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The use of 5-hydroxytryptophan in a child with Lesch-Nyhan syndrome

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Abstract

The effects of 5-hydroxytryptophan (5-HTP), a serotonin precursor, were studied in a boy with the Lesch-Nyhan syndrome. During the course of drug treatment, self-mutilation, crying, sleep state architecture, serum dopamine B-hydroxylase (DBH), and cerebrospinal fluid levels of 5-hydroxyindolylacetic acid and homovanillic acid were studied. Treatment with 5-HTP failed to affect this child's biting behavior. However, the drug significantly reduced irritability as measured by crying time. Moreover, 5-HTP may have affected sleep state architecture, making it more normal in character. Serum DBH levels were normal throughout the study. Neither the patient's mother nor his maternal grandmother showed a hypertensive response on the cold pressor test.

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Authors and Affiliations

  1. Stanford University School of Medicine, 94305, Stanford, California

    Roland D. Ciaranello MD (Resident in Psychiatry), Thomas F. Anders MD, Jack D. Barchas MD, Philip A. Berger MD & Howard M. Cann MD

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  1. Roland D. Ciaranello MD
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  2. Thomas F. Anders MD
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  3. Jack D. Barchas MD
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  4. Philip A. Berger MD
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  5. Howard M. Cann MD
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Additional information

The authors wish to express their gratitude to Mrs. Pamela Angwin, Mrs. Martha Bowden, and Miss Donna Wong for performing the biochemical assays; to Ms. Debbie Zarin and Dr. Helena Kraemer for assistance in the statistical analyses; to the nursing staff of the Clinical Research Center for their outstanding clinical care of the patient; and to Dr. William Nyhan for performing the HGPRT assays required to establish the diagnosis of Lesch-Nyhan syndrome and for calling the authors' attention to the use of 5-HTP in this illness. This investigation was supported by Public Health Service Research Grant R.R. 70 from the General Clinical Research Center, Division of Research Resources, and in part by National Institute of Health Grants MH23861 and MH 25998.

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Ciaranello, R.D., Anders, T.F., Barchas, J.D. et al. The use of 5-hydroxytryptophan in a child with Lesch-Nyhan syndrome. Child Psych Hum Dev 7, 127–133 (1976). https://doi.org/10.1007/BF01464036

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  • DOI: https://doi.org/10.1007/BF01464036

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