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      Congenital neuroglial choristoma of the foot

      case-report

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          Abstract

          Neuroglial choristomas are rare malformations of heterotopic neural tissue that have been previously reported predominantly in the head and neck. Competing theories of embryogenesis propose their origin as encephaloceles that have undergone resorption of their cranial connection or displaced neuroectodermal cells which have undergone ectopic proliferation. Most cases occur in midline or para-midline structures. There have been no prior published cases of a neuroglial choristoma in the extremities. We present a case of a 13-month-old otherwise healthy child who presented to our institution with a slowly growing foot mass who was found to have a neuroglial choristoma. This case suggests an early embryological migration defect as the etiology and offers a unique differential consideration for a benign extremity mass.

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          Most cited references8

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          A clinicopathological study of 15 patients with neuroglial heterotopias and encephaloceles of the middle ear and mastoid region.

          Heterotopic masses of neuroglial tissue involving non-midline structures, specifically, the middle ear region, are exceptional. The pathogenesis of these lesions and, in particular, their relation to encephaloceles, is uncertain. H&E-stained sections from 15 lesions diagnosed as neuroglial heterotopias or encephaloceles involving the middle ear region were reviewed. Radiographic or operative evidence of a central nervous system (CNS) relation and clinical factors possibly related to pathogenesis were analyzed. All 15 lesions (from six men and nine women; mean age, 49 y; range, 16-67 y), regardless of their relation to the CNS, were composed of varying proportions of neurons and glia with associated chronic inflammatory cells and reactive gliosis. No significant ependymal or choroid plexus component was present. Operative findings revealed that two lesions had definite CNS connections and two were unrelated to the CNS; this relation could not be determined in the remaining cases. Seven of 10 patients for whom clinical information was available had a history of chronic otitis media or mastoiditis or both; four of these seven patients also had a history of previous trauma or surgery. Three patients, including both patients whose lesions had no demonstrable CNS attachment, had no predisposing factors. Most neuroglial heterotopias of the middle ear are probably acquired encephaloceles. These lesions occur in older patients than do their midline counterparts. Determination of the relation of these lesions to adjacent CNS structures must be done radiographically or using operative findings, because histology alone cannot be reliably used to render an accurate diagnosis.
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            Pulmonary glial heterotopia in a monoamniotic twin.

            Brain heterotopia in the lungs is rare, but when it occurs in an otherwise healthy newborn, it presents a difficult diagnostic problem and uncertain pathophysiology. We report on a 2-week-old premature infant who presented with respiratory distress and widespread cystic lung changes identified by chest imaging studies. Autopsy demonstrated that the cyst walls were composed of well-differentiated neuroglial tissue, which was confirmed by immunohistochemistry. The cysts were partially lined by bronchial epithelium and contained keratinous debris. For the first time, we demonstrate that the debris stain for human chorionic gonadotropin, compatible with aspirated amnion. There were no other congenital abnormalities. Her monoamniotic twin was anencephalic and died at birth. Although the etiology of glial heterotopia in the lungs is unknown, the majority of cases are associated with anencephalic newborns. Some authors postulated that this heterotopia may therefore be a consequence of fetal aspiration of brain tissue. Other possibilities include glial predominant teratomas, hamartomatous malformations, and abnormal neural crest migration. Our review of the 21 cases reported over the past century suggests that in utero aspiration of glial cells, or abnormal neural crest migration, are the most likely explanations for this rare and fatal disease.
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              Rudimentary cephalocele or neural crest remnant?

              A Drapkin (1990)
              Three cases of the so-called rudimentary cranial meningocele/encephalocele are presented. A review of the literature yielded a number of similar cases which were tabulated and analyzed. It is suggested that this group of lesions represents neural crest remnants for which there is a good prognosis, distinct from the true meningocele/encephalocele complex.
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                Author and article information

                Contributors
                Journal
                Radiol Case Rep
                Radiol Case Rep
                Radiology Case Reports
                Elsevier
                1930-0433
                02 April 2019
                June 2019
                02 April 2019
                : 14
                : 6
                : 718-722
                Affiliations
                [a ]Department of Radiological Sciences, University of California Irvine, Orange, CA, USA
                [b ]Department of Diagnostic Imaging, Children's Hospital of Orange County, Orange, CA, USA
                [c ]Department of Oncology, Children's Hospital of Orange County, Orange, CA, USA
                [d ]Department of Pathology, Children's Hospital of Orange County, Orange, CA, USA
                Author notes
                [* ]Corresponding author. tdao@ 123456choc.org
                Article
                S1930-0433(19)30049-4
                10.1016/j.radcr.2019.03.025
                6447744
                30988863
                13607259-9064-4b00-8a19-9841f3e4d904
                © 2019 The Authors

                This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

                History
                : 13 February 2019
                : 21 March 2019
                : 24 March 2019
                Categories
                Pediatric

                neuroglial choristoma,glial heterotopia,neural heterotopia

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