|Year : 2015 | Volume
| Issue : 4 | Page : 217-219
Angiolymphoid hyperplasia with eosinophilia in parotid gland: A diagnostic challenge
Bifica Sofia Lyngdoh1, Anadi Roy Chowdhury1, Soumya Ghatak2, Gautam Bandyopadhyay1
1 Department of Pathology, RG Kar Medical College and Hospital, Kolkata, West Bengal, India
2 Department of ENT, College of Medicine and Sagore Dutta Hospital, Kolkata, West Bengal, India
|Date of Web Publication||21-Jan-2016|
Dr. Bifica Sofia Lyngdoh
Department of Pathology, 5th Floor, Academic Building, 1, RG Kar Medical College, Khudiram Bose Sarani, Kolkata - 700 004, West Bengal
Source of Support: None, Conflict of Interest: None
Angiolymphoid hyperplasia with eosinophilia (ALHE) rarely involves the parotid glands or other major glands. ALHE and Kimura's disease (KD) share many clinical and histopathological features. Ill-defined, deeply seated lesions involving the parotid glands are frequently observed in KD, and only three cases of intraparotid ALHE have been reported in the literature. Often these benign angioproliferative lesions of unknown etiology will be misdiagnosed as parotid tumors clinically. We report a case of ALHE in a 29-year-old Indian male who presented with unilateral parotid enlargement. The previous fine-needle aspiration cytology (FNAC) report indicated parotid sialadenitis but histopathological examination following superficial parotidectomy of the lesion allowed us to make a definitive diagnosis.
Keywords: Angiolymphoid hyperplasia with eosinophilia (ALHE), Kimura′s disease (KD), parotid gland
|How to cite this article:|
Lyngdoh BS, Chowdhury AR, Ghatak S, Bandyopadhyay G. Angiolymphoid hyperplasia with eosinophilia in parotid gland: A diagnostic challenge. Arch Int Surg 2015;5:217-9
|How to cite this URL:|
Lyngdoh BS, Chowdhury AR, Ghatak S, Bandyopadhyay G. Angiolymphoid hyperplasia with eosinophilia in parotid gland: A diagnostic challenge. Arch Int Surg [serial online] 2015 [cited 2019 Dec 15];5:217-9. Available from: http://www.archintsurg.org/text.asp?2015/5/4/217/174670
| Introduction|| |
Angiolymphoid hyperplasia with eosinophilia (ALHE) is also known as epithelioid hemangioma.  ALHE and Kimura's disease (KD) share many clinical and histopathological features. Often these benign angioproliferative lesions of unknown etiology will be misdiagnosed as parotid tumors clinically. Ill-defined, deeply seated lesions involving the parotid gland are frequently observed in KD,  and only three cases of intraparotid ALHE have been reported in the literature. , The objective of this study was to make a note that on cytology, ALHE may be falsely diagnosed as chronic sialadenitis.
| Case Report|| |
A 29-year-old male patient presented with gradually increasing painless swelling on the right parotid region for 3 months. There was no history of trauma, weight loss, low-grade fever or night sweating. Medical, surgical, and family histories were noncontributory.
On examination, the swelling measured 6 × 3.5 cm 2 and was firm, nontender, and mobile [Figure 1]. The overlying skin was normal with respect to both color and temperature, without any abnormal facial nerve function. A provisional diagnosis of pleomorphic adenoma of the parotid gland was made and the patient was advised for fine-needle aspiration cytology (FNAC), which revealed features suggestive of right parotid sialadenitis.
|Figure 1: Clinical picture of the patient showing a swelling at the right parotid region not fixed to overlying skin|
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Computed tomography (CT) revealed enlarged superficial lobe of the right parotid gland containing a heterogenous space-occupying lesion measuring 4 × 2.5 cm 2 [Figure 2].
|Figure 2: CT scan demonstrating a mass inside the right superficial parotid gland with no overlying skin involvement|
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Laboratory studies revealed the hemoglobin concentration, white cell count, and absolute eosinophil count to be within normal limits. As the swelling was gradually increasing in size, superficial parotidectomy of the right parotid gland was done. Intraoperative findings were that of a highly vascular tumor, which led to the suspicion of malignant condition.
