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Title: Functionally active ganglioneuroma with increased plasma and urinary catecholamines and positive iodine 131-meta-iodobenzylguanidine scintigraphy

Abstract

Ganglioneuromas are usually considered not to be functionally active. Studies of their catecholamine excretory pattern and of their imaging by means of the adrenergic tracing agent 131-I-MIBG have been therefore sparse. We report on a case of secretory ganglioneuroma, as demonstrated by the increased urinary excretion of the catecholamine metabolites HVA and VMA, increased plasma dopamine and epinephrine levels, and positive 131-I-MIBG scintigraphy. We must therefore be aware that a functionally active tumor is not necessarily a neuroblastoma, and that the diagnosis should be biopsy proven.

Authors:
; ; ; ; ;  [1]
  1. (Univ. La Sapienza, Rome (Italy))
Publication Date:
OSTI Identifier:
5381460
Resource Type:
Journal Article
Resource Relation:
Journal Name: Medical and Pediatric Oncology; (United States); Journal Volume: 19:4
Country of Publication:
United States
Language:
English
Subject:
62 RADIOLOGY AND NUCLEAR MEDICINE; ADRENALINE; BIOSYNTHESIS; CATECHOLAMINES; RENAL CLEARANCE; DOPAMINE; NEOPLASMS; DIAGNOSIS; IODINE 131; SCINTISCANNING; ADRENAL HORMONES; AMINES; AROMATICS; AUTONOMIC NERVOUS SYSTEM AGENTS; BETA DECAY RADIOISOTOPES; BETA-MINUS DECAY RADIOISOTOPES; CARDIOTONICS; CARDIOVASCULAR AGENTS; CLEARANCE; COUNTING TECHNIQUES; DAYS LIVING RADIOISOTOPES; DIAGNOSTIC TECHNIQUES; DISEASES; DRUGS; EXCRETION; HORMONES; HYDROXY COMPOUNDS; INTERMEDIATE MASS NUCLEI; IODINE ISOTOPES; ISOTOPES; NEUROREGULATORS; NUCLEI; ODD-EVEN NUCLEI; ORGANIC COMPOUNDS; PHENOLS; POLYPHENOLS; RADIOISOTOPE SCANNING; RADIOISOTOPES; SYMPATHOMIMETICS; SYNTHESIS; 550601* - Medicine- Unsealed Radionuclides in Diagnostics

