Limited dorsal myeloschisisの病態と外科治療
Limited dorsal myeloschisis (LDM) は, 2010年にPangらが提唱した一次神経管形成時の障害で, 神経・皮膚外胚葉の限局的な不分離が起こり, 両者の連続性がfibroneural stalkとして残存し, 脊髄係留をきたすものである. この部位に一致した背側正中部に, 扁平上皮の陥凹性 (cigarette-burning), もしくは囊胞性の皮膚病変を有し, それぞれflat, saccular LDMと呼ばれる. Flat LDMは従来のmeningocele manquéに, saccular LDMはsegmental myelocystoceleに...
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| Published in | 脳神経外科ジャーナル Vol. 30; no. 6; pp. 424 - 431 |
|---|---|
| Main Authors | , , , , , , , , |
| Format | Journal Article |
| Language | Japanese |
| Published |
日本脳神経外科コングレス
2021
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| Subjects | |
| Online Access | Get full text |
| ISSN | 0917-950X 2187-3100 |
| DOI | 10.7887/jcns.30.424 |
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| Abstract | Limited dorsal myeloschisis (LDM) は, 2010年にPangらが提唱した一次神経管形成時の障害で, 神経・皮膚外胚葉の限局的な不分離が起こり, 両者の連続性がfibroneural stalkとして残存し, 脊髄係留をきたすものである. この部位に一致した背側正中部に, 扁平上皮の陥凹性 (cigarette-burning), もしくは囊胞性の皮膚病変を有し, それぞれflat, saccular LDMと呼ばれる. Flat LDMは従来のmeningocele manquéに, saccular LDMはsegmental myelocystoceleに相当すると考えられている. 近年, human tail様の皮膚突起物であるtail-like LDMも報告されている. 皮膚病変から脊髄背側に連続するstalkを, 3D-heavily T2強調画像を中心としたMRIで診断する. 治療は早期の係留解除が勧められるが, このstalk内に先天性皮膚洞が10～20%に合併し, これを硬膜内に残存させると将来dermoidが発生することが知られている. Fibroneural stalkの病理診断には, GFAP陽性神経グリア組織の存在が重視されてきたが, この陽性率は50～60%程度と低く, われわれは末梢神経やメラノサイトなどの神経堤由来の組織の証明が診断に有用であると考えている. これらLDMの病態と外科治療について考察する. |
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| AbstractList | Limited dorsal myeloschisis (LDM) は, 2010年にPangらが提唱した一次神経管形成時の障害で, 神経・皮膚外胚葉の限局的な不分離が起こり, 両者の連続性がfibroneural stalkとして残存し, 脊髄係留をきたすものである. この部位に一致した背側正中部に, 扁平上皮の陥凹性 (cigarette-burning), もしくは囊胞性の皮膚病変を有し, それぞれflat, saccular LDMと呼ばれる. Flat LDMは従来のmeningocele manquéに, saccular LDMはsegmental myelocystoceleに相当すると考えられている. 近年, human tail様の皮膚突起物であるtail-like LDMも報告されている. 皮膚病変から脊髄背側に連続するstalkを, 3D-heavily T2強調画像を中心としたMRIで診断する. 治療は早期の係留解除が勧められるが, このstalk内に先天性皮膚洞が10～20%に合併し, これを硬膜内に残存させると将来dermoidが発生することが知られている. Fibroneural stalkの病理診断には, GFAP陽性神経グリア組織の存在が重視されてきたが, この陽性率は50～60%程度と低く, われわれは末梢神経やメラノサイトなどの神経堤由来の組織の証明が診断に有用であると考えている. これらLDMの病態と外科治療について考察する. |
| Author | 村上, 信哉 迎, 伸孝 庄野, 禎久 森岡, 隆人 下川, 能史 黒木, 愛 溝口, 昌弘 鈴木, 諭 金田, 章子 |
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| References | 26) Quon JL, Grant GA : Commentary : Limited dorsal myeloschisis : Reconsideration of its embryological origin. Neurosurgery 86 : E13-E14, 2019. 16) Morioka T, Suzuki SO, Murakami N, Mukae N, Shimogawa T, Haruyama H, Kira R, Iihara K : Surgical histopathology of limited dorsal myeloschisis with flat skin lesion. Childs Nerv Syst 35 : 119-128, 2019. 14) Morioka T, Hashiguchi K, Yoshida F, Nagata S, Miyagi Y, Mihara F, Sasaki T : Dynamic morphological changes in lumbosacral lipoma during the first months of life revealed by constructive interference in steady-state (CISS) MR imaging. Childs Nerv Syst 23 : 415-420, 2007. 23) Pang D, Diaz MS : Cervical myelomenigoceles. Neurosurgery 33 : 363-373, 1993. 1) Abe K, Mukae N, Morioka T, Shimogawa T, Suzuki SO, Mizoguchi M : Tail-like cutaneous appendage at the upper thoracic region with a continuous stalk of limited dorsal myeloschisis. Interdiscip Neurosurg 22 : 100823, 2020. 