Gross pathology showed the superficial lobe of the parotid gland measuring 6 × 4 × 2.5 cm 3 . Serial cut section revealed an ill-defined solid whitish mass measuring 4 × 3 × 1.2 cm 3 [Figure 3]. Two associated lymph nodes were identified also.
|Figure 3: Gross picture of the tissue after superficial right-side parotidectomy|
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Histopathological study of the specimen demonstrated numerous blood vessels along with dense lymphoid aggregates with germinal center, prominent eosinophilic and plasma cell infiltrates, and attenuation of parotid acini. The endothelium of the blood vessels was plump and vacuolated with vesicular nuclei. A diagnosis of ALHE of the parotid gland was made [Figure 4]. The section examined from the lymph nodes showed only reactive follicular hyperplasia.
|Figure 4: Histopathological features of ALHE in parotid gland. (a) Showing vascular proliferation along with lymphoid aggregates and part of normal parotid gland (Hematoxylin and eosin (H&E), 40×) (b) Showing vascular proliferation along with lymphoid aggregates (H&E, 100×) (c) Showing blood vessel with eosinophilic plump and vacuolated endothelial cells typically accompanied by lymphoid cells and eosinophils (H&E 400×)|
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| Discussion|| |
First described by Wells and Whimster,  ALHE is an unusual but distinctive vascular tumor. Young adults are usually affected, and the lesions are found mainly in the subcutis with occasional intradermal extension. They may resolve spontaneously or persist for several years with no malignant change reported for long. ,
Ussmüller  reported a case of ALHE in the parotid gland and presented the opinion that ALHE might be a primary arterial disorder with a secondary vascular proliferation and chronic inflammation. The most frequent clinical findings are of an epidermal cyst or angioma,  but in our case, a provisional diagnosis of pleomorphic salivary adenoma was given.
The etiology and pathogenesis of ALHE are unknown. The diagnosis was based on the typical histopathological features of an unencapsulated mass with exuberant proliferation of immature vessels, which are lined by plump, epithelioid endothelial cells with amphophilic or eosinophilic cytoplasm, and a large nucleus with an open chromatin pattern and central nucleolus. Most cases have a prominent inflammatory component, but a notable eosinophilic reaction may not be essential to ALHE. ,
There is a lot of confusion between KD and ALHE in the literature, but Rosai et al. (1979)  in their study eventually established these lesions as two separate entities. Males are typically affected in KD, while ALHE shows roughly equal sex distribution.  The lesions of KD are usually ill defined, large, and deeply seated.  Regional lymph node and salivary gland involvement is more frequent in KD and very rare in ALHE.  Increased serum immunoglobulin E (IgE) and eosinophilia in peripheral blood are rare, usually occurring in lesions with a longer duration. 
In our case, the lesion was limited to the intraparotid region. Two regional lymph nodes that showed reactive hyperplasia were also found. A diagnosis of ALHE can be challenging because clinically it can mimic a parotid neoplasm. FNAC can only suggest a reactive or inflammatory lesion, and radiological findings can only assess the extent of the lesion.
Complete local excision and follow-up form the treatment of choice for AHLE.
| Conclusion|| |
AHLE is not uncommon in the head and neck region. However, it is very rare in the parotid glands. That is why it may be falsely diagnosed as chronic sialadenitis on cytology and treatment may be delayed.
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Conflicts of interest
There are no conflicts of interest.
| References|| |
Fletcher CD. Vascular tumours. In: Fletcher CD, editor. Diagnostic Histopathology of Tumors. 4 th
ed. Philladelphia, PA: Churchill Livingstone; 2013. p. 54-5.
Ussmüller J, Donath K, Shimizu M, Bergmann I. Differential diagnosis of tumorous space-occupying lesions of the parotid gland: Angiolymphoid hyperplasia with eosinophilia and Kimura disease. Laryngorhinootologie 1997;76:110-5.
Hung T, Gillet D, Gluckman P. Angiolymphoid hyperplasia with eosinophilia of the parotid gland. J Otolaryngol 2001;30:359-60.
Wells GC, Whimster IW. Subcutaneous angiolymphoid hyperplasia with eosinophilia. Br J Dermatol 1969;81:1-14.
Liu X, Wang R, Guan W, Wang L. Epithelioid hemangioma of the testis. Indian J Pathol Microbiol 2013;56:422-4.
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.
Iguchi Y, Inoue T, Shimono M, Yamamura T, Shigematsu T, Takahashi S. Kimura′s disease and its relation to angiolymphoid hyperplasia with eosinophilia: Report of three cases and review of the literature. J Oral Pathol 1986;15:132-7.
Kuo T, Shih LY, Chan HL. Kimura′s disease. Involvement of regional lymph nodes and distinction from angiolymphoid hyperplasia with eosinophilia. Am J Surg Pathol 1988;12:843-56.
Ramchandani PL, Sabesan T, Hussein K. Angiolymphoid hyperplasia with eosinophilia masquerading as Kimura disease. Br J Oral Maxillofac Surg 2005;43:249-52.
[Figure 1], [Figure 2], [Figure 3], [Figure 4]