Middle meningeal artery embolization for refractory chronic subdural hematoma in a pediatric victim of nonaccidental trauma: illustrative case

Zsombor T Gal Department of Neurosurgery, Boston Children’s Hospital, Harvard Medical School, Boston, Massachusetts

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Alfred P See Department of Neurosurgery, Boston Children’s Hospital, Harvard Medical School, Boston, Massachusetts

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BACKGROUND

Chronic subdural hematoma (cSDH) in children is associated with several etiologies, one of which is nonaccidental trauma (NAT). Performed alone or as an adjunct to surgical intervention, middle meningeal artery (MMA) embolization may be effective in resolving and preventing the recurrence of cSDH in adult patients. However, MMA embolization for pediatric cSDH has not been adequately evaluated, and there exists no consensus on the appropriate selection of pediatric patients for this minimally invasive procedure. Appreciating the variable underlying pathophysiology of pediatric cSDH will enable the development of guidelines for patient selection.

OBSERVATIONS

A 14-month-old female patient presented with depressed consciousness, seizure-like activity, and apneic episodes. She was diagnosed with bilateral subacute on chronic SDH associated with NAT, and imaging revealed diffuse brain atrophy. Despite undergoing burr hole drainage, she developed refractory cSDH and was treated with bilateral MMA embolization. At her 10-month follow-up, magnetic resonance imaging revealed a decrease in the size of the subdural collections, and she was tolerating feeds, at her neurological baseline, and seizure-free.

LESSONS

Infants and young children with symptomatic cSDH secondary to NAT, whose tissue dynamics are reminiscent of cSDH in elderly patients with diffuse brain atrophy, may derive the most benefit from MMA embolization.

ABBREVIATIONS

cSDH = chronic subdural hematoma; CSF = cerebrospinal fluid; MMA = middle meningeal artery; MRI = magnetic resonance imaging; NAT = nonaccidental trauma

BACKGROUND

Chronic subdural hematoma (cSDH) in children is associated with several etiologies, one of which is nonaccidental trauma (NAT). Performed alone or as an adjunct to surgical intervention, middle meningeal artery (MMA) embolization may be effective in resolving and preventing the recurrence of cSDH in adult patients. However, MMA embolization for pediatric cSDH has not been adequately evaluated, and there exists no consensus on the appropriate selection of pediatric patients for this minimally invasive procedure. Appreciating the variable underlying pathophysiology of pediatric cSDH will enable the development of guidelines for patient selection.

OBSERVATIONS

A 14-month-old female patient presented with depressed consciousness, seizure-like activity, and apneic episodes. She was diagnosed with bilateral subacute on chronic SDH associated with NAT, and imaging revealed diffuse brain atrophy. Despite undergoing burr hole drainage, she developed refractory cSDH and was treated with bilateral MMA embolization. At her 10-month follow-up, magnetic resonance imaging revealed a decrease in the size of the subdural collections, and she was tolerating feeds, at her neurological baseline, and seizure-free.

LESSONS

Infants and young children with symptomatic cSDH secondary to NAT, whose tissue dynamics are reminiscent of cSDH in elderly patients with diffuse brain atrophy, may derive the most benefit from MMA embolization.

ABBREVIATIONS

cSDH = chronic subdural hematoma; CSF = cerebrospinal fluid; MMA = middle meningeal artery; MRI = magnetic resonance imaging; NAT = nonaccidental trauma

Chronic subdural hematoma (cSDH) most commonly occurs in elderly adults and is often caused by single-event traumas in the setting of diffuse brain atrophy, which places bridging veins under tension and results in a potential space into which resolved SDH membranes can bleed.1,2 Patients with cSDH can be treated with conservative management or surgical intervention, depending on whether the cSDH is symptomatic.3 Neuroendovascular management of cSDH can be undertaken via middle meningeal artery (MMA) embolization, which can be performed in isolation or as an adjunct to surgical intervention.4 MMA embolization is believed to occlude the vessels that supply the inner and outer cSDH membranes, devascularize the source of accumulating blood products, and promote resolution of the cSDH via resorption.5

Case series of adult patients with cSDH have shown that MMA embolization, compared with conventional surgical treatment, is associated with lower rates of cSDH recurrence, lower complication rates, and lower rates of surgical rescue.6–10 Thus, MMA embolization has become an established practice in the management of adult cSDH at many centers, and several clinical trials comparing the safety and efficacy of MMA embolization versus traditional surgical management are underway.11

cSDH in the pediatric population, unlike that in elderly adults, is rare and typically not associated with single-event traumas.12 Instead, pediatric cSDH cases are often associated with nonaccidental trauma (NAT), ruptured arachnoid cysts, coagulopathy, or cerebrospinal fluid (CSF) overdrainage via ventriculoperitoneal shunting.12,13 Management is highly variable, given the lack of consensus regarding optimal treatment, and includes an array of invasive surgical options.14 However, MMA embolization for cSDH has not been adequately studied in children. Given the variety of etiologies and differences in the underlying pathophysiology of pediatric cSDH, it is important to determine which pediatric patients with cSDH may derive the most benefit from MMA embolization. Hence, we describe a case of refractory pediatric cSDH associated with NAT treated with MMA embolization and propose several hypotheses regarding appropriate patient selection for this minimally invasive procedure.

