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Immune reconstitution inflammatory syndrome following allo-SCT in a patient with Dipodascus capitatus spondylodiscitis

Bone Marrow Transplantation volume 46, pages 1265–1267 (2011)Cite this article

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We describe a man with allogeneic BMT who developed Dipodascus capitatus (anamorph Geotrichum capitatum) spondylodiscitis. Relapse of the bone and joint lesions remained culture negative and paralleled the immune restitution, suggesting an immune reconstitution inflammatory syndrome (IRIS).

D. capitatus (anamorph G. capitatum) is a filamentous yeast-like fungus that composes a part of the normal human flora of the skin, the gastrointestinal and respiratory tracts.1 Infections in immunocompromised patients often mimic invasive candidiasis but are more likely associated with multiorgan involvement and fatal course.2, 3 Here, we report a case of disseminated D. capitatus infection with spondylodiscitis and probable pneumonia in a patient with allogeneic BMT. Only two other patients with D. capitatus spondylodiscitis have been described.4, 5 Our patient experienced significant improvement of the L5–S1 bone and disc infections after prolonged L-AmB therapy and the subsequent deterioration observed after 3 months of voriconazole was unexpected. First, the drug blood levels, which were repeatedly measured, were persistently above 1 mg/L, a threshold that has been linked to appropriate absorption and therapeutic success.6 These drug levels were satisfactory regarding the minimal inhibitory concentration (MIC) values exhibited by the isolates (voriconazole MICs ranged from 0.125 to 0.25 mg/L). However, it must be stressed that no interpretative criteria for the in vitro tests have been proposed for this fungus. Second, the patient experienced, concurrent with the relapse, a decrease (even more a discontinuation of corticosteroids) of the immunosuppressive therapy and control of the GVHD, two conditions that usually favor the recovery of invasive fungal infections in general, and D. capitatus infections in particular.2 Third, as voriconazole has shown high ability to penetrate bone and is orally available, it appears a drug of choice in fungal osteomyelitis management.7 The temporal association of new vertebral lesions that remained culture-negative despite a three-week period free of any antifungal therapy, histological findings showing unspecific osteitis with fibrosis and inflammatory cells, and immune restoration is highly reminiscent of IRIS. This syndrome was initially described in AIDS patients with fungal infections such as cryptococcosis, histoplasmosis and pneumocystosis, and who responded to antiretroviral therapy.8 IRIS has been also reported in neutropenic patients with exacerbation of invasive aspergillosis and in chronic disseminated candidiasis.9, 10

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Acknowledgements

We thank Dr C Plainvert and G Collobert (Microbiology Laboratory, Hôtel-Dieu) for molecular identification (ITS sequencing) of the Dipodascus isolates. We also thank Dr H Yera (Mycology and Parasitology Laboratory, Cochin Hospital) for the mycological and molecular analysis of the L4–L5 biopsy specimens.

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Authors and Affiliations

  1. Department of Haematology, Hôtel-Dieu, Assistance-Publique Hôpitaux de Paris, Paris, France

    S Lapusan, S Dimicoli, Z Marjanovic & B Rio

  2. University of Paris Descartes, Paris, France

    S Dimicoli, F Lanternier, M-E Bougnoux & O Lortholary

  3. Department of Pneumology, Hôtel-Dieu, Assistance-Publique Hôpitaux de Paris, Paris, France

    A Lefebvre

  4. Department of Radiology, Hôtel-Dieu, Assistance-Publique Hôpitaux de Paris, Paris, France

    C Bordonné

  5. Department of Infectious Diseases, Necker Hospital, Assistance-Publique Hôpitaux de Paris, Paris, France

    F Lanternier & O Lortholary

  6. Laboratory of Parasitology-Mycology, Saint-Louis Hospital, Assistance-Publique Hôpitaux de Paris, University of Paris Diderot, Paris, France

    C Lacroix

  7. Laboratory of Microbiology, Necker Hospital, Assistance-Publique Hôpitaux de Paris, Paris, France

    M-E Bougnoux

  8. Laboratory of Microbiology, Hôtel-Dieu, Assistance-Publique Hôpitaux de Paris, Paris, France

    M Cornet

  9. Laboratory of Parasitology-Mycology, Grenoble Hospital, University of Joseph Fourier, Grenoble, France

    M Cornet

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  1. S Lapusan
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Corresponding author

Correspondence to M Cornet.

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Competing interests

O Lortholary is a member of the speaker bureau of Pfizer, MSD, Astellas and Gilead Sciences. All other authors declare no conflict of interest.

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Lapusan, S., Dimicoli, S., Marjanovic, Z. et al. Immune reconstitution inflammatory syndrome following allo-SCT in a patient with Dipodascus capitatus spondylodiscitis. Bone Marrow Transplant 46, 1265–1267 (2011). https://doi.org/10.1038/bmt.2010.267

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