13) Lee JY, Park SH, Chong S, Phi JH, Kim SK, Cho BK, Wang KC : Congenital dermal sinus and limited dorsal myeloschisis : “Spectrum disorders” of incomplete dysjunction between cutaneous and neural ectoderms. Neurosurgery 84 : 428-434, 2019. 8) Hiraoka A, Morioka T, Murakami N, Suzuki SO, Mizoguchi M : Limited dorsal myeloschisis with no extradural stalk linking to a flat skin lesion : A case report. Childs Nerv Syst 34 : 2497-2501, 2018. 27) Rajpal S, Salamat MS, Tubbs RS, Kelly DR, Oakes WJ, Iskandar BJ : Tethering tracts in spina bifida occulta : revisiting an established nomenclature. J Neurosurg Spine 7 : 315-322, 2007. 21) Murakami N, Morioka T, Suzuki SO, Mukae N, Shimogawa T, Matsuo Y, Sasaguri T, Mizoguchi M : Clinicopathological findings of limited dorsal myeloschisis associated with spinal lipoma of dorsal-type. Interdiscip Neurosurg 21 : 100781, 2020. 15) Morioka T, Suzuki SO, Murakami N, Shimogawa T, Mukae N, Inoha S, Sasaguri T, Iihara K : Neurosurgical pathology of the limited dorsal myeloschisis. Childs Nerv Syst 34 : 293-303, 2018. 30) Sarukawa M, Morioka T, Murakami N, Shimogawa T, Mukae N, Kuga N, Suzuki SO, Iihara K : Human tail-like cutaneous appendage with a contiguous stalk of limited dorsal myeloschisis. Childs Nerv Syst 35 : 973-978, 2019. 12) Lee JY, Chong S, Choi YH, Phi JH, Cheon JE, Kim SK, Park SH, Kim IO, Wang KY : Modification of surgical procedure for “probable” limited dorsal myeloschisis. J Neurosurg Pediatr 19 : 616-619, 2017. 24) Pang D, Zovickian J, Oviedo A, Moes GS : Limited dorsal myeloschisis : A distinctive clinicopathological entity. Neurosurgery 67 : 1555-1580, 2010. 17) Morioka T, Murakami N, Suzuki SO, Takada A, Tajiri S, Shimogawa T, Mukae N, Iihara K : Neurosurgical pathology and management of limited dorsal myeloschisis with congenital dermal sinus in infancy. Pediatr Neurosurg 55 : 113-125, 2020. 11) Lassman LP, James CC : Meningocoele manque. Childs Brain 3 : 1-11, 1977. 19) Morioka T, Murakami N, Yanagida H, Yamaguchi T, Noguchi Y, Takahata Y, Tsukamoto A, Suzuki SO : Terminal syringomyelia associated with lumbar limited dorsal myeloschisis. Childs Nerv Syst 36 : 819-826, 2020. 22) Nishio S, Morioka T, Hikino S, Fukui M : Cervical (myelo) meningocoele : report of 2 cases. J Clin Neurosci 8 : 586-587, 2001. 5) Eibach S, Moes G, Zovickian J, Pang D : Limited dorsal myeloschisis associated with dermoid elements. Childs Nerv Syst 33 : 55-67, 2017. 25) Pang D, Zovickian J, Wong ST, Hou YJ, Moes GS : Limited dorsal myeloschisis : a not-so-rare form of primary neurulation defect. Childs Nerv Syst 29 : 1459-1484, 2013. 10) Lafitte AS, Blouet M, Frederique B, Borha A, Benoist G : A case of prenatally dignosed limited dorsal myeloschisis with good prognosis. J Clin Ultrasound 46 : 282-285, 2017. 3) Chattterjee S, Rao KSM : Missed limited dorsal myeloschisis : an unfortunate cause for recurrent tethered cord syndrome. Childs Nerv Syst 31 : 1553-1557, 2015. 31) Steinbok P : Comment : Congenital dermal sinus and limited dorsal myeloschisis : “Spectrum disorders” of incomplete dysjunction between cutaneous and neural ectoderms. Neurosurgery 84 : 434, 2019. 