Citation Formats

Clerico, A., Jenkner, A., Castello, M.A., Ciofetta, G., Lucarelli, C., and Codini, M. Functionally active ganglioneuroma with increased plasma and urinary catecholamines and positive iodine 131-meta-iodobenzylguanidine scintigraphy. United States: N. p., 1991. Web. doi:10.1002/mpo.2950190421.
Clerico, A., Jenkner, A., Castello, M.A., Ciofetta, G., Lucarelli, C., & Codini, M. Functionally active ganglioneuroma with increased plasma and urinary catecholamines and positive iodine 131-meta-iodobenzylguanidine scintigraphy. United States. doi:10.1002/mpo.2950190421.
Clerico, A., Jenkner, A., Castello, M.A., Ciofetta, G., Lucarelli, C., and Codini, M. 1991. "Functionally active ganglioneuroma with increased plasma and urinary catecholamines and positive iodine 131-meta-iodobenzylguanidine scintigraphy". United States. doi:10.1002/mpo.2950190421.
@article{osti_5381460,
title = {Functionally active ganglioneuroma with increased plasma and urinary catecholamines and positive iodine 131-meta-iodobenzylguanidine scintigraphy},
author = {Clerico, A. and Jenkner, A. and Castello, M.A. and Ciofetta, G. and Lucarelli, C. and Codini, M.},
abstractNote = {Ganglioneuromas are usually considered not to be functionally active. Studies of their catecholamine excretory pattern and of their imaging by means of the adrenergic tracing agent 131-I-MIBG have been therefore sparse. We report on a case of secretory ganglioneuroma, as demonstrated by the increased urinary excretion of the catecholamine metabolites HVA and VMA, increased plasma dopamine and epinephrine levels, and positive 131-I-MIBG scintigraphy. We must therefore be aware that a functionally active tumor is not necessarily a neuroblastoma, and that the diagnosis should be biopsy proven.},
doi = {10.1002/mpo.2950190421},
journal = {Medical and Pediatric Oncology; (United States)},
number = ,
volume = 19:4,
place = {United States},
year = 1991,
month = 1
}
  • Iodine-123 (I-123) meta-iodobenzylguanidine (MIBG) imaging was performed in 31 patients. Three patients were without cardiac disease and 28 had idiopathic dilated cardiomyopathy with various degrees of left ventricular dysfunction. The qualitatively assessed myocardial I-123 MIBG scintigrams and the myocardial versus mediastinal I-123 MIBG uptake ratio were related to I-123 MIBG activity and norepinephrine concentration determined from endomyocardial biopsy samples taken from the right side of the interventricular septum. Scintigrams and the MIBG uptake ratio were also related to plasma catecholamine concentrations, left ventricular ejection fraction and New York Heart Association functional class. Patients with distinct myocardial I-123 MIBG uptake (scoremore » 1) had a normal ejection fraction (58 +/- 16%). Patients with diffusely reduced uptake or scintigraphic defects (score 2) had a significantly lower ejection fraction (38 +/- 9%, p less than 0.05), whereas patients with shadowy or no visible myocardial uptake (score 3) had the lowest ejection fraction (23 +/- 6%, p less than 0.002 versus patients with score 2). The scintigraphically determined I-123 MIBG activity in the septal region correlated significantly with I-123 MIBG activity from the endomyocardial biopsy samples (r = 0.78, p less than 0.001, n = 9). The myocardial versus mediastinal I-123 MIBG activity ratio was significantly related to myocardial norepinephrine concentration (r = 0.63, n = 28) and to left ventricular ejection fraction (r = 0.74, n = 31). These data suggest that myocardial I-123 MIBG scintigraphy is a useful noninvasive method for the assessment of myocardial adrenergic nervous system disintegrity in patients with idiopathic dilated cardiomyopathy.« less
  • Heart intensity (HI) in the 24- and 48-hr images of meta-(/sup 131/I)iodobenzylguanidine (I-131 MIBG), a pheochromocytoma-seeking guanethidine analog, were compared with concentrations of plasma and urinary catecholamines and their metabolites in nonpheochromocytoma and pheochromocytoma patients. HI was inversely related to plasma concentrations and urinary excretion rates of the hormones. Plasma norepinephrine had the highest inverse correlation with HI (r = -0.73 at 24 hr, -0.63 at 48 hr) and urinary metanephrine the lowest (r = -0.23 at 24 hr, -0.28 at 48 hr). A similar relationship was observed in the intensity of salivary-gland visualization, but with less marked variations. HImore » was much higher in ninpheochromocytoma patients than in pheochromocytoma patients. HI in an I-131 MIGB image provides useful information in the diagnosis of pheochromocytoma, and may provide a tool for the study of the influence of catecholamines on the heart.« less
  • Radioiodinated m-iodobenzylguanidine has been applied mainly for the diagnosis of pheochromocytoma and blastoma. In this paper the authors show that an ontogenetically related tumor, the neuroblastoma, is also scintigraphically visualized by its high uptake of I-131 MIBG. Because of the kinetic findings and the high uptake of more than 30% of the injected activity, it is likely that the neuroblastoma, by analogy with pheochromocytoma, is susceptible to specific radionuclide therapy.
  • Sixteen neuroblastoma patients have been studied by /sup 131/I-meta-iodobenzylguanidine (MIBG) scintigraphy. Three patients were possibly cured, and their scintigraphy results were normal. Thirteen patients had tumors and metastases demonstrated by /sup 131/I-MIBG, two of these patients had a normal vanillylmandelic acid (VMA) excretion level. One patient has been treated by /sup 131/I-MIBG, but died. /sup 131/I-MIBG was concentrated in other cells too, eg, in erythrocytes and platelets.
  • Radioiodinated m-iodobenzylguanidine has been applied mainly for the diagnosis of pheochromocytoma and blastoma. In this paper the author shows that an ontogenetically related tumor, the neuroblastoma, is also scintigraphically visualized by its high uptake of I-131 MIBG. Because of the kinetic findings and the high uptake of more than 30% of the injected activity, it is likely that the neuroblastoma, by analogy with pheochromocytoma, is susceptible to specific radionuclide therapy.