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• W2921762482 abstract "We present an interesting case of hemophagocytic lymphohistiocytosis (HLH) presenting as jaundice and acute liver failure in a 59-year-old male with a history of sarcoidosis. HLH is rare, and unusual to be triggered by sarcoidosis. Only a limited number of such cases have been reported in the literature. Our patient is a previously healthy 59-year-old gentleman who presented to our hospital with a one-day history of altered mental status, two-week history of subjective fevers, malaise, jaundice, and several months of intermittent syncopal episodes requiring hospitalizations. During that time he was diagnosed with sarcoidosis. Steroid treatment was initiated with clinical improvement, until two weeks prior to his admission to our hospital when he ran out of medication. Clinical examination was remarkable for a profoundly jaundiced, febrile, ill-appearing, and confused gentleman. Laboratory tests were notable for abnormal liver enzymes and pancytopenia. Imaging with MRCP was without biliary obstruction, showing hepatic and splenic granulomas. Initially the differential diagnosis was broad. Work up was unrevealing for infectious or malignant causes. Additional lab work revealed a ferritin of 24,000, significantly elevated soluble CD25 level, and bone marrow biopsy had features consistent with infiltrative sarcoidosis and hemophagocytosis. At this point based on clinical data and test results, our patient met seven of eight criteria determined by the histiocyte society guidelines, qualifying him for a diagnosis of HLH. A review of the limited number of published case reports suggests that HLH is rare in adults, and is uncommon to be triggered by a rheumatologic condition. Furthermore jaundice is a poor prognostic marker for HLH, and prompt diagnosis and treatment is imperative. Only a few cases of hepatic sarcoidosis have been reported as the cause of HLH, and in adults the most common causes are infections and drugs. Among rheumatologic causes, juvenile idiopathic arthritis is most common. Another unique aspect of the case is that the patient initially improved with steroid treatment, which may have delayed the diagnosis of HLH. It is interesting to postulate whether HLH may be occurring more in the course of rheumatic diseases but is likely underdiagnosed because of overlapping symptomatology. Our patient was started on a modified HLH-94 protocol with significant clinical improvement and was eventually discharged from the hospital to a rehabilitation facility.2334_A Figure 1. MRCP with no evidence of biliary obstruction. Hepatomegaly and splenomegaly. Noncaseating granulomas noted in both liver and spleen.2334_B Figure 2. Bone marrow biopsy image showing single macrophage that has engulfed 3 nRBCs, platelets and 5-6 RBCs clustered together, consistent with hemophagocytosis.2334_C Figure 3. Significant improvement in bilirubin trend after initiation of treatment with a modified HLH-94 protocol." @default.
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• W2921762482 date "2018-10-01" @default.
• W2921762482 modified "2023-09-27" @default.
• W2921762482 title "A Rare Case of Hepatic Sarcoidosis Leading to the Development of Hemophagocytic Lymphohistiocytosis (HLH)" @default.
• W2921762482 doi "https://doi.org/10.14309/00000434-201810001-02333" @default.
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