Abstract
We measured strikingly increased urinary and serum levels of catecholamines in a newborn with gangrene of the extremities, hypertension (BP 200/160), myocarditis, enterocolitis, hematologic abnormalities (thrombocytopenia, hyperkininemia with edema, disseminated intravascular coagulation) renal disease (proteinuria, oliguria), seizures and coma. Total serum catechols were 6.0 ng/ml (normal 0.2-0.5 ng/ml). Urine catechol levels in ug/mg creatinine were determined before and after 12 hrs. of therapy with α-methyl-paratyrosine (α-MPT), an inhibitor of endogenous catechol synthesis.
Phentolamine test was positive; treatment with peripheral alpha-blockade normalized blood pressure. During one month of α-MPT therapy, all chemical and physical abnormalities were largely reversed. Two attempts to taper α-MPT were followed by hypertension. Following death attributable to sepsis and DIC, extensive autopsy failed to reveal a catechol source. Because catechol excess is a potentially treatable cause of this catastrophic circulatory disorder, it should be considered in the differential diagnosis of hypertension and of peripheral gangrene in infants.
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Stalcup, S., Pang, L., Driscoll, J. et al. CATECHOLAMINE EXCESS IN THE NEWBORN: A NEW SYNDROME. Pediatr Res 11, 432 (1977). https://doi.org/10.1203/00006450-197704000-00375
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DOI: https://doi.org/10.1203/00006450-197704000-00375