Intraspinal lesions associated with sacrococcygeal dimples

Harada, Atsuko; Nishiyama, Kenichi; Yoshimura, Junichi; Sano, Masakazu; Fujii, Yukihiko

doi:10.3171/2014.4.peds13431

Cited by 23 publications

(21 citation statements)

References 22 publications

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“…In the only other series examining MRI screening of infants with intergluteal dimples, Harada et al found 16.7% of 84 patients to have FFT (Table 6). 9 No other OSD lesions were identified. In their series, deep dimples were more commonly associated with OSD.…”

Section: Dimplesmentioning

confidence: 97%

“…21 Cutaneous stigmata associated with OSD include dimples, hemangiomas, subcutaneous lipomas, hairy patches, deviated gluteal fold, and skin appendages. 3,4,9,10,15,16,18,26,27 The true prevalence of OSD has not been established. Magnetic resonance imaging is the gold standard for diagnosing the disorder but is expensive and time consuming and often requires the patient to undergo sedation or general anesthesia.…”

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confidence: 99%

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Use of magnetic resonance imaging to detect occult spinal dysraphism in infants

O’Neill¹,

Gallegos

Herron³

et al. 2017

PED

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OBJECTIVE Cutaneous stigmata or congenital anomalies often prompt screening for occult spinal dysraphism (OSD) in asymptomatic infants. While a number of studies have examined the results of ultrasonography (US) screening, less is known about the findings when MRI is used as the primary imaging modality. The object of this study was to assess the results of MRI screening for OSD in infants. METHODS The authors undertook a retrospective review of all infants who had undergone MRI of the lumbar spine to screen for OSD over a 6-year period (September 2006–September 2012). All images had been obtained on modern MRI scanners using sequences optimized to detect OSD, which was defined as any fibrolipoma of the filum terminale (FFT), a conus medullaris ending at or below the L2–3 disc space, as well as more complex lesions such as lipomyelomeningocele (LMM). RESULTS Five hundred twenty-two patients with a mean age of 6.2 months at imaging were included in the study. Indications for imaging included isolated dimple in 235 patients (45%), asymmetrically deviated gluteal cleft in 43 (8%), symmetrically deviated (Y-shaped) gluteal cleft in 38 (7%), hemangioma in 28 (5%), other isolated cutaneous stigmata (subcutaneous lipoma, vestigial tail, hairy patch, and dysplastic skin) in 31 (6%), several of the above stigmata in 97 (18%), and congenital anomalies in 50 (10%). Twenty-three percent (122 patients) of the study population had OSD. Lesions in 19% of these 122 patients were complex OSD consisting of LMM, dermal sinus tract extending to the thecal sac, and lipomeningocele. The majority of OSD lesions (99 patients [81%]) were filar abnormalities, a group including FFT and low-lying conus. The rate of OSD ranged from 12% for patients with asymmetrically deviated gluteal crease to 55% for those with other isolated cutaneous stigmata. Isolated midline dimple was the most common indication for imaging. Among this group, 20% (46 of 235) had OSD. There was no difference in the rate of OSD based on dimple location. Those with OSD had a mean dimple position of 15 mm (SD 11.8) above the coccyx. Those without OSD had a mean dimple position of 12.2 mm (SD 19) above the coccyx (p = 0.25). CONCLUSIONS The prevalence of OSD identified with modern high-resolution MRI screening is significantly higher than that reported with US screening, particularly in patients with dimples. The majority of OSD lesions identified are FFT and low conus. The clinical significance of such lesions remains unclear.

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Section: Dimplesmentioning

confidence: 97%

mentioning

confidence: 99%

Use of magnetic resonance imaging to detect occult spinal dysraphism in infants

O’Neill¹,

Gallegos

Herron³

et al. 2017

PED

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“…A tiny dimple in the sacrococcygeal region often provides a clue for diagnosis, although its pathological significance is still disputed. 5,6 Surgery, if indicated, is straightforward in the majority of cases. On these grounds, Type 4 spinal lipoma is considered distinct from Type 3 in our classification, although both are considered to be instances of "terminal lipoma" in Pang's classification.…”

Section: Figmentioning

confidence: 99%

New classification of spinal lipomas based on embryonic stage

Morota

Ihara

Ogiwara

2017

PED

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OBJECTIVE Spinal lipomas are generally thought to occur as a result of failed primary neurulation. However, some clinical features cannot be explained by this theory. The authors propose a novel classification of spinal lipomas based on embryonic changes seen during primary and secondary neurulation. METHODS A total of 677 patients with occult spinal dysraphism underwent 699 surgeries between August 2002 and May 2015 at the National Center for Child Health and Development and Tokyo Metropolitan Children's Medical Center. This group of patients had 378 spinal lipomas, including 119 conus spinal lipomas, 27 lipomyelomeningoceles, and 232 filum lipomas, which the authors classified into 4 types based on neural tube formation during embryonic development. Type 1 is defined as pure primary neurulation failure; Type 2 ranges from primary to secondary neurulation failure; Type 3 consists of secondary neurulation failure (early phase); and Type 4 is defined as secondary neurulation failure (late phase). The authors also review embryogenesis in secondary neurulation and analyze the clinical utility of the new classification. RESULTS There were 55 Type 1 spinal lipomas, 29 Type 2, 62 Type 3, and 232 Type 4. All filum lipomas fell into the Type 4 spinal lipoma category. Association with anorectal and/or sacral anomalies was seen in none of the Type 1 cases, 15 (52%) of Type 2, 35 (56%) of Type 3, and 31 (13%) of Type 4. Urogenital anomalies were observed in none of the Type 1 or Type 2 cases, 1 (2%) of Type 3, and 28 (12%) of Type 4. Anomaly syndromes were present in none of the Type 1 cases, 6 (21%) of Type 2, 3 (5%) of Type 3, and 16 (7%) of Type 4. Associated anomalies or anomaly syndromes were clearly observed only for Type 2–4 spinal lipomas encompassing failed secondary neurulation. Radical resection was feasible for Type 1 spinal lipomas. CONCLUSIONS Secondary neurulation of the spinal cord gives rise to the conus medullaris and filum terminale, which are often involved in spinal lipomas. Formation of spinal lipomas seems to be a continuous process overlapping primary and secondary neurulation in some cases. Association with other anomalies was higher in Type 2–4 spinal lipomas, which included failed secondary neurulation, than in Type 1 lipomas, with failed primary neurulation. On the other hand, radical resection was indicated for Type 1, but not for Type 2, spinal lipomas. The new classification of spinal lipomas based on embryonic stage has the potential for clinical use and agrees well with both clinical and surgical findings. The classification proposed here is still preliminary. Further studies and verification are necessary to establish its clinical utility.

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“…The search also resulted in one additional paper that was a survey of practice patterns and three review and, or, opinion papers. MRI rather than ultrasound was used in two of the remaining papers . The remaining nine papers addressed the routine use of spine ultrasound in patients with sacral dimples and, or, other cutaneous stigmata.…”

Section: Resultsmentioning

confidence: 99%

“…When these seven patients were excluded, the incidence of MRI abnormalities in the children with isolated simple sacral dimples fell to 8.3%. The authors did not comment on whether any of these individuals required surgery .…”

Section: Discussionmentioning

confidence: 99%