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      Bilateral cerebellopontine angle lipomas in an infant with encephalocele: illustrative case

      case-report

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          Abstract

          BACKGROUND

          Bilateral cerebellopontine angle (CPA) lipomas are extremely rare. Herein the authors present a case of bilateral CPA lipomas in an infant along with a literature review of bilateral CPA lipomas.

          OBSERVATIONS

          A newborn girl was incidentally found to have bilateral CPA lipomas during the workup for an occipital encephalocele. The encephalocele was repaired primarily on day 2 after birth. The patient demonstrated no symptoms associated with the bilateral CPA lipomas. Eight cases of bilateral CPA lipomas were identified in the literature review and are summarized. Conservative management is the consensus strategy, given minimum growth of the tumor and the high risk of surgical intervention.

          LESSONS

          This is the first reported case of bilateral CPA lipomas in an infant as well as the first with a coexisting intracranial malformation. Intracranial lipomas share an extremely low growth rate and typically do not cause severe symptoms. The management of asymptomatic or mildly symptomatic bilateral CPA lipomas is usually conservative.

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          Most cited references14

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          Pathogenesis of intracranial lipoma: an MR study in 42 patients.

          C Truwit, A Barkovich (2015)
          Intracranial lipomas are uncommon lesions whose development remains poorly understood. To clarify the anatomic and embryologic features of intracranial lipomas, we retrospectively reviewed the MR scans of 42 patients with 44 intracranial lipomas. Interhemispheric lipomas were the most common, accounting for 45% of cases. The remainder of the lesions were clustered in the quadrigeminal/superior cerebellar (25%), suprasellar/interpeduncular (14%), cerebellopontine angle (9%), and sylvian (5%) cisterns. Fifty-five percent of the lesions were associated with brain malformations of varying degrees. Intracranial vessels and nerves were noted to course through 16 (36%) of the lesions. The relative frequencies of the locations of the lipomas correspond to the temporal sequence of dissolution of the meninx primitiva, the mesenchymal anlage of the meninges. This finding supports the concept of lipoma formation as a result of abnormal persistence and maldifferentiation of the meninx. This embryologic concept of the development of intracranial lipomas explains the high frequency of callosal and other brain hypoplasias. Intracranial lipomas are neither hamartomas nor true neoplasms; rather, they are congenital malformations.
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            Cerebellopontine angle lipomas: report of four cases and review of the literature.

            E Vitte, F Tankéré, J Soudant … (2002)
            To define the management of internal acoustic meatus and cerebellopontine angle (CPA) lipomas according to their clinical, histological, and surgical characteristics. We report four new cases of CPA lipomas diagnosed in the Department of Otorhinolaryngology-Head and Neck Surgery of Hôpital Pitié-Salpêtrière and review 94 cases reported previously in the literature. Lipomas represented 0.14% of CPA and internal acoustic meatus tumors. Localization was on the left side in 59.9%, on the right side in 37%, and bilateral in 3.1% of the patients. The diagnosis was confirmed radiologically in 33 of 98 patients, surgically in 60 patients, and by autopsy in 5 patients. The most frequent associated symptoms were of cochleovestibular origin, such as hearing loss (62.2%), dizziness (43.3%), and unilateral tinnitus (42.2%). Other associated symptoms involved the facial nerve (9%) or the trigeminal nerve (14.4%). Complete resection was performed in only 32.8% of the patients with frequent cranial nerve involvement. Frequent cranial nerve involvement was seen in 95.4% of all patients. After surgery, patient symptomatology was unchanged in 9.2% of the patients, and 50% were improved; however, new postoperative deficits occurred in two-thirds of the patients. Overall, 72.2% of the patients experienced new postoperative deficits such as hearing loss (64.8%). Preservation of hearing was possible in only 26% of the patients. Only 18% of patients were improved after surgery without any new postoperative deficits. Preoperative diagnosis of internal acoustic meatus/CPA lipomas is based on magnetic resonance imaging. The aim of surgery in these cases is not tumor removal but cranial nerve decompression or vestibular transection, and surgery is performed only in patients with disabling and uncontrolled symptoms.
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              Development of the cerebrospinal fluid pathway in the normal and abnormal human embryos.

              S. Matsumoto, H Handa, M Yasuda … (1979)
              The subarachnoid space, the chorioid plexus and the arachnoid villi are microscopically studied in 60 normal human embryos and in 3 abnormal human embryos with rhombencephaloschisis and cervical myeloschisis. The subarachnoid space has been generally considered to be developed by outflow of cerebrospinal fluid (CSF) of the choroid-plexus origin from the IVth ventricle. This generally accepted concept does not meet with our findings: (1) cavity formation in the meninx primitiva is seen before appearance of the choroid plexus; (2) the primitive subarachnoid space is developed earlier in the prepontine region than in the area dorsal to the rhombic roof, and (3) the primitive subarachnoid space is formed in the embryos with dysraphism where the perineural subarachnoid space is separated from the ventricles. Apparently the embryonic pattern of CSF circulation should be much different from the generally believed pattern of adult, since the arachnoid villi are absent in the embryos and the ability of production of CSF in the embryonic choroid plexus is questionable. It is suggested that such embryonic pattern of CSF production and absorption may partly persist in adult human being.
              Article 0 comments Cited 9 times – based on 0 reviews     Review now
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                Author and article information

                Journal
                Journal ID (nlm-ta): J Neurosurg Case Lessons
                Journal ID (iso-abbrev): J Neurosurg Case Lessons
                Journal ID (publisher-id): J Neurosurg Case Lessons
                Title: Journal of Neurosurgery: Case Lessons
                Publisher: American Association of Neurological Surgeons
                ISSN (Electronic): 2694-1902
                Publication date Collection: 25 September 2023
                Publication date (Electronic): 25 September 2023
                Volume: 6
                Issue: 13
                Electronic Location Identifier: CASE23315
                Affiliations
                [1]Division of Pediatric Neurosurgery, Department of Neurosurgery, Oklahoma Children’s Hospital, University of Oklahoma Health Sciences Center, Oklahoma City, Oklahoma
                Author notes
                Correspondence Joanna E. Gernsback: University of Oklahoma Health Sciences Center, Oklahoma City, OK. joanna-gernsback@ 123456ouhsc.edu .

                INCLUDE WHEN CITING Published September 25, 2023; DOI: 10.3171/CASE23315.

                Disclosures The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.

                Article
                Publisher ID: CASE23315
                DOI: 10.3171/CASE23315
                PMC ID: 10555580
                PubMed ID: 37773761
                SO-VID: c0028baf-9147-49c9-aee8-cb203c7738b0
                Copyright © © 2023 The authors
                License:

                CC BY-NC-ND 4.0 ( http://creativecommons.org/licenses/by-nc-nd/4.0/)

                History
                Date received : 13 June 2023
                Date accepted : 15 August 2023
                Page count
                Figures: 1, Tables: 1, References: 14, Pages: 3
                Categories
                Compound subject: Congenital, Congenital
                Compound subject: Pediatric, Pediatric
                Compound subject: Skull-Base, Skull Base
                Subject: Case Lesson

                Keywords: case report,cerebellopontine angle,encephalocele,lipoma,pediatric neurosurgery,cpa = cerebellopontine angle,mri = magnetic resonance imaging
                Data availability:
                Keywords: case report, cerebellopontine angle, encephalocele, lipoma, pediatric neurosurgery, cpa = cerebellopontine angle, mri = magnetic resonance imaging

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