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儿童慢性复发性多灶性骨髓炎(CRMO)的脊柱受累及帕米膦酸盐的作用。

Spinal involvement in chronic recurrent multifocal osteomyelitis (CRMO) in childhood and effect of pamidronate.

机构信息

Pediatric Rheumatology, Olgahospital, Bismarckstr 8, 70176 Stuttgart, Germany.

出版信息

Eur J Pediatr. 2010 Sep;169(9):1105-11. doi: 10.1007/s00431-010-1188-5. Epub 2010 Mar 27.

Abstract

There are only a few studies that address the frequency and type of spinal involvement in patients with chronic recurrent multifocal osteomyelitis (CRMO) as well as the outcome of these patients treated with pamidronate (PAM). We performed a retrospective study on patients with CRMO and analyzed clinical and pain assessments as well as regional and whole body MRI findings and compared with posttreatment findings. Of 102 children and adolescents with CRMO, 27 (26%) had involvement of the spine. Vertebral deformities were seen in 14 of these 27 patients, scoliosis or kyphosis in 6. After routine whole body MRI, 19 complained of back pain, whereas eight were asymptomatic with spinal lesions detected incidentally. A total of 72 spinal lesions were detected, thoracic vertebrae being the most commonly affected. Seven patients were treated with PAM; all of whom had vertebral deformities and ongoing back pain. Pain resolution was achieved within 3 months of PAM treatment in every case. One patient subsequently developed a pain amplification syndrome. Repeat MRI performed at a mean interval of 13 months revealed partial or complete resolution of vertebral hyperintensities in every patient. Improvement of vertebral height was seen in a total of three vertebrae in two patients. Severe side effects were not observed. In conclusion, we demonstrated that spinal involvement and associated vertebral deformities with or without kyphoscoliosis are not rare in CRMO, and PAM appears to be an effective and safe treatment for this condition. Although controlled studies are urgently needed, the use of PAM for refractory CRMO with extended spinal involvement (vertebral deformities, kyphosis, and scoliosis) should be considered, especially after failing of conventional therapy.

摘要

仅有少数研究探讨了慢性复发性多灶性骨髓炎(CRMO)患者脊柱受累的频率和类型,以及用帕米膦酸(PAM)治疗这些患者的结果。我们对 CRMO 患者进行了回顾性研究,分析了临床和疼痛评估以及局部和全身 MRI 结果,并与治疗后结果进行了比较。在 102 例 CRMO 患儿和青少年中,27 例(26%)有脊柱受累。这 27 例患者中有 14 例存在椎体畸形,其中 6 例存在脊柱侧凸或后凸。在常规全身 MRI 后,19 例有背痛,而 8 例无症状但意外发现脊柱病变。共发现 72 个脊柱病变,胸椎最常受累。7 例患者接受 PAM 治疗;所有患者均有椎体畸形和持续背痛。在接受 PAM 治疗后 3 个月内,所有患者的疼痛均得到缓解。1 例患者随后出现疼痛放大综合征。在平均 13 个月的随访中,重复 MRI 显示每位患者的椎体高信号均部分或完全缓解。在 2 例患者中,共 3 个椎体的椎体高度增加。未观察到严重的副作用。总之,我们证明 CRMO 中并不罕见脊柱受累和相关的椎体畸形,伴或不伴后凸侧凸,PAM 似乎是这种疾病的有效和安全的治疗方法。尽管迫切需要进行对照研究,但对于广泛脊柱受累(椎体畸形、后凸和脊柱侧凸)的难治性 CRMO,尤其是在常规治疗失败后,应考虑使用 PAM。

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