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

Child's Nervous System volume 35, pages 973–978 (2019)Cite this article

A Correction to this article was published on 02 March 2019

This article has been updated

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.

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Change history

  • 02 March 2019

    The article was recently published, contained error. Author name “Nobutaka Mukai” should be “Nobutaka Mukae”. Given in this article is the correct name.

References

  1. Dao AH, Netsky MG (1984) Human tails and pseudotails. Hum Pathol 15:449–453

    Article  CAS  PubMed  Google Scholar 

  2. Donovan DJ, Pedersen RC (2005) Human tail with noncontiguous intraspinal lipma and spinal cord tethering: case report and embryologic discussion. Pediatr Neurosurg 41:35–40

    Article  PubMed  Google Scholar 

  3. Gaskill SJ, Marlin AE (1989) Neuroectodermal appendages: the human tail explained. Pediatr Neurosci 15:95–99

    Article  CAS  PubMed  Google Scholar 

  4. 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 34:2497–2501

    Article  PubMed  Google Scholar 

  5. 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 19:616–619

    Article  PubMed  Google Scholar 

  6. 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. 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 37:385–392

    Article  PubMed  Google Scholar 

  8. 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 34:293–303

    Article  PubMed  Google Scholar 

  9. 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. 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 29:1905–1914

    Article  PubMed  Google Scholar 

  11. Pang D, Zovickian J, Oviedo A, Moes GS (2010) Limited dorsal myeloschisis: a distinctive clinicopathological entity. Neurosurgery 67:1555–1580

    Article  PubMed  Google Scholar 

  12. 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 29:1459–1484

    Article  PubMed  Google Scholar 

  13. 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 25:137–141

    Article  PubMed  Google Scholar 

  14. 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. 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 29:430–438

    Article  PubMed  Google Scholar 

  16. Turk CC, Kara NN, Bacanli A (2016) The human tail: a simple appendage or cutaneous stigma of an anomaly? Turk Neurosurg 26:140–145

    PubMed  Google Scholar 

  17. Wilkinson CC, Boylan AJ (2017) Proposed caudal appendage classification system; spinal cord tethering associated with sacrococcygeal eversion. Childs Nerv Syst 33:69–89

    Article  PubMed  Google Scholar 

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Acknowledgments

We thank Drs. Nobuko Kawamura, Ayumi Tsukamoto, and Ryutaro Kira for supporting our study. We also thank Edanz Group (www.edanzediting.com/ac) for editing a draft of this manuscript.

Funding

This work was partly supported by the Research Foundation of Fukuoka Children’s Hospital.

Author information

Affiliations

  1. Department of Pediatric Neurology, Fukuoka Children’s Hospital, Fukuoka, Japan

    Mio Sarukawa

  2. Department of Neurosurgery, Fukuoka Children’s Hospital, 5-1-1 Kashii-teriha, Higashi-ku, Fukuoka, 813-0017, Japan

    Takato Morioka & Nobuya Murakami

  3. Department of Neurosurgery, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan

    Takafumi Shimogawa, Nobutaka Mukae & Koji Iihara

  4. Department of General Pediatrics & Interdisciplinary Medicine, Fukuoka Children’s Hospital, Fukuoka, Japan

    Noriko Kuga

  5. Department of Neuropathology, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan

    Satoshi O. Suzuki

Authors
  1. Mio Sarukawa
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  2. Takato Morioka
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  3. Nobuya Murakami
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  4. Takafumi Shimogawa
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  5. Nobutaka Mukae
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  6. Noriko Kuga
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  7. Satoshi O. Suzuki
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  8. Koji Iihara
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Corresponding author

Correspondence to Takato Morioka.

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The original version of this article was revised: The article was recently published, contained error. Author name “Nobutaka Mukai” should be “Nobutaka Mukae”. Given in this article is the correct name.

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Sarukawa, M., Morioka, T., Murakami, N. et al. Human tail-like cutaneous appendage with a contiguous stalk of limited dorsal myeloschisis. Childs Nerv Syst 35, 973–978 (2019). https://doi.org/10.1007/s00381-019-04071-w

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  • DOI: https://doi.org/10.1007/s00381-019-04071-w

Keywords

  • Glial fibrillary acidic protein
  • Fibroneural stalk
  • Untethering
  • Fibroadipose tissue