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      Congenital basal meningoceles with different outcomes: a case series

      case-report

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          Abstract

          Background

          Basal meningoceles are rare congenital defects and often clinically occult until they result in life-threatening complications. Therefore, it is important to know the diagnostic clues to early diagnosis.

          Case presentation

          We describe three cases of congenital basal meningocele in a 3-year-old Japanese boy, a 1-month-old Japanese baby boy, and a 10-month-old Japanese baby girl. One of our patients died of sepsis due to traumatic rupture of the meningocele during nasal suction. His meningocele remained undiagnosed until it resulted in the fatal complication. The other patients underwent surgical repair without any complications. Their meningoceles were complicated by midfacial anomalies including cleft palate and hypertelorism, or a sign of nasal obstruction such as snoring.

          Conclusions

          These clinical features may be a clue to the early diagnosis of congenital basal meningocele, which enables its safe preoperative management and provides an opportunity for surgical repair before the condition results in serious complications.

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          Most cited references10

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          Anterior basal encephalocele of the neonatal and infantile period.

          K Moroki, Y Matsukado, I Fuwa … (1986)
          Diagnosis of occult basal encephalocele is not difficult if the peculiar clinical and radiological signs of this anomaly are borne in mind. Recent surgery for transsphenoidal encephalocele has had better results than realized. However, high surgical risks may still be encountered in transsphenoidal encephalocele of the early infantile period, because the pituitary-hypothalamic structures are usually incorporated in the herniated encephalocele of this age group. Surgical indications for and operative approaches to transsphenoidal encephalocele in the infantile period are discussed on the basis of the authors' failure in transcranial repair, which resulted in early postoperative death due to hypothalamic dysfunction. Reviewing the reported cases of anterior basal encephalocele, a high correlation between transsphenoidal encephalocele, particularly in the pediatric age group, and allied malformations of the face, eye, and brain was disclosed. This characteristic malformation complex may be explained by a common pathogenetic mechanism operating in the embryonal period at about the stage of the anterior neuropore closure and occurring in the ventral surface of the cephalic end of the neural tube. Three cases of transsphenoidal encephalocele diagnosed in the neonatal period with progressive obstruction in the nasopharyngeal airway are also reported. A characteristic malformation complex consisting of median cleft face syndrome, optic nerve dysplasia, and agenesis of the corpus callosum was associated in two cases; the other patient had an extremely rare combination of septooptic dysplasia. Two patients died pre- and postoperatively, respectively; the other patient did not undergo operation because of grave multiple anomalies.
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            Frontonasal malformation as a field defect and in syndromic associations.

            H O Sedano, R J Gorlin (1988)
            Several new "syndromes" have been described that have in common facial findings identical or similar to those seen in frontonasal malformation (FNM), previously termed frontonasal dysplasia. Some of those new syndromes are inherited, whereas FNM is an isolated finding. Thus there is a need for differentiation among those conditions. A review of the literature was undertaken to identify and classify the various reports that describe FNM as it occurs alone and in association with a syndrome.
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              Transalar sphenoidal encephalocele and respiratory distress in a neonate: a case report.

              P Wesseling, E. Mylanus, L Kollée … (1998)
              We present a full-term newborn infant who suffered from immediate postpartum severe respiratory distress. The infant had an inspiratory stridor as a result of a swelling of the soft palate, extending from the roof of the nasopharynx. Transoral endotracheal intubation resulted in normal saturation levels. Histologic examination after an open biopsy showed mature neuroglial tissue. Radiology demonstrated the presence of a right parapharyngeal process obstructing the nasopharynx and oropharynx and extending to the right middle and posterior fossa, via the foramen ovale. After transoral debulking, the infant was extubated successfully. After an uneventful period of 5 months, the patient was readmitted at our hospital for treatment of meningitis. Subsequently, the inspiratory stridor recurred, and staged surgery was performed. First, a transcranial approach was used to remove a large intradural part of the process and close the defect at Meckel's cave. Two weeks later the retro- and parapharyngeal part of the process were removed transorally. Given the site of the defect of the skull base and the intradural location of the process, the diagnosis is a transalar sphenoidal encephalocele. This is a rare type of basal encephalocele, and has never been reported in an infant nor known to present with respiratory distress. The pathogenesis, clinical presentation, pathology, and therapeutic implications of basal encephaloceles are discussed.
              Article 0 comments Cited 7 times – based on 0 reviews     Review now
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                Author and article information

                Contributors
                Satomi Okano: s-okano@ceres.dti.ne.jp
                Ryosuke Tanaka: ryot5p@asahikawa-med.ac.jp
                Akie Okayama: oakie5p@asahikawa-med.ac.jp
                Etsushi Tsuchida: etsu5p@asahikawa-med.ac.jp
                Fumikatsu Nohara: nohara5p@asahikawa-med.ac.jp
                Nao Suzuki: nao5p@asahikawa-med.ac.jp
                Toshio Okamoto: toshio5p@asahikawa-med.ac.jp
                Ken Nagaya: nagaya5p@asahikawa-med.ac.jp
                Satoru Takahashi: satoru5p@asahikawa-med.ac.jp
                Hiroshi Azuma: azuma5p@asahikawa-med.ac.jp
                Journal
                Journal ID (nlm-ta): J Med Case Rep
                Journal ID (iso-abbrev): J Med Case Rep
                Title: Journal of Medical Case Reports
                Publisher: BioMed Central (London )
                ISSN (Electronic): 1752-1947
                Publication date (Electronic): 27 December 2017
                Publication date PMC-release: 27 December 2017
                Publication date Collection: 2017
                Volume: 11
                Electronic Location Identifier: 359
                Affiliations
                ISNI 0000 0000 8638 2724, GRID grid.252427.4, Department of Pediatrics, , Asahikawa Medical University, ; 2-1-1-1 Midorigaoka-Higashi, Asahikawa, Hokkaido 078-8510 Japan
                Article
                Publisher ID: 1497
                DOI: 10.1186/s13256-017-1497-7
                PMC ID: 5744424
                PubMed ID: 29279052
                SO-VID: ef2e4baa-546a-4044-a0c4-5afce27ca412
                Copyright © © The Author(s). 2017
                License:

                Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License ( http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

                History
                Date received : 20 October 2016
                Date accepted : 26 October 2017
                Categories
                Subject: Case Report
                Custom metadata
                issue-copyright-statement © The Author(s) 2017

                ScienceOpen disciplines: Medicine
                Keywords: basal meningocele,meningitis,suction,midfacial anomalies,snore
                Data availability:
                ScienceOpen disciplines: Medicine
                Keywords: basal meningocele, meningitis, suction, midfacial anomalies, snore

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