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Table of Contents
CLINICAL NOTE
Year : 2001  |  Volume : 3  |  Issue : 1  |  Page : 19-20

Angiolymphoid hyperplasia with eosinophilia diagnosis and treatment (case study)


1 Senior Registrar, Assir Central Hospital, Abha, Saudi Arabia
2 Consultant ENT Surgeons, Assir Central Hospital, Abha, Saudi Arabia
3 Department of Otolaryngology - Assir Central Hospital, Abha, Saudi Arabia

Date of Web Publication9-Jul-2020

Correspondence Address:
FRCS (Ed) Yinka Fawehinmi
Department of Otolaryngology,Assir Central Hospital, P.O. Box 34, Abha
Saudi Arabia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1319-8491.289355

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  Abstract 


Angiolymphoid hyperplasia with eosinophil is a reactive vascular hyperproliferative response of unknown etiology. It has a predilection for the head and neck region. This paper highlights the difference between angiolymphoid hyperplasia with eosinophilia and Kimura’s disease. It also highlights the modality for treatment and diagnosis.

Keywords: Angiolymphoid hyperplasia with eosinophilia diagnosis, treatment


How to cite this article:
Ardi T, Fawehinmi Y, Alabidi A. Angiolymphoid hyperplasia with eosinophilia diagnosis and treatment (case study). Saudi J Otorhinolaryngol Head Neck Surg 2001;3:19-20

How to cite this URL:
Ardi T, Fawehinmi Y, Alabidi A. Angiolymphoid hyperplasia with eosinophilia diagnosis and treatment (case study). Saudi J Otorhinolaryngol Head Neck Surg [serial online] 2001 [cited 2021 May 5];3:19-20. Available from: https://www.sjohns.org/text.asp?2001/3/1/19/289355




  Introduction Top


Angiolymphoid hyperplasia with eosinophilia presents usually in adults as single or multiple nodules. It is commonly found on the face, ears, scalp and salivary gland regions. It occurs in all races in comparison to Kimura’s disease that occurs in orientals of mongoloid origin [1] where it is endemic. The disease presents as single or multiple nodules. It is believed to be a reactive vascular hyperproliferative response, although the etiology and pathogenesis are unknown.

They are usually benign lesions though histologically they may resemble Kaposi sarcoma or malignant angiosarcoma. It may also be confused with benign reactive eosinophilic ulcer in the oral mucosa that is of unknown origin [2]. We report a case of retroparotid mass, the diagnosis of which could not be confirmed by sialogram and computerized tomogram scan.


  Case Report: Top


An 18-year-old Saudi boy was referred from a peripheral hospital to the ENT Clinic at Asir Central hospital, Abha, Saudi Arabia complaining of a swelling of the right retroparotid area for over 6 months. The mass was 6x2cm in size, tender and adherent to the skin. There was no regional lyrn- phadenopathy and the facial nerve was intact.

The parotid duct orifice was viewed. The character of the secretion was normal. Bimanual palpation of the gland showed a localized discrete mass near the tail of the right parotid gland.

A diagnosis of retroparotid mass was made and fine needle biopsy was done in the clinic but the histopathology report was inconclusive.

Sialogram was also negative for calculus and computerized tomogram scan showed soft tissue mass near the mastoid process behind the tail of the right parotid gland (see [Figure 1].
Figure 1:

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The mass was excised under general anesthesia and sent for histology. Histology report confirmed the diagnosis of angiolymphoid hyperplasia with eosinophilia (see [Figure 2].
Figure 2:

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The patient had an uneventful post- operative recovery and was discharged three days later. He was followed up for one year and there was no sicn of recurrence.


  Discussion: Top


Angiolymphoid hyperplasia with eosinophilia is an uncommon benign slow growing lesion found primarily in the head and neck region with an onset usually between the third and fourth decade and a female predominance in the non oriental population [3]. Kimura’s disease occurring primarily in young oriental males is a similar lesion with peripheral blood eosinophilia, regional lym- phadenopathy and occasional nephrotic syndrome. A review of the literature revealed that only two cases have been reported so far in the Gulf states The first one was reported in an Arab female from Tabuk, Saudi Arabia [4], and the second one also in an arab patient from Bahrain[5], Both were Kimura’s disease. Our patient is a male Arab and the diagnosis was angiolymphoid hyperplasia with eosinophilia. This is in contrast to female predominance of angiolymphoid hyperplasia with eosinophilia, which is characterized microscopically by marked proliferation of large endothelial cells with a mixed inflammatory infiltrate consisting of lymphocytes, histocytes and eosinophils [6].

In comparison, Kimura’s disease has more endothelial proliferation but lacks the atypical histocytoid endothelial cells and has less extracellular granule protein deposition [7]. The mass was surgically excised and there has been no recurrence.

Some authors advocated intralesional or systemic steroids without surgery and some have advocated irradiation where surgery is not possible [8]. Excision biopsy and histological study established a diagnosis of angiolymphoid hyperplasia with eosinophilia in our case.


  Conclusion: Top


We present a case of angiolymphoid hyperplasia, which was successfully treated by excision although treatments reported in the literature consist of a combination of surgery, steroids and radiotherapy. We also highlight the typical diagnostic histological features of angiolymphoid hyperplasia with eosinophilia as compared to Kimura’s disease. We propose that angiolymphoid hyperplasia with eosinophilia represents an angiomatous neoplasm similar to hemangioma but characterized by a marked reactive appearance and the diagnosis can be only confirmed by histology.



 
  References Top

1.
Helander SD, Peters MS, Kao TT, Su WP. Kimura’s disease and angiolymphoid hyperplasia with eosinophilia J Antan Pathol 1995 ; 22 (4): 319-26.  Back to cited text no. 1
    
2.
Mesei MM, Tvou VA, Stewart WD., Rivens JK. Eosinophilic Ulcer of the oral mucosa J Am Acad Dermatol 1995 ; 33 (5 Pt 1): 734 n740.  Back to cited text no. 2
    
3.
Archer KF, Hunnitz JJ, Heathcote G. Orbital angiolymphoid Hyperplasia with eosinophilia. Presentation as chalazion. Opthal Plast Reconstr Sur 1991; 7 (3): 208-21.  Back to cited text no. 3
    
4.
Jaber K. Kimura’s disease in an Arab Female. Histopathology 1996 ; 29 (1): 76-78  Back to cited text no. 4
    
5.
Pamargin N, Khalifa SA, Darwish A, Ahmed N, Yousef H. Kimura’s disease J larynoo Otol 1996’ 110(1 IV 1084-87. '  Back to cited text no. 5
    
6.
Haas AF, La Penere R, King E. Angiolymphoid hyperplasia with eosinophilia of the hand J Dermatol Sura Oncol 1991; 17(9): 731 n4.  Back to cited text no. 6
    
7.
Luguams D R, Stanford N D, Creagh T M. Angiolymphoid hyperplasia with eosinophilia J Laryneo Otol 1995; 109 (3): 262-4. F J b  Back to cited text no. 7
    
8.
Gnumb M A, Pan N B, SavvayaRJ, Rubustein J, Vithilliugan L, Choi YJ. Kimura’s disease, a case report and lit erature review. J Surg Oncol 1999; 70(3): 190-193.H  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2]



 

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