The objectives of this retrospective study were to evaluate the effect of direct vertebral derotation on the sagittal alignment of the spine after selective posterior thoracic fusion for Lenke Type I adolescent idiopathic scoliosis (AIS). Preservation of the sagittal alignment has become critical in the management of spinal deformity. Better coronal and rotational corrections in posterior selective thoracic fusion for AIS have been reported with direct vertebral derotation as compared with the simple rod rotation technique. A greater lordogenic effect has been anticipated with direct vertebral derotation; however, data comparing those two techniques in terms of correction in the sagittal plane are still lacking. Standing full-spine PA and standard lateral serial X-rays of a total of 30 consecutive patients with adolescent idiopathic scoliosis treated between 2002 and 2008 at a single institution were evaluated. All the patients had Lenke Type I curves and underwent selective posterior thoracic fusion with pedicle screw instrumentation. Patients who were treated with additional osteotomies and concave or convex thoracoplasty or concomitant anterior releases were excluded. Minimum follow-up period was 24 months. Preoperative and postoperative coronal and sagittal spinal alignments in both the groups were compared. In 13 patients, the correction was achieved by means of a simple rod rotation (SRR). In 17 patients, the technique of direct vertebral derotation (DVD) was used. Scoliosis correction averaged 67 and 69%, respectively, and was similar in both groups (p > 0.05). Thoracic kyphosis and lumbar lordosis remained unchanged in the SRR group (p > 0.1). In the direct vertebral derotation group, a significant decrease of both thoracic kyphosis and lumbar lordosis of 8.1° and 11.8°, respectively, was observed (p < 0.0001). Global sagittal balance remained within normal limits in all the patients at the latest follow-up. Decrease in thoracic kyphosis and lumbar lordosis should be taken into account when using direct vertebral derotation for selective posterior thoracic fusion in AIS. In order to preserve sagittal alignment in these patients, ultra hard rods or maneuvers that pull posteriorly the concave side of the spine, thus avoiding the application of additional flattening forces should be considered.
The modified Waldenström classification system for staging of Legg-Calvé-Perthes disease demonstrated substantial to almost perfect agreement between and within observers across multiple rounds of study. In doing so, the results of this study provide a foundation for future validation studies, in which the classification stage will be associated with clinical outcomes.
Skeletal pathologies are frequently observed in lysosomal storage disorders, yet the relevance of specific lysosomal enzymes in bone remodeling cell types is poorly defined. Two lysosomal enzymes, ie, cathepsin K (Ctsk) and Acp5 (also known as tartrate-resistant acid phosphatase), have long been known as molecular marker proteins of differentiated osteoclasts. However, whereas the cysteine protease Ctsk is directly involved in the degradation of bone matrix proteins, the molecular function of Acp5 in osteoclasts is still unknown. Here we show that Acp5, in concert with Acp2 (lysosomal acid phosphatase), is required for dephosphorylation of the lysosomal mannose 6-phosphate targeting signal to promote the activity of specific lysosomal enzymes. Using an unbiased approach we identified the glycosaminoglycan-degrading enzyme arylsulfatase B (Arsb), mutated in mucopolysaccharidosis type VI (MPS-VI), as an osteoclast marker, whose activity depends on dephosphorylation by Acp2 and Acp5. Similar to Acp2/Acp5 mice, Arsb-deficient mice display lysosomal storage accumulation in osteoclasts, impaired osteoclast activity, and high trabecular bone mass. Of note, the most prominent lysosomal storage accumulation was observed in osteocytes from Arsb-deficient mice, yet this pathology did not impair production of sclerostin (Sost) and Fgf23. Because the influence of enzyme replacement therapy (ERT) on bone remodeling in MPS-VI is still unknown, we additionally treated Arsb-deficient mice by weekly injection of recombinant human ARSB from 12 to 24 weeks of age. We found that the high bone mass phenotype of Arsb-deficient mice and the underlying bone cell deficits were fully corrected by ERT in the trabecular compartment. Taken together, our results do not only show that the function of Acp5 in osteoclasts is linked to dephosphorylation and activation of lysosomal enzymes, they also provide an important proof-of-principle for the feasibility of ERT to correct bone cell pathologies in lysosomal storage disorders. © 2018 The Authors. Journal of Bone and Mineral Research Published by Wiley Periodicals Inc.
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