Orbital ALHE Case
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Dr. Rohini Waghmare ( Post graduate student), Dr .Sagar Chaudhari (MS Ophthalmology).
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Orbital ALHE Case
- 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.