Acute presentations of intradural lipomas: case reports and a review of the literature.
Natural history
Prophylactic surgery
Spina bifida occulta
Spinal lipoma
Surgical indications
Journal
BMC neurology
ISSN: 1471-2377
Titre abrégé: BMC Neurol
Pays: England
ID NLM: 100968555
Informations de publication
Date de publication:
08 Aug 2019
08 Aug 2019
Historique:
received:
11
03
2019
accepted:
25
07
2019
entrez:
10
8
2019
pubmed:
10
8
2019
medline:
30
11
2019
Statut:
epublish
Résumé
Lumbosacral lipomas (LLs) may remain asymptomatic or lead to progressive neurological deterioration. However, sudden neurological deterioration is a rare and severe event. Herein, we report rare occurrences of sudden clinical deterioration in two previously asymptomatic children harbouring intradural LLs without dermal sinus tracts or signs of occult dysraphism. A review of the pertinent literature is also included. One child exhibited acute deterioration because of an epidural abscess associated with a filar lipoma without a sinus tract (probably caused by haematogenous spreading from a respiratory tract multiple infection), and the other child exhibited acute deterioration because of a very large, holocord syringomyelia-like cyst associated with a small conus lipoma. Both patients were 4 years old. In case #2, a previously undetected, severe tethered cord (conus at the S3-S4 level) was also present. A complete recovery was attained after an urgent surgical operation in both cases (in addition to targeted antibiotic therapy in case #1). All cases of deterioration in the literature were caused by abscess formation in dermal sinus tracts. Prophylactic surgery may be indicated even in asymptomatic children that have tethered cord and surgically favourable LLs (small dorsal and filar LLs), especially if the conditions are associated with progressive syringomyelia. Similarly, intradural dermal sinus tracts should be regarded as surgery-indicated, even if the conus is in its normal position and the patient is asymptomatic because there is a consistent risk of severe, infection-related complications. Finally, asymptomatic patients with filar LLs and a normally located conus can be candidates for surgery or an accurate clinical and radiological follow-up.
Sections du résumé
BACKGROUND
BACKGROUND
Lumbosacral lipomas (LLs) may remain asymptomatic or lead to progressive neurological deterioration. However, sudden neurological deterioration is a rare and severe event. Herein, we report rare occurrences of sudden clinical deterioration in two previously asymptomatic children harbouring intradural LLs without dermal sinus tracts or signs of occult dysraphism. A review of the pertinent literature is also included.
CASE PRESENTATION
METHODS
One child exhibited acute deterioration because of an epidural abscess associated with a filar lipoma without a sinus tract (probably caused by haematogenous spreading from a respiratory tract multiple infection), and the other child exhibited acute deterioration because of a very large, holocord syringomyelia-like cyst associated with a small conus lipoma. Both patients were 4 years old. In case #2, a previously undetected, severe tethered cord (conus at the S3-S4 level) was also present. A complete recovery was attained after an urgent surgical operation in both cases (in addition to targeted antibiotic therapy in case #1). All cases of deterioration in the literature were caused by abscess formation in dermal sinus tracts.
CONCLUSIONS
CONCLUSIONS
Prophylactic surgery may be indicated even in asymptomatic children that have tethered cord and surgically favourable LLs (small dorsal and filar LLs), especially if the conditions are associated with progressive syringomyelia. Similarly, intradural dermal sinus tracts should be regarded as surgery-indicated, even if the conus is in its normal position and the patient is asymptomatic because there is a consistent risk of severe, infection-related complications. Finally, asymptomatic patients with filar LLs and a normally located conus can be candidates for surgery or an accurate clinical and radiological follow-up.
