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Cavernous angioma after chemotherapy for desmoplastic/nodular medulloblastoma associated with anhidrotic ectodermal dysplasia

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Abstract

Purpose

While cavernous angioma (CVA) after cranial irradiation has been documented, its development after high-dose chemotherapy with autologous peripheral blood stem cell transplantation (PBSCT) has not. We present a patient with desmoplastic/nodular medulloblastoma (DNMB) associated with anhidrotic ectodermal dysplasia (AED) who developed CVA 2 years after high-dose chemotherapy and PBSCT.

Methods

A 1-year-old boy with ingravescent vomiting was admitted to our institute. He presented with a large head, a depressed nasal bridge, low-set ears, thick lips with peg-shaped teeth, hypohidrosis, sparse hair, thin atrophic skin, scaly dermatitis with frontal bossing, and a bulging anterior fontanel. Neuroradiological examination revealed multiple cerebellar masses with heterogeneous enhancement and speckled calcifications and severe obstructive hydrocephalus. The histological diagnosis of surgical specimens was DNMB, and he underwent postoperative multiple-drug chemotherapy with autologous PBSCT. The outcome was favorable and he did not undergo radiotherapy.

Results

After 2 years, intracranial hemorrhage was detected at his regular radiological check-up and he again underwent surgery. The histological diagnosis was CVA.

Conclusions

To our knowledge, this is the first report of AED-associated DNMB and CVA.

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Acknowledgments

We thank Ursula Petralia for editorial review. This study was partly supported by the Japan Society for the Promotion of Science Grant-in-Aid for Scientific Research (C) number 25462262.

Conflict of interest

The authors have no conflicts of interest to disclose.

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Authors and Affiliations

  1. Department of Neurosurgery, Institute of Biomedical and Health Sciences, Hiroshima University, 1-2-3 Kasumi, Minami-ku, Hiroshima, 734-8551, Japan

    Fumiyuki Yamasaki, Takeshi Takayasu, Ryo Nosaka & Kaoru Kurisu

  2. Department of Pediatrics, Graduate School of Biomedical and Health Sciences, Hiroshima University, Hiroshima, 734-8551, Japan

    Hiroshi Kawaguchi & Masao Kobayashi

  3. Department of Clinical Oncology and Neuro-oncology Program, Hiroshima University Hospital, Hiroshima, 734-8551, Japan

    Kazuhiko Sugiyama

Authors
  1. Fumiyuki Yamasaki
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  2. Takeshi Takayasu
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  3. Ryo Nosaka
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  4. Hiroshi Kawaguchi
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  5. Kazuhiko Sugiyama
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  6. Masao Kobayashi
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  7. Kaoru Kurisu
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Corresponding author

Correspondence to Fumiyuki Yamasaki.

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Yamasaki, F., Takayasu, T., Nosaka, R. et al. Cavernous angioma after chemotherapy for desmoplastic/nodular medulloblastoma associated with anhidrotic ectodermal dysplasia. Childs Nerv Syst 32, 395–398 (2016). https://doi.org/10.1007/s00381-015-2848-6

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  • DOI: https://doi.org/10.1007/s00381-015-2848-6

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