Congenital oculo-bulbar palsy.

نویسندگان

  • F G Jennekens
  • H Veldman
  • L J Vroegindeweij-Claessens
  • P C Molenaar
  • A A Op de Coul
چکیده

A girl developed progressive weakness of bulbar and ocular muscles starting before the age of two years. Electromyography revealed a widespread subclinical myopathy. An intercostal muscle biopsy showed complex abnormalities including occasional neurofilamentous accumulations and honeycomb-like membranous material in terminal axons. Endplates were small and some secondary synaptic clefts were abnormally deep. Acetylcholine receptors extended unusually deeply into the clefts of the junctional folds. Muscle fibres showed subsarcolemmal vacuolation at some places. This form of congenital oculo-bulbar palsy does not appear to have been described previously.

برای دانلود متن کامل این مقاله و بیش از 32 میلیون مقاله دیگر ابتدا ثبت نام کنید

ثبت نام

اگر عضو سایت هستید لطفا وارد حساب کاربری خود شوید

منابع مشابه

A rare case report showing direct association between hepatitis B and bulbar palsy.

Bulbar Palsy is B/L impairment of function of cranial N. 9, 10, 11, 12 at lower motor neuron level either at nuclear or fascicular level in medulla or B/L lesion of lower cranial N. outside brainstem. We present case of viral hepatitis who initially presented with classical signs and symptoms of hepatitis B followed by characteristic features of bulbar palsy in form of difficulty in swallowing ...

متن کامل

Co-existence of Two Rare Conditions: Oculo-Palato-Cerebral Syndrome and Congenital Chylothorax

Background: Oculo-palato-cerebral syndrome is an extremely rare condition characterized by various features, including low-birth weight, microcephaly, cerebral atrophy, mild-to-severe developmental delay, cleft palate, persistent hyperplastic primary vitreous, microphthalmia, small hands and feet, joint laxity, and large ears with thick helices. Diagnosis of this syndrome is based on the clinic...

متن کامل

Oculo-auriculovertebral Spectrum with Radial Anomaly: A Rare Case Report

Oculo-auriculovertebral spectrum (OAVS) or the Goldenhar syndrome is a rare developmental disorder with plethora of congenital anomalies which mostly affects structures arising from the fi rst and the second branchial arches. The affected structures include cheekbone, jaws, mouth eyes, ear and vertebrae. The case which we are presenting here had classical features such as microsomia, unilateral...

متن کامل

Observations on the Pathology of the Moebius Syndrome.

The first report of the congenital facial diplegia syndrome, or Moebius syndrome, was that of Graefe (1880). Moebius reported cases in 1888 and 1892 and reviewed the prior case reports, thus gaining eponymic distinction. Since that time, some authors have broadened the definition of the Moebius syndrome, as for example, Henderson (1939), who considered some cases of congenital unilateral facial...

متن کامل

CT of severe inner ear anomalies, including aplasia, in a case of Wildervanck syndrome.

The Wildervanck (or cervico-oculo-acoustic) syndrome consists of the characteristic triad of Klippel-Feil anomaly, abducens nerve palsy, and congenital deafness. It is a rare "disorder of polygenic heredity with sex limitation predominant to the female" (female-male ratio is 10: 1) ( 1 ). Hearing loss is most commonly neurosensory due to developmental inner ear anomalies. Only a single case of ...

متن کامل

ذخیره در منابع من


  با ذخیره ی این منبع در منابع من، دسترسی به آن را برای استفاده های بعدی آسان تر کنید

برای دانلود متن کامل این مقاله و بیش از 32 میلیون مقاله دیگر ابتدا ثبت نام کنید

ثبت نام

اگر عضو سایت هستید لطفا وارد حساب کاربری خود شوید

عنوان ژورنال:
  • Journal of neurology, neurosurgery, and psychiatry

دوره 55 5  شماره 

صفحات  -

تاریخ انتشار 1992