Abdelmageed Sunny, Bae Gloria H, Mossner James M, Trierweiler Robin, Keen Mary E, Katholi Benjamin, Raskin Jeffrey S
Department of Neurosurgery, Northwestern University Feinberg School of Medicine, Chicago, Illinois.
Division of Pediatric Neurosurgery, Ann & Robert H. Lurie Children's Hospital, Chicago, Illinois.
J Neurosurg Case Lessons. 2025 Jan 27;9(4). doi: 10.3171/CASE24599.
Medically refractory hypertonia (MRH) within the pediatric population causes severe disability and is difficult to treat. Neurosurgery for mixed MRH includes intrathecal baclofen (ITB) and lumbosacral ventral-dorsal rhizotomy (VDR). Surgical efficacy limitations can be mitigated by combining the two into a multimodal strategy. The authors examined outcomes following a multimodal neurosurgical strategy combining intraspinal ITB catheter revision to the cervical level and lumbosacral VDR.
Two patients with severe MRH resistant to ITB delivered through a thoracic catheter tip were identified: 1) a 16-year-old boy with quadriplegic mixed hypertonia and 2) a 17-year-old girl with secondary dystonia. The patient in case 1 experienced improvement in his Barry-Albright Dystonia Scale (BADS) score from 29 to 17 and lower-extremity modified Ashworth Scale score from 4 to 0 at 18 months postoperatively; the patient in case 2 experienced a decrease in her BADS score from 30 to 13 at 6 months postoperatively. Significant improvement in caregiving provisions, including patient positioning and transfers, was reported.
The authors highlight favorable outcomes using multimodal surgery in pediatric patients. Multimodal therapy is surgically feasible and better addresses MRH, particularly in patients in whom ITB monotherapy and polypharmacy have failed. Future studies with larger patient volumes are necessary to optimize indications and make more definitive outcome conclusions. https://thejns.org/doi/10.3171/CASE24599.
儿科难治性高张力(MRH)会导致严重残疾且难以治疗。针对混合型MRH的神经外科手术包括鞘内注射巴氯芬(ITB)和腰骶部腹背根切断术(VDR)。将两者结合成多模式策略可减轻手术疗效的局限性。作者研究了将脊髓内ITB导管改为颈段水平并联合腰骶部VDR的多模式神经外科策略后的结果。
确定了两名对经胸导管尖端给药的ITB耐药的严重MRH患者:1)一名16岁的四肢瘫痪混合型高张力男孩和2)一名17岁的继发性肌张力障碍女孩。病例1中的患者术后18个月时,其巴里-奥尔布赖特肌张力障碍量表(BADS)评分从29分提高到17分,下肢改良Ashworth量表评分从4分降至0分;病例2中的患者术后6个月时,其BADS评分从30分降至13分。据报告,在护理措施方面有显著改善,包括患者体位和转移。
作者强调了在儿科患者中使用多模式手术取得的良好结果。多模式治疗在手术上是可行的,能更好地治疗MRH,特别是在ITB单一疗法和联合药物治疗失败的患者中。需要开展更大样本量患者的未来研究,以优化适应症并得出更明确的结果结论。https://thejns.org/doi/10.3171/CASE24599