Association between cloacal exstrophy and two limited dorsal myeloschisis lesions: A case report and literature review

Background: The term limited dorsal myeloschisis (LDM) was coined by Pang in 1993. It involves incomplete obliteration of the dorsal neural fold in primary neurulation. On the other hand, Cloacal Exstrophy is one of the most complex endodermal anomalies and is usually associated with spinal anomali...

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Main Authors: Abdulrahman Jafar Sabbagh, Rana Moshref, Faisal Asiri, Abdulaziz Basurrah, Abdulrahman Arafah, Jamal Kamal
Format: Article
Language:English
Published: Knowledge E 2022-07-01
Series:Journal of Spine Practice
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Online Access:https://knepublishing.com/index.php/jsp/article/view/10492
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author Abdulrahman Jafar Sabbagh
Rana Moshref
Faisal Asiri
Abdulaziz Basurrah
Abdulrahman Arafah
Jamal Kamal
author_facet Abdulrahman Jafar Sabbagh
Rana Moshref
Faisal Asiri
Abdulaziz Basurrah
Abdulrahman Arafah
Jamal Kamal
author_sort Abdulrahman Jafar Sabbagh
collection DOAJ
description Background: The term limited dorsal myeloschisis (LDM) was coined by Pang in 1993. It involves incomplete obliteration of the dorsal neural fold in primary neurulation. On the other hand, Cloacal Exstrophy is one of the most complex endodermal anomalies and is usually associated with spinal anomalies; hence, the term OEIS complex was coined (Omphalocele, Exstrophy of the cloaca, Imperforate anus, and Spinal deformities complex). Case Description: We report the coexistence of two LDM lesions in tandem (originating at L2 and S2 levels) in a child diagnosed with cloacal exstrophy. Initially, prenatal ultrasound detected the lumbar but not the sacral lesion. The patient was surgically untethered under intraoperative neurophysiologic monitoring (IOM) at four years of age, and this paper reports his one-year follow-up. Conclusion: Cases of cloacal exstrophy must always be investigated for spinal cord malformations. LDM is rare and requires careful diagnosis. MRI should be done for the whole neuroaxis to rule out associated conditions, including multiplicity. Surgery should be done under IOM to avoid long-term complications.
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spelling doaj-art-17907de3ffb34c13a4a4c1ddbe81cbcd2025-08-20T02:04:53ZengKnowledge EJournal of Spine Practice2789-94542789-94622022-07-011210.18502/jsp.v1i2.10492Association between cloacal exstrophy and two limited dorsal myeloschisis lesions: A case report and literature reviewAbdulrahman Jafar Sabbagh0Rana Moshref1Faisal Asiri2Abdulaziz Basurrah3Abdulrahman Arafah4Jamal Kamal5Neurosurgery Division, Department of Surgery, College of Medicine, King Abdulaziz University, Jeddah, Saudi ArabiaNeurosurgery Division, Department of Surgery, College of Medicine, King Abdulaziz University, Jeddah, Saudi ArabiaNeurosurgery Division, Department of Surgery, College of Medicine, King Abdulaziz University, Jeddah, Saudi ArabiaNeurosurgery Division, Department of Surgery, College of Medicine, King Abdulaziz University, Jeddah, Saudi ArabiaDepartment of Surgery, Faculty of Medicine of Rabigh, King Abdulaziz University, Saudi ArabiaDivision of Pediatric Surgery, Department of Surgery, College of Medicine, King Abdulaziz University, Saudi Arabia Background: The term limited dorsal myeloschisis (LDM) was coined by Pang in 1993. It involves incomplete obliteration of the dorsal neural fold in primary neurulation. On the other hand, Cloacal Exstrophy is one of the most complex endodermal anomalies and is usually associated with spinal anomalies; hence, the term OEIS complex was coined (Omphalocele, Exstrophy of the cloaca, Imperforate anus, and Spinal deformities complex). Case Description: We report the coexistence of two LDM lesions in tandem (originating at L2 and S2 levels) in a child diagnosed with cloacal exstrophy. Initially, prenatal ultrasound detected the lumbar but not the sacral lesion. The patient was surgically untethered under intraoperative neurophysiologic monitoring (IOM) at four years of age, and this paper reports his one-year follow-up. Conclusion: Cases of cloacal exstrophy must always be investigated for spinal cord malformations. LDM is rare and requires careful diagnosis. MRI should be done for the whole neuroaxis to rule out associated conditions, including multiplicity. Surgery should be done under IOM to avoid long-term complications. https://knepublishing.com/index.php/jsp/article/view/10492Limited dorsal myeloschisisTethered cordCloacal exstrophyOEIS complex
spellingShingle Abdulrahman Jafar Sabbagh
Rana Moshref
Faisal Asiri
Abdulaziz Basurrah
Abdulrahman Arafah
Jamal Kamal
Association between cloacal exstrophy and two limited dorsal myeloschisis lesions: A case report and literature review
Journal of Spine Practice
Limited dorsal myeloschisis
Tethered cord
Cloacal exstrophy
OEIS complex
title Association between cloacal exstrophy and two limited dorsal myeloschisis lesions: A case report and literature review
title_full Association between cloacal exstrophy and two limited dorsal myeloschisis lesions: A case report and literature review
title_fullStr Association between cloacal exstrophy and two limited dorsal myeloschisis lesions: A case report and literature review
title_full_unstemmed Association between cloacal exstrophy and two limited dorsal myeloschisis lesions: A case report and literature review
title_short Association between cloacal exstrophy and two limited dorsal myeloschisis lesions: A case report and literature review
title_sort association between cloacal exstrophy and two limited dorsal myeloschisis lesions a case report and literature review
topic Limited dorsal myeloschisis
Tethered cord
Cloacal exstrophy
OEIS complex
url https://knepublishing.com/index.php/jsp/article/view/10492
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