Literature DB >> 29872110

Cervical spinal cord compression in infants with achondroplasia: should neuroimaging be routine?

Victoria R Sanders1, Stephen H Sheldon2, Joel Charrow3.   

Abstract

PURPOSE: To examine results of magnetic resonance imaging (MRI), polysomnograms (PSG), and patient outcomes in patients with achondroplasia in light of recent screening recommendations for infants with achondroplasia.
METHODS: We reviewed medical records of 49 patients with achondroplasia followed at our institution between September 1997 and January 2017, including physical exams, MRIs, PSGs (when available), and surgical histories. Appropriate PSG data were available for 39 of these patients.
RESULTS: Twenty-seven of 49 patients had cervical cord compression on MRI, and 20 of those patients required surgery. Central apnea was detected in 2/23 patients with cervical cord compression in whom PSG data was available. Physical exam revealed depressed deep-tendon reflexes in two patients with cord compression and one patient without cord compression. Besides hypotonia in some, the neurological exams of these patients were unremarkable.
CONCLUSIONS: Cervical cord compression is a common occurrence in infants with achondroplasia and necessitates surgical intervention in some patients. Physical exam and PSG are poor predictors of the presence of cord compression or the need for surgery. All infants with achondroplasia should have MRIs of the craniocervical junction in the first 6 months of life.

Entities:  

Keywords:  Achondroplasia; Central apnea; Cervical cord compression; Foramen magnum stenosis; Sudden death

Mesh:

Year:  2018        PMID: 29872110     DOI: 10.1038/s41436-018-0070-0

Source DB:  PubMed          Journal:  Genet Med        ISSN: 1098-3600            Impact factor:   8.822


  5 in total

1.  Mortality in achondroplasia.

Authors:  J T Hecht; C A Francomano; W A Horton; J F Annegers
Journal:  Am J Hum Genet       Date:  1987-09       Impact factor: 11.025

2.  Cervicomedullary junction compression in infants with achondroplasia: when to perform neurosurgical decompression.

Authors:  D L Rimoin
Journal:  Am J Hum Genet       Date:  1995-04       Impact factor: 11.025

Review 3.  Magnetic resonance imaging in the assessment of medullary compression in achondroplasia.

Authors:  I T Thomas; J L Frias; J L Williams; W A Friedman
Journal:  Am J Dis Child       Date:  1988-09

Review 4.  Acute neurological deficit after minor trauma in an infant with achondroplasia and cervicomedullary compression. Case report and review of the literature.

Authors:  David M Benglis; David I Sandberg
Journal:  J Neurosurg       Date:  2007-08       Impact factor: 5.115

5.  Prospective assessment of risks for cervicomedullary-junction compression in infants with achondroplasia.

Authors:  R M Pauli; V K Horton; L P Glinski; C A Reiser
Journal:  Am J Hum Genet       Date:  1995-03       Impact factor: 11.025

  5 in total
  5 in total

Review 1.  International Consensus Statement on the diagnosis, multidisciplinary management and lifelong care of individuals with achondroplasia.

Authors:  Ravi Savarirayan; Penny Ireland; Melita Irving; Dominic Thompson; Inês Alves; Wagner A R Baratela; James Betts; Michael B Bober; Silvio Boero; Jenna Briddell; Jeffrey Campbell; Philippe M Campeau; Patricia Carl-Innig; Moira S Cheung; Martyn Cobourne; Valérie Cormier-Daire; Muriel Deladure-Molla; Mariana Del Pino; Heather Elphick; Virginia Fano; Brigitte Fauroux; Jonathan Gibbins; Mari L Groves; Lars Hagenäs; Therese Hannon; Julie Hoover-Fong; Morrys Kaisermann; Antonio Leiva-Gea; Juan Llerena; William Mackenzie; Kenneth Martin; Fabio Mazzoleni; Sharon McDonnell; Maria Costanza Meazzini; Josef Milerad; Klaus Mohnike; Geert R Mortier; Amaka Offiah; Keiichi Ozono; John A Phillips; Steven Powell; Yosha Prasad; Cathleen Raggio; Pablo Rosselli; Judith Rossiter; Angelo Selicorni; Marco Sessa; Mary Theroux; Matthew Thomas; Laura Trespedi; David Tunkel; Colin Wallis; Michael Wright; Natsuo Yasui; Svein Otto Fredwall
Journal:  Nat Rev Endocrinol       Date:  2021-11-26       Impact factor: 47.564

2.  Literature review and expert opinion on the impact of achondroplasia on medical complications and health-related quality of life and expectations for long-term impact of vosoritide: a modified Delphi study.

Authors:  Ravi Savarirayan; Wagner Baratela; Thomas Butt; Valérie Cormier-Daire; Melita Irving; Bradley S Miller; Klaus Mohnike; Keiichi Ozono; Ron Rosenfeld; Angelo Selicorni; Dominic Thompson; Klane K White; Michael Wright; Svein O Fredwall
Journal:  Orphanet J Rare Dis       Date:  2022-06-13       Impact factor: 4.303

3.  Identification of clinical and radiographic predictors of central nervous system injury in genetic skeletal disorders.

Authors:  Antônio L Cunha; Ana P S Champs; Carla M Mello; Mônica M M Navarro; Frederico J C Godinho; Cássia M B Carvalho; Teresa C A Ferrari
Journal:  Sci Rep       Date:  2021-05-31       Impact factor: 4.379

4.  The (extended) achondroplasia foramen magnum score has good observer reliability.

Authors:  Nathan Jenko; Daniel J A Connolly; Ashok Raghavan; James A Fernandes; Shungu Ushewokunze; Heather E Elphick; Paul Arundel; Utku Alhun; Amaka C Offiah
Journal:  Pediatr Radiol       Date:  2022-04-09

5.  Apparently benign craniocervical signs in achondroplasia: "neurologic leftovers" identified through a retrospective dataset.

Authors:  Cory J Smid; Janet M Legare; Peggy Modaff; Richard M Pauli
Journal:  Orphanet J Rare Dis       Date:  2020-10-23       Impact factor: 4.123

  5 in total

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