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Ruptured AComA aneurysm and asymptomatic bilateral ACA vasospasm in an infant: surgical case report

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Abstract

Intracranial aneurysms (IAs) are localized dilations of the cerebral vasculature, representing the leading cause for non-traumatic subarachnoid hemorrhage and an important source of morbidity and mortality. Despite it being a frequent pathology and most often diagnosed incidentally, IAs in infants are a very rare occurrence, and the ruptured variant is exceptional. A 4-month-old boy with a negative family history was brought to our department because of several episodes of incoercible vomiting and fever. Upon examination, the child was somnolent, without any noticeable deficit. Transfontanellar ultrasonography and CT angiography revealed a ruptured aneurysm of the anterior communicating artery (AComA), whereas the pre-clipping MRI showed thin, almost angiographically invisible anterior cerebral arteries (ACAs) on both sides due to vasospasm. We intervened surgically by placing an external ventricular shunt in an emergency setting, followed by clipping of the IA in a delayed manner. The child was discharged a month after admission with no deficit, despite the paradoxical aspect of the ACA. Ruptured IAs can be safely treated via microsurgery, even in infants. However, this requires a great amount of experience and surgical expertise. Furthermore, the lack of proper management would most likely result in a severe deficit in the long term. Lastly, the lack of visibility of the ACA on angiographic studies may not have neurological consequences if they occur in this age group.

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References

  1. Afifi AK, Godersky JC, Menezes A, Smoker WR, Bell WE, Jacoby CG (1987) Cerebral hemiatrophy, hypoplasia of internal carotid artery, and intracranial aneurysm. A rare association occurring in an infant. Arch Neurol 44(2):232–235. https://doi.org/10.1001/archneur.1987.00520140090024

    Article  CAS  PubMed  Google Scholar 

  2. Ahmad M, Butt RM, Ahmad N (2008) Clipping of intracranial aneurysms--3 years study. J Ayub Med Coll Abbottabad 20(3):75–77

    PubMed  Google Scholar 

  3. Aldakkan A, Mansouri A, Jaja BN, Alotaibi NM, Macdonald RL (2017) Subarachnoid hemorrhage international trialists collaborators. Predictors of delayed cerebral ischemia in patients with aneurysmal subarachnoid hemorrhage with asymptomatic angiographic vasospasm on admission. World Neurosurg 97:199–204. https://doi.org/10.1016/j.wneu.2016.09.096

    Article  PubMed  Google Scholar 

  4. Allison JW, Davis PC, Sato Y, James CA, Haque SS, Angtuaco EJ, Glasier CM (1998) Intracranial aneurysms in infants and children. Pediatr Radiol 28(4):223–229. https://doi.org/10.1007/s002470050336

    Article  CAS  PubMed  Google Scholar 

  5. Aryan HE, Giannotta SL, Fukushima T, Park MS, Ozgur BM, Levy ML (2006) Aneurysms in children: review of 15 years experience. J Clin Neurosci 13(2):188–192. https://doi.org/10.1016/j.jocn.2005.07.006

    Article  PubMed  Google Scholar 

  6. Block RI, Magnotta VA, Bayman EO, Choi JY, Thomas JJ, Kimble KK (2017) Are anesthesia and surgery during infancy associated with decreased white matter integrity and volume during childhood? Anesthesiology. 127(5):788–799

    Article  Google Scholar 

  7. Bolander H, Hassler O, Liliequist B, West KA (1978) Cerebral aneurysm in an infant with fibromuscular hyperplasia of the renal arteries. Case report J Neurosurg 49(5):756–759. https://doi.org/10.3171/jns.1978.49.5.0756

    Article  CAS  PubMed  Google Scholar 

  8. Börcek AÖ, Egemen E, Güngör G, Baykaner MK (2013) Intracranial aneurysm in childhood and interrupted aortic arch. Childs Nerv Syst 29(1):11–15

    Article  Google Scholar 

  9. Chen R, Ren Y, Zhang S, You C, Liu Y (2018) Radiologic characteristics and high risk of seizures in infants with ruptured intracranial aneurysms: case report and review of the literature. World Neurosurg 118:e772–e777

