Two Cases of Large Filar Cyst Associated with Terminal Lipoma: Relationship with Retained Medullary Cord

Mukae, Nobutaka; Morioka, Takato; Suzuki, Satoshi; Murakami, Nobuya; Shimogawa, Takafumi; Kanata, Akiko; Tsukamoto, Haruhisa; Mizoguchi, Masahiro

doi:10.1016/j.wneu.2020.07.026

Cited by 11 publications

(22 citation statements)

References 15 publications

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“…Therefore, the histopathological coexistence of the E-LC w/NGT with filar lipoma and TFT demonstrated in this study, is not a surprising finding. [ 16 , 19 - 21 ] As the terminology for RMC, and lipoma and TFT was based on different backgrounds, namely, embryological and morphological backgrounds, respectively, it may be difficult in clinical practice to make a distinct diagnosis between these three entities.…”

Section: Discussionmentioning

confidence: 99%

“…Furthermore, the diameter of the C-LS with RMC was <2 mm, probably depending on the age of the patient. We previously reported two cases with cystic RMC associated with terminal lipoma,[ 16 ] which was difficult to make a correct preoperative diagnosis. The more important issue is to diagnose the filar anomaly involved as an additional tethering tract and make an appropriate decision regarding untethering procedures.…”

Section: Discussionmentioning

confidence: 99%

“…Our previous report demonstrated that some filar lipomas also contain E-LC w/NGT, which is the characteristic histopathology of the C-LS of RMC. [ 16 , 19 ] To clarify the embryological background of these filar anomalies, we evaluated the histopathological findings, paying special attention to E-LC w/NGT.…”

Section: Introductionmentioning

confidence: 99%

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Surgical histopathology of a filar anomaly as an additional tethering element associated with closed spinal dysraphism of primary neurulation failure

Morioka

Murakami

Suzuki³

et al. 2021

Surgical Neurology International

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Background: Closed spinal dysraphism of primary neurulation failure could be associated with filar anomalies, such as filar lipoma or thickened and tight filum terminale (TFT), resulting from impaired secondary neurulation. Retained medullary cord (RMC) is a remnant of the cavitary medullary cord originating from the secondary neurulation failure. Some filar lipomas are known to contain a central canal-like ependyma-lined lumen with surrounding neuroglial tissues (E-LC w/NGT), that is, a characteristic histopathology of RMC. To clarify the embryological background of these filar anomalies, we evaluated the histopathological findings. Methods: Among 41 patients with lesions of primary neurulation failure who underwent initial untethering surgery, the filum including cord-like structure (C-LS) was additionally resected in 10 patients (five dorsal and transitional lipomas; five limited dorsal myeloschisis). We retrospectively analyzed the clinical, neuroradiological, intraoperative, and histopathological findings. Results: Among 10 patients, two patients were diagnosed with RMC based on morphological features and intraoperative neurophysiological monitoring. The diagnosis of filar lipoma was made in six patients, since various amounts of fibroadipose tissue were histopathologically noted in the filum. Two patients were diagnosed with TFT, since the filum was composed solely of fibrocollagenous tissue. E-LC w/NGT was noted not only in both C-LSs of RMCs but also in two out of six fila both with filar lipomas and fila with TFTs. Conclusion: These findings provide further evidence for the idea that entities, such as filar lipoma, TFT, and RMC, can be considered consequences of a continuum of regression failure occurring during late secondary neurulation.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Surgical histopathology of a filar anomaly as an additional tethering element associated with closed spinal dysraphism of primary neurulation failure

Morioka

Murakami

Suzuki³

et al. 2021

Surgical Neurology International

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“…As nonfunctional RMC is indistinguishable from the functional conus on neuroimaging; moreover, in the intraoperative view, detecting the presence of a nonfunctional C-LS with intraoperative neurophysiological monitoring (IONM) is mandatory for the diagnosis of RMC. [ 4 , 6 , 9 , 11 , 12 , 14 , 17 , 18 , 20 ] The characteristic histopathological feature of RMC is the predominant presence of a central canal-like ependymal-lined lumen (CC-LELL) with surrounding glial fibrillary acidic protein (GFAP)-immunopositive neuroglial tissues, as a remnant of the medullary cord. [ 4 , 6 , 9 , 11 , 12 - 17 , 18 , 21 ] Some CC-LELLs show cystic dilatation, in which case, these are referred to as “cystic RMC” or “RMC of cystic type,”[ 4 - 6 , 14 , 18 ] though Pang et al .…”

