Dorsal midline cutaneous stigmata associated with occult spinal dysraphism in pediatric patients

Sung, Hyun Jung; Lee, Hyun-Seung

doi:10.3345/kjp.2018.06744

Cited by 14 publications

(9 citation statements)

References 29 publications

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“…The prevalence of OSD was significantly higher in patients with combined stigmata (10.5%) than those with a single stigmata (2.3%). This finding is consistent with the results of previous studies that reported that a combination of 2 or more midline cutaneous lesions is more likely to be associated with OSD than a single lesion . Therefore, when evaluating infants with dorsal midline skin lesions, meticulous screening for skin lesions is warranted to determine whether 2 or more lesions are present, given that this indicates a higher risk of OSD.…”

Section: Discussionsupporting

confidence: 90%

“…This finding is consistent with the results of previous studies that reported that a combination of 2 or more midline cutaneous lesions is more likely to be associated with OSD than a single lesion. 25,26 Therefore, when evaluating infants with dorsal midline skin lesions, meticulous screening for skin lesions is warranted to determine whether 2 or more lesions are present, given that this indicates a higher risk of OSD. However, some studies in our current review focused on specific skin lesions, such as spinal strawberry nevi 5 and dorsal midline vascular anomalies, 8 that are associated with other stigmata.…”

Section: Discussionmentioning

confidence: 99%

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Incidence of Occult Spinal Dysraphism Among Infants With Cutaneous Stigmata and Proportion Managed With Neurosurgery

et al. 2020

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IMPORTANCE Occult spinal dysraphism (OSD) is the most common congenital spinal anomaly.Cutaneous anomalies such as skin dimples or deviated gluteal folds are well known as stigmata of OSD and are indicators for further evaluation; however, the association between cutaneous anomalies and OSD has not been systemically evaluated.OBJECTIVE To evaluate the incidence of OSD and the proportion of OSD cases managed with a neurosurgical intervention among neonates or infants with various cutaneous stigmata.DATA SOURCES PubMed and Embase databases were searched for studies published up to July 25, 2018, that evaluated the proportion of OSD cases in neonates or infants with cutaneous stigmata.Search terms included ultrasound, dysraphism, dimple, and infant or neonate. The search was limited to English-language publications. STUDY SELECTION Two reviewers selected the studies evaluating the incidence of OSD among neonates or infants with cutaneous stigmata. DATA EXTRACTION AND SYNTHESIS The Preferred Reporting Items for Systematic Reviews andMeta-analyses (PRISMA) guidelines for data extraction were followed. Pooled proportions of OSD cases and OSD cases that were managed with a neurosurgical intervention were obtained using the generalized linear mixed model and maximum likelihood method. MAIN OUTCOME AND MEASURESThe pooled incidence of OSD and OSD cases managed with neurological surgery among patients with cutaneous stigmata was the primary outcome. This outcome was also evaluated in each subgroup, and heterogeneity was explored using subgroup analysis. RESULTSA total of 15 studies, involving 6558 neonate or infant patients with various cutaneous stigmata, were included. The pooled proportion of OSD cases among the patients with cutaneous stigmata was 2.8% (95% CI, 2.1%-3.8%; I 2 = 51.6%), and the proportion managed with neurological surgery was 0.6% (95% CI, 0.3%-1.3%; I 2 = 66.4%). Cases with combined stigmata showed a significantly higher association with OSD than those with a single stigma (10.5% [95% CI, 6.9%-15.8%] vs 2.3% [%, 95% CI, 1.5%-3.5%]; P < .001). The pooled proportion of OSD cases among patients with an atypical dimple was significantly higher than among those with simple dimple (8.8% [95% CI, 4.5%-16.6%] vs 0.6% [95% CI of 1.4%-2.1%]; P = .001). CONCLUSIONS AND RELEVANCEThe proportion of OSD in healthy, asymptomatic patients with midline cutaneous stigmata was low, and the proportion of patients who underwent a neurosurgical (continued) Key Points Question What is the incidence of occult spinal dysraphism (OSD) in neonates or infants with various cutaneous stigmata, and how many of these cases are managed with a neurosurgical intervention? Findings In this systematic review and meta-analysis of 15 studies involving 6558 patients, the pooled proportion of OSD among cases with cutaneous stigmata was 2.8%, and in 0.6% of patients, the condition was managed with neurological surgery. A stronger association with OSD was found in patients with combined stigmata and atypical dimple. Meaning The findings of this s...

