The spinal congenital dermal sinus is a unique form of spinal dysraphism lined by stratified squamous epithelium with innocuous external appearance, which results from the failure of neuroectoderm to separate from the cutaneous ectoderm during the process of neurulation.1-3 Infective complications associated with dermal sinuses are not uncommon.4-6 We report a case of congenital dermal sinus in a youngest patient (15-day female neonate) of world literature associated with huge lumbosacral abscess and discuss the management.
A 15-day female neonate presented with history of 2-day swelling over lumbosacral region associated with fever and listlessness and not accepting food. The neonate was delivered at home through full-term normal vaginal delivery and cried immediately after birth. There was no history of fever or any drug intake during pregnancy by mother, and she was full immunized. On examination, neonate was dull with poor cry. On painful stimuli, she was moving all 4 limbs. Head circumference was 33 cm, and anterior fontanel was open, lax, and pulsatile. Local examination revealed a huge swelling over lumbosacral region with red and shiny but intact skin with poor local hygiene (Fig. 1). There was a small opening just below the swelling. A diagnosis of congenital dermal sinus with abscess and septicemia was suspected. In view of poor general condition and lack of facilities to perform a magnetic resonance imaging (MRI) in such a small child, an ultrasound was performed. Ultrasound sound showed a hypoechoic lesion superficial to deep fascia without any evidence of intraspinal extension or sinus tract (Figs. 2A, B). The child was started on broad-spectrum antibiotics (Inj. Ceftriaxone, Inj. Amikacin, and Inj. Metronidazole) and planned for incision and drainage. After opening the abscess, thick foul smelling pus came out, and without manipulating the abscess cavity too much, it was drained (Fig. 3). Pus culture showed coagulase positive Staphylococcus aureus sensitive to all antibiotics. Baby started improving during the next 24 hours, her fever subsided, and she started accepting food. Antibiotics were continued fora week with daily dressing. Follow-up ultrasound also did not show the evidence of sinus tract, and the baby is doing well at follow-up.
Most spinal congenital dermal sinus (SCDS) lesions occur in the lumbar or lumbosacral region followed by the occipital and thoracic regions, respectively.3,7 It is estimated that 2% to 4% of children harbor intergluteal dorsal dermal sinuses.3 Spinal congenital dermal sinus may communicate between the surface of the skin and deeper structures and may occur anywhere along the craniospinal axis and may be often accompanied by other cutaneous stigmata, various dysraphic abnormalities, or intraspinal tumors.3 The inward extent of the tract depends on the extent of adhesions; the tract may actually end blindly in the subcutaneous tissue, or it may extend into the spinal canal.3,8,9 The lesions over and around sacrococcygeal region include coccygeal pits and dermal sinus tracts, and these tows are distinctly different clinical entities.10 It is important for clinicians to be able to distinguish between the benign coccygeal pits and the potentially more ominous SCDSs.11 The dermal sinus tract may end blindly in the subcutaneous tissue, or it may extend into the spinal canal.3 Intergluteal dorsal dermal sinuses are relatively common and do not seem to be associated with significant risk of spinal cord and intraspinal anomalies.3 Simple intergluteal dorsal dermal sinuses without other cutaneous findings do not require radiographic or surgical evaluation and treatment, unless other markers or neurological symptoms are present.3 However, one should have a high index of suspicion for all the dimples above the intergluteal fold, despite a normal examination or neuroradiological workup.12 MRI is the investigation of choice and can demonstrate extraspinal tract path, inclusion tumors, and associated spinal dysraphism.5,12 Although MRI can provide exquisite anatomic images, thermal instability, the need for sedation, and expense contribute to the impracticality of screening with MRI in most.13 Ultrasonography is a well-established method of investigating the spinal canal and cord as well as the meningeal coverings in newborns and infants.14,15 Ultrasonography can demonstrate the entire spectrum of intraspinal anatomy and pathologic conditions with high geometric resolution.16 The availability, portability, and high-resolution capabilities of spinal sonography and normal lack of ossification of the posterior spinal elements provide an acoustic window for