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Intracranial extracerebral glioneuronal heterotopia

Case report and review of the literature

Soichi Oya, Nobutaka Kawahara, Shigeki Aoki, Naoto Hayashi, Junji Shibahara, Masafumi Izumi and Takaaki Kirino

P The authors report a case of intracranial extracerebral glioneuronal heterotopia (IEGH) in an infant. Glioneuronal heterotopias are rare congenital disorders that arise in the head, face, spine, and thoracic cavity. They consist of nodular accumulations of neuronal and glial cells that have developed abnormally, ranging in size from small lesions to large masses. Among heterotopias, IEGHs are relatively rare. They cause various clinical symptoms, depending on their size and location. The neuroimaging studies, histological examinations, and intraoperative findings presented provide insight into the pathogenesis of this disorder. The findings support the separation and detachment theory, which proposes that IEGHs originate from a third telencephalon that erroneously forms between the 4th and 6th week of embryogenesis. More detailed case reports are necessary to understand fully the pathogenesis of IEGHs.

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Toru Doi, Ryuji Sakamoto, Chiaki Horii, Naoki Okamoto, Koji Nakajima, Shima Hirai, Fumihiko Oguchi, So Kato, Yuki Taniguchi, Yoshitaka Matsubayashi, Naoto Hayashi, Sakae Tanaka and Yasushi Oshima

OBJECTIVE

The incidence and risk factors for the progression of ossification of the posterior longitudinal ligament (OPLL) have been previously reported in surgically and nonsurgically treated symptomatic patients. However, the correlates of OPLL progression in asymptomatic subjects with OPLL are not well characterized. This study aimed to clarify the incidence and risk factors for OPLL progression in asymptomatic subjects based on whole-body CT.

METHODS

The authors retrospectively reviewed 2585 healthy subjects who underwent whole-body CT at a single health center from September 2007 to December 2011. This study included asymptomatic subjects with OPLL who underwent CT scans twice with an interval of at least 5 years. Progression of OPLL was assessed based on initial and final CT scan. Subjects were divided into two groups: nonprogression (OPLL-NP) and progression (OPLL-P) groups. Clinical characteristics, bone mineral density status, OPLL types, and OPLL involvement of multiple vertebral levels between the two groups were compared. Risk factors for progression of OPLL were identified by logistic regression analysis after propensity score adjustment.

RESULTS

Of the 109 subjects with OPLL (91 men and 18 women), 20 (18.3%) exhibited OPLL progression (OPLL-P group). Subjects in the OPLL-P group were significantly younger (p = 0.031), had higher prevalence of multilevel OPLL involvement (p = 0.041) and continuous type of OPLL (p = 0.015), and had higher uric acid (UA) levels (p = 0.004) at the time of initial CT examination compared to the OPLL-NP group. Younger age (adjusted odds ratio [aOR] 0.95, 95% CI 0.90–0.99), OPLL involvement of multiple vertebral levels (aOR 2.88, 95% CI 1.06–7.83), continuous type of OPLL (aOR 4.21, 95% CI 1.35–13.10), and higher UA levels (aOR 2.09, 95% CI 1.24–3.53) were significant risk factors for OPLL progression.

CONCLUSIONS

Younger age, OPLL involvement of multiple vertebral levels, continuous type of OPLL, and higher UA levels are significant risk factors for OPLL progression in asymptomatic subjects.