Literature DB >> 7577667

Association of infantile neuroaxonal dystrophy and osteopetrosis: a rare autosomal recessive disorder.

H Rees1, L C Ang, R Casey, D H George.   

Abstract

The association of neuroaxonal dystrophy and osteopetrosis is reported in 2 siblings born to non-consanguineous parents. The 1st child was diagnosed as having infantile osteopetrosis shortly after delivery. A computed tomography scan of the head revealed agenesis of the corpus callosum. She died at the age of 9 months. Post-mortem examination showed pneumonia and bony sclerosis. Neuropathological examination revealed cerebral atrophy, ventricular dilation, absence of the corpus callosum, and a small hippocampus. Neuroaxonal spheroids were found in hippocampus, basal ganglia, pons, medulla, spinal cord, cranial nerves, cerebellum, and peripheral nerves. Ultrastructural examination revealed membranous cytoplasmic bodies and electron-dense granular deposits within the neuroaxonal spheroids as well as the soma of neurons. The 2nd child was delivered at 36 weeks of gestation because of intrauterine fetal distress. The diagnosis of osteopetrosis and partial agenesis of the corpus callosum was made shortly after delivery. The child died at 1 month without an autopsy. There are rare cases reported previously with the association of neuroaxonal dystrophy and osteopetrosis. We review these cases and compare them with ours.

Entities:  

Mesh:

Year:  1995        PMID: 7577667     DOI: 10.1159/000120923

Source DB:  PubMed          Journal:  Pediatr Neurosurg        ISSN: 1016-2291            Impact factor:   1.162


  6 in total

Review 1.  Autosomal recessive osteopetrosis: diagnosis, management, and outcome.

Authors:  C J Wilson; A Vellodi
Journal:  Arch Dis Child       Date:  2000-11       Impact factor: 3.791

2.  Importance of neurological assessment before bone marrow transplantation for osteopetrosis.

Authors:  M Abinun; T Newson; P W Rowe; T J Flood; A J Cant
Journal:  Arch Dis Child       Date:  1999-03       Impact factor: 3.791

Review 3.  Osteopetrosis: genetics, treatment and new insights into osteoclast function.

Authors:  Cristina Sobacchi; Ansgar Schulz; Fraser P Coxon; Anna Villa; Miep H Helfrich
Journal:  Nat Rev Endocrinol       Date:  2013-07-23       Impact factor: 43.330

4.  Anesthesia Management of a Child with Osteopetrosis.

Authors:  Hashem Jarineshin; Fereydoon Fekrat; Mehdi Feiz Dowlat Abadi
Journal:  Anesth Essays Res       Date:  2017 Jul-Sep

5.  SNX10 gene mutation leading to osteopetrosis with dysfunctional osteoclasts.

Authors:  Eva-Lena Stattin; Petra Henning; Joakim Klar; Emma McDermott; Christina Stecksen-Blicks; Per-Erik Sandström; Therese G Kellgren; Patrik Rydén; Göran Hallmans; Torsten Lönnerholm; Adam Ameur; Miep H Helfrich; Fraser P Coxon; Niklas Dahl; Johan Wikström; Ulf H Lerner
Journal:  Sci Rep       Date:  2017-06-07       Impact factor: 4.379

6.  Arthrogryposis multiplex congenital in a child manifesting phenotypic features resembling dysosteosclerosis/osteosclerosis malformation complex; 3DCT scan analysis of the skull base.

Authors:  Ali Al Kaissi; Georg Kalchhauser; Franz Grill; Klaus Klaushofer
Journal:  Cases J       Date:  2008-07-23
  6 in total

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