Illustrative Case

Presentation and Initial Treatment

A 25-month-old female with a history of NAT and global developmental delay initially presented at the age of 14 months with depressed consciousness, seizure-like activity, and apneic episodes. Computed tomography revealed no evidence of skull fracture but did show bilateral subacute on chronic SDH (Fig. 1A). The patient was emergently taken for bilateral parietal burr hole drainage.

FIG. 1
FIG. 1

A: Computed tomography at initial presentation displaying bilateral subdural collections. The dashed lines indicate an older, isodense component on the left side, and the arrow points to an acute, hyperdense component along the posterior right convexity. B: MRI on postoperative day (POD) 1 revealed a slight decrease in the size of the subdural collections with associated pneumocephalus. C: MRI on POD 18 showed reaccumulation with bilateral increases in the size of the subdural collections. D: An angiogram of the left MMA revealed a typical branching pattern, including a sphenoidal branch; angiography of the right MMA (not shown) revealed a similar pattern. E: Radiographic image with arrows indicating successful embolization of the left and right parietal branches of the MMA. F: MRI 10 days after MMA embolization showed a slight decrease in the size of the subdural collections bilaterally. G: MRI at 4-month follow-up showed further decreases in the size of the left and right collections. H: MRI at 10-month follow-up also showed bilateral decreases in the size of the subdural collections. Note the moderate ventriculomegaly and pronounced loss of cerebral volume in these images.

Investigations

The following day, magnetic resonance imaging (MRI) revealed a slight decrease in the size of the subdural collections and showed internal membranes (Fig. 1B). MRI also showed extensive cerebral and cerebellar volume loss.

Five-day interval MRI showed stable collections, but the patient became increasingly irritable and continued to experience apneic episodes, desaturations, and seizure activity over the subsequent 2 weeks. Repeat MRI conducted 18 days after surgery revealed reaccumulation of subdural fluid collections and increased layering of blood products (Fig. 1C).

Endovascular Treatment

Repeat burr hole drainage was performed for the clinical and radiographic recurrence. Immediately following this procedure, the patient was transported to the angiography suite for bilateral MMA embolization to devascularize the putative source of the cSDH.

A 4-French Vert catheter (Cordis) was positioned in the right external carotid artery. A 1.3-French Headway DUO microcatheter (MicroVention) was then advanced over a 0.014-inch Asahi Chikai microguidewire (Asahi Intecc) into the parietal branch of the right MMA. Embolization was achieved with 0.6 mL of Onyx 18 liquid embolic agent (Medtronic). This was repeated in the left parietal MMA (Fig. 1D and E). A liquid embolic agent was used instead of microparticles; this was partially because of institutional preference but also because the catheters for liquid embolisates are sometimes smaller and more distally navigable than the larger catheters needed to avoid clogging when using particles. Thus, it is often easier to access the infant vasculature with catheters for liquid embolization.

Outcome and Follow-Up

Brain MRI revealed a slight decrease in the subdural collections at 10 days (Fig. 1F). She was discharged to a rehabilitation facility 19 days after endovascular embolization. Work-up ruled out a mitochondrial disorder, suggesting her diffuse brain atrophy was likely secondary to NAT. At her 4-month follow-up, MRI showed a decrease in the size of the subdural collections (Fig. 1G), and the patient was noted to be tolerating feeds, at her neurological baseline, and seizure-free. At her 10-month follow-up, MRI revealed a further decrease in the size of the subdural collections (Fig. 1H), and the patient was tolerating feeds, gaining weight, and seizure-free, although she was still developmentally delayed.

Patient Informed Consent

The necessary patient informed consent was obtained in this study.

