Skip to main content

Advertisement

SpringerLink
Log in

Bleeding pediatric AVM with negative pre-operative angiography: the first case report

  • Case Report
  • Published:
Child's Nervous System Aims and scope Submit manuscript

Abstract

Introduction

Pediatric arteriovenous malformation (pAVM) is one of the most common vascular entities in non-traumatic intracerebral hemorrhage (ICH) in children. To diagnose arteriovenous malformation (AVM), digital subtraction angiography (DSA) is the gold standard investigation because it can provide sufficient dynamic information about the AVM. In extremely rare occasions, angiography is unable to detect an AVM because the AVM is spontaneously occluded. All reported cases found by authors in the literature had already been diagnosed with AVM by angiography or other vascular studies before the AVM occlusion.

Case Presentation

We present a case of a 4-year-old girl who presented with left occipital ICH with atypical calcification. Based on history and investigation, pAVM was the most likely diagnosis. However, preoperative angiography was negative for pAVM and for shunting. Bleeding tumor was then suspected instead. After resection, pathological diagnosis confirmed pAVM.

Conclusion

Our case demonstrates that DSA, despite being considered the gold standard, cannot always diagnose pAVM. The mechanism of spontaneous AVM occlusion remains unknown.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2
Fig. 3
Fig. 4
Fig. 5

Similar content being viewed by others

Abbreviations

AVM:

Arteriovenous malformation

CT:

Computed tomography

DNET:

Dysembryonic neuroepithelial tumor

DSA:

Digital subtraction angiography

GFAP:

Glial fibrillary acidic protein

ICH:

Intracerebral hemorrhage

MRI:

Magnetic resonance imaging

pAVM:

Pediatric arteriovenous malformation

References

  1. Boulouis G, Blauwblomme T, Hak JF, Benichi S, Kirton A, Meyer P, Chevignard M, Tournier-Lasserve E, Mackay MT, Chabrier S, Cordonnier C, Kossorotoff M, Naggara O (2019) Nontraumatic Pediatric Intracerebral Hemorrhage. Stroke 50:3654–3661. https://doi.org/10.1161/STROKEAHA.119.025783

    Article  PubMed  Google Scholar 

  2. Lu AY, Winkler EA, Garcia JH, Raygor KP, Fullerton HJ, Fox CK, Kim H, Auguste KI, Sun PP, Hetts SW, Lawton MT, Abla AA, Gupta N (2023) A comparison of incidental and symptomatic unruptured brain arteriovenous malformations in children. J Neurosurg Pediatr 31:463–468. https://doi.org/10.3171/2023.1.Peds22541

    Article  PubMed  Google Scholar 

  3. El-Ghanem M, Kass-Hout T, Kass-Hout O, Alderazi YJ, Amuluru K, Al-Mufti F, Prestigiacomo CJ, Gandhi CD (2016) Arteriovenous malformations in the pediatric population: review of the existing literature. Interv Neurol 5:218–225. https://doi.org/10.1159/000447605

    Article  PubMed Central  Google Scholar 

  4. Krapf H, Siekmann R, Freudenstein D, Küker W, Skalej M (2001) Spontaneous occlusion of a cerebral arteriovenous malformation: angiography and MR imaging follow-up and review of the literature. AJNR Am J Neuroradiol 22:1556–1560

    CAS  PubMed Central  Google Scholar 

  5. Ciochon UM, Bindslev JBB, Hoei-Hansen CE, Truelsen TC, Larsen VA, Nielsen MB, Hansen AE (2022) Causes and risk factors of pediatric spontaneous intracranial hemorrhage-a systematic review. Diagnostics (Basel) 12. https://doi.org/10.3390/diagnostics12061459

  6. Ajiboye N, Chalouhi N, Starke RM, Zanaty M, Bell R (2014) Cerebral arteriovenous malformations: evaluation and management. Sci World J 2014:649036. https://doi.org/10.1155/2014/649036

  7. Gibb PA, Gullan RW, Sandhu N (1988) Spontaneous angiographic disappearance of an intracranial arteriovenous malformation. Br J Neurosurg 2:529–533. https://doi.org/10.3109/02688698809029610

    Article  CAS  PubMed  Google Scholar 

  8. Golev D, Larrazabal R, Gunnarson T, Algird A (2012) Spontaneous occlusion of the temporal AVM associated with tinnitus. Can J Neurol Sci 39:255–257. https://doi.org/10.1017/S0317167100022204

    Article  Google Scholar 

  9. Goyal N, Hoit D, Elijovich L (2014) Spontaneous thrombosis of a ruptured brain arteriovenous malformation: the argument for early conservative management. Interv Neurol 3:122–128. https://doi.org/10.1159/000381035

    Article  Google Scholar 

  10. Kobari T, Nagai M, Miyata S, Oguma H, Watanabe E (2014) Spontaneous occlusion of the frontal arteriovenous malformation and the following progression of the associated aneurysm: a case report. J Stroke Cerebrovasc Dis 23:1253–1255. https://doi.org/10.1016/j.jstrokecerebrovasdis.2013.08.024

    Article  Google Scholar 

  11. Kumar R, Madhugiri V, Ramesh A, Sathiaprabhu A (2014) Spontaneous thrombosis of a cerebral arteriovenous malformation. Neurol India 62:565–567. https://doi.org/10.4103/0028-3886.144500

