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Mitochondrial Neurogastrointestinal Encephalomyopathy Syndrome Treated with Stem Cell Transplant: A Case Series and Literature Review.
- Source :
-
Experimental and clinical transplantation : official journal of the Middle East Society for Organ Transplantation [Exp Clin Transplant] 2018 Dec; Vol. 16 (6), pp. 773-778. Date of Electronic Publication: 2017 Apr 14. - Publication Year :
- 2018
-
Abstract
- Mitochondrial neurogastrointestinal encephalomyopathy syndrome is a rare autosomal recessive multisystem disorder caused by nuclear TYMP gene mutations, which leads to deficiency in thymidine phosphorylase enzyme. This deficiency then leads to mitochondrial dysfunction, which causes the features characteristic of this syndrome, including severe muscle wasting, gastrointestinal dysmotility, leukoencephalopathy, peripheral neuropathy, and ophthalmoplegia. Here, we present a case series of 3 patients with mitochondrial neurogastrointestinal encephalomyopathy from Saudi Arabia who underwent allogeneic stem cell transplant at King Faisal Specialist Hospital (Riyadh, Saudi Arabia). Two patients died within the first year of transplant, and the third is still alive but without improvement in clinical features. Allogeneic hematopoietic stem cell transplant-related mortality appears to be high; this may at least be partially related to established end-organ effects with decreased performance status. Although allogeneic hematopoietic stem cell transplant clearly affects correction of genetic and biochemical defects in mitochondrial neurogastrointestinal encephalomyopathy, its ability to reverse or improve established clinical manifestations has not been proven.
- Subjects :
- Adolescent
Adult
Fatal Outcome
Genetic Predisposition to Disease
Humans
Intestinal Pseudo-Obstruction diagnosis
Intestinal Pseudo-Obstruction enzymology
Intestinal Pseudo-Obstruction genetics
Male
Muscular Dystrophy, Oculopharyngeal diagnosis
Muscular Dystrophy, Oculopharyngeal enzymology
Muscular Dystrophy, Oculopharyngeal genetics
Mutation
Ophthalmoplegia congenital
Phenotype
Thymidine Phosphorylase deficiency
Thymidine Phosphorylase genetics
Time Factors
Transplantation, Homologous
Treatment Outcome
Hematopoietic Stem Cell Transplantation
Intestinal Pseudo-Obstruction surgery
Muscular Dystrophy, Oculopharyngeal surgery
Subjects
Details
- Language :
- English
- ISSN :
- 2146-8427
- Volume :
- 16
- Issue :
- 6
- Database :
- MEDLINE
- Journal :
- Experimental and clinical transplantation : official journal of the Middle East Society for Organ Transplantation
- Publication Type :
- Academic Journal
- Accession number :
- 28411356
- Full Text :
- https://doi.org/10.6002/ect.2016.0109