Discussion

Observations

Pediatric cSDH, compared with cSDH in elderly adults, is relatively rare and associated with many etiologies, each with a distinct underlying pathophysiology.12,13 Elderly patients typically have diffuse brain atrophy, which generates a brain-calvarial volume mismatch that is also observed in some children. Correspondingly, we propose that the pediatric patients with cSDH who may derive the most benefit from MMA embolization are infants and young children who are victims of NAT—their brains undergo atrophy from repeated injury, but their head volumes decrease only minimally, manifesting as increased potential space. CSF shunt overdrainage may also produce a comparable scenario wherein the ventricular CSF space is accounted for but iatrogenically removed from the intracranial space. The rupture of an arachnoid cyst likewise involves an acute change in the initial long-term balance of calvarial volume to the sum of the brain and the arachnoid cyst compartment volume. This volume mismatch is not observed when children have anticoagulation-associated hemorrhage, unlike the situation in which elderly patients with a history of anticoagulant use have diffuse brain atrophy contributing to cSDH. However, some children in the cardiac intensive care unit and children receiving long chemotherapy regimens may also have delayed or decreased growth of cerebral volume associated with chronic illness.15,16

Although brain-calvarial volume mismatch may present a useful heuristic in patient selection for MMA embolization, there exist highly effective and low-risk alternative treatment options for CSF shunt overdrainage and for rupture of arachnoid cysts. Shunt overdrainage can be resolved by removing or revising a malfunctioning shunt. cSDH from ruptured arachnoid cysts often resolves spontaneously with conservative management, and there are many standard or minimally invasive surgical strategies as well.17,18 In these cases, we would argue that MMA embolization is a riskier treatment option for pediatric cSDH. In contrast, recurrence is a known risk when treating cSDH after NAT, and, in that setting, there is considerable variability in success rates of drainage with or without temporary drains or subdural shunts.14 Accordingly, the comparative effectiveness against MMA embolization for NAT-associated cSDH may merit formal prospective studies.

Although the authors of a recent systematic review proposed that challenging vascular access may preclude MMA embolization for infant victims of NAT,13 we note that neuroendovascular access of young children (3 months to 1 year old) is routinely performed, and young age (<1 year) is not a risk factor for complications of neuroendovascular procedures at experienced centers.19 Thus, we emphasize that infants and young children who present with symptomatic cSDH secondary to NAT, such as our patient, may actually derive the most benefit from MMA embolization, given that their clinical picture is anatomically reminiscent of cSDH in elderly adults with diffuse brain atrophy, patients for whom MMA embolization is often highly effective and prevents cSDH recurrence, and because neuroendovascular access is feasible in this age group.

Lessons

MMA embolization is an emerging treatment modality for cSDH in elderly adults, one that is minimally invasive and effective in promoting the resolution of cSDH and preventing its recurrence. In elderly adults, cSDH is most often a result of single-event traumas in the setting of diffuse brain atrophy; the presence of brain atrophy generates a brain-calvarial volume mismatch that promotes bleeding into the subdural space. MMA embolization addresses this pathophysiology by occluding the vessels that supply the putative source of bleeding in cSDH—resolved SDH membranes.

A similar pathophysiology can be observed in patients with pediatric cSDH secondary to NAT, because these patients also display diffuse brain atrophy (from abusive head trauma) with minimal changes in overall head volume. Increased potential space promotes bleeding from cSDH membranes into the subdural space, which likely contributes to the high rate of cSDH recurrence in these patients. Given the pathophysiological and anatomical similarities between cSDH in elderly adults and that in children who are victims of NAT, MMA embolization may be the most appropriate for children with symptomatic cSDH secondary to NAT. Likewise, children with chronic illnesses who display decreased or delayed brain growth and present with cSDH may also derive substantial benefit from MMA embolization.

For patients with pediatric cSDH not associated with a brain-calvarial volume mismatch, including those with anticoagulation-associated hemorrhage, MMA embolization may not be indicated, given the pathophysiological incompatibility. Despite the presence of a volume mismatch, patients with cSDH due to shunt overdrainage and ruptured arachnoid cysts may benefit the most by addressing the underlying cause; MMA embolization would present an unnecessary risk to these patients.

Given these pathophysiological considerations and the paucity of literature on MMA embolization for pediatric cSDH, formal prospective studies comparing the safety and efficacy of conventional surgical approaches versus MMA embolization (standalone and/or adjunctive) for NAT-associated pediatric cSDH are warranted.

Author Contributions

Conception and design: both authors. Acquisition of data: both authors. Analysis and interpretation of data: both authors. Drafting the article: both authors. Critically revising the article: both authors. Reviewed submitted version of manuscript: both authors. Approved the final version of the manuscript on behalf of both authors: See. Study supervision: See.

References

  • 1

    Adhiyaman V, Asghar M, Ganeshram KN, Bhowmick BK. Chronic subdural haematoma in the elderly. Postgrad Med J. 2002;78(916):7175.