    Article  PubMed  Google Scholar 

  12. Liew JA, Yang W, Mashouf LA, Li S, Caplan JM, Tamargo RJ, Huang J (2020) Incidence of spontaneous obliteration in untreated brain arteriovenous malformations. Neurosurgery 86:139–149. https://doi.org/10.1093/neuros/nyz047

    Article  PubMed  Google Scholar 

  13. Lim S, Foo A, Karlsson B, Yeo T (2016) Spontaneous obliteration highlights the dynamic nature of cerebral arteriovenous malformations: a case report and review of the literature. Surg Neurol Int 7:45

    Article  PubMed Central  Google Scholar 

  14. Panciani PP, Fontanella M, Carlino C, Bergui M, Ducati A (2008) Progressive spontaneous occlusion of a cerebellar AVM: pathogenetic hypothesis and review of literature. Clin Neurol Neurosurg 110:502–510. https://doi.org/10.1016/j.clineuro.2008.01.009

    Article  CAS  Google Scholar 

  15. Rajasekaran V, Gutmann L, Hogg J (2012) Spontaneous occlusion of arteriovenous malformation (P05.271). Neurology 78:P05.271

  16. Shchehlov DV, Svyrydiuk OE, Chebanyuk SV, Slobodian OV, Vyval MB (2022) Spontaneous occlusion of the cerebral arteriovenous malformations. Ukrainian Interv Neuroradiol Surg 39:34–39. https://doi.org/10.26683/2786-4855-2022-1(39)-34-39

    Article  Google Scholar 

  17. Sukoff MH, Barth B, Moran T (1972) Spontaneous occlusion of a massive arteriovenous malformation—Case report. Neuroradiology 4:121–123. https://doi.org/10.1007/BF00344441

    Article  CAS  Google Scholar 

  18. Barr JC, Ogilvy CS (2012) Selection of treatment modalities or observation of arteriovenous malformations. Neurosurg Clin N Am 23:63–75. https://doi.org/10.1016/j.nec.2011.09.010

    Article  Google Scholar 

  19. Agosti E, Graepel S, Lanzino G (2022) Principles and strategies for step-by-step AVM excision. Neurosurg Focus 53:E5. https://doi.org/10.3171/2022.4.FOCUS21786

    Article  PubMed  Google Scholar 

  20. Ghali MG, Srinivasan VM, Mohan AC, Jones JY, Kan PT, Lam S (2016) Pediatric cerebral cavernous malformations: genetics, pathogenesis, and management. Surg Neurol Int 7:S1127-s1134. https://doi.org/10.4103/2152-7806.196921

    Article  PubMed Central  Google Scholar 

  21. Pawane P, A, Gangane N, (2014) Hemangiomas versus arterio-venous malformations: role of elastic stains and mast cell density. Indian J Pathol Microbiol 57:191–195. https://doi.org/10.4103/0377-4929.134661

    Article  PubMed  Google Scholar 

Download references

Acknowledgements

We thank the Language Editing Center under Clinical Research Center, Faculty of Medicine, Thammasat University, for their kind help in editing the English language.

Author information

Authors and Affiliations

  1. Division of Neurosurgery, Department of Surgery, Thammasat University Hospital, Faculty of Medicine, Thammasat University, Pathumthani, Thailand

    Vich Yindeedej, Gahn Duangprasert, Raywat Noiphithak, Pree Nimmannitya, Prachya Punyarat & Dilok Tantongtip

  2. Department of Pathology, Thammasat University Hospital, Faculty of Medicine, Thammasat University, Pathumthani, Thailand

    Chatchai Thamwongskul

Authors
  1. Vich Yindeedej
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Gahn Duangprasert
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Raywat Noiphithak
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Pree Nimmannitya
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Prachya Punyarat
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. Dilok Tantongtip
    View author publications

    You can also search for this author in PubMed Google Scholar

  7. Chatchai Thamwongskul
    View author publications

    You can also search for this author in PubMed Google Scholar

Contributions

Conceptual and design: Yindeedej. Acquisition of data: Yindeedej, Duangprasert, Thamwongskul. Drafting the article: Yindeedej. Critically revising: Duangprasert. Review submitted version of manuscript: All authors. Approved the final version: Yindeedej.

Corresponding author

Correspondence to Vich Yindeedej.

Ethics declarations

Ethical approval

All procedures performed in studies involving human participants were in accordance with the ethical standards of the Ethics Committee of Faculty of Medicine, Thammasat University and with the 1964 Declaration of Helsinki and its later amendments or comparable ethical standards.

Informed consent

The patient consented to the procedure and written informed consent was obtained.

Conflict of Interest

All authors certify that they have no affiliations with or involvement in any organization or entity with any financial interest (such as honoraria; educational grants; participation in speakers’’ bureaus; membership, employment, consultancies, stock ownership, or other equity interest; and expert testimony or patent-licensing arrangements) or non-financial interest (such as personal or professional relationships, affiliations, knowledge or beliefs) in the subject matter or materials discussed in this manuscript. The authors have no personal, financial, or institutional interest in any of the drugs, materials, or devices described in this article.

Additional information

Publisher's Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Rights and permissions

Springer Nature or its licensor (e.g. a society or other partner) holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Yindeedej, V., Duangprasert, G., Noiphithak, R. et al. Bleeding pediatric AVM with negative pre-operative angiography: the first case report. Childs Nerv Syst 39, 3327–3331 (2023). https://doi.org/10.1007/s00381-023-06055-3

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00381-023-06055-3

Keywords

Search

Navigation