Evidence map›Paper›PMID 40195250›Full record

ArticleNeurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology2025

Two distinct phenotypes and a novel mutation in limb-girdle muscular dystrophy R7 telethonin-related patients from Thai neuromuscular center.

Tanitnun Paprad, Jakkrit Amornvit, Thippamas Pobsuk, Manasawan Santananukarn, Chamaiporn Taychargumpoo, Worawan Sirichana, Chupong Ittiwut, Rungnapa Ittiwut, Kanya Suphapeetiporn, Nath Pasutharnchat and 1 more

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Article in Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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1 · What the graph read from it

What it found

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2 · The registry

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3 · Its place in the literature

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4 · The record

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5 · Who and what money

Authors and funding

11 authors.

Tanitnun PapradDivision of Neurology, Department of Pediatrics, Faculty of Medicine, Chulalongkorn University, Bangkok, Thailand.ORCID http://orcid.org/0000-0003-2854-4029
Jakkrit AmornvitDivision of Neurology, Department of Medicine, Faculty of Medicine, Chulalongkorn University, Bhumisiri Mangkhalanusorn Building, 7th Floor Zone C, 1873 Rama 4 Road, Pathumwan, Bangkok, 10330, Thailand. jakkrit.a@chula.ac.th.
Thippamas PobsukKing Chulalongkorn Memorial Hospital, Thai Red Cross Society, Bhumisiri Mangkhalanusorn Building, 7th Floor Zone C, 1873 Rama 4 Road, Pathumwan, Bangkok, 10330, Thailand.
Manasawan SantananukarnKing Chulalongkorn Memorial Hospital, Thai Red Cross Society, Bhumisiri Mangkhalanusorn Building, 7th Floor Zone C, 1873 Rama 4 Road, Pathumwan, Bangkok, 10330, Thailand.
Chamaiporn TaychargumpooKing Chulalongkorn Memorial Hospital, Thai Red Cross Society, Bhumisiri Mangkhalanusorn Building, 7th Floor Zone C, 1873 Rama 4 Road, Pathumwan, Bangkok, 10330, Thailand.
Worawan SirichanaDivision of Pulmonary and Critical Care Medicine, Department of Medicine, Faculty of Medicine, Chulalongkorn University, Bangkok, Thailand.
Chupong IttiwutExcellence Center for Genomics and Precision Medicine, King Chulalongkorn Memorial Hospital, the Thai Red Cross Society, Bangkok, 10330, Thailand.
Rungnapa IttiwutExcellence Center for Genomics and Precision Medicine, King Chulalongkorn Memorial Hospital, the Thai Red Cross Society, Bangkok, 10330, Thailand.
Kanya SuphapeetipornExcellence Center for Genomics and Precision Medicine, King Chulalongkorn Memorial Hospital, the Thai Red Cross Society, Bangkok, 10330, Thailand.
Nath PasutharnchatDivision of Neurology, Department of Medicine, Faculty of Medicine, Chulalongkorn University, Bhumisiri Mangkhalanusorn Building, 7th Floor Zone C, 1873 Rama 4 Road, Pathumwan, Bangkok, 10330, Thailand.
Numphung NumkarunarunroteDepartment of Radiology, Faculty of Medicine, Chulalongkorn University, Bangkok, Thailand.

Funding

Ratchadapisek Sompotch GA65/01
6 · The paper itself

Abstract

Limb-girdle muscular dystrophy R7 telethonin-related (LGMDR7) is a rare autosomal recessive disorder caused by TCAP gene mutations. This study described the phenotypic spectrum, genetic characteristics, and muscle magnetic resonance imaging (MRI) findings of patients with LGMDR7. Five patients from three unrelated families with TCAP mutations were retrospectively identified at the Neuromuscular Center at King Chulalongkorn Memorial Hospital. Demographic, clinical, laboratory, and muscle MRI data were collected and analyzed. We observed a mild phenotype associated with asymptomatic/paucisymptomatic hyperCKemia in one family and a classic limb-girdle muscular dystrophy phenotype in two unrelated patients. The novel deletion variant c.136_137del was identified in a compound heterozygous state with c.26_33dup in a family with a mild phenotype. Muscle MRI of four patients revealed consistent sparing of the sartorius muscle in all patients. This study expands the clinical and genetic spectrum of LGMDR7 by demonstrating an asymptomatic/paucisymptomatic hyperCKemia phenotype and identifying the novel c.136_137del variant. The muscle MRI findings highlight a characteristic pattern in which the sartorius muscle is consistently uninvolved. These findings contribute to a better understanding of the disease and assist in developing future diagnostic strategies for affected individuals, specifically by using clinical profiles in conjunction with the characteristics of muscle MRI.

Indexed as

Muscular Dystrophies, Limb-GirdleMutationAdolescentAdultChildConnectinCreatine KinaseFemaleHumansMagnetic Resonance ImagingMaleMiddle AgedMuscle, SkeletalPedigreePhenotypeRetrospective StudiesConnectinCreatine KinaseTCAP protein, humanAsymptomatic/paucisymptomatic hyperCKemiaLimb-girdle muscular dystrophyMuscle magnetic resonance imagingNovel mutationTCAP geneTelethonin

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.