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A New Understanding of Dorsal Dysraphism with Lipoma (Lipomyeloschisis): Radiologic Evaluation and Surgical Correction

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A New Understanding of Dorsal Dysraphism with Lipoma (Lipomyeloschisis): Radiologic Evaluation and Surgical Correction 103 A New Understanding of Dorsal Dysraphism with Lipoma (Lipomyeloschisis): Radiologic Evaluation and Surgical Correction Thomas P. Naidich 1 The spinal anomaly designated dorsal dysraphism with lipoma (Iipomyeloschisis) David G. McLone2 consists of skin-covered, focal spina bifida; focal partial clefting of the dorsal half of 2 Saffet Mutluer . 3 the spinal cord; continuity of the dorsal cleft with the central canal of the cord above (and occasionally below) the cleft; deficiency of the dura underlying the spina bifida; deep extension of subcutaneous lipoma through the spina bifid a and the dural defi­ ciency to insert directly into the cleft on the dorsal half of the cord; variable cephalic extension of lipoma into the contiguous central canal of the cord; and variable balloon­ ing of the subarachnoid space to form an associ3ted meningocele. The variable individual expressions of the anomaly are best understood by reference to their archetypal concept. Careful analysis of radiographic and surgical findings in human lipomyeloschisis and correlation with an animal model of lipomyeloschisis indicate that plain spine radiographs and high-resolution metrizamide computed tomographic my­ elography successfully delineate the precise anatomic derangements associated with lipomyeloschisis and provide the proper basis for planning surgical therapy of this condition. Spinal lipomas are distinct collections of fat associated with an abnormally large amount of connective tissue; they appear at least parti ally encapsulated, and have a definite connection with the leptomeninges or spinal cord [1). Intradural lipoma and lipomyelome(l ingocele are two forms of lipomyeloschisis (dorsal dysraphism with lipoma) [2-41). Analysis of a series of 14 patients suggests that these conditions may be understood most easil y in term s of variations about an archetype of the anomaly. Di splay of the specific anatomic derangements at every level by high- resolution transverse metrizamide computed tomographic (eT) myelography provides an important basis for pl anning correc­ tive surgery and for assessing both operative risk and the likelihood of successful outcome. This article appears in the March/ April 1983 issue of AJNR and the June 1983 issue of AJR. Received June 14. 1982; accepted after revi­ Materials and Methods sion October 16, 1982. The study material includes the presurgical pl ain spine radiographs, routine metrizamide Presented at the annual meeting of the Ameri­ can Society of Neuroradiology, Ch icago, April myelograms, and metrizamide CT myelograms from each of 14 pati ents with lipomyelo­ 1981. schisis. There were 11 females and three males. Six patients were between 1 month and , Departm ent of Radiology, Northwestern Uni­ 1 year of age. Seven were 2 -1 2 years old, and one was 35 years old. Th e lipoma was versity Medical School, and Children 's Memorial situated precisely in the midline in two pati ents and extended asymmetricall y away from the Hospital, 2300 Children's Plaza, Ch icago, IL midline in 10 pati ents (to th e left : seven; to the right: three). In two oth er pati ents, the site 60614. Address reprint requests to T. P. Naidich. of lipoma could not be determined because of pri or surgery. One pati ent had a skin tag 2 Department of Neurosurgery, Children's Me­ associated with the lipoma. morial Hospital and Northwestern University Med­ Five of the 14 pati ents had prior partial surgical correcti on of th e superficial extradural ical School, Chicago, IL 60614. component of the lipomyelomeningocele. All 14 patients underwent subsequent definitive 3 Present ad dress: Departm ent of Neurosur­ gery, Eg e Unive rsity Medical School, Bornova, surgery, at which time surgical observations were recorded, and intraoperati ve photograph s Izmir, Turkey. of the surgical dissecti on were obtained to document the correlation between radiography and in vivo anatomy. The fin al diagnoses were lipomyelomeningocele (seven pati ents), AJNR 4:103-116, March/ April 1983 0195-6108/ 83/ 0402- 0103 $00.00 intradural lipoma (two), and lipomyeloschi sis (exact nature obscured by partial pri or © American Roentgen Ray Society treatm ent) (five). 104 NAIDICH ET AL. AJNR:4, Mar. l Apr. 1983 A B c Fig. 1 .