Identifiants
pubmed: 31395022
doi: 10.1186/s12883-019-1413-4
pii: 10.1186/s12883-019-1413-4
pmc: PMC6688213
doi:
Types de publication
Case Reports
Journal Article
Review
Langues
eng
Sous-ensembles de citation
IM
Pagination
189Références
Childs Nerv Syst. 1999 Sep;15(9):439-42; discussion 443
pubmed: 10502000
Radiographics. 2000 Jul-Aug;20(4):923-38
pubmed: 10903684
Neuroradiology. 2000 Jul;42(7):471-91
pubmed: 10952179
Eur J Pediatr Surg. 2000 Dec;10 Suppl 1:13-7
pubmed: 11214824
Acta Neurochir (Wien). 2001 Sep;143(9):857-64
pubmed: 11685617
Joint Bone Spine. 2002 Oct;69(5):511-4
pubmed: 12477239
Neurosurgery. 2004 Apr;54(4):868-73; discussion 873-5
pubmed: 15046652
Pediatr Neurosurg. 2004 Mar-Apr;40(2):51-7; discussion 58
pubmed: 15292632
Eur J Radiol. 2004 Sep;51(3):234-40
pubmed: 15294330
Arch Dermatol. 2004 Sep;140(9):1109-15
pubmed: 15381552
Surg Neurol. 2005;63 Suppl 1:S8-12
pubmed: 15629346
Acta Neurochir (Wien). 2009 Mar;151(3):189-97; discussion 197
pubmed: 19240973
J Neurosurg Pediatr. 2009 May;3(5):412-9
pubmed: 19409021
Neurosurgery. 2009 Sep;65(3):511-28; discussion 528-9
pubmed: 19687697
Neurosurgery. 2010 Feb;66(2):253-72; discussion 272-3
pubmed: 20042988
Childs Nerv Syst. 2012 Oct;28(10):1731-9
pubmed: 22562193
Childs Nerv Syst. 2012 Oct;28(10):1741-2
pubmed: 22661145
J Neurosurg. 1990 Jul;73(1):48-52
pubmed: 2352021
J Pediatr Neurosci. 2014 Jan;9(1):21-6
pubmed: 24891897
J Neurosurg Pediatr. 2014 Sep;14(3):245-54
pubmed: 24971607
J Paediatr Child Health. 2015 Feb;51(2):223-5
pubmed: 25099316
AJNR Am J Neuroradiol. 2015 Feb;36(2):317-22
pubmed: 25300986
J Pediatr Neurosci. 2015 Jan-Mar;10(1):73-5
pubmed: 25878753
Neurol Med Chir (Tokyo). 2015;55(9):695-721
pubmed: 26345666
J Neurosurg Pediatr. 2016 Jan;17(1):86-93
pubmed: 26431244
Surg Neurol Int. 2015 Oct 07;6(Suppl 17):S429-34
pubmed: 26539316
Childs Nerv Syst. 2016 Jul;32(7):1265-72
pubmed: 27060067
J Neurosurg Pediatr. 2016 Aug;18(2):171-6
pubmed: 27082975
Case Rep Neurol Med. 2016;2016:8918954
pubmed: 28050293
Childs Nerv Syst. 2017 Apr;33(4):561-562
pubmed: 28236066
Childs Nerv Syst. 2018 Feb;34(2):285-291
pubmed: 29075839
Eur J Pediatr Surg. 2019 Jun;29(3):229-238
pubmed: 29857348
SAGE Open Med Case Rep. 2018 May 31;6:2050313X18775584
pubmed: 29899987
Rev Med Chil. 2018 Jul;146(7):899-906
pubmed: 30534890
J Neurosurg. 1985 Jan;62(1):1-8
pubmed: 3964839
J Clin Microbiol. 1983 Apr;17(4):628-30
pubmed: 6853690
Neurosurg Clin N Am. 1995 Apr;6(2):311-23
pubmed: 7620356
J Neurosurg. 1995 May;82(5):752-5
pubmed: 7714598
Neurosurgery. 1995 Dec;37(6):1212-5
pubmed: 8584165
Childs Nerv Syst. 1997 Jun;13(6):298-334; discussion 335
pubmed: 9272285