{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,30]],"date-time":"2026-10-30T03:49:58Z","timestamp":1793332198859,"version":"4.5.0"},"reference-count":28,"publisher":"Archives of Pathology and Laboratory Medicine","issue":"12","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2009,12]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>This article provides an overview of the pathologic features of adult orbital xanthogranulomatous disease, a rare heterogeneous group of disorders that includes 4 clinical syndromes: adult-onset xanthogranuloma, necrobiotic xanthogranuloma, adult-onset asthma and periocular xanthogranuloma, and Erdheim-Chester disease. The diagnosis is made by biopsy of the lesion, demonstrating tissue infiltration by the hallmarks of xanthoma cells and Touton giant cells. The differential diagnosis is broad, including syndromes within the adult xanthogranulomatous disease category as well as other entities involving the eyelid and the orbital tissues. Because of its rarity and sometimes close similarity to other disease entities, it is often misdiagnosed initially. This article focuses on the morphology and immunohistochemical patterns in diagnosis of adult orbital xanthogranulomatous disease with emphasis on adult-onset asthma and periocular xanthogranuloma in particular, its clinical features and associated systemic manifestations in differential diagnosis, as well as the current management strategy.<\/jats:p>","DOI":"10.5858\/133.12.1994","type":"journal-article","created":{"date-parts":[[2021,2,12]],"date-time":"2021-02-12T09:18:46Z","timestamp":1613121526000},"page":"1994-1997","source":"Crossref","is-referenced-by-count":83,"title":["Adult Orbital Xanthogranulomatous Disease: Review of the Literature"],"prefix":"10.5858","volume":"133","author":[{"given":"Jin","family":"Guo","sequence":"first","affiliation":[{"name":"From the Department of Pathology and Laboratory Medicine, Loma Linda University Medical Center, Loma Linda, California"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jun","family":"Wang","sequence":"additional","affiliation":[{"name":"From the Department of Pathology and Laboratory Medicine, Loma Linda University Medical Center, Loma Linda, California"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"3906","reference":[{"key":"i1543-2165-133-12-1994-b1","series-title":"Trans Am Ophthalmol Soc","first-page":"99","article-title":"Periocular xanthogranulomas associated with severe adult-onset asthma.","volume":"91","author":"Jakobiec, F. A.","year":"1993","unstructured":"Jakobiec, F. A., M. D.Mills, and A. A.Hidayat. et al. Periocular xanthogranulomas associated with severe adult-onset asthma.Trans Am Ophthalmol Soc1993. 91:99\u2013125."},{"key":"i1543-2165-133-12-1994-b2","series-title":"Br J Ophthalmol","first-page":"602","article-title":"Adult xanthogranulomatous disease of the orbit and ocular adnexa: new immunohistochemical findings and clinical review.","volume":"90","author":"Sivak-Callcott, J. A.","year":"2006","unstructured":"Sivak-Callcott, J. A., J.Rootman, and S. L.Rasmussen. et al. Adult xanthogranulomatous disease of the orbit and ocular adnexa: new immunohistochemical findings and clinical review.Br J Ophthalmol2006. 90:602\u2013608."},{"key":"i1543-2165-133-12-1994-b3","series-title":"Ophthal Plast Reconstr Surg","first-page":"329","article-title":"Adult orbital xanthogranuloma with associated adult-onset asthma.","volume":"20","author":"Hammond, M. D.","year":"2004","unstructured":"Hammond, M. D., E. W.Niemi, T. P.Ward, and A. S.Eiseman. Adult orbital xanthogranuloma with associated adult-onset asthma.Ophthal Plast Reconstr Surg2004. 20:329\u2013332."},{"key":"i1543-2165-133-12-1994-b4","series-title":"Orbit","first-page":"221","article-title":"Orbital and eyelid manifestations of xanthogranulomatous diseases.","volume":"25","author":"Vick, V. L.","year":"2006","unstructured":"Vick, V. L., M. W.Wilson, J. C.Fleming, and B. G.Haik. Orbital and eyelid manifestations of xanthogranulomatous diseases.Orbit2006. 25:221\u2013225."},{"key":"i1543-2165-133-12-1994-b5","series-title":"Orbit","first-page":"191","article-title":"Necrobiotic xanthogranuloma occurring in an eyelid scar.","volume":"27","author":"Rayner, S. A.","year":"2008","unstructured":"Rayner, S. A., A. S.Duncombe, M.Keefe, J.Theaker, and R. M.Manners. Necrobiotic xanthogranuloma occurring in an eyelid scar.Orbit2008. 