4) Dupin E, Calloni GW, Coelho-Aguiar JM, Le Douarin NM : The issue of the multipotency of the neural crest cells. Dev Biol 444 : S47-S59, 2018. 28) Rossi A, Piatelli G, Gandolfo C, Pavanello M, Hoffmann C, Van Goethem JW, Cama A, Tortori-Donati P : Spectrum of nonterminal myelocystoceles. Neurosurgery 58 : 509-515, 2006. 6) Friszer S, Dhombres F, Morel B, Zerah M, Jouannic JM, Garel C : Limited dorsal myeloschisis : A diagnostic pitfall in the prenatal ultrasound of fetal dysraphism. Fetal Diagn Ther 41 : 136-144, 2017. 20) Murakami N, Morioka T, Hashiguchi K, Yoshiura T, Hiwatashi K, Suzuki SO, Nakamizo A, Amano T, Hata N, Sasaki T : 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, 2013. 33) Wong ST, Kan A, Pang D : Limited dorsal spinal nondisjunctional disorders : Limited dorsal myeloschisis, congenital spinal dermal sinus tract, and mixed lesions. in Di Rocco C, Pang D, Rutka JT (eds) : Textbook of Pediatric Neurosurgery, 1st ed. Switzerland, Springer, 2020, pp.1-64. 7) Gaskill SJ, Marlin AE : Neuroectodermal appendages : the human tail explained. Pediatr Neurosci 15 : 95-99, 1989. 9) Kim JW, Wang KC, Chong S, Kim SK, Lee JY : Limited dorsal myeloschisis : Reconsideration of its embryological origin. Neurosurgery 86 : 93-100, 2020. 2) Batista Cezar-Junior A, Faquini IV, Frank K, Euripedes Almondes S Lemos L, de Carvalho EV J, Almeida NS, Azevedo-Filho HRC : Limited dorsal myeloschisis with a contiguous stalk to human tail-like cutaneous appendage, associated with a lipoma of conus medullaris : A case report. Int J Surg Case Rep 71 : 303-306, 2020. 18) Morioka T, Murakami N, Ichiyama M, Kusuda T, Suzuki SO : Congenital dermal sinus elements in each tethering stalk of coexisting thoracic limited dorsal myeloschisis and retained medullary cord. Pediatr Neurosurg 55 : 380-387, 2020. 29) Russell NE, Chalouhi GE, Dirocco F, Zerah M, Ville Y : Not all large neural tube defects have a poor prognosis : a case or prenatally diagnosed limited dorsal myeloschisis. Ultrasound Obstet Gynecol 42 : 238-239, 2013. 32) Tomita Y, Morioka T, Murakami N, Noguchi Y, Sato Y, Suzuki SO : Slender stalk with combined features of saccular limited dorsal myeloschisis and congenital dermal sinus in a neonate. Pediatr Neurosurg 54 : 125-131, 2019. |
| References_xml | – reference: 28) Rossi A, Piatelli G, Gandolfo C, Pavanello M, Hoffmann C, Van Goethem JW, Cama A, Tortori-Donati P : Spectrum of nonterminal myelocystoceles. Neurosurgery 58 : 509-515, 2006. – reference: 11) Lassman LP, James CC : Meningocoele manque. Childs Brain 3 : 1-11, 1977. – reference: 30) Sarukawa M, Morioka T, Murakami N, Shimogawa T, Mukae N, Kuga N, Suzuki SO, Iihara K : Human tail-like cutaneous appendage with a contiguous stalk of limited dorsal myeloschisis. Childs Nerv Syst 35 : 973-978, 2019. – reference: 23) Pang D, Diaz MS : Cervical myelomenigoceles. Neurosurgery 33 : 363-373, 1993. – reference: 33) Wong ST, Kan A, Pang D : Limited dorsal spinal nondisjunctional disorders : Limited dorsal myeloschisis, congenital spinal dermal sinus tract, and mixed lesions. in Di Rocco C, Pang D, Rutka JT (eds) : Textbook of Pediatric Neurosurgery, 1st ed. Switzerland, Springer, 2020, pp.1-64. – reference: 4) Dupin E, Calloni GW, Coelho-Aguiar JM, Le