Evidence map›Paper›PMID 42117555›Full record

ArticleThe Journal of infectious diseases2026

Cryptosporidiosis in inborn errors of immunity: a single center cohort and review of the literature.

Chen Wang, Evgenii Filippov, Amir Seyedmousavi, David E Kleiner, Anjali Rai, Theo Heller, Ajoy L Dias, Corina E Gonzalez, Danielle E Arnold, Dimana Dimitrova and 8 more

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In one paragraph

Article in The Journal of infectious diseases, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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0citing papers in PubMed
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1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

0 citing papers in PubMed.

No citing paper in PubMed yet.

4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

18 authors.

Chen WangLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA.ORCID 0000-0003-4055-7391
Evgenii FilippovLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA.
Amir SeyedmousaviMicrobiology Service, Department of Laboratory Medicine, Clinical Center, National Institutes of Health, Bethesda, MD 20852, USA.
David E KleinerLaboratory of Pathology, National Cancer Institute, National Institutes of Health, Bethesda, MD, USA.
Anjali RaiLiver Diseases Branch, National Institute of Diabetes and Digestive and Kidney Diseases, National Institutes of Health, Bethesda, MD, USA.ORCID 0000-0002-8343-4132
Theo HellerLiver Diseases Branch, National Institute of Diabetes and Digestive and Kidney Diseases, National Institutes of Health, Bethesda, MD, USA.ORCID 0000-0002-2643-6289
Ajoy L DiasImmune Deficiency Cellular Therapy Program, Center for Cancer Research, National Cancer Institute, National Institute of Health, Bethesda, MD, USA.
Corina E GonzalezImmune Deficiency Cellular Therapy Program, Center for Cancer Research, National Cancer Institute, National Institute of Health, Bethesda, MD, USA.
Danielle E ArnoldImmune Deficiency Cellular Therapy Program, Center for Cancer Research, National Cancer Institute, National Institute of Health, Bethesda, MD, USA.
Dimana DimitrovaCenter for Immuno-Oncology, National Cancer Institute, National Institutes of Health, Bethesda, MD, USA.ORCID 0000-0002-4868-6852
Harry L MalechLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA.ORCID 0000-0001-5874-5775
Sung-Yun PaiImmune Deficiency Cellular Therapy Program, Center for Cancer Research, National Cancer Institute, National Institute of Health, Bethesda, MD, USA.ORCID 0000-0002-0158-8147
Luigi D NotarangeloLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA.
Steven M HollandLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA.
Gulbu UzelLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA.ORCID 0009-0003-0700-694X
Jennifer Cuellar-RodriguezLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA.ORCID 0000-0001-5298-6463
Alexandra F FreemanLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA.
Mary M CzechLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA.

Funding

Intramural NIH HHS Z99 AI999999
6 · The paper itself

Abstract

backgroundSevere intestinal or hepatobiliary cryptosporidiosis affects patients with inborn errors of immunity (IEI), particularly those with CD40 ligand or CD40 deficiency, major histocompatibility complex class II deficiency, interleukin-21 receptor deficiency, and DOCK8 deficiency. Susceptibility to Cryptosporidium reflects defects in lymphocyte development, activation, and effector function.

methodsWe conducted a retrospective chart review of IEI patients with cryptosporidiosis, focusing on 17 individuals with chronic infection who underwent hematopoietic cell transplant (HCT), to characterize their clinical course, diagnostic evaluation, management, and outcomes. A literature review was also performed to summarize current knowledge of cryptosporidiosis in IEI patients.

resultsPatients frequently presented with secondary sclerosing cholangitis, posing significant diagnostic and therapeutic challenges. Molecular diagnostic assays offered higher sensitivity than conventional microscopy. Antiparasitic agents had limited efficacy against hepatobiliary disease. Durable immune reconstitution through HCT remained the mainstay of management, though it carries risk, especially in patients with established liver disease. Prophylactic strategies for at-risk individuals remain underexplored but should include exposure avoidance and consideration of antiparasitic prophylaxis.

conclusionsCryptosporidium-related sclerosing cholangitis imposes a substantial disease burden in susceptible IEI patients, underscoring the importance of early recognition and proactive management, particularly timely immune reconstitution in this population.

Indexed as

cryptosporidiosishematopoietic cell transplantationinborn errors of immunity

Identifiers

PMID42117555
PMCPMC13223774

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.