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Section: Discussionsupporting

confidence: 90%

Section: Discussionmentioning

confidence: 99%

Incidence of Occult Spinal Dysraphism Among Infants With Cutaneous Stigmata and Proportion Managed With Neurosurgery

et al. 2020

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“…Our findings concur with previous studies that >1 LsCM portends a significantly higher risk of OSD than a single LsCM. 3,10–12…”

Section: Discussionmentioning

confidence: 99%

Risk of occult spinal dysraphism based on lumbosacral cutaneous manifestations

et al. 2021

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Objectives: Occult spinal dysraphism is a congenital failure of fusion of the posterior vertebral arches with intact skin overlying the defect. Lumbosacral cutaneous manifestations are associated with a variable risk of occult spinal dysraphism. Tethered cord syndrome is a type of occult spinal dysraphism that puts abnormal traction on the spinal cord. This study analyzed neonates and infants who were referred to our pediatric urology practice and had evidence of lumbosacral cutaneous manifestation on physical examination. Methods: We reviewed the presence of lumbosacral cutaneous manifestations in neonates and infants evaluated in our pediatric urology clinic at our Institution over a 6-year period (1 March, 2015–28 February, 2021) with no prior diagnosis of lumbosacral cutaneous manifestation. All patients underwent a spinal ultrasound. Results: The most common lumbosacral cutaneous manifestations were bifurcated/duplicated gluteal folds (33%), gluteal asymmetry (19%), and sacral dimples (14%). A total of 34 (24%) patients had an abnormal spinal ultrasound; 15 (44%) of these infants underwent a lumbar magnetic resonance imaging. A coccygeal pit was statistically marginally higher in abnormal versus normal spinal ultrasound (p = 0.07). Patients with only one lumbosacral cutaneous manifestation (N = 121) were significantly more likely to have a normal spinal ultrasound compared to those with two or more lumbosacral cutaneous manifestation (N = 17) (79% vs 53%, p = 0.03). Conclusion: Due to the varying risk of certain lumbosacral cutaneous manifestations with occult spinal dysraphism, all patients with a lumbosacral cutaneous manifestation should undergo spinal ultrasound. This study also highlights the importance of urodynamic studies when there are abnormal cutaneous findings. Routine physical examinations of the lumbar region for cutaneous manifestations of occult spinal dysraphism are vital to ensure prompt management of tethered cord syndrome and avoid potentially devastating consequences.

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“…[6] Penelitian melaporkan bahwa jika ditemukan stigmata kulit di garis tengah punggung, misalnya massa fibrofatty, lesung kulit di daerah sakral, aplasia kulit, dan hipertrikosis maka perlu diwaspadai adanya SB okulta, terutama jika terdapat kelainan kongenital lainnya. [7] Spina bifida aperta (SBA) disebut juga SB cystica, saat lahir terlihat sebagai jaringan saraf yang terbuka dengan atau tanpa kantung yang menonjol di lokasi lesi. SBA dapat berupa myeloschisis atau myelomeningocele.…”

Section: Etiologiunclassified

Kelainan Kongenital Multipel Pada Neonatus Dengan Spina Bifida: Serial Kasus

Mukminin

Diarsvitri²

2022

SBJ

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Pendahuluan: Spina bifida (SB) merupakan suatu kelainan kongenital sistem saraf pusat dengan prevalensi tertinggi dalam spektrum cacat tabung saraf (neural tube defect) akibat gangguan penutupan tulang belakang janin pada bulan pertama kehamilan. Laporan kasus: Dilaporkan dua kasus SB pada bayi laki-laki berusia dua hari dan tiga hari yang lahir prematur. Pada kedua pasien ditemukan massa di area lumbosakral disertai rembesan cairan serebrospinal (cerebro spinal fluid) serta kelainan kongenital multipel antara lain berupa congenital talipes equinovarus (CTEV) bilateral, hipospadia dan atresia ani. Kedua pasien menjalani pemeriksaan darah, ekokardiografi, CT scan tanpa kontras dan babygram. Hasil pemeriksaan darah menunjukkan peningkatan prothrombine time, thromboplastine time dan kalium. Hasil CT scan menunjukkan ventriculomegali dan edema otak. Echocardiography menunjukkan adanya PDA. Pasien menjalani kolostomi, repair spina bifida dan pemasangan ventriculoperitoneal (VP) shunt. Pembahasan: SB sering disertai dengan kelainan kongenital lain yang berasal dari lapisan neuroektoderm saat embriogenesis. Segera setelah bayi lahir perlu dilakukan pemeriksaan dan manajemen secara komprehensif dari tim multidisiplin terkait adanya bagian neural placode yang terbuka untuk mengetahui lokasi, ukuran, apakah ada kebocoran cairan serebrospinal serta kelainan kongenital lain yang menyertai.

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Dorsal midline cutaneous stigmata associated with occult spinal dysraphism in pediatric patients

Cited by 14 publications

References 29 publications

Incidence of Occult Spinal Dysraphism Among Infants With Cutaneous Stigmata and Proportion Managed With Neurosurgery

Incidence of Occult Spinal Dysraphism Among Infants With Cutaneous Stigmata and Proportion Managed With Neurosurgery

Risk of occult spinal dysraphism based on lumbosacral cutaneous manifestations

Kelainan Kongenital Multipel Pada Neonatus Dengan Spina Bifida: Serial Kasus

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