  • 2

    Shen J, Gao Y, Li Q, et al. Risk factors predicting recurrence of bilateral chronic subdural hematomas after initial bilateral evacuation. World Neurosurg. 2019;130:e133e139.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 3

    Feghali J, Yang W, Huang J. Updates in chronic subdural hematoma: epidemiology, etiology, pathogenesis, treatment, and outcome. World Neurosurg. 2020;141:339345.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 4

    Fiorella D, Arthur AS. Middle meningeal artery embolization for the management of chronic subdural hematoma. J Neurointerv Surg. 2019;11(9):912915.

  • 5

    Moshayedi P, Liebeskind DS. Middle meningeal artery embolization in chronic subdural hematoma: implications of pathophysiology in trial design. Front Neurol. 2020;11:923.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 6

    Di Cristofori A, Remida P, Patassini M, et al. Middle meningeal artery embolization for chronic subdural hematomas. A systematic review of the literature focused on indications, technical aspects, and future possible perspectives. Surg Neurol Int. 2022;13:94.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 7

    Srivatsan A, Mohanty A, Nascimento FA, et al. Middle meningeal artery embolization for chronic subdural hematoma: meta-analysis and systematic review. World Neurosurg. 2019;122:613619.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 8

    Joyce E, Bounajem MT, Scoville J, et al. Middle meningeal artery embolization treatment of nonacute subdural hematomas in the elderly: a multiinstitutional experience of 151 cases. Neurosurg Focus. 2020;49(4):E5.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 9

    Ban SP, Hwang G, Byoun HS, et al. Middle meningeal artery embolization for chronic subdural hematoma. Radiology. 2018;286(3):992999.

  • 10

    Ironside N, Nguyen C, Do Q, et al. Middle meningeal artery embolization for chronic subdural hematoma: a systematic review and meta-analysis. J Neurointerv Surg. 2021;13(10):951957.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 11

    Catapano JS, Nguyen CL, Wakim AA, Albuquerque FC, Ducruet AF. Middle meningeal artery embolization for chronic subdural hematoma. Front Neurol. 2020;11:557233.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 12

    Deora H, Mishra A, Gupta R, et al. Paediatric chronic subdural haematoma: what are the predisposing factors and outcomes in management of these cases? Childs Nerv Syst. 2022;38(1):123132.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 13

    Paro MR, Ollenschleger MD, Fayad MF, et al. Middle meningeal artery embolization for primary treatment of a chronic subdural hematoma in a pediatric patient: a systematic review of the literature and case report. Oper Neurosurg (Hagerstown). 2023;24(1):310.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 14

    Melo JRT, Di Rocco F, Bourgeois M, et al. Surgical options for treatment of traumatic subdural hematomas in children younger than 2 years of age. J Neurosurg Pediatr. 2014;13(4):456461.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 15

    Wernovsky G, Licht DJ. Neurodevelopmental outcomes in children with congenital heart disease—what can we impact? Pediatr Crit Care Med. 2016;17(8)(suppl 1):S232S242.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 16

    Prassopoulos P, Cavouras D, Evlogias N, Golfinopoulos S. Brain atrophy in children undergoing systemic chemotherapy for extracranial solid tumors. Med Pediatr Oncol. 1997;28(3):228233.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 17

    Maher CO, Garton HJL, Al-Holou WN, Trobe JD, Muraszko KM, Jackson EM. Management of subdural hygromas associated with arachnoid cysts. J Neurosurg Pediatr. 2013;12(5):434443.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 18

    Li B, Ng C, Feldstein E, Muh C, Mohan A, Tobias M. Non-operative management of a pediatric patient with bilateral subdural hematomas in the setting of ruptured arachnoid cyst. Cureus. 2021;13(12):e20099.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 19

    Lin N, Smith ER, Scott RM, Orbach DB. Safety of neuroangiography and embolization in children: complication analysis of 697 consecutive procedures in 394 patients. J Neurosurg Pediatr. 2015;16(4):432438.

    • PubMed
    • Search Google Scholar
    • Export Citation
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  • FIG. 1

    A: Computed tomography at initial presentation displaying bilateral subdural collections. The dashed lines indicate an older, isodense component on the left side, and the arrow points to an acute, hyperdense component along the posterior right convexity. B: MRI on postoperative day (POD) 1 revealed a slight decrease in the size of the subdural collections with associated pneumocephalus. C: MRI on POD 18 showed reaccumulation with bilateral increases in the size of the subdural collections. D: An angiogram of the left MMA revealed a typical branching pattern, including a sphenoidal branch; angiography of the right MMA (not shown) revealed a similar pattern. E: Radiographic image with arrows indicating successful embolization of the left and right parietal branches of the MMA. F: MRI 10 days after MMA embolization showed a slight decrease in the size of the subdural collections bilaterally. G: MRI at 4-month follow-up showed further decreases in the size of the left and right collections. H: MRI at 10-month follow-up also showed bilateral decreases in the size of the subdural collections. Note the moderate ventriculomegaly and pronounced loss of cerebral volume in these images.