    Article  Google Scholar 

  10. Chen R, Zhang S, You C, Guo R, Ma L (2018) Pediatric intracranial aneurysms: changes from previous studies. Childs Nerv Syst 34(9):1697–1704

    Article  Google Scholar 

  11. Cheng Q, Li Z, Wang R, Zhang H, Cao H, Chen F, Li H, Xia Z, Feng S, Zhang H, Rui Y, Fan F (2019) Genetic profiles related to pathogenesis in sporadic intracranial aneurysm patients. World Neurosurg 131:e23–e31

    Article  Google Scholar 

  12. Chowdhary S, Garg P, Sawhney V, Pandya A, Sambhav K, Gupta S (2020) Unusual missed diagnosis of foreign body: a case report. Int Med Case Rep J 13:187–190. https://doi.org/10.2147/IMCRJ.S246924

    Article  PubMed  PubMed Central  Google Scholar 

  13. Ciurea AV, Munteanu V, Nica DA, Mohan A, Moisa H (2015) An aggressive management of anterior communicating artery aneurysms: predictive outcome factors. J Neurol Surg A Cent Eur Neurosurg 76 - A007. https://doi.org/10.1055/s-0035-1566326

  14. Condette-Auliac S, Bracard S, Anxionnat R, Schmitt E, Lacour JC, Braun M, Meloneto J, Cordebar A, Yin L, Picard L (2001) Vasospasm after subarachnoid hemorrhage: interest in diffusion-weighted MR imaging. Stroke. 32(8):1818–1824. https://doi.org/10.1161/01.str.32.8.1818

    Article  CAS  PubMed  Google Scholar 

  15. Del Santo MA, Cordina SM (2016) Infantile intracranial aneurysm of the superior cerebellar artery. BMJ Case Rep 2016:bcr2015012175 Published 2016 Feb 29

    Article  Google Scholar 

  16. Deora H, Rao KVLN, Somanna S, Srinivas D, Shukla DP, Bhat DI (2017) Surgically managed pediatric intracranial aneurysms: how different are they from adult intracranial aneurysms? Pediatr Neurosurg 52(5):313–317

    Article  Google Scholar 

  17. Elgamal EA, Murshid WR, Abu-Rahma HM, Samir D (2004) Aneurysmal subarachnoid hemorrhage in the first year of life: case report and review of the literature. Childs Nerv Syst 20(7):489–493. https://doi.org/10.1007/s00381-003-0891-1

    Article  PubMed  Google Scholar 

  18. Finitsis S, Anxionnat R, Lebedinsky A, Albuquerque PC, Clayton MF, Picard L, Bracard S (2010) Endovascular treatment of ACom intracranial aneurysms. Report on series of 280 patients. Interv Neuroradiol 16:7–16

    Article  CAS  Google Scholar 

  19. Florian IA, Timis TL, Aldea CC, Florian IS. Preventing rupture: clipping of unruptured intracranial aneurysms In: Ambrosi PB, Ahmad R, Abdulahi A, Agrawal A, editors. New insight into cerebrovascular diseases - an updated comprehensive review. IntechOpen. 2019; https://doi.org/10.5772/intechopen.88038. Available from: https://www.intechopen.com/books/new-insight-into-cerebrovascular-diseases-an-updated-comprehensive-review/preventing-rupture-clipping-of-unruptured-intracranial-aneurysms

  20. Garg K, Singh PK, Sharma BS, Chandra PS, Suri A, Singh M, Kumar R, Kale SS, Mishra NK, Gaikwad SK, Mahapatra AK (2014) Pediatric intracranial aneurysms—our experience and review of literature. Childs Nerv Syst 30(5):873–883

    Article  Google Scholar 

  21. Gonçalves VM, Cristino N, Cunha E, Sá M (2014) Spontaneous thrombosis in giant aneurysm of the anterior communicating artery complex in pediatric age: five-year follow-up. Case Rep Vasc Med 2014:236041. https://doi.org/10.1155/2014/236041

    Article  PubMed  PubMed Central  Google Scholar 

  22. Gross BA, Smith ER, Scott RM, Orbach DB (2015) Intracranial aneurysms in the youngest patients: characteristics and treatment challenges. Pediatr Neurosurg 50(1):18–25

