Literature DB >> 11504608

Encephalocraniocutaneous lipomatosis associated with iris coloboma, chorioretinitis and spinal cord involvement: a case report.

G Deda1, H Caksen, G Yavuzer, T Arasil.   

Abstract

A 9-year-old boy with a history of lipoma excision and laminectomy at the Th10-11 levels, resulting in incomplete paraparesis and neurogenic bladder, was admitted for a comprehensive rehabilitation programme. Physical examination revealed an ipsilateral focal dermal hypoplastic defect within an area of alopecia and a subcutaneous lipomatous tissue on the left temporo-parietal region of the scalp. Iris coloboma and chorioretinitis were diagnosed on the left eye. He also had mild mental retardation and triparesis. Magnetic resonance imaging of the brain and the spine demonstrated hyperintense masses which were consistent with lipoma. Although in the literature three cases of encephalocraniocutaneous lipomatosis (ECCL) concomitant with spinal cord involvement have been reported, to our knowledge iris coloboma and chorioretinitis in ECCL have not been reported previously. In conclusion, we would like to stress that aside from known ophthalmological malformations, iris coloboma and chorioretinitis may also be observed in ECCL and that all patients who have been diagnosed as having ECCL should be examined for spinal cord involvement.

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Year:  2001        PMID: 11504608     DOI: 10.1016/s0387-7604(01)00219-4

Source DB:  PubMed          Journal:  Brain Dev        ISSN: 0387-7604            Impact factor:   1.961


  3 in total

Review 1.  Encephalocraniocutaneous lipomatosis: a case with unilateral odontomas and review of the literature.

Authors:  Kristina Hauber; Monika Warmuth-Metz; Christian Rose; Eva-Bettina Bröcker; Henning Hamm
Journal:  Eur J Pediatr       Date:  2003-07-22       Impact factor: 3.183

2.  Encephalocraniocutaneous lipomatosis, a rare neurocutaneous disorder: report of additional three cases.

Authors:  O Kocak; C Yarar; K B Carman
Journal:  Childs Nerv Syst       Date:  2015-08-01       Impact factor: 1.475

Review 3.  Clinical manifestation and neurosurgical intervention of encephalocraniocutaneous lipomatosis--a case report and review of the literature.

Authors:  Chia-Chun Chiang; Shih-Chieh Lin; Hsiu-Mei Wu; Jia-Chi Wang; Tsui-Fen Yang; Hsin-Hung Chen; Donald M Ho; Tai-Tong Wong
Journal:  Childs Nerv Syst       Date:  2013-08-24       Impact factor: 1.475

  3 in total

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