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- W2018889506 abstract "A 10-YEAR-OLD Iraqichildpresentedwithabilateral exophthalmos (Figure1). An ophthalmologist had partially stitched up his eyelids to avoid subluxation of the globes. Exophthalmosbeganatage3yearsandwas diagnosed as pseudotumor orbitae. Attempted control of progressiveproptosiswithsteroid therapyhadbeenunsuccessful. Health status was otherwise normal. Erythrocyte sedimentationrateandserumimmunoglobulin levelswere elevated.Acranialmagneticresonanceimagingscanshowed abnormal tissue completely occupying the orbital spaces (Figure2), the sphenoidal and maxillar sinuses, the right retrostyloidspace,andslightly infiltrating theanterior fossa uptothesella.Chestx-rayfilms, radionucleotidebonescan, andbonemarrowaspirate showednoabnormalities.Anorbital tissue biopsy specimen showed sheets of large, pale, eosinophilic polygonal histiocytic cells with large vesicularnucleiandsmallnucleoli;occasionalmultinucleatedhistiocytes and spindle-shaped histiocytes were also present. No intact lymphoid cells were detected in the cytoplasm of largehistiocytes (ie,noemperipolesis),mitosiswas rare, and small lymphocytes and plasma cells were frequently scatteredthroughtthelesionassinglecellsandalsoarranged in clusters (Figure3, left). By immunohistochemistry the histiocytic cells stained positive for S100 protein (Figure 3, right) and CD68, but negative for CD1a (not shown); a presumptivediagnosisofRosai-Dorfmandiseaseof soft tissue was made. Diagnosis of multifocal Rosai-Dorfman diseaseofthesoft tissuewasthenconfirmedwithabiopsyspecimenofanasymptomatic,mildlyenlargedsubmaxillarlymph node. At histological examination, the lymph node architecture was preserved and the sinuses were distended by distinctive large histiocytes with abundant pale cytoplasm, oftenfilledwithapparentlyintact lymphocytes(emperipolesis).By immunohistochemistry thehistiocytic cells stained positive for S100 protein and CD68, but negative for CD1a (not shown). These cells showed the same phenotype observed inorbital tissue(Figure3). Sixteenweeksof chemotherapy(vinblastine,6mg/m perweek), gavepoor results. Furtherchemotherapywithmercaptopurine(60mg/m per day) and methotrexate (12 mg/m per week) for another 16 weeks also gave poor results." @default.
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- W2018889506 date "1999-11-01" @default.
- W2018889506 modified "2023-09-27" @default.
- W2018889506 title "Pathological Case of the Month" @default.
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- W2018889506 doi "https://doi.org/10.1001/archpedi.153.11.1199" @default.
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