Complete recovery from Cryptosporidium parvum infection with gastroenteritis and sclerosing cholangitis after successful bone marrow transplantation in two brothers with X-linked hyper-IgM syndrome

Dimicoli, Sophie; Bensoussan, D.; Latger‐Cannard, Véronique; Straczek, J.; Antunes, Laurent; Mainard, L.; Dao, Anne; Barbé, Françoise; Araújo, Carlo Scognamiglio Renner; Clément, Laurence; Feugier, Pierre; Lecompte, T.; Stoltz, J F; Bordigoni, Pierre

doi:10.1038/sj.bmt.1704211

Cited by 43 publications

(22 citation statements)

References 16 publications

(18 reference statements)

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“…While a number of pathogens have been implicated, C. parvum is the single most common identifiable pathogen associated with this disease. Although biliary cryptosporidiosis is also seen in boys with X-linked immunodeficiency (18,22), in adults it has been reported only in patients with AIDS (12,17,21,25,30). In contrast, C. parvum infection of the intestine has been found in both immunocompetent and immunocompromised subjects (40).…”

mentioning

confidence: 89%

The Human Immunodeficiency Virus Type 1 Tat Protein EnhancesCryptosporidium parvum-Induced Apoptosis in Cholangiocytes via a Fas Ligand-Dependent Mechanism

et al. 2007

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While Cryptosporidium parvum infection of the intestine has been reported in both immunocompetent and immunocompromised individuals, biliary infection is seen primarily in adult AIDS patients and is associated with development of AIDS cholangiopathy. However, the mechanisms of pathogen-induced AIDS cholangiopathy remain unclear. Since we previously demonstrated that the Fas/Fas ligand (FasL) system is involved in paracrine-mediated C. parvum cytopathicity in cholangiocytes, we also tested the potential synergistic effects of human immunodeficiency virus type 1 (HIV-1) transactivator of transcription (Tat)-mediated FasL regulation on C. parvum-induced apoptosis in cholangiocytes by semiquantitative reverse transcription-PCR, immunoblotting, immunofluorescence analysis, and immunogold electron microscopy. H69 cells do not express CXCR4 and CCR5, which are receptors required for direct HIV-1 viral infection. However, recombinant biologically active HIV-1-associated Tat protein increased FasL expression in the cytoplasm of cholangiocytes without a significant increase in apoptosis. We found that C. parvum-induced apoptosis was associated with translocation of intracellular FasL to the cell membrane surface and release of full-length FasL from infected H69 cells. Tat significantly (P < 0.05) increased C. parvum-induced apoptosis in bystander cells in a dose-dependent manner. Moreover, Tat enhanced both C. parvum-induced FasL membrane translocation and release of full-length FasL. In addition, the FasL neutralizing antibody NOK-1 and the caspase-8 inhibitor Z-IETD-fmk both blocked C. parvum-induced apoptosis in cholangiocytes. The data demonstrated that HIV-1 Tat enhances C. parvum-induced cholangiocyte apoptosis via a paracrine-mediated, FasL-dependent mechanism. Our results suggest that concurrent active HIV replication, with associated production of Tat protein, and C. parvum infection synergistically increase cholangiocyte apoptosis and thus jointly contribute to AIDS-related cholangiopathies.

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mentioning

confidence: 89%

The Human Immunodeficiency Virus Type 1 Tat Protein EnhancesCryptosporidium parvum-Induced Apoptosis in Cholangiocytes via a Fas Ligand-Dependent Mechanism

et al. 2007

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“…It is routinely used to treat haematological malignancy but case reports document improvement in a variety of other conditions in those treated with this procedure [79][80][81]. Cancer is an increasing problem and the evidence outlined in this review points to the tumour stroma as a more dynamic component of tumours than previously appreciated.…”

Section: Therapeutic Implications Of Bm-derived Tumour Stromamentioning

confidence: 91%

Bone marrow and tumour stroma: an intimate relationship

Direkze

Alison

2006

Hematological Oncology

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In recent years the bone marrow has become recognized as a potential source of cells for non-haematopoietic wound healing, in some instances demonstrating surprising plasticity in providing new epithelial cells. On the other hand, the contribution of bone marrow derived cells to fibrosis and blood vessel formation is more widely acknowledged. Tumour stroma has a vital role to play in determining cancer growth and spread, and there is a growing realization that the bone marrow has a significant input into this desmoplastic response. This review focuses on the contribution of bone marrow cells to tumour stroma, highlighting the bone marrow as a potential new portal through which to direct anti-tumour therapies.

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“…commun.]. Dimicoli et al [4 ]described two brothers with CD40L deficiency affected with C. parvum infection and cholangitis. After BMT, expression of CD40L on activated T lymphocytes was normal and both C. parvum infection and cholangitis resolved [4].…”

Section: Discussionmentioning

confidence: 99%

“…Children with CD40L deficiency have been known to develop sclerosing cholangitis associated with cryptosporidium infection [3,4]. However, the development of cholangiocarcinoma arising from sclerosing cholangitis secondary to cryptosporidial infection has not been previously reported in an adult with this combined immune deficiency.…”

Section: Introductionmentioning

confidence: 94%

“…Dimicoli et al [4 ]described two brothers with CD40L deficiency affected with C. parvum infection and cholangitis. After BMT, expression of CD40L on activated T lymphocytes was normal and both C. parvum infection and cholangitis resolved [4]. However, in one study of CD40L deficiency in children with pre-existing liver disease, 5 out of 12 patients were found to be chronically colonised though only 1 had a known history of infection.…”

Section: Discussionmentioning

confidence: 99%

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Cholangiocarcinoma Complicating Secondary Sclerosing Cholangitis from Cryptosporidiosis in an Adult Patient with CD40 Ligand Deficiency: Case Report and Review of the Literature

Rahman¹,

Chapel

Chapman³

et al. 2012

Int Arch Allergy Immunol

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A 43-year-old man with a hyper-immunoglobulin M syndrome due to CD40 ligand deficiency presented with insidious onset of recurrent diarrhoea and deranged liver function tests. Standard stool microscopy was repeatedly negative for cryptosporidia but immunofluorescent testing and polymerase chain reaction demonstrated the presence of infection eventually. Despite both paromomycin and nitazoxanide, he developed sclerosing cholangitis secondary to cryptosporidial infection. Whilst being considered for dual bone marrow and liver transplantation, he was found to have cholangiocarcinoma on imaging after three biopsies of a suspicious lesion. This is a rare complication of this combined immune deficiency predominantly in children that has not been reported previously in a long-term survivor with this condition.

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Complete recovery from Cryptosporidium parvum infection with gastroenteritis and sclerosing cholangitis after successful bone marrow transplantation in two brothers with X-linked hyper-IgM syndrome

Cited by 43 publications

References 16 publications

The Human Immunodeficiency Virus Type 1 Tat Protein EnhancesCryptosporidium parvum-Induced Apoptosis in Cholangiocytes via a Fas Ligand-Dependent Mechanism

The Human Immunodeficiency Virus Type 1 Tat Protein EnhancesCryptosporidium parvum-Induced Apoptosis in Cholangiocytes via a Fas Ligand-Dependent Mechanism

Bone marrow and tumour stroma: an intimate relationship

Cholangiocarcinoma Complicating Secondary Sclerosing Cholangitis from Cryptosporidiosis in an Adult Patient with CD40 Ligand Deficiency: Case Report and Review of the Literature

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