Family tree intertwined with DNA strands, symbolizing hereditary genetic diseases and diagnostic power of genetic sequencing.

Unlocking the Mystery of Hereditary Gelsolin Amyloidosis: A Korean Family's Genetic Journey

"Whole-exome sequencing (WES) reveals a rare p.D214Y mutation in the GSN gene, marking the first reported case of HGA in a Korean family and offering new hope for diagnosis and treatment."


Hereditary gelsolin amyloidosis (HGA), also known as familial amyloidosis of the Finnish type (FAF), is a rare, autosomal dominant amyloidosis. This means it's passed down through families and only one copy of the mutated gene is needed to cause the condition. HGA is characterized by a trio of distinctive symptoms: corneal lattice dystrophy (an eye disorder), peripheral neuropathy (nerve damage affecting the limbs), and cutis laxa (loose, sagging skin).

While HGA has been identified in various ethnic groups worldwide, including Finnish, Japanese, and Danish families, it had never been reported in Korean patients until now. The typical onset of HGA occurs in the third or fourth decade of life, initially presenting with corneal lattice dystrophy. Over time, individuals develop cutis laxa and slowly progressive neuropathy, leading to a range of debilitating symptoms.

This article delves into the groundbreaking case of a Korean family diagnosed with HGA through advanced whole-exome sequencing (WES). We'll explore how the identification of a specific genetic mutation, p.D214Y in the gelsolin (GSN) gene, not only provided a definitive diagnosis for this family but also highlights the increasing role of WES in diagnosing rare and complex neuropathies.

The Diagnostic Puzzle: Unraveling the Symptoms

Family tree intertwined with DNA strands, symbolizing hereditary genetic diseases and diagnostic power of genetic sequencing.

The index patient, a 58-year-old Korean male, presented with a constellation of concerning symptoms, including involuntary facial twitching, progressive bilateral facial weakness, and tongue atrophy. These symptoms had been gradually worsening over four years, since the age of 54. Compounding the diagnostic challenge, his mother, maternal uncle, two sisters, and son exhibited similar symptoms, pointing towards a possible hereditary condition.

Neurological examination revealed subtle yet significant findings. The patient displayed mild weakness in the orbicularis oculi (muscles around the eyes) and orbicularis oris muscles (muscles around the mouth). He also reported paresthesia (abnormal sensations) in his right hand, intermittent cramps in his right calf, and decreased sweating in both hands. Notably, he had no ophthalmic complaints, which is atypical for HGA.

To further investigate the cause of these symptoms, a series of electrophysiological studies were conducted:
  • Nerve conduction study (NCS): Revealed reduced compound muscle action potential (CMAP) amplitudes and conduction velocity in the left facial nerve.
  • Electromyography (EMG): Showed reduced recruitment of motor unit action potential in specific muscles.
  • Autonomic function tests (AFTs): Indicated sympathetic skin response abnormalities.
Given the complex presentation and family history, the medical team turned to whole-exome sequencing (WES), a powerful tool for identifying genetic mutations across the entire protein-coding region of the genome. WES revealed a heterozygous p.D214Y mutation in the gelsolin (GSN) gene in affected family members. This mutation has been previously linked to HGA in other populations, confirming the diagnosis in this Korean family.

The Power of WES: A New Era for Diagnosing Rare Diseases

This case marks the first reported instance of HGA in a Korean family and highlights the crucial role of WES in diagnosing rare and often misdiagnosed conditions. While traditional diagnostic approaches rely on recognizing specific clinical features, the phenotypic manifestations of HGA can vary, making diagnosis challenging.

WES offers a powerful alternative, allowing clinicians to identify the underlying genetic cause, even in the absence of classic symptoms. In this case, WES not only confirmed the diagnosis of HGA but also facilitated genetic counseling for the family, enabling them to understand the inheritance pattern and assess the risk for future generations.

The successful diagnosis of HGA in this Korean family underscores the transformative potential of genomic medicine. As WES becomes more accessible and affordable, it is poised to play an increasingly important role in diagnosing and managing a wide range of rare and complex genetic disorders, offering hope for improved outcomes and personalized treatment strategies.

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This article is based on research published under:

DOI-LINK: 10.3343/alm.2016.36.3.259, Alternate LINK

Title: The First Korean Family With Hereditary Gelsolin Amyloidosis Caused By P.D214Y Mutation In The Gsn Gene

Subject: Biochemistry (medical)

Journal: Annals of Laboratory Medicine

Publisher: Annals of Laboratory Medicine

Authors: Kyoung-Jin Park, Jong-Ho Park, June-Hee Park, Eun Bin Cho, Byoung Joon Kim, Jong-Won Kim

Published: 2016-05-01

Everything You Need To Know

1

What is Hereditary Gelsolin Amyloidosis (HGA)?

Hereditary Gelsolin Amyloidosis (HGA), also known as familial amyloidosis of the Finnish type (FAF), is a rare genetic disorder. It's characterized by a set of symptoms including corneal lattice dystrophy, peripheral neuropathy, and cutis laxa. This means that the body produces an abnormal form of the protein gelsolin, leading to the accumulation of amyloid deposits in various tissues. This can lead to the trio of distinctive symptoms.

2

What is the p.D214Y mutation in the GSN gene, and what is its role?

The p.D214Y mutation in the GSN gene is a specific change in the DNA sequence of the gelsolin gene (GSN). This mutation leads to the production of an abnormal gelsolin protein that forms amyloid deposits. This mutation was found in the Korean family affected by Hereditary Gelsolin Amyloidosis (HGA). The identification of this mutation was key to confirming the diagnosis in this family.

3

How did whole-exome sequencing (WES) help in this case?

Whole-exome sequencing (WES) is a powerful genetic testing method that analyzes the protein-coding regions of genes. It was used to diagnose Hereditary Gelsolin Amyloidosis (HGA) in a Korean family. Because symptoms of HGA can be variable, WES helped to identify the specific genetic mutation (p.D214Y in the GSN gene) that caused the condition. This highlights the increasing role of WES in diagnosing rare and complex conditions like HGA.

4

Why is this case of Hereditary Gelsolin Amyloidosis (HGA) significant?

The significance of this case lies in the groundbreaking diagnosis of Hereditary Gelsolin Amyloidosis (HGA) in a Korean family, marking the first reported instance in this population. This diagnosis was made possible through Whole-exome sequencing (WES). The identification of the p.D214Y mutation in the GSN gene provides a definitive diagnosis and opens avenues for early detection and potential treatment strategies, improving the understanding of the disease worldwide.

5

What are the common symptoms of Hereditary Gelsolin Amyloidosis (HGA) and how did they present in this case?

The symptoms of Hereditary Gelsolin Amyloidosis (HGA) typically begin in the third or fourth decade of life. The early symptoms include corneal lattice dystrophy, followed by the development of cutis laxa, and progressive neuropathy. In the case described, the Korean patient presented with involuntary facial twitching, weakness, and tongue atrophy, which is consistent with the neuropathy associated with the condition. The family history and Whole-exome sequencing (WES) results helped confirm the diagnosis despite the atypical presentation.

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