils, lymphocytes and neutrophils. Surgical excision of the lesion was recommended and patient refused surgery. Follow up of the case revealed a slight increase in size of the lesion.

Figure 1

Multiple nodular lesions in the temporal region of left sided scalp.

https://s3-us-west-2.amazonaws.com/typeset-prod-media-server/a24d3f0c-0941-4fda-977b-cdb23cac6c7fimage1.png
Figure 2

Microphotograph showing skin with underlying dermis exhibiting abnormal proliferation of blood vessels (a. hematoxylin and eosin, 10 X). Microphotograph showing vessel lumen lined by plump endothelial cells with dense infiltration of eosinophils and lymphocytes.

https://s3-us-west-2.amazonaws.com/typeset-prod-media-server/a24d3f0c-0941-4fda-977b-cdb23cac6c7fimage2.png

Discussion

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare slow growing tumor with unknown etiopathogenesis. It present as single or multiple red nodules ranging in size from 2 – 3 cm. It is most common in middle aged adults with predilection to females. ALHE most commonly occur in head and neck region especially in proximity to ears, however it can also occur at other sites like trunk and limbs, oral mucosa, tongue, breast, lymph node, bone and testis.5, 6

Its pathogenesis is unknown, however the possible contributing factors are arteriovenous shunting, local trauma and elevated serum estrogen levels. The inflammatory cells are thought to play an important role in the proliferation of endothelial cells. Thus the theories of origin of this tumor is controversial and the leading members are a reactive process, infectious etiology, immunologic or neoplastic process.5, 7

The differential diagnosis of concern is kimura’s disease and is most common in Asian men. Here the patient present with lymphadenopathy, peripheral eosinophilia, increased serum IgE levels and immunologically mediated systemic involvement like membranous nephropathy. Histologically it presents in the deeper part with numerous lymphoid follicle formation, dense fibrosis and eosinophil microabscess. In contrast the ALHE shows numerous abnormal vascular proliferation with dense lymphocyte aggregates and eosinophils. These capillaries are lined by plump endothelial cells with hobnailing, with occasional cytoplasmic vacuoles.8

Other differential diagnosis considered are Injection site granuloma, bacillary angiomatosis, epithelioid hemangioendothelioma, cylindroma and angiosarcoma.9

The treatment of choice is surgical excision, however the other modalities that can be considered like cryotherapy, photodynamic therapy and application of imiquimod, which are providing promising results.10, 11

Conclusion

Angiolymphoid hyperplasia with eosinophilia though a rare entity should be considered when abnormal proliferation of blood vessels are seen. Though it is a benign lesion it does not regress without intervention, so prompt diagnosis is essential for further intervention.

Authors’ Contributions

Dr.P.P, collected the data, collected the reference, and typed the article. Dr.P.J provided gross and histopathological opinion and collected and compiled the data.Dr.P.P.B edited the article.

Acknowledgments

We thank the Faculty of Department of Pathology,Sri Lakshmi Narayana institute of medical sciences, Puducherry, for their support.

Source of Funding

No financial support was received for the work within this manuscript.

Conflict of Interest

The authors declare they have no conflict of interest.

References

1 

Y Park J Chung C Chang-Gun Angiolymphoid hyperplasia with eosinophilia of the tongue: report of a case and review of the literatureOral Oncol2002381103610.1016/s1368-8375(01)00020-3

2 

GC Wells IW Whimster Subcutaneous angiolymphoid hyperplasia with eosinophiliaBr J Dermatol 19698111510.1111/j.1365-2133.1969.tb15914.x

3 

Sap Sampaio E A Rivitti Hiperplasia angiolinfóide com eosinofiliaArtes Médicas200812212

4 

C M Quattrocchi R Jankovic M Jacquier A Sánchez A Bergero Raf Bussy Hiperplasia angiolinfoide con eosinofilia: reporte de un casoArch Argent Dermatol20126218992

5 

A Nogueira J W Accioly Filho M C Ribeiro de Castro JP Maceira M Ramos-e-Silva Hiperplasia angiolinfóide com eosinofilia: relato de dois casosAnais Bras de Dermatol2003781798510.1590/s0365-05962003000100008

6 

H. Tsuboi T. Fujimura K. Katsuoka Angiolymphoid hyperplasia with eosinophilia in the oral mucosa.Br J Dermatol20011452365610.1046/j.1365-2133.2001.04364.x

7 

I Zaraa M Mlika S Chouk I Chelly M Mokni M Zitouna Angiolymphoid hyperplasia with eosinophilia: a study of 7 casesDermatol Online J2011171

8 

J Ti de Bastos C R M da Rocha P M C Silva B M P de Freitas F de Freire Cassia J C R Avelleira Angiolymphoid hyperplasia with eosinophilia versus Kimura's disease: a case report and a clinical and histopathological comparisonAnais Bras de Dermatol2017923392410.1590/abd1806-4841.20175318

9 

S W Weiss J Goldblum Soft tissue tumors4th Edn.Mosby, Philadelphia2001593

10 

E. Sotiriou Z. Apalla A. Patsatsi D. Devliotou Panagiotidou D. Ioannides Angiolymphoid hyperplasia with eosinophilia: good response to photodynamic therapyClin Exp Dermatol2009348e6293110.1111/j.1365-2230.2009.03348.x

11 

A Youssef A R Hasan Y Youssef L Al-soufi Y Elshimali Z Alshehabi Angiolymphoid hyperplasia with eosinophilia: a case reportJ Med Case Rep201812110.1186/s13256-018-1599-x



jats-html.xsl

© 2020 Published by Innovative Publication Creative Commons Attribution 4.0 International License (creativecommons.org)


Article type

Case Report


Article page

317-319


Authors Details

Priyadharsini J, Partho Protim Barman, Priyatharsini Pari


Article Metrics


View Article As

 


Downlaod Files