-Cross-sectional anatomy. A and B, Axial sections of 2 1-day-old dorsal halves of placode and projects posteri or to neural tissue within dorsal myeloschisti c chicken embryo with dorsal glycogen body. Myelin stain. dural defic iency. White Gs and Ms in dicate dorsal root ganglia and paraspinal Ventral lies toward top. Relations of normal av ian lumbosacral glycogen body musculature, respecti vely. B, 5 mm cephalad. Intact neural arch (A). Paired and dura depicted here are believed to be a paradigm for human lipomyelo­ ventral (V) and dorsal (D) nerve roots arise from pial surface of neural tissue. schisis. A, Intact skin (single crossed arrow) covers spina bifida. Bifid laminae Glycogen body ascends within dorsal deficiency to underlie intact laminae of (L) are joined by fibrovascular band (open arrowheads ). Dura (closed arrow­ more cephalic vertebra. Glycogen body only appears to be intradural at this heads) underlies entire in ner surface of vertebral canal except directly level. C, Anatomi c relati ons in human lipomyeloschi sis. Ventral at top. Dura beneath spina bifida. Instead of crossing under spina bifida, dura is reflected (small arrowheads ), placode (P), and subarachnoid space (SAS) extend out onto posterolateral aspect of placode (P) at ipsilateral dorsal nerve root entry of spinal canal between th e bifid laminae (L). Dura inserts onto placode just zone (asterisks ), leaving a median dorsal dural deficiency (DOD). Left and dorsal to entry of dorsal nerve roots (D). Pia-arachnoid forms single contin­ right halves of neural tissue are joined only ventral to remnant of central canal uous membrane whic h lines dura and is refl ected over ventral surface of (double crossed arrow) of spinal cord. Ventrat surface of placode is placode. Skin (crossed arrows ) is intact. Subcutaneous lipoma (U) inserts lined by pia mater, whic h is continuou s with arachnoid mater at dorsal root onto dorsal face of lipoma via fibrous subareolar layer (large arrowhead). G entry zones. Subarachnoid space (SAS) is c rossed by multiple arachnoid = dorsal root ganglion; M = paraspinal muscle. (Reprinted from [22].) trabeculae. Norm al avian glycogen body (black G) occupies midline between In each pati ent, preoperative myelography was performed in the CT and correlation with surgical findings, we present an decubitus position with the lipoma side dependent via lateral C1- archetype of lipomyeloschisis against which the details of C2 puncture monitored by vertical-beam fluoroscopy using metri­ each individual may be viewed as variations on a theme (fig . zamide in a concentration of 190-220 mg 11 m!. About 2 hr after 1). myelography, each patient underwent CT using an EMI 5005 gen­ Patients with dorsal dysraphism and lipoma typically ex­ eral scann er. In 11 of the 14 patients, CT was performed with the hibit a subcutaneous soft-tissue mass that occupies the high-resolution sector scan modification [42], using contiguous 5- mm-thick axial sections, 25.4 cm wedges, 140 kVp, 28 mA, and 70 midline and usually extends asymmetrically toward one side. sec scan time. Direct coronal im ages and computer reformatted Deep to this mass is a relatively focal spina bifid a that coronal and sagittal images were obtained in selected cases. usually involves the lumbosacral spine. The transition from Surgical and radiographic data were th en correlated with the normal to bifid vertebrae may be abrupt or gradual. A thick anatomic derangements observed in myelin-stained transverse sec­ fibrovascular band joins the laminae of the most cephalic tions of chicken embryos in which spina bifida was induced by vertebra with widely bifid laminae. Patients in whom a normal dorsal midline incision of the caudal spine and neural tube at 72- spinal canal reforms below the spina bifida frequently exhibit 90 hr after fertilizati on (just after closure of the posterior neuropore); a fibrovascular band joining the laminae of the most caudal the embryos were sacrificed at term (2 1 days) (fig. 1) [43 -47]. vertebra with widely bifid laminae as well. The band is Detail s of this technique for inducing myeloschisis and the results continuous with the periosteum of the laminae. The bands of that study are to be the subject of a separate report. are not present in the center of the spina bifida, and appear to be transitional elements that roof-over the canal at the Anatomic Relations in Lipomyeloschisis cranial and caudal ends of the spina bifida. Dossetor et al. [9], James and Oliff [14], Kaiser et al. In patients with lipomyelomeningocele, the meningocele [17], Kapl an and Quencer [18], Lohkamp et al. [23], Resjo and spinal cord herniate posteriorly into the subcutaneous et al. [30], Scatliff et al. [33], Schroeder et al. [34], and tissue under the fibrovascular band (NOT under the last Wolpert et al. [40] have contributed to our understanding of intact arch nor under narrowly bifid laminae). The fibrovas­ the CT appearance of dorsal dysraphism with lipoma. Bulcke cular band kinks and notches the superior surface of the et al. [48], Osborn and Koehl er [49], Chiu and Schapiro spinal cord and meningocele in all cases of lipomyelomen­ [50], and Schapiro and Chiu [51] have contributed to our ingocele. This band appears to tether the meningocele sac understanding of the CT appearance of the paraspinal mus­ and neural tissue, since they relax after surgical section of cul ature. On th e basis of our experience with high-resolution this band, and since, in at least some cases, cortical-evoked AJNR:4, Mar. / Apr. 1983 LlPOMYELOSCHISIS 105 Fig. 2.-Lipomyeloschisis in 10-year-old girl wilh previous, purely extradural repair of lipomye­ loschi sis. A and B, Axial secti on metrizamide CT.
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