Literature DB >> 8409269

Möbius syndrome: evidence for a vascular etiology.

O F D'Cruz1, C N Swisher, S Jaradeh, T Tang, R J Konkol.   

Abstract

We report five infants with restricted lateral gaze, facial diplegia, feeding difficulty, and/or respiratory disorders without significant pulmonary disease. Viral studies were negative in all patients. Two children had radiologic findings that included brain-stem hypoplasia and symmetric calcification in the dorsal tectum at the junction of the midbrain and pons. Autopsy of one of these two children demonstrated capillary telangiectasia in the mesencephalon and pons. The other three children had normal computed tomographic (CT) scans. However, their autopsies revealed focal brain-stem necrosis with calcifications but without vascular malformation. We suggest that the capillary malformations in one of our patients directly resulted in a vascular-induced necrosis and the manifestation of Möbius sequence. The similarity of symmetric neuropathologic findings in the three other patients and the CT scan in the one surviving patient suggest focal hemodynamic changes restricted to the posterior circulation, indirectly supporting a vascular theory of embryopathogenesis.

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Year:  1993        PMID: 8409269     DOI: 10.1177/088307389300800310

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  7 in total

1.  Magnetic resonance imaging of the endophenotype of a novel familial Möbius-like syndrome.

Authors:  Sean Dumars; Caroline Andrews; Wai-Man Chan; Elizabeth C Engle; Joseph L Demer
Journal:  J AAPOS       Date:  2008-05-02       Impact factor: 1.220

2.  Möbius syndrome with brain stem calcification: prenatal and neonatal sonographic findings.

Authors:  K Yoon; S J Yoo; D C Suh; Y A Lee; K S Kim; G Choe
Journal:  Pediatr Radiol       Date:  1997-02

3.  Multigenic truncation of the semaphorin-plexin pathway by a germline chromothriptic rearrangement associated with Moebius syndrome.

Authors:  Lusine Nazaryan-Petersen; Inês R Oliveira; Mana M Mehrjouy; Juan M M Mendez; Mads Bak; Merete Bugge; Vera M Kalscheuer; Iben Bache; Dustin C Hancks; Niels Tommerup
Journal:  Hum Mutat       Date:  2019-05-14       Impact factor: 4.878

4.  Goldenhar, Möbius and hypoglossia-hypodactyly anomalies in a patient: syndrome or association?

Authors:  S Preis; F Majewski; R Hantschmann; H Schumacher; H G Lenard
Journal:  Eur J Pediatr       Date:  1996-05       Impact factor: 3.183

5.  A case of Moebius syndrome presenting with congenital bilateral vocal cord paralysis.

Authors:  Nobuko Kanemoto; Katsuyoshi Kanemoto; Tomohiro Kamoda; Makoto Hasegawa; Tadao Arinami
Journal:  Eur J Pediatr       Date:  2006-11-30       Impact factor: 3.183

Review 6.  Congenital aberrant tearing: a re-look.

Authors:  Marilyn T Miller; Kerstin Strömland; Liana Ventura
Journal:  Trans Am Ophthalmol Soc       Date:  2008

7.  Brain phenotyping in Moebius syndrome and other congenital facial weakness disorders by diffusion MRI morphometry.

Authors:  Neda Sadeghi; Elizabeth Hutchinson; Carol Van Ryzin; Edmond J FitzGibbon; John A Butman; Bryn D Webb; Flavia Facio; Brian P Brooks; Francis S Collins; Ethylin Wang Jabs; Elizabeth C Engle; Irini Manoli; Carlo Pierpaoli
Journal:  Brain Commun       Date:  2020-02-14
  7 in total

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