Centre for Child and Adolescent Medicine, Clinic I, Division of Child Neurology and Metabolic Medicine, Heidelberg University Hospital, Heidelberg, Germany.
Department of Neuroradiology, Heidelberg University Hospital, Heidelberg, Germany.
J Inherit Metab Dis. 2021 Nov;44(6):1343-1352. doi: 10.1002/jimd.12436. Epub 2021 Sep 30.
Subdural hematoma (SDH) was initially reported in 20% to 30% of patients with glutaric aciduria type 1 (GA1). A recent retrospective study found SDH in 4% of patients, but not in patients identified by newborn screening (NBS). 168 MRIs of 69 patients with GA1 (age at MRI 9 days - 73.8 years, median 3.2 years) were systematically reviewed for presence of SDH, additional MR and clinical findings in order to investigate the frequency of SDH and potential risk factors. SDH was observed in eight high-excreting patients imaged between 5.8 and 24.4 months, namely space-occupying SDH in two patients after minor accidental trauma and SDH as an incidental finding in six patients without trauma. In patients without trauma imaged at 3 to 30 months (n = 36, 25 NBS, 27/9 high/low excreters), incidence of SDH was 16.7% (16% in NBS). SDH was more common after acute (33.3%) than insidious onset of dystonia (14.3%) or in asymptomatic patients (5.9%). It was only seen in patients with wide frontoparietal CSF spaces and frontotemporal hypoplasia. High excreters were over-represented among patients with SDH (6/27 vs 0/9 low excreters), acute onset (10/12), and wide frontoparietal CSF spaces (16/19). Incidental SDH occurs despite NBS and early treatment in approximately one in six patients with GA1 imaged during late infancy and early childhood. Greater risk of high excreters is morphologically associated with more frequent enlargement of external CSF spaces including frontotemporal hypoplasia, and may be furthered aggravated by more pronounced alterations of cerebral blood volume and venous pressure.
硬膜下血肿(SDH)最初在 20%至 30%的 1 型戊二酸血症(GA1)患者中报道。最近一项回顾性研究发现,4%的患者存在 SDH,但新生儿筛查(NBS)识别的患者没有。系统回顾了 69 名 GA1 患者的 168 次 MRI(MRI 时的年龄为 9 天至 73.8 岁,中位数为 3.2 岁),以了解 SDH 的存在、其他 MRI 和临床发现,以调查 SDH 的频率和潜在危险因素。在 5.8 至 24.4 个月之间成像的 8 名高排泄患者中观察到 SDH,即 2 名患者在轻微意外创伤后出现占位性 SDH,6 名无创伤患者出现 SDH 作为偶然发现。在 3 至 30 个月之间成像且无创伤的患者(n=36,25 名 NBS,27/9 名高/低排泄者),SDH 的发生率为 16.7%(NBS 中为 16%)。SDH 在急性(33.3%)起病的患者中比隐匿性起病的肌张力障碍(14.3%)或无症状患者(5.9%)更为常见。仅在额顶和额颞区脑脊液空间宽和额颞部发育不良的患者中可见。SDH 患者中高排泄者更为常见(27 例中有 6 例,9 例低排泄者中无 0 例),急性起病(12 例中有 10 例),额顶和额颞区脑脊液空间宽(19 例中有 16 例)。尽管有 NBS 和早期治疗,大约六分之一在婴儿后期和幼儿期成像的 GA1 患者仍会偶然发生 SDH。高排泄者的风险更高,与更频繁的外部脑脊液空间扩大有关,包括额颞部发育不良,可能由于脑血容量和静脉压的更明显改变而进一步加重。