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Histochemical and molecular genetic study of MELAS and MERRF in Korean patients.

Mitochondrial myopathy, encephalopathy, lactic acidosis and stroke-like episode (MELAS) and myoclonic epilepsy and ragged-red fibers (MERRF) are rare disorders caused by point mutation of the tRNA gene of the mitochondrial genome. To understand the pathogenetic mechanism of MELAS and MERRF, we studi...

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Autores principales: Kim, Dae Seong, Jung, Dae Soo, Park, Kyu Hyun, Kim, In Joo, Kim, Cheol Min, Lee, Won Ho, Rho, Soon Ki
Formato: Texto
Lenguaje:English
Publicado: Korean Academy of Medical Sciences 2002
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3054831/
https://www.ncbi.nlm.nih.gov/pubmed/11850598
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author Kim, Dae Seong
Jung, Dae Soo
Park, Kyu Hyun
Kim, In Joo
Kim, Cheol Min
Lee, Won Ho
Rho, Soon Ki
author_facet Kim, Dae Seong
Jung, Dae Soo
Park, Kyu Hyun
Kim, In Joo
Kim, Cheol Min
Lee, Won Ho
Rho, Soon Ki
author_sort Kim, Dae Seong
collection PubMed
description Mitochondrial myopathy, encephalopathy, lactic acidosis and stroke-like episode (MELAS) and myoclonic epilepsy and ragged-red fibers (MERRF) are rare disorders caused by point mutation of the tRNA gene of the mitochondrial genome. To understand the pathogenetic mechanism of MELAS and MERRF, we studied four patients. Serially sectioned frozen muscle specimens with a battery of histochemical stains were reviewed under light microscope and ultrastructural changes were observed under electron microscope. The polymerase chain reaction-restriction fragment length polymorphism (PCR-RFLP) analysis was performed and the tRNA genes were sequenced to confirm mutations. In two patients with MELAS, strongly succinyl dehydrogenase positive blood vessels (SSVs) and many cytochrome oxidase (COX) positive ragged-red fibers (RRFs) were observed, and A3243G mutations were found from the muscle samples. In two patients with MERRF, neither SSV nor COX positive RRFs were seen and A8344G mutations were found from both muscle and blood samples. In the two MERRF families, the identical mutation was observed among family members. The failure to detect the mutation in blood samples of the MELAS suggests a low mutant load in blood cells. The histochemical methods including COX stain are useful for the confirmation and differentiation of mitochondrial diseases. Also, molecular biological study using muscle sample seems essential for the confirmation of the mtDNA mutation.
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spelling pubmed-30548312011-03-15 Histochemical and molecular genetic study of MELAS and MERRF in Korean patients. Kim, Dae Seong Jung, Dae Soo Park, Kyu Hyun Kim, In Joo Kim, Cheol Min Lee, Won Ho Rho, Soon Ki J Korean Med Sci Research Article Mitochondrial myopathy, encephalopathy, lactic acidosis and stroke-like episode (MELAS) and myoclonic epilepsy and ragged-red fibers (MERRF) are rare disorders caused by point mutation of the tRNA gene of the mitochondrial genome. To understand the pathogenetic mechanism of MELAS and MERRF, we studied four patients. Serially sectioned frozen muscle specimens with a battery of histochemical stains were reviewed under light microscope and ultrastructural changes were observed under electron microscope. The polymerase chain reaction-restriction fragment length polymorphism (PCR-RFLP) analysis was performed and the tRNA genes were sequenced to confirm mutations. In two patients with MELAS, strongly succinyl dehydrogenase positive blood vessels (SSVs) and many cytochrome oxidase (COX) positive ragged-red fibers (RRFs) were observed, and A3243G mutations were found from the muscle samples. In two patients with MERRF, neither SSV nor COX positive RRFs were seen and A8344G mutations were found from both muscle and blood samples. In the two MERRF families, the identical mutation was observed among family members. The failure to detect the mutation in blood samples of the MELAS suggests a low mutant load in blood cells. The histochemical methods including COX stain are useful for the confirmation and differentiation of mitochondrial diseases. Also, molecular biological study using muscle sample seems essential for the confirmation of the mtDNA mutation. Korean Academy of Medical Sciences 2002-02 /pmc/articles/PMC3054831/ /pubmed/11850598 Text en
spellingShingle Research Article
Kim, Dae Seong
Jung, Dae Soo
Park, Kyu Hyun
Kim, In Joo
Kim, Cheol Min
Lee, Won Ho
Rho, Soon Ki
Histochemical and molecular genetic study of MELAS and MERRF in Korean patients.
title Histochemical and molecular genetic study of MELAS and MERRF in Korean patients.
title_full Histochemical and molecular genetic study of MELAS and MERRF in Korean patients.
title_fullStr Histochemical and molecular genetic study of MELAS and MERRF in Korean patients.
title_full_unstemmed Histochemical and molecular genetic study of MELAS and MERRF in Korean patients.
title_short Histochemical and molecular genetic study of MELAS and MERRF in Korean patients.
title_sort histochemical and molecular genetic study of melas and merrf in korean patients.
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3054831/
https://www.ncbi.nlm.nih.gov/pubmed/11850598
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