  • 1

    Adhiyaman V, Asghar M, Ganeshram KN, Bhowmick BK. Chronic subdural haematoma in the elderly. Postgrad Med J. 2002;78(916):7175.

  • 2

    Shen J, Gao Y, Li Q, et al. Risk factors predicting recurrence of bilateral chronic subdural hematomas after initial bilateral evacuation. World Neurosurg. 2019;130:e133e139.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 3

    Feghali J, Yang W, Huang J. Updates in chronic subdural hematoma: epidemiology, etiology, pathogenesis, treatment, and outcome. World Neurosurg. 2020;141:339345.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 4

    Fiorella D, Arthur AS. Middle meningeal artery embolization for the management of chronic subdural hematoma. J Neurointerv Surg. 2019;11(9):912915.

  • 5

    Moshayedi P, Liebeskind DS. Middle meningeal artery embolization in chronic subdural hematoma: implications of pathophysiology in trial design. Front Neurol. 2020;11:923.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 6

    Di Cristofori A, Remida P, Patassini M, et al. Middle meningeal artery embolization for chronic subdural hematomas. A systematic review of the literature focused on indications, technical aspects, and future possible perspectives. Surg Neurol Int. 2022;13:94.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 7

    Srivatsan A, Mohanty A, Nascimento FA, et al. Middle meningeal artery embolization for chronic subdural hematoma: meta-analysis and systematic review. World Neurosurg. 2019;122:613619.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 8

    Joyce E, Bounajem MT, Scoville J, et al. Middle meningeal artery embolization treatment of nonacute subdural hematomas in the elderly: a multiinstitutional experience of 151 cases. Neurosurg Focus. 2020;49(4):E5.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 9

    Ban SP, Hwang G, Byoun HS, et al. Middle meningeal artery embolization for chronic subdural hematoma. Radiology. 2018;286(3):992999.

  • 10

    Ironside N, Nguyen C, Do Q, et al. Middle meningeal artery embolization for chronic subdural hematoma: a systematic review and meta-analysis. J Neurointerv Surg. 2021;13(10):951957.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 11

    Catapano JS, Nguyen CL, Wakim AA, Albuquerque FC, Ducruet AF. Middle meningeal artery embolization for chronic subdural hematoma. Front Neurol. 2020;11:557233.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 12

    Deora H, Mishra A, Gupta R, et al. Paediatric chronic subdural haematoma: what are the predisposing factors and outcomes in management of these cases? Childs Nerv Syst. 2022;38(1):123132.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 13

    Paro MR, Ollenschleger MD, Fayad MF, et al. Middle meningeal artery embolization for primary treatment of a chronic subdural hematoma in a pediatric patient: a systematic review of the literature and case report. Oper Neurosurg (Hagerstown). 2023;24(1):310.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 14

    Melo JRT, Di Rocco F, Bourgeois M, et al. Surgical options for treatment of traumatic subdural hematomas in children younger than 2 years of age. J Neurosurg Pediatr. 2014;13(4):456461.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 15

    Wernovsky G, Licht DJ. Neurodevelopmental outcomes in children with congenital heart disease—what can we impact? Pediatr Crit Care Med. 2016;17(8)(suppl 1):S232S242.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 16

    Prassopoulos P, Cavouras D, Evlogias N, Golfinopoulos S. Brain atrophy in children undergoing systemic chemotherapy for extracranial solid tumors. Med Pediatr Oncol. 1997;28(3):228233.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 17

    Maher CO, Garton HJL, Al-Holou WN, Trobe JD, Muraszko KM, Jackson EM. Management of subdural hygromas associated with arachnoid cysts. J Neurosurg Pediatr. 2013;12(5):434443.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 18

    Li B, Ng C, Feldstein E, Muh C, Mohan A, Tobias M. Non-operative management of a pediatric patient with bilateral subdural hematomas in the setting of ruptured arachnoid cyst. Cureus. 2021;13(12):e20099.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 19

    Lin N, Smith ER, Scott RM, Orbach DB. Safety of neuroangiography and embolization in children: complication analysis of 697 consecutive procedures in 394 patients. J Neurosurg Pediatr. 2015;16(4):432438.

    • PubMed
    • Search Google Scholar
    • Export Citation

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