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

Sarukawa, Mio; Morioka, Takato; Murakami, Nobuya; Shimogawa, Takafumi; Mukae, Nobutaka; Kuga, Noriko; Suzuki, Satoshi; Iihara, Koji

doi:10.1007/s00381-019-04071-w

Cited by 10 publications

(7 citation statements)

References 17 publications

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“…Because neural crest cells are separated from the epidermal ectoderm and neuroectoderm during primary neurulation, a nondisjunctional stalk might drag with it neural crest progenitor cells without neuroglial progenitor cells. Therefore, a diagnosis of LDM can also be applied to patients with tissues of neural crest origin in the stalk but with no neuroglial tissue [3, 14-18].…”

Section: Discussionmentioning

confidence: 99%

Congenital Dermal Sinus Elements in Each Tethering Stalk of Coexisting Thoracic Limited Dorsal Myeloschisis and Retained Medullary Cord

et al. 2020

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Introduction: The embryogenesis of limited dorsal myeloschisis (LDM) likely involves impaired disjunction between the cutaneous and neural ectoderms during primary neurulation. Because LDM and congenital dermal sinus (CDS) have a shared origin in this regard, CDS elements can be found in the LDM stalk. Retained medullary cord (RMC) is a closed spinal dysraphism involving a robust, elongated, cord-like structure extending from the conus medullaris to the dural cul-de-sac. Because the RMC is assumed to be caused by impaired secondary neurulation, concurrent RMC and CDS cannot be explained embryologically. In the present article, we report a case in which CDS elements were noted in each tethering stalk of a coexisting LDM and RMC. Case Presentation: A 2.5-month-old boy with left clubfoot and frequent urinary and fecal leakage had 2 tethering tracts. The upper tract, which ran from the thoracic tail-like cutaneous appendage, had CDS elements in the extradural stalk and a tiny dermoid cyst in the intradural stalk immediately after the dural entry. In the lower tract, which ran from the lumbosacral dimple, the CDS as an extradural stalk continued to the RMC at the dural cul-de-sac. Both stalks were entirely resected through skip laminotomy/laminectomy at 1 stage to untether the cord and resect the CDS elements. Conclusion: Surgeons should be aware that CDS elements, in addition to LDM, may coexist with RMC that extends out to the extradural space.

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Section: Discussionmentioning

confidence: 99%

Congenital Dermal Sinus Elements in Each Tethering Stalk of Coexisting Thoracic Limited Dorsal Myeloschisis and Retained Medullary Cord

et al. 2020

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“…Insets indicate the gross anatomy of each stalk. Although almost the entire tract was submitted for histopathological examination in all 3 patients, extensive examination of the entire stalk could not be performed in a strict sense [8]. Note that only positive histopathological findings obtained are demonstrated.…”

Section: Discussionmentioning

confidence: 99%

“…From April 2015 to June 2019, 19 Japanese patients with LDM underwent initial untethering surgery at Fukuoka Children’s Hospital and related hospitals. The clinical diagnosis of LDM was based on a comprehensive histological examination and the patients’ clinical manifestations, as described previously [4, 6-8, 11, 13]. The external skin lesions were flat lesions in 13 patients, saccular lesions in 3, and human tail-like cutaneous appendages [8] in 3.…”

Section: Methodsmentioning

confidence: 99%

“…1a, b). Immunohistochemical detection of glial fibrillary acidic protein (GFAP)-expressing tissue is commonly performed for histopathological confirmation of the neuroglial tissues in the stalk [1-8]. LDM is classified as saccular or nonsaccular (flat) based on its external skin manifestations [1, 2].…”

Section: Introductionmentioning

confidence: 99%

“…The surgical treatment of LDM involves a relatively straightforward untethering of the stalk from the cord [4-8, 13]. A coexistent CDS can be microscopic and escape intraoperative observation [9, 10], especially in infancy [11]; however, such a CDS can develop into an inclusion tumor such as a dermoid or epidermoid cyst if part of the CDS invested in the LDM stalk is left inside the dura mater [1-3, 9, 10] (Fig.…”

Section: Introductionmentioning

confidence: 99%

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Neurosurgical Pathology and Management of Limited Dorsal Myeloschisis Associated with Congenital Dermal Sinus in Infancy

et al. 2020

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• Because of the shared origin of limited dorsal myeloschisis (LDM) and congenital dermal sinus (CDS), CDS elements may be found within the LDM stalk. • The recommendation for excision of the entire length of the LDM stalk in all infants should be more carefully made. Novel Insights • Once the histological diagnosis of coexistent CDS is made in the resected end, the entire length of the remnant stalk should be excised as soon as possible.

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Terminal syringomyelia associated with lumbar limited dorsal myeloschisis

et al. 2019

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

Cited by 10 publications

References 17 publications

Congenital Dermal Sinus Elements in Each Tethering Stalk of Coexisting Thoracic Limited Dorsal Myeloschisis and Retained Medullary Cord

Congenital Dermal Sinus Elements in Each Tethering Stalk of Coexisting Thoracic Limited Dorsal Myeloschisis and Retained Medullary Cord

Neurosurgical Pathology and Management of Limited Dorsal Myeloschisis Associated with Congenital Dermal Sinus in Infancy

Terminal syringomyelia associated with lumbar limited dorsal myeloschisis

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