Douarin NM : The issue of the multipotency of the neural crest cells. Dev Biol 444 : S47-S59, 2018. – reference: 6) Friszer S, Dhombres F, Morel B, Zerah M, Jouannic JM, Garel C : Limited dorsal myeloschisis : A diagnostic pitfall in the prenatal ultrasound of fetal dysraphism. Fetal Diagn Ther 41 : 136-144, 2017. – reference: 15) Morioka T, Suzuki SO, Murakami N, Shimogawa T, Mukae N, Inoha S, Sasaguri T, Iihara K : Neurosurgical pathology of the limited dorsal myeloschisis. Childs Nerv Syst 34 : 293-303, 2018. – reference: 12) Lee JY, Chong S, Choi YH, Phi JH, Cheon JE, Kim SK, Park SH, Kim IO, Wang KY : Modification of surgical procedure for “probable” limited dorsal myeloschisis. J Neurosurg Pediatr 19 : 616-619, 2017. – reference: 3) Chattterjee S, Rao KSM : Missed limited dorsal myeloschisis : an unfortunate cause for recurrent tethered cord syndrome. Childs Nerv Syst 31 : 1553-1557, 2015. – reference: 19) Morioka T, Murakami N, Yanagida H, Yamaguchi T, Noguchi Y, Takahata Y, Tsukamoto A, Suzuki SO : Terminal syringomyelia associated with lumbar limited dorsal myeloschisis. Childs Nerv Syst 36 : 819-826, 2020. – reference: 24) Pang D, Zovickian J, Oviedo A, Moes GS : Limited dorsal myeloschisis : A distinctive clinicopathological entity. Neurosurgery 67 : 1555-1580, 2010. – reference: 20) Murakami N, Morioka T, Hashiguchi K, Yoshiura T, Hiwatashi K, Suzuki SO, Nakamizo A, Amano T, Hata N, Sasaki T : 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, 2013. – reference: 7) Gaskill SJ, Marlin AE : Neuroectodermal appendages : the human tail explained. Pediatr Neurosci 15 : 95-99, 1989. – reference: 16) Morioka T, Suzuki SO, Murakami N, Mukae N, Shimogawa T, Haruyama H, Kira R, Iihara K : Surgical histopathology of limited dorsal myeloschisis with flat skin lesion. Childs Nerv Syst 35 : 119-128, 2019. – reference: 18) Morioka T, Murakami N, Ichiyama M, Kusuda T, Suzuki SO : Congenital dermal sinus elements in each tethering stalk of coexisting thoracic limited dorsal myeloschisis and retained medullary cord. Pediatr Neurosurg 55 : 380-387, 2020. – reference: 13) Lee JY, Park SH, Chong S, Phi JH, Kim SK, Cho BK, Wang KC : Congenital dermal sinus and limited dorsal myeloschisis : “Spectrum disorders” of incomplete dysjunction between cutaneous and neural ectoderms. Neurosurgery 84 : 428-434, 2019. – reference: 21) Murakami N, Morioka T, Suzuki SO, Mukae N, Shimogawa T, Matsuo Y, Sasaguri T, Mizoguchi M : Clinicopathological findings of limited dorsal myeloschisis associated with spinal lipoma of dorsal-type. Interdiscip Neurosurg 21 : 100781, 2020. – reference: 8) Hiraoka A, Morioka T, Murakami N, Suzuki SO, Mizoguchi M : Limited dorsal myeloschisis with no extradural stalk linking to a flat skin lesion : A case report. Childs Nerv Syst 34 : 2497-2501, 2018. – reference: 22) Nishio S, Morioka T, Hikino S, Fukui M : Cervical (myelo) meningocoele : report of 2 cases. J Clin Neurosci 8 : 586-587, 2001. – reference: 27) Rajpal S, Salamat MS, Tubbs RS, Kelly DR, Oakes WJ, Iskandar BJ : Tethering tracts in spina bifida occulta : revisiting an established nomenclature. J Neurosurg Spine 7 : 315-322, 2007. – reference: 10) Lafitte AS, Blouet M, Frederique B, Borha