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Sarcoidosis as an unusual cause of hepatic dysfunction following reduced intensity conditioned allogeneic stem cell transplantation

Bone Marrow Transplantation volume 39, pages 511–512 (2007)Cite this article

Sarcoidosis is a multisystem, chronic granulomatous disorder, commonly affecting young adults and frequently presenting with bilateral hilar lymphadenopathy, pulmonary infiltrates and skin or eye lesions.1, 2 Although of unknown aetiology, sarcoidosis has been postulated to result from either a T-cell-mediated immune response against an environmental trigger or to be an abnormal immunological host response to a common antigenic trigger.3 Sarcoidosis has been associated with infections, inflammatory conditions, and/or drugs, but it has rarely been reported following haemopoietic stem cell transplantation (HSCT).4, 5 We report two patients who developed liver sarcoidosis following a reduced intensity conditioned (RIC) allogeneic HSCT.

The second patient was a 51-year-old Caucasian male, who underwent a FBC-conditioning RIC HSCT from a HLA-matched unrelated donor for fibrotic MDS with complex cytogenetics. A year before his transplant, he developed a blistering skin rash diagnosed as porphyria cutanea tarda on biopsy, with quantitation of porphyrins in blood, urine and faeces. No serologic evidence of hepatotropic-virus infection was found. Immediate transplant recovery was uneventful. Elevated indices of hepatobiliary injury were found 20 months after transplant (ALP 200 IU/l, AST 63 IU/l, GGT 273 IU/l). His only medications were penicillin V 250 mg twice daily orally and lansoprazole 30 mg once daily orally. Liver biopsy found non-caseating granulomata, with mild fibrosis of portal tracts and moderate siderosis of hepatocytes (Figure 1c). Mycobacteria and fungi were not identified by microscopy or on culture. Serum ACE levels were elevated at 160 U/l. Sarcoidosis was diagnosed. CT and routine radiography of the chest revealed no lesions. After oral prednisolone (30 mg once daily) was begun, ACE values normalised to 50 U/l. He is currently maintained on 5 mg of prednisolone once daily.

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Authors and Affiliations

  1. Department of Haematological Medicine, King's College London and King's College Hospital, London, UK

    L Gooneratne, Z Y Lim, A Y L Ho, A Pagliuca & G J Mufti

  2. Department of Dermatology, King's College London and King's College Hospital, London, UK

    A du Vivier

  3. Department of Histopathology, King's College London and King's College Hospital, London, UK

    J R Salisbury

  4. Institute of Liver Studies, King's College London and King's College Hospital, London, UK. E-mail: ghulam.mufti@kcl.ac.uk,

    A S Knisely

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Gooneratne, L., Lim, Z., Vivier, A. et al. Sarcoidosis as an unusual cause of hepatic dysfunction following reduced intensity conditioned allogeneic stem cell transplantation. Bone Marrow Transplant 39, 511–512 (2007). https://doi.org/10.1038/sj.bmt.1705606

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