This case report describes a 20-year-old male who presented with a 4-month history of a swelling in his right upper eyelid. Examination revealed a firm, round mass measuring 2x2cm. MRI could not provide a conclusive diagnosis. The patient underwent surgery to remove the mass, which was diagnosed through histopathology as Angiolymphoid Hyperplasia with Eosinophilia (ALHE), a rare condition characterized by proliferation of endothelial cells and infiltration of lymphocytes and eosinophils. The patient recovered well after surgery with no recurrence after 6 months.

1. Dr. Rohini Waghmare. ( Post graduate
student)
Dr .Sagar Chaudhari (MS
Ophthalmology)
Angiolymphoid hyperplasia with
eosinophilia of orbit in an young
male

2. INTRODUCTION:
 Angiolymphoid Hyperplasia with Eosinophilia
(ALHE) is a rare clinical entity characterized by
the presence of a variable number of papules,
plaques or nodules of the dermis and
subcutaneous tissues2.
 ALHE shows a predilection for the head and neck
area1. Orbital involvement is unusual.

3. CASE REPORT:
 HISTORY
A twenty year old male presented with a
four-month history of fluctuating swelling of the
upper medial aspect of right upper lid.
Fig- 1

4. EXAMINATION
 On palpation ,a well-defined, firm to hard lesion
over the supero-medial region of the right upper
eyelid close to the orbital margin was felt with
mild to moderate signs of inflammation.
 It measured about 2x2cm and was round to oval
in shape. .
 MRI of right orbit with contrast could not give
any conclusive diagnosis except some
inflammatory signs.
Fig- 2

5. TREATMENT
 Patient underwent anterior orbitotomy under
general anesthesia (fig 3-5)and the mass was
sent for histopathology which confirmed the
diagnosis of Angiolymphoid hyperplasia with
eosinophillia .
 Patient had uneventful recovery and had no
signs of recurrence over a period of 6 months

6. Fig- 5
Fig- 4Fig- 3

7. DISCUSSION:
 Wells and Whimster were the first to describe
ALHE in 1969.3
 This condition is characterized by abnormal
proliferation of endothelial cells with infiltrates of
lymphocytes and eosinophils, as demonstrated
by this case.
 ALHE is also known eosinophilic lymphofolliculitis
or Histiocytoid hemangioma.4

8.  Histologically multiple fibrocollagenous tissue
showing many blood vessels and intense
lymphoplasmacytic and eosinophilic infiltrate.
 ALHE should be distinguished from a variety of
benign and malignant vascular proliferations
including pyogenic granuloma, epithelioid
hemangioendothelioma and Kaposi’s sarcoma –
all of which lack a noticeable eosinophil infiltrate.
Fig- 6

9.  ALHE usually presents as benign nodules with
erythematous
papules mostly in the head and neck region,
however the
orbital involvement seen in this case is unusual.
 Features of orbital involvement may include
proptosis, watering, pruritus around the eyes and
blurring of peripheral vision.
 Our patient was successfully treated surgically
before she developed any such symptoms.

10.  Surgical excision may be beneficial in the case of
a solitary small tumour but there is a risk of
recurrence at the surgical site2.
 Systemic and intralesional steroid administration,
interferon therapy, cryotherapy, laser therapy and
topical application of Tacrolimus have been used
with success2.

11. CONCLUSION:
 ALHE with orbital involvement is a rare clinical
entity, and further work is required to accurately
describe its incidence, aetiology and
presentation.

12. REFERENCES:
 1. Gonzalez-Cuyar LF, Tavora F, Zhao XF, Wang G,
Auerbach A, Aguilera N, et al. Angiolymphoid hyperplasia
with eosinophilia developing in a patient with history of
peripheral T-cell lymphoma: evidence for multicentric T-cell
lymphoproliferative process. Diagn Pathol,2008,-; 29-;( -3):
-22
 2. Wolff K, Goldsmith L, Katz S,Gilchrest B,Paller A, Lafell
D. Fitzpatricks textbook of dermatology in general
medicine, 2008,-; 7th ed. 313-15.Fryer RH, Reinke KR.
Pyogenic granuloma: a complication of transconjunctival
incisions. Plast Reconstr Surg. 2000;105:1565-1566.
 3. 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(10):-1023-29.
 4. Jacob J, George S, Roy RB ,Dildeepa S. Angiolymphoid
hyperplasia with eosinophilia – A case report. Indian Jl of
Otolaryngol Head Neck Surg.2006,-; 58(3):- 285-87.