A, Benoist G : A case of prenatally dignosed limited dorsal myeloschisis with good prognosis. J Clin Ultrasound 46 : 282-285, 2017. – reference: 14) Morioka T, Hashiguchi K, Yoshida F, Nagata S, Miyagi Y, Mihara F, Sasaki T : Dynamic morphological changes in lumbosacral lipoma during the first months of life revealed by constructive interference in steady-state (CISS) MR imaging. Childs Nerv Syst 23 : 415-420, 2007. – reference: 17) Morioka T, Murakami N, Suzuki SO, Takada A, Tajiri S, Shimogawa T, Mukae N, Iihara K : Neurosurgical pathology and management of limited dorsal myeloschisis with congenital dermal sinus in infancy. Pediatr Neurosurg 55 : 113-125, 2020. – reference: 1) Abe K, Mukae N, Morioka T, Shimogawa T, Suzuki SO, Mizoguchi M : Tail-like cutaneous appendage at the upper thoracic region with a continuous stalk of limited dorsal myeloschisis. Interdiscip Neurosurg 22 : 100823, 2020. – reference: 31) Steinbok P : Comment : Congenital dermal sinus and limited dorsal myeloschisis : “Spectrum disorders” of incomplete dysjunction between cutaneous and neural ectoderms. Neurosurgery 84 : 434, 2019. – reference: 32) Tomita Y, Morioka T, Murakami N, Noguchi Y, Sato Y, Suzuki SO : Slender stalk with combined features of saccular limited dorsal myeloschisis and congenital dermal sinus in a neonate. Pediatr Neurosurg 54 : 125-131, 2019. – reference: 5) Eibach S, Moes G, Zovickian J, Pang D : Limited dorsal myeloschisis associated with dermoid elements. Childs Nerv Syst 33 : 55-67, 2017. – reference: 9) Kim JW, Wang KC, Chong S, Kim SK, Lee JY : Limited dorsal myeloschisis : Reconsideration of its embryological origin. Neurosurgery 86 : 93-100, 2020. – reference: 2) Batista Cezar-Junior A, Faquini IV, Frank K, Euripedes Almondes S Lemos L, de Carvalho EV J, Almeida NS, Azevedo-Filho HRC : Limited dorsal myeloschisis with a contiguous stalk to human tail-like cutaneous appendage, associated with a lipoma of conus medullaris : A case report. Int J Surg Case Rep 71 : 303-306, 2020. – reference: 26) Quon JL, Grant GA : Commentary : Limited dorsal myeloschisis : Reconsideration of its embryological origin. Neurosurgery 86 : E13-E14, 2019. – reference: 29) Russell NE, Chalouhi GE, Dirocco F, Zerah M, Ville Y : Not all large neural tube defects have a poor prognosis : a case or prenatally diagnosed limited dorsal myeloschisis. Ultrasound Obstet Gynecol 42 : 238-239, 2013. – reference: 25) Pang D, Zovickian J, Wong ST, Hou YJ, Moes GS : Limited dorsal myeloschisis : a not-so-rare form of primary neurulation defect. Childs Nerv Syst 29 : 1459-1484, 2013. |
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| Snippet | Limited dorsal myeloschisis (LDM) は, 2010年にPangらが提唱した一次神経管形成時の障害で, 神経・皮膚外胚葉の限局的な不分離が起こり, 両者の連続性がfibroneural stalkとして残存し, 脊髄係留をきたすものである. この部位に一致した背側正中部に,... |
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| SubjectTerms | congenital dermal sinus fibroneural stalk glial fibrillary acidic protein neural crest cells primary neurulation |
| Title | Limited dorsal myeloschisisの病態と外科治療 |
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| ispartofPNX | 脳神経外科ジャーナル, 2021, Vol.30(6), pp.424-431 |
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