Nonketotic hyperglycinemia in two siblings with neonatal seizures.

نویسندگان

  • Duangrurdee Wattanasirichaigoon
  • Anannit Visudtibhan
  • Suchart Phudhichareonrat
  • Surang Chiemchanya
  • Preeya Leelahagul
  • Kannika Suwan
  • Sarayut Supapannachart
چکیده

Seizures are a common problem in neonates. Differential diagnoses include infection, trauma, hypoxia and congenital metabolic disorders. Among these, congenital metabolic disorder is less familiar to general pediatricians. We report two patients with nonketotic hyperglycinemia (NKH), a rare and lethal congenital metabolic disease. Transient hyperammonemia and transient hypouricemia, uncommon features found in NKH, were detected in one patient. High doses of sodium benzoate and dextromethorphan failed to modify the clinical course. Neuropathology denoted characteristic diffuse vacuolization and changes in reactive and gliotic astrocytes. The clinical course, biochemical findings, diagnostic approaches and diagnostic tests are discussed in detail. Recent modalities of treatment are reviewed. Because of its rarity and rapidly progressive course, it maybe underdiagnosed resulting in death before being recognized. Awareness of the possibility of congenital metabolic disorder in early neonatal catastrophe will increase the diagnostic rate.

برای دانلود متن کامل این مقاله و بیش از 32 میلیون مقاله دیگر ابتدا ثبت نام کنید

ثبت نام

اگر عضو سایت هستید لطفا وارد حساب کاربری خود شوید

منابع مشابه

A novel mutation in the glycine decarboxylase gene in patient with non-ketotic hyperglycinemia

Non-ketotic hyperglycinemia (NKH) is a rare inborn error of metabolism and is caused by a glycine cleavage system deficiency. Eighty-five percent of patients present with the neonatal type of NKH, the infants initially develop lethargy, seizures, and episodes of apnea, and most often death. Between 60-90% of cases are caused by mutations in the glycine decarboxylase (GLDC). We believed that mor...

متن کامل

Biochemical and molecular predictors for prognosis in nonketotic hyperglycinemia

OBJECTIVE Nonketotic hyperglycinemia is a neurometabolic disorder characterized by intellectual disability, seizures, and spasticity. Patients with attenuated nonketotic hyperglycinemia make variable developmental progress. Predictive factors have not been systematically assessed. METHODS We reviewed 124 patients stratified by developmental outcome for biochemical and molecular predictive fac...

متن کامل

Neonates with inborn errors of metabolism: spectrum and short-term outcomes at a tertiary care hospital.

We aimed to evaluate the neonates diagnosed as IEM in our neonatal intensive care unit and their outcomes. Among 2994 neonates hospitalized, 51 were diagnosed as IEM (1.7%). Admission complaints were poor feeding, decreased activity, jaundice, seizures, abnormal screening and respiratory problems. Phenylketonuria (11), organic acidemias (8), maple syrup urine disease (5), citrullinemia (5), gal...

متن کامل

Two novel mutations in the glycine decarboxylase gene in a boy with classic nonketotic hyperglycinemia: case report.

Nonketotic hyperglycinemia is an extremely rare autosomal recessively inherited glycine encephalopathy caused by a deficiency in the mitochondrial glycine cleavage system, which leads to severe clinical symptoms. Nonketotic hyperglycinemia is characterized by complex and diverse phenotypes, such as hypotonia, seizures, cognitive impairment, developmental delays and myoclonic jerks that may lead...

متن کامل

A Baby with Nonketotic Hyperglycinaemia

Nonketot ic hyperglyc inaemia or nonketo t ic hyperglycinaemic encephalopathy (NKH) is an autosomal recessive inborn error of glycine metabolism with poor prognosis. The inherited defect of the glycine cleavage system leads to accumulation of glycine in body fluids, such as plasma, urine and particularly, cerebrospinal fluid (CSF). In the neonatal type, NKH generally presents with intractable s...

متن کامل

ذخیره در منابع من


  با ذخیره ی این منبع در منابع من، دسترسی به آن را برای استفاده های بعدی آسان تر کنید

برای دانلود متن کامل این مقاله و بیش از 32 میلیون مقاله دیگر ابتدا ثبت نام کنید

ثبت نام

اگر عضو سایت هستید لطفا وارد حساب کاربری خود شوید

عنوان ژورنال:
  • The Southeast Asian journal of tropical medicine and public health

دوره 34 1  شماره 

صفحات  -

تاریخ انتشار 2003