Clinicopathological findings of limited dorsal myeloschisis associated with spinal lipoma of dorsal-type
Purpose: Limited dorsal myeloschisis (LDM) is thought to arise from focal incomplete disjunction between the cutaneous and neural ectoderm during primary neurulation, while spinal lipoma of dorsal-type (dorsal lipoma) arises from premature disjunction. Thus, simultaneous occurrence of an LDM and dor...
| Main Authors: | , , , , , , , |
|---|---|
| Format: | Article |
| Language: | English |
| Published: |
Elsevier
2020-09-01
|
| Series: | Interdisciplinary Neurosurgery |
| Subjects: | |
| Online Access: | http://www.sciencedirect.com/science/article/pii/S2214751920303388 |
| _version_ | 1818849933243973632 |
|---|---|
| author | Nobuya Murakami Takato Morioka Satoshi O. Suzuki Nobutaka Mukae Takafumi Shimogawa Yoshihiro Matsuo Takakazu Sasaguri Masahiro Mizoguchi |
| author_facet | Nobuya Murakami Takato Morioka Satoshi O. Suzuki Nobutaka Mukae Takafumi Shimogawa Yoshihiro Matsuo Takakazu Sasaguri Masahiro Mizoguchi |
| author_sort | Nobuya Murakami |
| collection | DOAJ |
| description | Purpose: Limited dorsal myeloschisis (LDM) is thought to arise from focal incomplete disjunction between the cutaneous and neural ectoderm during primary neurulation, while spinal lipoma of dorsal-type (dorsal lipoma) arises from premature disjunction. Thus, simultaneous occurrence of an LDM and dorsal lipoma are not surprising, and may represent slightly different perturbations of disjunction caused by the same insult in neighboring loci. However, the clinicopathological findings of the LDM with dorsal lipoma have not been fully determined. Methods: Of 21 patients with LDM, 3 (14.3%) had dorsal lipoma. We retrospectively analyzed the clinicopathological findings of these 3 patients, especially the histopathological distribution of the fibrocollagenous LDM tract and fibroadipose tissue of the lipoma. Results: Patients 1 and 2 had flat skin lesions, while patient 3 had a human tail-like cutaneous appendage. In the tethering stalks linking the skin lesion at the lumbosacral lesion to the low-lying conus medullaris of the three patients, fibrocollagenous tissues embedding adipose tissues at the subcutaneous site, and with abundant adipose tissues at the extradural site, were changed to fibroadipose tissue at the intradural site. While glial fibrillary acidic protein-immunopositive neuroglial tissues were observed in 2 (patients 1 and 2), peripheral nerve fibers were observed in every stalk. Smooth muscle fibers were noted in patient 1, while a large amount of striated muscle fibers were seen in patients 2 and 3. Conclusion: These cases showed various tissues with different origins in the stalk. There may also be a seamless continuation between fibrocollagenous LDM tissue at the distal site and lipomatous tissue at the proximal site. Peripheral nerve fibers and smooth muscle fibers of neural crest origin may be dragged into the stalk during incomplete disjunction, while the striated muscle fibers of mesodermal origin may enter the stalk along with the lipomatous tissues during premature disjunction. |
| first_indexed | 2024-12-19T06:41:06Z |
| format | Article |
| id | doaj.art-9ebc58449969443387971a53c6dd11b8 |
| institution | Directory Open Access Journal |
| issn | 2214-7519 |
| language | English |
| last_indexed | 2024-12-19T06:41:06Z |
| publishDate | 2020-09-01 |
| publisher | Elsevier |
| record_format | Article |
| series | Interdisciplinary Neurosurgery |
| spelling | doaj.art-9ebc58449969443387971a53c6dd11b82022-12-21T20:32:05ZengElsevierInterdisciplinary Neurosurgery2214-75192020-09-0121100781Clinicopathological findings of limited dorsal myeloschisis associated with spinal lipoma of dorsal-typeNobuya Murakami0Takato Morioka1Satoshi O. Suzuki2Nobutaka Mukae3Takafumi Shimogawa4Yoshihiro Matsuo5Takakazu Sasaguri6Masahiro Mizoguchi7Department of Neurosurgery, Fukuoka Children’s Hospital, 5-1-1 Kashii-teriha, Higashi-ku, Fukuoka 813-0017, Japan; Corresponding author at: Department of Neurosurgery, Fukuoka Children’s Hospital, 5-1-1 Kashii-teriha, Higashi-ku, Fukuoka 813-0017, Japan.Department of Neurosurgery, Hara-Sanshin Hospital, 1-8 Taihakumachi, Hakata-ku, Fukuoka 812-0033, JapanShourai Hospital, 4304-1 Kagami, Karatu City, Saga 847-0022, JapanDepartment of Neurosurgery, Graduate School of Medical Sciences, Kyushu University, 3-1-1 Maidashi, Higashi-ku, Fukuoka 812-8582, JapanDepartment of Neurosurgery, Graduate School of Medical Sciences, Kyushu University, 3-1-1 Maidashi, Higashi-ku, Fukuoka 812-8582, JapanDepartment of Neurosurgery, Japan Community Health Care Organization (JCHO), Kyushu Hospital, 1-8-1 