27:191\u2013194."},{"key":"i1543-2165-133-12-1994-b6","series-title":"Hong Kong Med J","first-page":"238","article-title":"Orbital involvement in Erdheim-Chester disease.","volume":"13","author":"Lau, W. W.","year":"2007","unstructured":"Lau, W. W., E.Chan, and C. W.Chan. Orbital involvement in Erdheim-Chester disease.Hong Kong Med J2007. 13:238\u2013240."},{"key":"i1543-2165-133-12-1994-b7","series-title":"Am J Dermatopathol","first-page":"104","article-title":"\u201cJuvenile\u201d xanthogranuloma: an immunophenotypic study with a reappraisal of histogenesis.","volume":"23","author":"Kraus, M. D.","year":"2001","unstructured":"Kraus, M. D., J. C.Haley, R.Ruiz, L.Essary, C. A.Moran, and C. D.Fletcher. \u201cJuvenile\u201d xanthogranuloma: an immunophenotypic study with a reappraisal of histogenesis.Am J Dermatopathol2001. 23:104\u2013111."},{"key":"i1543-2165-133-12-1994-b8","series-title":"Surv Ophthalmol","first-page":"608","article-title":"Juvenile xanthogranuloma of the corneoscleral limbus: case report and review of the literature.","volume":"49","author":"Chaudhry, I. A.","year":"2004","unstructured":"Chaudhry, I. A., Z.Al-Jishi, F. A.Shamsi, and F.Riley. Juvenile xanthogranuloma of the corneoscleral limbus: case report and review of the literature.Surv Ophthalmol2004. 49:608\u2013614."},{"key":"i1543-2165-133-12-1994-b9","series-title":"Cytojournal","first-page":"19","article-title":"Cytodiagnosis of multiple myeloma presenting as orbital involvement: a case report.","volume":"3","author":"Sharma, A.","year":"2006","unstructured":"Sharma, A., M.Kaushal, N. K.Chaturvedi, and R.Yadav. Cytodiagnosis of multiple myeloma presenting as orbital involvement: a case report.Cytojournal2006. 3:19. doi:10.1186\/1742-6413-3-19."},{"key":"i1543-2165-133-12-1994-b10","series-title":"Trans Am Ophthalmol Soc","first-page":"69","article-title":"Local corticosteroid treatment of eyelid and orbital xanthogranuloma.","volume":"103","author":"Elner, V. M.","year":"2005","unstructured":"Elner, V. M., R.Mintz, H.Demirci, and A. S.Hassan. Local corticosteroid treatment of eyelid and orbital xanthogranuloma.Trans Am Ophthalmol Soc2005. 103:69\u201374."},{"issue":"3","key":"i1543-2165-133-12-1994-b11","series-title":"Hong Kong Med J","first-page":"238","article-title":"Orbital involvement in Erdhein-Chester disease.","volume":"13","author":"Winnie, W. Y.","year":"2007","unstructured":"Winnie, W. Y., W. W.Lau, and E.Chan. Orbital involvement in Erdhein-Chester disease.Hong Kong Med J2007. 133:238\u2013240."},{"key":"i1543-2165-133-12-1994-b12","series-title":"Ophthal Plast Reconstr Surg","first-page":"36","article-title":"Local corticosteroid treatment of eyelid and orbital xanthogranuloma.","volume":"22","author":"Elner, V. M.","year":"2006","unstructured":"Elner, V. M., R.Mintz, H.Demirci, and A. S.Hassan. Local corticosteroid treatment of eyelid and orbital xanthogranuloma.Ophthal Plast Reconstr Surg2006. 22:36\u201340."},{"key":"i1543-2165-133-12-1994-b13","series-title":"Br J Ophthalmol","first-page":"1375","article-title":"Xanthogranulomatous disease in the lacrimal gland.","volume":"89","author":"Sivak-Callcott, J. A.","year":"2005","unstructured":"Sivak-Callcott, J. A., W. K.Lim, and L. L.Seah. et al. Xanthogranulomatous disease in the lacrimal gland.Br J Ophthalmol2005. 89:1375\u20131377."},{"key":"i1543-2165-133-12-1994-b14","series-title":"Br J Ophthalmol","first-page":"434","article-title":"Management of orbital xanthogranuloma with methotrexate.","volume":"91","author":"Hayden, A.","year":"2007","unstructured":"Hayden, A., D. J.Wilson, and J. T.Rosenbaum. Management of orbital xanthogranuloma with methotrexate.Br J Ophthalmol2007. 91:434\u2013436."},{"key":"i1543-2165-133-12-1994-b1","series-title":"Trans Am Ophthalmol Soc","first-page":"99","article-title":"Periocular xanthogranulomas associated with severe adult-onset asthma.","volume":"91","author":"Jakobiec, F. A.","year":"1993","unstructured":"Jakobiec, F. A., M. D.Mills, and A. A.Hidayat. et al. 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