TY - JOUR
T1 - Report on a Case with Moreno-Nishimura-Schmidt Overgrowth Syndrome
T2 - A Clinically Delineated Disease Yet of an Unknown Origin!
AU - Mehawej, Cybel
AU - Chouery, Eliane
AU - Al Hage Chehade, Ghada
AU - Bejaoui, Yosra
AU - Mahfoud, Daniel
AU - Gerges, Maya
AU - Delague, Valérie
AU - El Hajj, Nady
AU - Megarbane, Andre
N1 - Publisher Copyright:
© 2023 S. Karger AG. All rights reserved.
PY - 2023/6/1
Y1 - 2023/6/1
N2 - Introduction: Overgrowth syndromes are a heterogeneous group of genetic disorders characterized by excessive growth, often accompanied by additional clinical features, such as facial dysmorphism, hormonal imbalances, cognitive impairment, and increased risk for neoplasia. Moreno-Nishimura-Schmidt (M-N-S) overgrowth syndrome is a very rare overgrowth syndrome characterized by severe pre- and postnatal overgrowth, dysmorphic facial features, kyphoscoliosis, large hands and feet, inguinal hernia, and distinctive skeletal features. The clinical and radiological features of the disorder have been well delineated, yet its molecular pathogenesis remains unclear. Case Presentation: We report on a Lebanese boy with M-N-S syndrome, whose clinical manifestations were compared with those of previously reported 5 affected individuals. Whole-exome sequencing combined with comparative genome hybridization analysis failed to delineate the molecular basis of the phenotype. However, epigenetic studies revealed a different methylation status of several CpG sites between him and healthy controls, with methyltransferase activity showing the most significant enrichment. Conclusion: An additional case of M-N-S syndrome recapitulated the clinical and radiological manifestations described in the previous reports. The data in the epigenetic studies implicated that abnormal methylations might play an essential role in development of the disease phenotype. However, additional studies in a clinically homogeneous cohort of patients are crucial to confirm this hypothesis.
AB - Introduction: Overgrowth syndromes are a heterogeneous group of genetic disorders characterized by excessive growth, often accompanied by additional clinical features, such as facial dysmorphism, hormonal imbalances, cognitive impairment, and increased risk for neoplasia. Moreno-Nishimura-Schmidt (M-N-S) overgrowth syndrome is a very rare overgrowth syndrome characterized by severe pre- and postnatal overgrowth, dysmorphic facial features, kyphoscoliosis, large hands and feet, inguinal hernia, and distinctive skeletal features. The clinical and radiological features of the disorder have been well delineated, yet its molecular pathogenesis remains unclear. Case Presentation: We report on a Lebanese boy with M-N-S syndrome, whose clinical manifestations were compared with those of previously reported 5 affected individuals. Whole-exome sequencing combined with comparative genome hybridization analysis failed to delineate the molecular basis of the phenotype. However, epigenetic studies revealed a different methylation status of several CpG sites between him and healthy controls, with methyltransferase activity showing the most significant enrichment. Conclusion: An additional case of M-N-S syndrome recapitulated the clinical and radiological manifestations described in the previous reports. The data in the epigenetic studies implicated that abnormal methylations might play an essential role in development of the disease phenotype. However, additional studies in a clinically homogeneous cohort of patients are crucial to confirm this hypothesis.
KW - Clinical genetics
KW - Epigenetics
KW - Moreno-Nishimura-Schmidt overgrowth Syndrome
KW - Rare syndrome
UR - http://www.scopus.com/inward/record.url?scp=85147435472&partnerID=8YFLogxK
U2 - 10.1159/000527215
DO - 10.1159/000527215
M3 - Article
AN - SCOPUS:85147435472
SN - 1661-8769
VL - 14
SP - 219
EP - 224
JO - Molecular Syndromology
JF - Molecular Syndromology
IS - 3
ER -