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Published in: Child's Nervous System 6/2019

01-06-2019 | Magnetic Resonance Imaging | Original Article

Human tail-like cutaneous appendage with a contiguous stalk of limited dorsal myeloschisis

Authors: Mio Sarukawa, Takato Morioka, Nobuya Murakami, Takafumi Shimogawa, Nobutaka Mukae, Noriko Kuga, Satoshi O. Suzuki, Koji Iihara

Published in: Child's Nervous System | Issue 6/2019

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Abstract

Purpose

Limited dorsal myeloschisis (LDM) is characterized by a fibroneural stalk linking the skin lesion to the underlying spinal cord. On account of the external skin lesion, all LDMs are either flat (nonsaccular) or saccular, and a human tail-like cutaneous appendage has not been reported.

Methods

In our 14 LDM patients, 2 had tail-like appendages. We retrospectively analyzed the relationship between the appendage and the LDM tract from the clinicopathological findings of these 2 patients.

Results

Preoperative magnetic resonance imaging including three-dimensional heavily T2-weighted images demonstrated an intradural tethering tract, but failed to reveal the precise communication with the appendage. However, surgery revealed the extradural and intradural slender stalk, starting at the base of appendage and running through the myofascial defect. Histological examination demonstrated that there was a tight anatomical relationship between the fibroadipose tissue of the appendage and the fibrocollagenous LDM stalk.

