Literature DB >> 14872341

Ehlers-Danlos syndrome type VI with cystic malformations of the meninges in a 7-year-old girl.

Irene Brunk1, Brigitte Stöver, Chrysanthy Ikonomidou, Jürgen Brinckmann, Luitgard M Neumann.   

Abstract

UNLABELLED: A 7-year-old girl with thoracolumbar kyphoscoliosis was admitted for further diagnostic evaluation after a spinal MRI scan had shown several intraspinal extramedullary lesions. The clinical features including joint hypermobility and cigarette-paper like scars led to the presumptive diagnosis of Ehlers-Danlos syndrome type VI (EDS VI). Analysis of urinary lysyl- and hydroxylysyl-pyridinoline cross-links excretion confirmed a deficiency of lysylhydroxylase 1 and the diagnosis of EDS VIA. Findings on the spinal MRI scan were interpreted as spinal meningeal cysts. Over a period of 2 years, the patient developed no neurological deficits and no radiological signs of progression of the spinal lesions.
CONCLUSION: We assume cystic malformations of the meninges to be most likely the result of connective tissue weakness in Ehlers-Danlos syndrome type VI.

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Year:  2004        PMID: 14872341     DOI: 10.1007/s00431-004-1407-z

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  16 in total

1.  Ehlers-Danlos Syndrome Type VI (EDS VI): problems of diagnosis and management.

Authors:  P Heim; M Raghunath; L Meiss; U Heise; R Myllylä; A Kohlschütter; B Steinmann
Journal:  Acta Paediatr       Date:  1998-06       Impact factor: 2.299

2.  Multiple meningeal cysts in Marfan syndrome.

Authors:  L Robinson; R Dominguez; J Cabrera; J W Yeakley; M J Fenstermacher; M E Milner
Journal:  AJNR Am J Neuroradiol       Date:  1989 Nov-Dec       Impact factor: 3.825

3.  Urinary pyridinium cross-links: a noninvasive diagnostic test for Ehlers-Danlos syndrome type VI.

Authors:  M Pasquali; P P Dembure; M J Still; L J Elsas
Journal:  N Engl J Med       Date:  1994-07-14       Impact factor: 91.245

4.  Urinary pyridinoline cross-links in Ehlers-Danlos syndrome type VI.

Authors:  B Steinmann; D R Eyre; P Shao
Journal:  Am J Hum Genet       Date:  1995-12       Impact factor: 11.025

5.  Ehlers-Danlos syndrome type VI: cross-link pattern in tissue and urine sample as a diagnostic marker.

Authors:  Y Açil; U Vetter; R Brenner; P K Müller; J Brinckmann
Journal:  J Am Acad Dermatol       Date:  1995-09       Impact factor: 11.527

6.  Different pattern of collagen cross-links in two sclerotic skin diseases: lipodermatosclerosis and circumscribed scleroderma.

Authors:  J Brinckmann; C M Neess; Y Gaber; H Sobhi; H Notbohm; N Hunzelmann; P P Fietzek; P K Müller; J Risteli; R Gebker; K Scharffetter-Kochanek
Journal:  J Invest Dermatol       Date:  2001-08       Impact factor: 8.551

7.  [A case of sacral meningeal cyst with Marfan syndrome].

Authors:  H Doi; S Sakurai; M Ida; S Sora; S Asamoto; H Sugiyama
Journal:  No Shinkei Geka       Date:  1999-09

Review 8.  Mutations in the lysyl hydroxylase 1 gene that result in enzyme deficiency and the clinical phenotype of Ehlers-Danlos syndrome type VI.

Authors:  H N Yeowell; L C Walker
Journal:  Mol Genet Metab       Date:  2000 Sep-Oct       Impact factor: 4.797

9.  Lysyl-protocollagen hydroxylase deficiency in fibroblasts from siblings with hydroxylysine-deficient collagen.

Authors:  S M Krane; S R Pinnell; R W Erbe
Journal:  Proc Natl Acad Sci U S A       Date:  1972-10       Impact factor: 11.205

10.  Biochemical characterization of variants of the Ehlers-Danlos syndrome type VI.

Authors:  A Ihme; L Risteli; T Krieg; J Risteli; U Feldmann; K Kruse; P K Müller
Journal:  Eur J Clin Invest       Date:  1983-08       Impact factor: 4.686

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  6 in total

1.  Enlarged Dural Sac in Idiopathic Bronchiectasis Implicates Heritable Connective Tissue Gene Variants.

Authors:  M Leigh Anne Daniels; Katherine R Birchard; Jared R Lowe; Michael V Patrone; Peadar G Noone; Michael R Knowles
Journal:  Ann Am Thorac Soc       Date:  2016-10

Review 2.  Ehlers-Danlos syndrome and neurological features: a review.

Authors:  Salvatore Savasta; Pietro Merli; Martino Ruggieri; Lucia Bianchi; Maria Valentina Spartà
Journal:  Childs Nerv Syst       Date:  2010-08-10       Impact factor: 1.475

Review 3.  Neurological manifestations of Ehlers-Danlos syndrome(s): A review.

Authors:  Marco Castori; Nicol C Voermans
Journal:  Iran J Neurol       Date:  2014-10-06

4.  A Case of a Tarlov Cyst in a Pediatric Patient With Ehlers-Danlos Syndrome.

Authors:  Shadi Shams; Riddhima Issar; Tanya Kadrmas-Iannuzzi
Journal:  Cureus       Date:  2022-09-10

Review 5.  Vascular phenotypes in nonvascular subtypes of the Ehlers-Danlos syndrome: a systematic review.

Authors:  Sanne D'hondt; Tim Van Damme; Fransiska Malfait
Journal:  Genet Med       Date:  2017-10-05       Impact factor: 8.822

6.  Agenesis of the corpus callosum: a rare association with Ehlers-Danlos syndrome.

Authors:  Motahareh Afrakhteh; Mostafa Almasi-Dooghaee; Fahimeh Haji Akhoundi
Journal:  Iran J Child Neurol       Date:  2020
  6 in total

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