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Characterization of Hyperinsulinism in Infancy Assessed with PET and ¹⁸F-Fluoro-L-DOPA

¹ Service Hospitalier Frédéric Joliot, Département de Recherche Médicale, Direction des Sciences du Vivant, Commissariat à l’Energie Atomique, Orsay, France
² Département de Métabolisme et Pédiatrie, Hôpital Necker-Enfants Malades, Paris, France
³ Service de Radiologie Pédiatrique, Hôpital Necker-Enfants Malades, Paris, France
⁴ Laboratoire de Anatomopathologie, Hôpital Necker-Enfants Malades, Paris, France
⁵ Département de Chirurgie Infantile, Hôpital Necker-Enfants Malades, Paris, France

Hyperinsulinism (HI) of infancy is a neuroendocrine disease secondary to either focal adenomatous hyperplasia or a diffuse abnormality of insulin secretion of the pancreas. HI with focal lesions can revert by selective surgical resection in contrast to the diffuse form, which requires subtotal pancreatectomy when resistant to medical treatment. Neuroendocrine diseases are a heterogeneous group of entities with the ability to take up amine precursors and to convert them into biogenic amines. Therefore, the aim of this study was (a) to evaluate the use of PET with ¹⁸F-fluoro-L-dihydroxyphenylalanine (¹⁸F-fluoro-L-DOPA) and (b) to distinguish between focal and diffuse HI. Methods: Fifteen patients (11 boys, 4 girls) with neonatal HI were enrolled in this study. All patients fasted for at least 6 h before the PET examination and their medication was discontinued for at least 72 h. The examination was performed under light sedation (pentobarbital associated with or without chloral). The dynamic acquisition started 45–65 min after the injection of ¹⁸F-fluoro-L-DOPA (4.0 MBq/kg weight). Four or 6 scans of 5 min each (2 or 3 steps according to the height of the patient) were acquired from the neck to the upper legs. Results: An abnormal focal pancreatic uptake of ¹⁸F-fluoro-L-DOPA was observed in 5 patients, whereas a diffuse uptake of the radiotracer was observed in the pancreatic area of the other patients. All patients with focal radiotracer uptake and also 4 of 10 patients with pancreatic diffuse radiotracer accumulation, unresponsive to medical treatment, underwent surgery. The histopathologic results confirmed the PET findings—that is, focal versus diffuse HI. Conclusion: The results of this study suggest that ¹⁸F-fluoro-L-DOPA could be an accurate noninvasive technique to distinguish between focal and diffuse forms of HI.

Key Words: hyperinsulinism of infancy • PET • ¹⁸F-fluoro-L-DOPA

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