    Article  Google Scholar 

  23. Heiskanen O, Vilkki J (1981) Intracranial arterial aneurysms in children and adolescents. Acta Neurochir 59(1-2):55–63. https://doi.org/10.1007/BF01411191

    Article  CAS  PubMed  Google Scholar 

  24. Hetts SW, Narvid J, Sanai N, Lawton MT, Gupta N, Fullerton HJ, Dowd CF, Higashida RT, Halbach VV (2009) Intracranial aneurysms in childhood: 27-year single-institution experience. AJNR Am J Neuroradiol 30(7):1315–1324. https://doi.org/10.3174/ajnr.A1587

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  25. Hoffman CE, Santillan A, Rotman L, Gobin YP, Souweidane MM (2014) Complications of cerebral angiography in children younger than 3 years of age. J Neurosurg Pediatr 13(4):414–419. https://doi.org/10.3171/2013.12.PEDS13172

    Article  PubMed  Google Scholar 

  26. Huang J, McGirt MJ, Gailloud P, Tamargo RJ (2005) Intracranial aneurysms in the pediatric population: case series and literature review. Surg Neurol 63(5):424–433

    Article  Google Scholar 

  27. Koroknay-Pál P, Laakso A, Lehto H, Seppä K, Kivisaari R, Hernesniemi J, Niemelä M (2012) Long-term excess mortality in pediatric patients with cerebral aneurysms. Stroke. 43(8):2091–2096

    Article  Google Scholar 

  28. Koroknay-Pál P, Lehto H, Niemelä M, Kivisaari R, Hernesniemi J (2012) Long-term outcome of 114 children with cerebral aneurysms. J Neurosurg Pediatr 9(6):636–645

    Article  Google Scholar 

  29. Krishna H, Wani AA, Behari S, Banerji D, Chhabra DK, Jain VK (2005) Intracranial aneurysms in patients 18 years of age or under, are they different from aneurysms in adult population? Acta Neurochir 147(5):469–476

    Article  CAS  Google Scholar 

  30. Kuchelmeister K, Schulz R, Bergmann M, Schwuchow R, Vollmer E (1993) A probably familial saccular aneurysm of the anterior communicating artery in a neonate. Childs Nerv Syst 9(5):302–305. https://doi.org/10.1007/BF00306281

    Article  CAS  PubMed  Google Scholar 

  31. Kurtelius A, Väntti N, Rezai Jahromi B, Tähtinen O, Manninen H, Koskenvuo J, Tulamo R, Kotikoski S, Nurmonen H, Kämäräinen OP, Huttunen T, Huttunen J, von Und Zu Fraunberg M, Koivisto T, Jääskeläinen JE, Lindgren AE (2019) Association of intracranial aneurysms with aortic aneurysms in 125 patients with fusiform and 4253 patients with saccular intracranial aneurysms and their family members and population controls. J Am Heart Assoc 8(18):e013277

    Article  Google Scholar 

  32. Lai LT, Gragnaniello C, Morgan MK (2013) Outcomes for a case series of unruptured anterior communicating artery aneurysm surgery. J Clin Neurosci 20:1688–1692

    Article  Google Scholar 

  33. Lasjaunias P, Wuppalapati S, Alvarez H, Rodesch G, Ozanne A (2005) Intracranial aneurysms in children aged under 15 years: review of 59 consecutive children with 75 aneurysms. Childs Nerv Syst 21(6):437–450. https://doi.org/10.1007/s00381-004-1125-x

    Article  PubMed  Google Scholar 

  34. Latorre JG, Lodi Y, El-Zammar Z, Devasenapathy A (2011) Is asymptomatic vasospasm associated with poor outcome in subarachnoid hemorrhage? Neurohospitalist 1(4):165–171. https://doi.org/10.1177/1941875211413134

    Article  PubMed  PubMed Central  Google Scholar 

  35. Lu VM, Kaszuba MC, Murphy ME, Lanzino G, Daniels DJ (2019) Near-fatal acute giant intracranial aneurysm rerupture in 7-month-old infant. World Neurosurg 128:191–195

    Article  Google Scholar 

  36. Lyon KA, Arrey EN, Haider AS, Jeevan DS, Benardete EA (2017) Endovascular treatment of a large ruptured middle cerebral artery bifurcation aneurysm in a 5-week-old infant: case report. J Neurosurg Pediatr 20(4):357–363