Conclusion

When there is potential for an LDM stalk in patients with an appendage, a meticulous exploration of the stalk leading from an appendage is required. Clinicians should be aware of possible morphological variations of skin lesions associated with LDM.
Literature
2.
go back to reference Donovan DJ, Pedersen RC (2005) Human tail with noncontiguous intraspinal lipma and spinal cord tethering: case report and embryologic discussion. Pediatr Neurosurg 41:35–40CrossRefPubMed Donovan DJ, Pedersen RC (2005) Human tail with noncontiguous intraspinal lipma and spinal cord tethering: case report and embryologic discussion. Pediatr Neurosurg 41:35–40CrossRefPubMed
3.
go back to reference Gaskill SJ, Marlin AE (1989) Neuroectodermal appendages: the human tail explained. Pediatr Neurosci 15:95–99CrossRefPubMed Gaskill SJ, Marlin AE (1989) Neuroectodermal appendages: the human tail explained. Pediatr Neurosci 15:95–99CrossRefPubMed
4.
go back to reference Hiraoka A, Morioka T, Murakami N, Suzuki SO, Mizoguchi M (2018) Limited dorsal myeloschisis with no extradural stalk linking to a flat skin lesion: a case report. Childs Nerv Syst 34:2497–2501CrossRefPubMed Hiraoka A, Morioka T, Murakami N, Suzuki SO, Mizoguchi M (2018) Limited dorsal myeloschisis with no extradural stalk linking to a flat skin lesion: a case report. Childs Nerv Syst 34:2497–2501CrossRefPubMed
5.
go back to reference Lee JY, Chong S, Choi YH, Phi JH, Cheon J-E, Kim S-K, Park SH, Kim I-O, Wang K-C (2017) Modification of surgical procedure for “probable” limited dorsal myeloschisis. J Neurosurg Pediatr 19:616–619CrossRefPubMed Lee JY, Chong S, Choi YH, Phi JH, Cheon J-E, Kim S-K, Park SH, Kim I-O, Wang K-C (2017) Modification of surgical procedure for “probable” limited dorsal myeloschisis. J Neurosurg Pediatr 19:616–619CrossRefPubMed
6.
go back to reference Lee JY, Park S-H, Chong S, Phi JH, Kim S-K, Cho B-K, Wang K-C (2019) Congenital dermal sinus and limited dorsal myeloschisis: “Spectrum disorders” of imcomplete dysjunction between cutaneous and neural ectoderms. Neurosurgery 84:428–434 Lee JY, Park S-H, Chong S, Phi JH, Kim S-K, Cho B-K, Wang K-C (2019) Congenital dermal sinus and limited dorsal myeloschisis: “Spectrum disorders” of imcomplete dysjunction between cutaneous and neural ectoderms. Neurosurgery 84:428–434
7.
go back to reference Morioka T, Murakami N, Shimogawa T, Mukae N, Hashiguchi K, Suzuki SO, Iihara K (2017) Neurosurgical management and pathology of the lumbosacral lipomas with tethered cord. Neuropathology 37:385–392CrossRefPubMed Morioka T, Murakami N, Shimogawa T, Mukae N, Hashiguchi K, Suzuki SO, Iihara K (2017) Neurosurgical management and pathology of the lumbosacral lipomas with tethered cord. Neuropathology 37:385–392CrossRefPubMed
8.
go back to reference Morioka T, Suzuki SO, Murakami N, Shimogawa T, Mukae N, Inoha S, Sasaguri T, Iihara K (2018) Neurosurgical pathology of limited dorsal myeloschisis. Childs Nerv Syst 34:293–303CrossRefPubMed Morioka T, Suzuki SO, Murakami N, Shimogawa T, Mukae N, Inoha S, Sasaguri T, Iihara K (2018) Neurosurgical pathology of limited dorsal myeloschisis. Childs Nerv Syst 34:293–303CrossRefPubMed
9.
go back to reference Morioka T, Suzuki SO, Murakami N, Mukae N, Shimogawa T, Haruyama H, Kira R, Iihara K (2019) Surgical histopathology of limited dorsal myeloschisis with flat skin lesion. Childs Nerv Syst 35:119–128 Morioka T, Suzuki SO, Murakami N, Mukae N, Shimogawa T, Haruyama H, Kira R, Iihara K (2019) Surgical histopathology of limited dorsal myeloschisis with flat skin lesion. Childs Nerv Syst 35:119–128
10.
go back to reference Murakami N, Morioka T, Hashiguchi K, Yoshiura T, Hiwatashi K, Suzuki SO, Nakamizo A, Amano T, Hata N, Sasaki T (2013) Usefulness of three-dimensional T1-weighted spoiled gradient-recalled echo and three-dimensional heavily T2-weighted images in preoperative evaluation of spinal dysraphism. Childs Nerv Syst 29:1905–1914CrossRefPubMed Murakami N, Morioka T, Hashiguchi K, Yoshiura T, Hiwatashi K, Suzuki SO, Nakamizo A, Amano T, Hata N, Sasaki T (2013) Usefulness of three-dimensional T1-weighted spoiled gradient-recalled echo and three-dimensional heavily T2-weighted images in preoperative evaluation of spinal dysraphism. Childs Nerv Syst 29:1905–1914CrossRefPubMed
11.
go back to reference Pang D, Zovickian J, Oviedo A, Moes GS (2010) Limited dorsal myeloschisis: a distinctive clinicopathological entity. Neurosurgery 67:1555–1580CrossRefPubMed Pang D, Zovickian J, Oviedo A, Moes GS (2010) Limited dorsal myeloschisis: a distinctive clinicopathological entity. Neurosurgery 67:1555–1580CrossRefPubMed
12.
go back to reference Pang D, Zovickian J, Wong S-T, Hou YJ, Moes GS (2013) Limited dorsal myeloschisis: a not-so-rare form of primary neurulation defect. Childs Nerv Syst 29:1459–1484CrossRefPubMed Pang D, Zovickian J, Wong S-T, Hou YJ, Moes GS (2013) Limited dorsal myeloschisis: a not-so-rare form of primary neurulation defect. Childs Nerv Syst 29:1459–1484CrossRefPubMed
13.
go back to reference Samura K, Morioka T, Hashiguchi K, Yoshida F, Miyagi Y, Yoshiura T, Suzuki SO, Sasaki T (2009) Coexistence of a human tail and congenital dermal sinus associated with lumbosacral lipoma. Childs Nerv Syst 25:137–141CrossRefPubMed Samura K, Morioka T, Hashiguchi K, Yoshida F, Miyagi Y, Yoshiura T, Suzuki SO, Sasaki T (2009) Coexistence of a human tail and congenital dermal sinus associated with lumbosacral lipoma. Childs Nerv Syst 25:137–141CrossRefPubMed
14.
go back to reference Tomita Y, Morioka T, Murakami N, Noguchi Y, Sato Y, Suzuki OS (2018) Slender stalk with combined features of saccular limited dorsal myeloschisis and congenital dermal sinus in a neonate. Pediatr Neurosurg. https://doi.org/10.1159/000495810 Tomita Y, Morioka T, Murakami N, Noguchi Y, Sato Y, Suzuki OS (2018) Slender stalk with combined features of saccular limited dorsal myeloschisis and congenital dermal sinus in a neonate. Pediatr Neurosurg. https://​doi.​org/​10.​1159/​000495810
15.
go back to reference Tubbs RS, Malefant J, Loukas M, Oakes WJ, Oskouian RJ, Fries FN (2016) Enigmatic human tails: a review of their history, embryology, classification, and clinical manifestations. Clin Anat 29:430–438CrossRefPubMed Tubbs RS, Malefant J, Loukas M, Oakes WJ, Oskouian RJ, Fries FN (2016) Enigmatic human tails: a review of their history, embryology, classification, and clinical manifestations. Clin Anat 29:430–438CrossRefPubMed
16.
go back to reference Turk CC, Kara NN, Bacanli A (2016) The human tail: a simple appendage or cutaneous stigma of an anomaly? Turk Neurosurg 26:140–145PubMed Turk CC, Kara NN, Bacanli A (2016) The human tail: a simple appendage or cutaneous stigma of an anomaly? Turk Neurosurg 26:140–145PubMed
17.
go back to reference Wilkinson CC, Boylan AJ (2017) Proposed caudal appendage classification system; spinal cord tethering associated with sacrococcygeal eversion. Childs Nerv Syst 33:69–89CrossRefPubMed Wilkinson CC, Boylan AJ (2017) Proposed caudal appendage classification system; spinal cord tethering associated with sacrococcygeal eversion. Childs Nerv Syst 33:69–89CrossRefPubMed
Metadata
Title
Human tail-like cutaneous appendage with a contiguous stalk of limited dorsal myeloschisis
Authors
Mio Sarukawa
Takato Morioka
Nobuya Murakami
Takafumi Shimogawa
Nobutaka Mukae
Noriko Kuga
Satoshi O. Suzuki
Koji Iihara
Publication date
01-06-2019
Publisher
Springer Berlin Heidelberg
Published in
Child's Nervous System / Issue 6/2019
Print ISSN: 0256-7040
Electronic ISSN: 1433-0350
DOI
https://doi.org/10.1007/s00381-019-04071-w

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