visualization of the entire intracanalicular contents until the infant is 5 or 6 months of age and makes ultrasound a valuable screening procedure in this age group.13,16,17 Scrupulously performed spinal ultrasound can also show the entire length of the dermal sinus tract from the skin to the spinal cord.13 In present case, ultrasound findings suggested the location of abscess confined to the subcutaneous plane without any intraspinal component and sinus tract. An inconspicuous ultrasound examination does not need a further imaging, but suspicious results of sonography need an MRI dependent of clinical conditions.18 The detail knowledge of SCDS is important because these lesionsmay become susceptible to local recurrent infection from trauma or hirsutism.3 Spinal congenital dermal sinus can be complicated by intramedullary abscess,5 meningitis,3,4,12 and spinal subdural abscess.4,6 In present case, proximity of sinus opening to anus and poor hygiene resulted in the infection and development of subcutaneous abscess. Classically, all midline skin dimples (or SCDS) above the intergluteal fold must be assumed to communicate intraspinally, and those below the top of crease are blind sacrococcygeal dimples not requiring exploration.12 In our case, the opening was located just above the intergluteal fold. In view of poor general condition and lack of facilities to perform MRI in such a small child, we used ultrasound to evaluate the location and extent of the lesion successfully. There is controversy regarding the evaluation and management of cutaneous defects in the coccygeal region.3 This controversy varies from radiographic and/or surgical evaluation in all cases,6,12,19,20 to investigations based on clinical findings,19 and some believe that all coccygeal dimples or sinuses are innocent and warrant no additional evaluation other than physical examination.13,21 Spinal congenital dermal sinus, although rarely, can present with subcutaneous abscess. In the present case, with the help of ultrasound and minimal surgical intervention in the form of incision and drainage in a sick neonate, we could achieve good result.
1. Wang KC, Yang HJ, Oh CW, et al. Spinal congenital dermal sinus-experience of 5 cases over a period of 10 years. J Korean Med Sci
2. Paul MK, Park TS. Dermoids and dermal sinus tracts of the spine. Neurosurg Clin N Am
3. Weprin BE, Oakes WJ. Coccygeal pits. Pediatrics
4. van Aalst J, Beuls EA, Cornips EM, et al. Anatomy and surgery of the infected dermal sinus of the lower spine. Childs Nerv Syst
5. Bunyaratavej K, Desudchit T, Pongpunlert W. Holocord intramedullary abscess due to dermal sinus in a 2-month-old child successfully treated with limited myelotomy and aspiration. Case report. J Neurosurg
. 2006;104(4 Suppl):269-274.
6. Park SW, Yoon SH, Cho KH, et al. Infantile lumbosacral spinal subdural abscess with sacral dermal sinus tract. Spine
7. Barkovich AJ, Edwards MSB, Cogen PH. MR evaluation of spinal dermal tracts in children. AJNR Am J Neuroradiol
8. Pang D. Split cord malformations-Part 2: clinical syndrome. Neurosurgery
9. McLone DG, Naidich TP. The tethered spinal cord. In: McLaurin RL, Venes JL, eds. Pediatric Neurosurgery
, 2nd ed. Philadelphia: WB Saunders; 1989;71-96.
10. Elton S, Oakes WJ. Dermal sinus tracts of the spine. Neurosurg Focus
11. Ackerman LL, Menezes AH. Spinal congenital dermal sinuses: a 30-year experience. Pediatrics
12. Jindal A, Mahapatra AK. Spinal congenital dermal sinus: an experience of 23 cases over 7 years. Neurol India
13. Korsvik HE, Keller MS. Sonography of occult spinal dysraphism in neonates and infants with MR imaging correlation. Radiographics
14. Cramer BC, Jequier SO, Gorman AM. Ultrasound of the neonatal craniocervical junction. AJNR Am J Neuroradiol
15. Zieger M, Dorr U, Schulz RD. Pediatric spinal sonography. II. Malformations and mass lesions. Pediatr Radiol
16. Unsinn KM, Geley T, Freund MC, et al. US of the spinal cord in newborns: spectrum of normal findings, variants, congenital anomalies, and acquired diseases. Radiographics
17. Raghavendra BN, Epstein FJ, Pinto RS, et al. The tethered spinal cord: diagnosis by high-resolution real-time ultrasound. Radiology
18. Schenk JP, Herweh C, Günther P, et al. Imaging of congenital anomalies and variations of the caudal spine and back in neonates and small infants. Eur J Radiol
19. Haworth JC, Zachary RB. Congenital dermal sinuses in children-their relation to pilonidal sinus. Lancet
20. Kajiwara H, Matsukado Y, Hiraki T, et al. Intraspinal communication of sacrococcygeal dermal sinuses. Childs Nerv Syst
21. Kanev PM, Park TS. Dermoids and dermal sinus tracts of the spine. Neurosurg Clin North Am