    Article  Google Scholar 

  37. Mehrotra A, Nair AP, Das KK, Srivastava A, Sahu RN, Kumar R (2012) Clinical and radiological profiles and outcomes in pediatric patients with intracranial aneurysms. J Neurosurg Pediatr 10(4):340–346

    Article  Google Scholar 

  38. Mitchell B, Dash D, Humphries W, Mawad M (2012) De novo intracranial aneurysm formation following endovascular treatment of giant aneurysm in an infant: case report. Childs Nerv Syst 28(4):645–648

    Article  Google Scholar 

  39. Moftakhar P, Cooke DL, Fullerton HJ, Ko NU, Amans MR, Narvid JA, Dowd CF, Higashida RT, Halbach VV, Hetts SW (2015) Extent of collateralization predicting symptomatic cerebral vasospasm among pediatric patients: correlations among angiography, transcranial Doppler ultrasonography, and clinical findings. J Neurosurg Pediatr 15(3):282–290. https://doi.org/10.3171/2014.9.PEDS14313

    Article  PubMed  Google Scholar 

  40. Nishio A, Sakaguchi M, Murata K, Egashira M, Yamada T, Izuo M, Nakanishi N (1991) Anterior communicating artery aneurysm in early childhood. Report of a case Surg Neurol 35(3):224–229. https://doi.org/10.1016/0090-3019(91)90075-kss

    Article  CAS  PubMed  Google Scholar 

  41. O’Neill AH, Chandra RV, Lai LT (2017) Safety and effectiveness of microsurgical clipping, endovascular coiling, and stent assisted coiling for unruptured anterior communicating artery aneurysms: a systematic analysis of observational studies. J Neurointerv Surg 9(8):761–765

    Article  Google Scholar 

  42. Pannu H, Tran-Fadulu V, Papke CL, Scherer S, Liu Y, Presley C, Guo D, Estrera AL, Safi HJ, Brasier AR, Vick GW, Marian AJ, Raman CS, Buja LM, Milewicz DM (2007) MYH11 mutations result in a distinct vascular pathology driven by insulin-like growth factor 1 and angiotensin II [published correction appears in Hum Mol Genet. 2008 Jan 1;17(1):158]. Hum Mol Genet 16(20):2453–2462

    Article  CAS  Google Scholar 

  43. Pasqualin A, Mazza C, Cavazzani P, Scienza R, DaPian R (1986) Intracranial aneurysms and subarachnoid hemorrhage in children and adolescents. Childs Nerv Syst 2(4):185–190. https://doi.org/10.1007/BF00706808

    Article  CAS  PubMed  Google Scholar 

  44. Phan TG, Huston J 3rd, Campeau NG, Wijdicks EF, Atkinson JL, Fulgham JR (2003) Value of diffusion-weighted imaging in patients with a nonlocalizing examination and vasospasm from subarachnoid hemorrhage. Cerebrovasc Dis 15(3):177–181. https://doi.org/10.1159/000068823

    Article  PubMed  Google Scholar 

  45. Proust F, Debono B, Hannequin D, Gerardin E, Clavier E, Langlois O, Fréger P (2003) Treatment of anterior communicating artery aneurysms: complementary aspects of microsurgical and endovascular procedures. J Neurosurg 99:3–14

    Article  Google Scholar 

  46. Pucci L, Pointet A, Good JM et al (2020) A new variant in the MYH11 gene in a familial case of thoracic aortic aneurysm. Ann Thorac Surg 109(4):e279–e281

    Article  Google Scholar 

  47. Ravindra VM, Karsy M, Schmidt RH, Taussky P, Park MS, Bollo RJ (2016) Rapid de novo aneurysm formation after clipping of a ruptured middle cerebral artery aneurysm in an infant with an MYH11 mutation. J Neurosurg Pediatr 18(4):463–470

    Article  Google Scholar 

  48. Shimamura N, Naraoka M, Matsuda N, Kakuta K, Ohkuma H (2014) Prophylactic intra-arterial injection of vasodilator for asymptomatic vasospasm converts the patient to symptomatic vasospasm due to severe microcirculatory imbalance. Biomed Res Int 2014:382484. https://doi.org/10.1155/2014/382484

