Extensive ossification of the paraspinal ligaments in a patient with vitamin D-resistant rickets: Case report with literature review

Hirao, Yujiro; Chikuda, Hirotaka; Oshima, Yasushi; Matsubayashi, Yoshitaka; Tanaka, Sakae

doi:10.1016/j.ijscr.2016.08.038

Cited by 10 publications

(8 citation statements)

References 19 publications

(19 reference statements)

Supporting

Mentioning

Contrasting

Order By: Relevance

“…26,27 A case report of vitamin D–resistant rickets found extensive ossification of the paraspinal ligaments. 28 The serum Pi concentration in most cases of rickets is lower than in normal controls. A recent study revealed that serum FGF-23 was higher in 41 male patients with OPLL than in 76 healthy male controls although the sample size was very small.…”

Section: Discussionmentioning

confidence: 97%

Increase of the Serum FGF-23 in Ossification of the Posterior Longitudinal Ligament

Kawaguchi

Kitajima

Nakano

et al. 2018

Global Spine Journal

View full text Add to dashboard Cite

Study Design: Case-control study. Objectives: To determine the possible pathogenesis of ossification of the posterior longitudinal ligament (OPLL) in regard to the serum concentration of fibroblast growth factor 23 (FGF-23). Methods: The study included 95 patients with OPLL and a control group of 73 age- and sex-matched volunteers. The serum concentrations of FGF-23, creatinine (Cre), alkaline phosphatase, calcium (Ca), inorganic phosphate (Pi), and hypersensitive C-reactive protein (hs-CRP) were analyzed from blood samples, and Cre, Ca, Pi, and tubular reabsorption of phosphate were measured using urine samples. We evaluated the severity of ossified spinal lesions in patients with OPLL according to the ossification index (the OP index and the OS index). Data was compared between the OPLL and control group and between the OPLL progression and no progression group. Results: Serum FGF-23 and hs-CRP were higher, and serum Pi was lower in patients with OPLL than in the controls. There was a positive correlation between FGF-23 and hs-CRP and a negative correlation between serum Pi and the OS index; however, the correlations were very weak. Overall, 31.7% of patients had progression of OPLL during follow-up. FGF-23 and hs-CRP were higher in the progression group than in the no progression group. Conclusions: These results might indicate that FGF-23 and hs-CRP are positive markers for OPLL. Phosphate metabolism via FGF-23 might be a target for future study on the pathogenesis of OPLL.

show abstract

Section: Discussionmentioning

confidence: 97%

Increase of the Serum FGF-23 in Ossification of the Posterior Longitudinal Ligament

Kawaguchi

Kitajima

Nakano

et al. 2018

Global Spine Journal

View full text Add to dashboard Cite

show abstract

“…The medical literature regarding the spinal manifestations of XLH and their surgical management and outcomes is restricted to case reports and short case series (Hirao et al 2016 ; Soehle and Casey 2002 ). Ossification of the paraspinal ligaments in XLH is well recognised and the published series document thickening of the laminae, calcification of the discs, facet joint hypertrophy and ossification of the ligamentum flavum and/or posterior longitudinal ligament (Hirao et al 2016 ; Soehle and Casey 2002 ; Dunlop and Stirling 1996 ; Velan et al 2001 ). These can result in spinal canal stenosis impinging on the spinal cord leading to myelopathy or on the lumbar nerve roots manifesting as neurogenic claudication/radiculopathy.…”

Section: Discussionmentioning

confidence: 99%

“…To date however, only 23 cases of XLH undergoing spinal surgery for myelopathy have been reported (Hirao et al 2016 ).…”

Section: Discussionmentioning

confidence: 99%

Outcome of adult patients with X‐linked hypophosphatemia caused by PHEX gene mutations

Chesher

Oddy

Darbar

et al. 2018

J of Inher Metab Disea

110

View full text Add to dashboard Cite

X-linked hypophosphatemia (XLH) is the most common monogenic disorder causing hypophosphatemia. This case-note review documents the clinical features and the complications of treatment in 59 adults (19 male, 40 female) with XLH. XLH is associated with a large number of private mutations; 37 different mutations in the PHEX gene were identified in this cohort, 14 of which have not been previously reported. Orthopaedic involvement requiring surgical intervention (osteotomy) was frequent. Joint replacement and decompressive laminectomy were observed in those older than 40 years. Dental disease (63%), nephrocalcinosis (42%), and hearing impairment (14%) were also common. The rarity of the disease and the large number of variants make it difficult to discern specific genotype-phenotype relationships. A new treatment, an anti-FGF23 antibody, that may affect the natural history of the disease is currently being investigated in clinical trials.Electronic supplementary materialThe online version of this article (10.1007/s10545-018-0147-6) contains supplementary material, which is available to authorized users.

show abstract

“…In fact, a recent case report revealed that 34-year-old female with vitamin D resistant rickets had extensive ossification of the paraspinal ligaments. The serum concentration of her Pi was low whereas that of Ca was normal [ 38 , 39 ].…”

Section: Discussionmentioning

confidence: 99%

Serum biomarkers in patients with ossification of the posterior longitudinal ligament (OPLL): Inflammation in OPLL

et al. 2017

View full text Add to dashboard Cite

BackgroudOssification of the posterior longitudinal ligament (OPLL) is characterized by replacement of ligamentous tissue by ectopic new bone formation. OPLL causes narrowing of the spinal canal, resulting in neurological impairment. However, the pathogenesis of OPLL has not been fully elucidated. We investigated whether inflammation occurs in OPLL or not using high-sensitivity CRP (hs-CRP) in a case-control study.Methods and findingsThis study included 103 patients with OPLL in the patient group and 95 age- and sex-matched volunteers with degenerative spinal disease in the control group. Of the 103 OPLL patients, 88 patients who were available for more than 2 years follow-up were checked for OPLL progression. A blood sample was obtained and Hs-CRP, and other routine data, including total protein (TP), albumin (ALB), lactate dehydrogenase (LDH), alkaline phosphatase (ALP), glucose (Glu), calcium (Ca), inorganic phosphate (Pi), white blood cell count (WBC), hemoglobin (Hb) and platelet (PLT), were analyzed. The data were compared between the patients with OPLL and the controls. The severity of the ossified lesions in the whole spine were evaluated by the ossification index (OS index) in patients with OPLL. The data were also compared between the patients with OPLL progression (the progression group) and the patients without OPLL progression (the non-progression group). In the results, the mean hs-CRP in the OPLL group was higher than that in the controls. The Pi in the OPLL group was lower than that in the control group. A negative correlation was found between the Pi and the OS index. The mean hs-CRP in the progression group was higher than that in the non-progression group. There was a positive correlation between the average length of the OPLL progression per year and the hs-CRP.ConclusionsThe results may suggest the occurrence of local inflammation in OPLL and the inflammation might cause OPLL progression. These facts are important for understanding the pathology of OPLL.

show abstract

Extensive ossification of the paraspinal ligaments in a patient with vitamin D-resistant rickets: Case report with literature review

Cited by 10 publications

References 19 publications

Increase of the Serum FGF-23 in Ossification of the Posterior Longitudinal Ligament

Increase of the Serum FGF-23 in Ossification of the Posterior Longitudinal Ligament

Outcome of adult patients with X‐linked hypophosphatemia caused by PHEX gene mutations

Serum biomarkers in patients with ossification of the posterior longitudinal ligament (OPLL): Inflammation in OPLL

Contact Info

Product

Resources

About