Journal List > J Korean Ophthalmol Soc > v.49(4) > 1008248

Choi, Kwon, and Kim: A Case of Post-traumatic Necrobiotic Xanthogranuloma of the Orbit

Abstract

Purpose

To investigate the role of nitric oxide (NO) on the migration of cultured human Tenon's capsule

Purpose

Necrobiotic xanthogranuloma is a very rare disease characterized by the presence of yellow subcutaneous skin lesions and systemic paraproteinemia. We report a case of post‐ traumatic necrobiotic xanthograuloma that presented with a preseptal mass.

Case summary

A 31-year-old man presented with ptosis and a mass on the left superior orbit that developed after trauma 3 weeks prior to admission. A computed tomography scan showed a contrast-enhancing preseptal mass, and surgical exploration was performed.

Conclusions

Histopathological examination revealed a tumor that had all the major features of necrobiotic xanthogranuloma, including fat necrosis with granulomatous inflammation, giant multinucleated cells, foamy cells, and several lymphoid follicles with germinal centers. Hematologic evaluation revealed polyclonal gammopathy.

References

1. Mehregan DA, Winkelmann RK. Necrobiotic xanthogranuloma. Arch Dermatol. 1992; 128:94–100.
crossref
2. Kossard S, Winkelmann RK. Necrobiotic xanthogranuloma with paraproteinemia. J Am Acad Dermatol. 1980; 3:257–70.
crossref
3. Winkelmann RK, Dahl PM, Perniciaro C. Asteroid and other cytoplasmic inclusions in necrobiotic xanthogranuloma with paraproteinemia. J Am Acad Dermatol. 1998; 38:967–70.
4. Novak PM, Robins TO, Winkelmann RK. Necrobiotic xanthogranuloma with myocardial and nodular transformation of the liver. Hum Pathol. 1995; 23:195–6.
5. Umbert I, Winkelmann RK. Necrobiotic xanthogranuloma with cardiac involvement. Br J Dermatol. 1995; 133:438–43.
crossref
6. Finan MC, Winkelmann RK. Necrobiotic xanthogranuloma with paraproteinaemia: a review of 22 cases. Medicine. 1986; 65:376–87.
7. Chave TA, Chowdhury MM, Holt PJ. Recalcitrant necrobiotic xanthogranuloma responding to pulsed highdose oral dexamethasone plus maintenance therapy with oral prednisolone. Br J Dermatol. 2001; 144:158–61.
crossref
8. Venencie PY, Le Bras P, Toan ND, et al. Recombinant interferon alfa‐2b treatment of necrobiotic xanthogranuloma with paraproteinemia. J Am Acad Dermatol. 1995; 32:666–7.
crossref
9. Finelli LG, Ratz JL. Plasmapheresis, a treatment modality for necrobiotic xanthogranuloma. J Am Acad Dermatol. 1987; 17:351–4.
crossref
10. Sivak Callcott JA, Rootman J, Rasmussen SL, et al. Adult xanthogranulomatous disease of the orbit and ocular adnexa: new immunohistochemical findings and clinical review. Br J Ophthalmol. 2006; 90:602–8.
11. Ugurlu S, Bartley GB, Gibson LE. Necrobiotic xanthogranuloma: Long‐ term outcome of ocular and systemic involvement. Am J Ophthalmol. 2000; 129:651–7.
12. Bullock JD, Bartley GB, Campbell RJ, et al. Necrobiotic xanthogranuloma with paraproteinemia. Case report and a pathogenetic theory. Ophthalmology. 1986; 93:1233–6.
13. Char DH, Leboit PE, Ljung BM, Wara W. Radiation therapy for ocular necrobiotic xanthogranuloma. Arch Ophthalmol. 1987; 105:174–5.
crossref
14. Rappersberger K, Wrba F, Heinz R, et al. Necrobiotic xanthogranuloma in paraproteinemia. Hautarzt. 1989; 40:358–63.
15. Stork J, Kodetova D, Vosmik F, Krejca M. Necrobiotic xanthogranuloma presenting as a solitary tumor. Am J Dermatopathol. 2000; 22:453–6.

Figure 1.
Clinical photograph of a 31-year-old man with 3-week history of ptosis of the left eye.
jkos-49-677f1.tif
Figure 2.
Orbital computed tomography scans showed ill-defined, contrast‐ enhancing mass in the preseptal space (arrows). (A) contrasted, (B) non-contrasted. (preinjection)
jkos-49-677f2.tif
Figure 3.
The granulomatous infiltrate consisted of histiocytes mixed with lymphocytes, foamy cells, multinucleated giant cells, and Touton cells (arrows). (HE stain, original magnifications ×100)
jkos-49-677f3.tif
Figure 4.
One-month postoperative photograph showed no mass lesion or ptosis.
jkos-49-677f4.tif
TOOLS
Similar articles