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Eight years of follow-up after laminectomy of calcium pyrophosphate crystal deposition in the cervical yellow ligament of patient with Coffin-Lowry syndrome: A case report.

Morino T, Ogata T, Horiuchi H, Yamaoka S, Fukuda M, Miura H - Medicine (Baltimore) (2016)

Bottom Line: CLS is a rare X-linked semidominant syndrome associated with growth and psychomotor retardation, general hypotonia, and skeletal abnormalities.At admission, the patient could not move his extremities, and tendon reflexes of the upper and lower extremities were significantly increased.The calcification was confirmed as calcium pyrophosphate by crystal analysis.

View Article: PubMed Central - PubMed

Affiliation: aSpine Center, Ehime University School of Medicine bDepartment of Pediatrics, Ehime University School of Medicine cDepartment of Orthopedic Surgery, Ehime University School of Medicine, Shitsukawa, Tohon City, Ehime, Japan.

ABSTRACT

Background: We report 8 years of follow-up after decompression to treat cervical myelopathy in a patient with Coffin-Lowry syndrome (CLS). CLS is a rare X-linked semidominant syndrome associated with growth and psychomotor retardation, general hypotonia, and skeletal abnormalities. In this patient, the spinal cord was compressed by calcium pyrophosphate crystal deposition in the cervical yellow ligament (YL). To date, only 1 report has described clinical features after surgery for calcified cervical YL in CLS.

Methods: A 15-year-old male with tetraplegia secondary to compression of the cervical spinal cord induced by a hypoplastic posterior arch of C1 and calcification of the YL from C2 to C7 was treated surgically with laminectomy from C1 to C7. The patient's history, clinical examination, imaging findings, and treatment are reported. The patient was incapable of speech because of mental retardation, so he could not describe his symptoms. Gait disturbance worsened over the 2 months before admission to our hospital. At admission, the patient could not move his extremities, and tendon reflexes of the upper and lower extremities were significantly increased. Computed tomography of the cervical spine showed YL calcification from C2 to C7. Magnetic resonance imaging showed consecutive compression of the cervical spinal cord. We diagnosed quadriplegia secondary to cervical cord damage and performed emergency surgery.

Results: During C1-C7 laminectomy, YL calcification in C2-C7 was observed. The calcification was confirmed as calcium pyrophosphate by crystal analysis. Quadriplegia gradually resolved, and almost disappeared by 2 weeks after the operation. Cervical hyperlordosis was observed in radiographs starting from 1 month after the operation, but it has not progressed and is not associated with any symptoms.

Conclusions: The efficacy of decompression continued, and no postoperative complications have occurred during at least 8 years of follow-up.

No MeSH data available.


Related in: MedlinePlus

Lateral whole spine radiograph showing congenital thoracolumbar kyphosis and cervical lordosis (A). Cervical lordosis did not change from 1 y (B) to 8 y after surgery (C).
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Figure 7: Lateral whole spine radiograph showing congenital thoracolumbar kyphosis and cervical lordosis (A). Cervical lordosis did not change from 1 y (B) to 8 y after surgery (C).

Mentions: The authors did not explain why they selected laminectomy instead of laminoplasty. It is well known that cervical laminectomy results in a high rate of kyphosis deformity.[9–11] However, we also selected laminectomy for this patient because there was a possibility of inadequate decompression and recurrent stenosis induced by progression of calcification, and recurrent stenosis is worse than a kyphosis deformity. Fortunately, kyphotic change did not develop during the 8-year follow-up period. However, lordosis occurred (Fig. 7). There was a kyphosis deformity in patient's thoracolumbar spine, and cervical hyperlordosis possibly developed in order to maintain sagittal balance.


Eight years of follow-up after laminectomy of calcium pyrophosphate crystal deposition in the cervical yellow ligament of patient with Coffin-Lowry syndrome: A case report.

Morino T, Ogata T, Horiuchi H, Yamaoka S, Fukuda M, Miura H - Medicine (Baltimore) (2016)

Lateral whole spine radiograph showing congenital thoracolumbar kyphosis and cervical lordosis (A). Cervical lordosis did not change from 1 y (B) to 8 y after surgery (C).
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4979837&req=5

Figure 7: Lateral whole spine radiograph showing congenital thoracolumbar kyphosis and cervical lordosis (A). Cervical lordosis did not change from 1 y (B) to 8 y after surgery (C).
Mentions: The authors did not explain why they selected laminectomy instead of laminoplasty. It is well known that cervical laminectomy results in a high rate of kyphosis deformity.[9–11] However, we also selected laminectomy for this patient because there was a possibility of inadequate decompression and recurrent stenosis induced by progression of calcification, and recurrent stenosis is worse than a kyphosis deformity. Fortunately, kyphotic change did not develop during the 8-year follow-up period. However, lordosis occurred (Fig. 7). There was a kyphosis deformity in patient's thoracolumbar spine, and cervical hyperlordosis possibly developed in order to maintain sagittal balance.

Bottom Line: CLS is a rare X-linked semidominant syndrome associated with growth and psychomotor retardation, general hypotonia, and skeletal abnormalities.At admission, the patient could not move his extremities, and tendon reflexes of the upper and lower extremities were significantly increased.The calcification was confirmed as calcium pyrophosphate by crystal analysis.

View Article: PubMed Central - PubMed

Affiliation: aSpine Center, Ehime University School of Medicine bDepartment of Pediatrics, Ehime University School of Medicine cDepartment of Orthopedic Surgery, Ehime University School of Medicine, Shitsukawa, Tohon City, Ehime, Japan.

ABSTRACT

Background: We report 8 years of follow-up after decompression to treat cervical myelopathy in a patient with Coffin-Lowry syndrome (CLS). CLS is a rare X-linked semidominant syndrome associated with growth and psychomotor retardation, general hypotonia, and skeletal abnormalities. In this patient, the spinal cord was compressed by calcium pyrophosphate crystal deposition in the cervical yellow ligament (YL). To date, only 1 report has described clinical features after surgery for calcified cervical YL in CLS.

Methods: A 15-year-old male with tetraplegia secondary to compression of the cervical spinal cord induced by a hypoplastic posterior arch of C1 and calcification of the YL from C2 to C7 was treated surgically with laminectomy from C1 to C7. The patient's history, clinical examination, imaging findings, and treatment are reported. The patient was incapable of speech because of mental retardation, so he could not describe his symptoms. Gait disturbance worsened over the 2 months before admission to our hospital. At admission, the patient could not move his extremities, and tendon reflexes of the upper and lower extremities were significantly increased. Computed tomography of the cervical spine showed YL calcification from C2 to C7. Magnetic resonance imaging showed consecutive compression of the cervical spinal cord. We diagnosed quadriplegia secondary to cervical cord damage and performed emergency surgery.

Results: During C1-C7 laminectomy, YL calcification in C2-C7 was observed. The calcification was confirmed as calcium pyrophosphate by crystal analysis. Quadriplegia gradually resolved, and almost disappeared by 2 weeks after the operation. Cervical hyperlordosis was observed in radiographs starting from 1 month after the operation, but it has not progressed and is not associated with any symptoms.

Conclusions: The efficacy of decompression continued, and no postoperative complications have occurred during at least 8 years of follow-up.

No MeSH data available.


Related in: MedlinePlus