    Article  PubMed  PubMed Central  Google Scholar 

  49. Song JK, Niimi Y, Brisman JL, Fernandez PM, Berenstein A (2005) Multiple cerebral aneurysms in a neonate: occlusion and rupture. J Neurosurg 102(1 Suppl):81–85. https://doi.org/10.3171/ped.2005.102.1.0081

    Article  PubMed  Google Scholar 

  50. Sudhir BJ, Honavalli Murali S, Gohil J, Poyuran R, Sethuraman M, Harihara VE (2021) Cyanosis and clipping: noninfectious aneurysm in a patient with 22q11.2 deletion syndrome. Pediatr Neurosurg 56(1):45–49. https://doi.org/10.1159/000512581

    Article  PubMed  Google Scholar 

  51. Sungarian A, Rogg J, Duncan JA 3rd. (2003) Pediatric intracranial aneurysm: a diagnostic dilemma solved with contrast-enhanced MR imaging. AJNR Am J Neuroradiol 24(3):370–372 Erratum in: AJNR Am J Neuroradiol. 2003 May;24(5):1032

    PubMed  PubMed Central  Google Scholar 

  52. Suzuki H, Muramatsu M, Kojima T, Taki W (2003) Intracranial heme metabolism and cerebral vasospasm after aneurysmal subarachnoid hemorrhage. Stroke. 34(12):2796–2800. https://doi.org/10.1161/01.STR.0000103743.62248.12

    Article  PubMed  Google Scholar 

  53. Takemoto K, Tateshima S, Golshan A, Gonzalez N, Jahan R, Duckwiler G, Vinuela F (2014) Endovascular treatment of pediatric intracranial aneurysms: a retrospective study of 35 aneurysms. J Neurointerv Surg 6(6):432–438

    Article  Google Scholar 

  54. Wong H, Banfield J, Hughes N, Shankar JJS. Are anterior communicating aneurysms truly anterior communicating aneurysms? An observational study. World Neurosurgery. 2019. pii: S1878-8750(19)30365-1

  55. Yasin JT, Wallace AN, Madaelil TP, Osbun JW, Moran CJ, Cross DT, Limbrick DD, Zipfel GJ, Dacey RG, Kansagra AP (2019) Treatment of pediatric intracranial aneurysms: case series and meta-analysis. J Neurointerv Surg 11(3):257–264. https://doi.org/10.1136/neurintsurg-2018-014001

    Article  PubMed  Google Scholar 

  56. Yatomi K, Oishi H, Yamamoto M, Suga Y, Nonaka S, Yoshida K, Arai H. Radiological changes in infantile dissecting anterior communicating artery aneurysm treated endovascularly. A case report and five-year follow-up. Interv Neuroradiol. 2014 Dec;20(6):796-803. 10.15274/INR-2014-10093. Epub 2014 Dec 5. PMID: 25496693; PMCID: PMC4295255.

  57. Yu L, Wang J, Wang S, Zhang D, Zhao Y, Wang R, Zhao J (2017) DNA Methylation regulates gene expression in intracranial aneurysms. World Neurosurg 105:28–36

    Article  Google Scholar 

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Acknowledgements

The authors would like to thank the family of the patient for their collaboration, support, and encouragement in writing this manuscript.

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Authors and Affiliations

  1. Clinic of Neurosurgery, Cluj County Emergency Clinical Hospital, Cluj-Napoca, Romania

    Cristina Caterina Aldea, Ioan Alexandru Florian, Zorinela Andrașoni & Ioan Stefan Florian

  2. Department of Neurosurgery, Iuliu Hatieganu University of Medicine and Pharmacy, Cluj-Napoca, Romania

    Ioan Alexandru Florian & Ioan Stefan Florian

  3. Department of Physiology, Iuliu Hatieganu University of Medicine and Pharmacy, Cluj-Napoca, Romania

    Teodora Larisa Timiș

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  1. Cristina Caterina Aldea
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Correspondence to Ioan Alexandru Florian.

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Aldea, C.C., Florian, I.A., Timiș, T.L. et al. Ruptured AComA aneurysm and asymptomatic bilateral ACA vasospasm in an infant: surgical case report. Childs Nerv Syst 38, 633–641 (2022). https://doi.org/10.1007/s00381-021-05215-7

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