Brain Dev. 1991;13(1):52-5.

A case of the Rett syndrome with acute encephalopathy induced during calcium hopantenate treatment.

Sasaki T, Minagawa M, Yamamoto T, Ichihashi H.

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Department of Pediatrics, Kyorin University School of Medicine, Tokyo, Japan.

Abstract

We report a girl with the Rett syndrome who had acute encephalopathy probably induced by calcium hopantenate (HOPA). This 4-year-6-month-old girl had a history of moderate developmental delay and had received HOPA administration when first admitted at 2 years 6 months of age with hypoglycemia, hyperammonemia, lactic and pyruvic acidemia, and non-ketotic dicarboxylic aciduria. After this episode, she showed the rapid destructive stage of the Rett syndrome, i.e., severe psychomotor retardation with loss of speech, peculiar stereotypic hand movements, autistic behavior and seizures. Despite subsequent investigations, including analysis of urinary metabolites of organic and amino acids, measurement of serum carnitine and a muscle biopsy, we could not clarify the primary metabolic abnormalities in this girl.

PMID:: 2063999; [PubMed - indexed for MEDLINE]

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Acute encephalopathy with hyperammonemia and dicarboxylic aciduria during calcium hopantenate therapy: a patient report. [Brain Dev. 1986]
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Kimura A, Yoshida I, Ono E, Matsuishi T, Yoshino M, Yamashita F, Yamamoto M, Hashimoto T, Shinka T, Kuhara T. Brain Dev. 1986; 8(6):601-5.
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[3 cases of acute encephalopathy associated with calcium hopantenate administration]. [No To Hattatsu. 1983]
[3 cases of acute encephalopathy associated with calcium hopantenate administration].
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Review [Clinical and biochemical studies in a case of acute encephalopathy associated with calcium hopanthenate administration].
Kajita M, Iwase K, Matsumoto M, Kuhara T, Sinka T, Matsumoto I. No To Hattatsu. 1990 May; 22(3):267-73.
Review [Delayed type malignant syndrome and Parkinson's syndrome due to tiapride, Reye-like syndrome induced by calcium hopantenate]. [Ryoikibetsu Shokogun Shirizu. 1999]
Review [Delayed type malignant syndrome and Parkinson's syndrome due to tiapride, Reye-like syndrome induced by calcium hopantenate].
Noda S. Ryoikibetsu Shokogun Shirizu. 1999; (27 Pt 2):538-41.

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pantogab

MW: 233.26 g/mol

MF: C₁₀H₁₉NO₅

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