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 Table of Contents  
Year : 2022  |  Volume : 2  |  Issue : 2  |  Page : 636

Adult-onset xanthogranuloma: Looking beyond the eye

1 Department of Ophthalmology, Guru Nanak Eye Centre, Maulana Azad Medical College, New Delhi, India
2 Department of Pathology, Govind Ballabh Pant Hospital, Maulana Azad Medical College, New Delhi, India

Date of Web Publication13-Apr-2022

Correspondence Address:
Ayushi Agarwal
Guru Nanak Eye Centre, Maulana Azad Medical College, New Delhi - 110 002
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijo.IJO_2518_21

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How to cite this article:
Agarwal A, Goel R, Shah S, Raut A, Raghav S, Saran RK. Adult-onset xanthogranuloma: Looking beyond the eye. Indian J Ophthalmol Case Rep 2022;2:636

How to cite this URL:
Agarwal A, Goel R, Shah S, Raut A, Raghav S, Saran RK. Adult-onset xanthogranuloma: Looking beyond the eye. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 May 26];2:636. Available from: https://www.ijoreports.in/text.asp?2022/2/2/636/342961

A 35-year-old male presented with bilateral, periorbital, yellowish papulonodular lesions for 4 months. No evidence of systemic illness, lymphadenopathy, asthma, abnormal cholesterol, or thyroid profile, was elicitable. Electrocardiography revealed left ventricular hypertrophy, corroborating with echocardiographic changes. Carotid doppler showed increased intima-media thickness for which medical therapy was initiated. Histopathological examination revealed S-100 and CD1a-negative foamy histiocytes and Touton giant cells, with no evidence of necrosis [Figure 1]. Based on clinicopathological correlation, diagnosis of adult-onset xanthogranuloma (AOX) was made. AOX is an uncommon non-Langerhans histiocytic xanthogranulomatous disorder.[1] Differential diagnoses include xanthelasma palpebrarum (which is more demarcated, superficial, and less indurated with foamy histiocytes), necrobiotic xanthogranuloma, and Erdheim–Chester disease, which are progressive lesions with multisystemic involvement, and Rosai–Dorfman disease, which exhibits S-100 positive histiocytes with emperipolesis. Meticulous evaluation in asymptomatic patients with this challenging entity may be lifesaving.[2] Surgical debulking and steroids, intralesional or intravenous, are beneficial in these cases.
Figure 1: (a) Clinical photograph showing periorbital, bilateral papulo-nodular lesions. (b) and (c) Histopathological examination (HES 40×) (b) Foamy macrophages (histiocytes) (arrow) in a background of fibrosis (c) Touton Giant cells (arrow)

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Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

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Conflicts of interest

There are no conflicts of interest.

  References Top

Kiratli H, Kiliç M, Tarlan B, Söylemezoglu F. Adult orbital xanthogranulomas: Clinical features and management. Eur J Ophthalmol 2015;25:288-92.  Back to cited text no. 1
Guo J, Wang J. Adult orbital xanthogranulomatous disease: Review of the literature. Arch Pathol Lab Med 2009;133:1994-7.  Back to cited text no. 2


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