Section: Introductionmentioning

confidence: 99%

“…[ 4 , 6 , 9 , 11 , 12 , 14 , 17 , 18 , 20 ] The characteristic histopathological feature of RMC is the predominant presence of a central canal-like ependymal-lined lumen (CC-LELL) with surrounding glial fibrillary acidic protein (GFAP)-immunopositive neuroglial tissues, as a remnant of the medullary cord. [ 4 , 6 , 9 , 11 , 12 - 17 , 18 , 21 ] Some CC-LELLs show cystic dilatation, in which case, these are referred to as “cystic RMC” or “RMC of cystic type,”[ 4 - 6 , 14 , 18 ] though Pang et al . [ 17 ] did not describe cystic RMCs in their initial article.…”

Section: Introductionmentioning

confidence: 99%

Intramedullary abscess at thoracolumbar region transmitted from infected dermal sinus and dermoid through retained medullary cord

Matsubara

Murakami

Kurogi

et al. 2022

Surgical Neurology International

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Background: A retained medullary cord (RMC) is a relatively newly defined entity of closed spinal dysraphism that is thought to originate from regression failure of the medullary cord during secondary neurulation. A congenital dermal sinus (CDS) may provide a pathway for intraspinal infections such as repeated meningitis. Intramedullary abscesses are the rarest but most serious complication of a CDS. Case Description: We treated a female infant with an intramedullary abscess in the thoracolumbar region, which was caused by infection of the CDS. Surgery revealed that the cord-like structure (C-LS) started from the cord with the intramedullary abscess, extended to the dural cul-de-sac, and further continued to the CDS tract and skin dimple. The boundary between the functional cord and the non-functional CL-S was electrophysiologically identified, and the entire length of the C-LS (the RMC) with an infected dermoid cyst was resected. As a result, the abscess cavity was opened and thorough irrigation and drainage of the pus could be performed. Histopathological examination of the C-LS revealed an infected dermoid cyst and abscess cavity with keratin debris in the fibrocollagenous tissue. The abscess cavity had a central canal-like ependymal lined lumen (CCLELL), with surrounding glial fibrillary acidic protein (GFAP)-immunopositive neuroglial tissues. Conclusion: We demonstrated that the transmission of an infection through the RMC was involved in the development of the intramedullary abscess. A good postoperative outcome was obtained because a terminal ventriculostomy for pus drainage could be achieved by excising the nonfunctional RMC.

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Spontaneous Disappearance of Filar Cysts Diagnosed by Transabdominal Ultrasonography: A Report of Three Cases

2021

J of Ultrasound Medicine

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Filar cysts (FCs) can be detected using ultrasound before or after delivery. They usually present as anechoic structures with a clear boundary just caudal to the lower end of the conus medullaris. They are generally considered a physiological variation and do not affect health. Some studies have pointed out that FCs in children can disappear spontaneously, as identified by ultrasound or magnetic resonance imaging. Three cases of FCs diagnosed by prenatal transabdominal ultrasound were reported in this study, and it was observed that FCs could disappear spontaneously in utero. The shortest time of disappearance was 4 weeks.

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Two Cases of Large Filar Cyst Associated with Terminal Lipoma: Relationship with Retained Medullary Cord

Cited by 11 publications

References 15 publications

Surgical histopathology of a filar anomaly as an additional tethering element associated with closed spinal dysraphism of primary neurulation failure

Surgical histopathology of a filar anomaly as an additional tethering element associated with closed spinal dysraphism of primary neurulation failure

Intramedullary abscess at thoracolumbar region transmitted from infected dermal sinus and dermoid through retained medullary cord

Spontaneous Disappearance of Filar Cysts Diagnosed by Transabdominal Ultrasonography: A Report of Three Cases

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