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Neurococcidiomycosis in children with hydrocephalus: assessment of functional outcome, quality of life and survival in relation to neuroimaging findings

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Abstract

Purpose

Coccidioidal meningitis (CM) is an uncommon disease frequently misdiagnosed. Neuroimaging and mortality are not considered in detail in previous pediatric CM series. Our objective is to evaluate outcome of pediatric neurococcidiomycosis in relation to neuroimaging findings.

Methods

We performed a prospective, observational, cross-sectional study in children with hydrocephalus and CM treated at Specialties Hospital in Torreon, Mexico (between 2015 and 2020). The outcome was evaluated by Hydrocephalus Outcome Questionnaire (HOQ) and the modified Rankin Scale (mRS). Follow-up was established at the first shunt surgery and survival since CM diagnosis confirmation. Neuroimaging was analyzed in relation to clinical data, outcome and survival. Kaplan-Meier analysis was performed with IBM-SPSS-25.

Results

Ten pediatric cases with CM and hydrocephalus were reported. Aged 6–228 months, 60% were female. Mean number of surgeries was 4.3 SD ± 3 (range 1–15). Asymmetric hydrocephalus was the most common neuroimaging finding (70%), followed by cerebral vasculitis (20%) and isolated fourth ventricle (IFV) (20%). The mean HOQ overall score was 0.338 SD ± 0.35. A minimum follow-up of 18 months was reported. Mean survival was 13.9 SD ± 6.15 months (range 3–24). Poor survival was correlated with asymmetric hydrocephalus (p = 0.335), cerebral vasculitis (p = 0.176), IFV (p < 0.001), bacterial superinfection (p = 0.017), lower mRS scores at hospital discharge (p = 0.017) and during follow-up (p = 0.004). The mortality rate was 20%.

Conclusions

We report the largest series in Latin America of pediatric CM and hydrocephalus. Asymmetric hydrocephalus, IFV and cerebral vasculitis are complications that increase mortality and must be early diagnosed for a timely surgical and medical treatment. HOQ and mRS could be alternative scales to evaluate outcome in these patients. After a long follow-up (18 months), survival remained poor after diagnosis confirmation in our series.

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Availability of data material

The datasets generated during and/or analyzed during the current study are available from the corresponding author upon reasonable request.

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Acknowledgements

We are grateful to Professor Abhaya V. Kulkarni for his advice on HOQ calculation. We express our acknowledgment to the National Council of Science and Technology (CONACyT) from Mexico.

Author information

Authors and Affiliations

  1. Department of Neurosurgery and Neurotechnologie, Universitätsklinik Tübingen, Tübingen, Germany

    Maria F. De la Cerda-Vargas

  2. Department of Neurosurgery, Specialties Hospital No. 71, Instituto Mexicano del Seguro Social, Torreon, Coahuila, Mexico

    Maria F. De la Cerda-Vargas, Jose A. Candelas-Rangel, Pedro Navarro-Dominguez & Luis H. Ramirez-Silva

  3. Associated Professor, Department of Neurosurgery, Specialties Hospital, Centro Médico Nacional (CMN) Siglo XXI, Instituto Mexicano del Seguro Social, Mexico City, Mexico

    Bayron A. Sandoval-Bonilla

  4. Head of Anatomical Pathology Department, Specialties Hospital No. 71, Instituto Mexicano del Seguro Social, Torreon, Coahuila, Mexico

    Elizabeth Meza-Mata

  5. Department of Health and Research, Specialties Hospital No. 71, Instituto Mexicano del Seguro Social, Torreón, Coahuila, Mexico

    Melisa A. Muñoz-Hernandez & F. K. Segura-Lopez

  6. Microbiology Department, Faculty of Medicine, Universidad Autónoma de Coahuila, Torreon, Coahuila, Mexico

    Marisela Del Rocio Gonzalez-Martinez

  7. Department of Transplants, Specialties Hospital No. 71, Instituto Mexicano del Seguro Social, Torreon, Coahuila, Mexico

    Delgado-Aguirre Hector A.

  8. Department of Neurosrugery, Specialties Hospital No. 71, Instituto Mexicano del Seguro Social, Torreon, Coahuila, Mexico

    Jose A. Candelas-Rangel

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  1. Maria F. De la Cerda-Vargas
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Contributions

María F. De la Cerda-Vargas wrote the main manuscript. Author MF had the conception of the study and was in charge of data collection and analysis. B.A. Sandoval-Bonilla, José A. Candelas-Rangel and Pedro Navarro-Domínguez were in charge of writing the manuscript and editing the images, tables, and figures. B.A. Sandoval-Bonilla and Luis H. Ramirez-Silva wrote and edited the manuscript in English. Elizabeth Meza-Mata provided images and descriptions of pathological studies. Melisa A. Muñoz- Hernández and F. K. Segura-López performed the statistical analysis and review of the results of the study. Marisela Del Rocio González-Martínez, and Héctor A. Delgado-Aguirre supported the review of the literature and preparation of discussion and analysis. All authors reviewed and approved the final manuscript.

Corresponding author

Correspondence to Maria F. De la Cerda-Vargas.

Ethics declarations

Ethics approval and consent to participate

This study was approved by the institutional research ethics committee (Local Health Research Committee 501, COFEPRIS 17 CI 05 035 078, Institutional registration ID R-2021-501-054). We certify that this study was performed in accordance with the ethical standards as laid down in the 1964 Declaration of Helsinki and its later amendments or comparable ethical standards. Children’s tutors signed informed consent before surgery, sampling for laboratory, and pathologic studies. No personally identifiable data or photographs of the participants are exposed in this study. Informed consent was not required because the information was anonymized and the submission does not include images that may identify the person. All children’s tutors signed informed consent to participate in the study (before surgery, sampling for laboratory and pathology, interviews (HOQ), etc.).

Consent for publication

Informed consent was obtained from legal guardians.

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There are no conflicts of interest.

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Appendices

Appendix 1

Fig. 7
figure 7

Box and whisker plots of asymmetric hydrocephalus (AH) in relation to age, hospital stay, diagnosis length, surgeries, and HOQ overall score. LoS = hospital length of stay

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Fig. 8
figure 8

Box and whisker plots of cerebral vasculitis in relation to age, hospital stay, diagnosis length, surgeries, and HOQ overall score. LoS = hospital length of stay

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Fig. 9
figure 9

Box and whisker plots of IFV in relation to age, hospital stay, diagnosis length, surgeries, and HOQ overall score. LoS = hospital length of stay

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Appendix 2

Fig. 10
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Univariate analysis (Kaplan-Meier) for overall survival

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De la Cerda-Vargas, M.F., Candelas-Rangel, J.A., Navarro-Dominguez, P. et al. Neurococcidiomycosis in children with hydrocephalus: assessment of functional outcome, quality of life and survival in relation to neuroimaging findings. Childs Nerv Syst 40, 303–319 (2024). https://doi.org/10.1007/s00381-023-06166-x

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