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Extensive brain infarction involving deep structures during an acetazolamide-challenged single-photon emission computed tomography scan in a patient with moyamoya disease

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Abstract

Background

Acetazolamide-challenged brain single-photon emission computed tomography (SPECT) is used for the evaluation of cerebral perfusion in cerebrovascular diseases including moyamoya disease (MMD). Not a few patients experience adverse side effects during the acetazolamide-challenged brain SPECT, but most of the symptoms are mild and transient. To our knowledge, this is the first case report of severe brain infarction leading to death during the examination of an acetazolamide-challenged brain SPECT in a patient with MMD.

Case presentation

An 11-year-old girl who had been diagnosed of MMD demonstrated sudden tonic movement during an acetazolamide-challenged brain SPECT as the preoperative examination for the second surgery. She had not experienced any adverse effect during the previous SPECT study and her first indirect bypass surgery on both left side and bifrontal area was uneventful. After she had seizures twice, she became unconscious and her pupils were dilated and fixed. Acute infarction involving bilateral occipital lobes, thalami, brainstem, and cerebellum was observed on brain magnetic resonance images which led to brain death.

Conclusion

We report a mortality case of patient with MMD after the administration of acetazolamide during the examination of brain SPECT that was accompanied by an extensive acute infarction involving the bilateral occipital lobes and thalami, brainstem, and cerebellum. Physicians should be aware of this rare but serious complication.

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Acknowledgments

This research was supported by the Original Technology Research Program for Brain Science through the National Research Foundation of Korea (NRF) funded by the Ministry of Science ICT and Future Planning (2015M3C7A1029034).

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Authors and Affiliations

  1. Division of Pediatric Neurosurgery, Seoul National University Children’s Hospital, 101 Daehak-ro, Jongno-gu, Seoul, 03080, Republic of Korea

    Sangjoon Chong, Seung-Ki Kim, Ji Hoon Phi, Ji Yeoun Lee & Kyu-Chang Wang

  2. Department of Pediatrics, Seoul National University Children’s Hospital, Seoul, Republic of Korea

    June Dong Park & Jong Hee Chae

  3. Division of Pediatric Radiology, Seoul National University Children’s Hospital, Seoul, Republic of Korea

    Jung-Eun Cheon

  4. Department of Anatomy, College of Medicine, Seoul National University, Seoul, Republic of Korea

    Ji Yeoun Lee

  5. Department of Nuclear Medicine, Seoul National University Hospital, 101 Daehak-ro, Jongno-gu, Seoul, 03080, Republic of Korea

    Jin Chul Paeng

Authors
  1. Sangjoon Chong
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  2. June Dong Park
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  3. Jong Hee Chae
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  4. Jung-Eun Cheon
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  5. Seung-Ki Kim
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  6. Ji Hoon Phi
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  7. Ji Yeoun Lee
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  8. Jin Chul Paeng
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  9. Kyu-Chang Wang
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Corresponding authors

Correspondence to Jin Chul Paeng or Kyu-Chang Wang.

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This case report was approved by Institutional Review Board (IRB) of Seoul National University Hospital (IRB No. 1701-074-824).

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The authors declare that they have no conflict of interest.

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Chong, S., Park, J.D., Chae, J.H. et al. Extensive brain infarction involving deep structures during an acetazolamide-challenged single-photon emission computed tomography scan in a patient with moyamoya disease. Childs Nerv Syst 33, 2029–2033 (2017). https://doi.org/10.1007/s00381-017-3512-0

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