Kishinoura, Yahatanishi-ku, Kitakyushu 806-8501, JapanDepartment of Pathology, Kitakyushu General Hospital, 1-1 Higashijyounomachi, Kokurakita-ku, Kitakyushu 802-8517, JapanDepartment of Neurosurgery, Graduate School of Medical Sciences, Kyushu University, 3-1-1 Maidashi, Higashi-ku, Fukuoka 812-8582, JapanPurpose: Limited dorsal myeloschisis (LDM) is thought to arise from focal incomplete disjunction between the cutaneous and neural ectoderm during primary neurulation, while spinal lipoma of dorsal-type (dorsal lipoma) arises from premature disjunction. Thus, simultaneous occurrence of an LDM and dorsal lipoma are not surprising, and may represent slightly different perturbations of disjunction caused by the same insult in neighboring loci. However, the clinicopathological findings of the LDM with dorsal lipoma have not been fully determined. Methods: Of 21 patients with LDM, 3 (14.3%) had dorsal lipoma. We retrospectively analyzed the clinicopathological findings of these 3 patients, especially the histopathological distribution of the fibrocollagenous LDM tract and fibroadipose tissue of the lipoma. Results: Patients 1 and 2 had flat skin lesions, while patient 3 had a human tail-like cutaneous appendage. In the tethering stalks linking the skin lesion at the lumbosacral lesion to the low-lying conus medullaris of the three patients, fibrocollagenous tissues embedding adipose tissues at the subcutaneous site, and with abundant adipose tissues at the extradural site, were changed to fibroadipose tissue at the intradural site. While glial fibrillary acidic protein-immunopositive neuroglial tissues were observed in 2 (patients 1 and 2), peripheral nerve fibers were observed in every stalk. Smooth muscle fibers were noted in patient 1, while a large amount of striated muscle fibers were seen in patients 2 and 3. Conclusion: These cases showed various tissues with different origins in the stalk. There may also be a seamless continuation between fibrocollagenous LDM tissue at the distal site and lipomatous tissue at the proximal site. Peripheral nerve fibers and smooth muscle fibers of neural crest origin may be dragged into the stalk during incomplete disjunction, while the striated muscle fibers of mesodermal origin may enter the stalk along with the lipomatous tissues during premature disjunction.http://www.sciencedirect.com/science/article/pii/S2214751920303388Fibrocollagenous tissueFibroadipose tissueGlial fibrillary acidic proteinPeripheral nerve |
| spellingShingle | Nobuya Murakami Takato Morioka Satoshi O. Suzuki Nobutaka Mukae Takafumi Shimogawa Yoshihiro Matsuo Takakazu Sasaguri Masahiro Mizoguchi Clinicopathological findings of limited dorsal myeloschisis associated with spinal lipoma of dorsal-type Interdisciplinary Neurosurgery Fibrocollagenous tissue Fibroadipose tissue Glial fibrillary acidic protein Peripheral nerve |
| title | Clinicopathological findings of limited dorsal myeloschisis associated with spinal lipoma of dorsal-type |
| title_full | Clinicopathological findings of limited dorsal myeloschisis associated with spinal lipoma of dorsal-type |
| title_fullStr | Clinicopathological findings of limited dorsal myeloschisis associated with spinal lipoma of dorsal-type |
| title_full_unstemmed | Clinicopathological findings of limited dorsal myeloschisis associated with spinal lipoma of dorsal-type |
| title_short | Clinicopathological findings of limited dorsal myeloschisis associated with spinal lipoma of dorsal-type |
| title_sort | clinicopathological findings of limited dorsal myeloschisis associated with spinal lipoma of dorsal type |
| topic | Fibrocollagenous tissue Fibroadipose tissue Glial fibrillary acidic protein Peripheral nerve |
| url | http://www.sciencedirect.com/science/article/pii/S2214751920303388 |
| work_keys_str_mv | AT nobuyamurakami clinicopathologicalfindingsoflimiteddorsalmyeloschisisassociatedwithspinallipomaofdorsaltype AT takatomorioka clinicopathologicalfindingsoflimiteddorsalmyeloschisisassociatedwithspinallipomaofdorsaltype AT satoshiosuzuki clinicopathologicalfindingsoflimiteddorsalmyeloschisisassociatedwithspinallipomaofdorsaltype AT nobutakamukae clinicopathologicalfindingsoflimiteddorsalmyeloschisisassociatedwithspinallipomaofdorsaltype AT takafumishimogawa clinicopathologicalfindingsoflimiteddorsalmyeloschisisassociatedwithspinallipomaofdorsaltype AT yoshihiromatsuo clinicopathologicalfindingsoflimiteddorsalmyeloschisisassociatedwithspinallipomaofdorsaltype AT takakazusasaguri clinicopathologicalfindingsoflimiteddorsalmyeloschisisassociatedwithspinallipomaofdorsaltype AT masahiromizoguchi clinicopathologicalfindingsoflimiteddorsalmyeloschisisassociatedwithspinallipomaofdorsaltype |