Adrenal ganglioneuromas: a retrospective multicentric study of 104 cases from the COMETE network

in European Journal of Endocrinology
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  • 1 Assistance Publique-Hôpitaux de Paris, Hôpital Européen Georges Pompidou, Centre de Référence des Maladies Rares de la Surrénale, Unité Fonctionnelle d’Hypertension Artérielle, Université de Paris, Paris, France
  • | 2 Service de Pathologie, HU Robert Debré, APHP, Paris, France
  • | 3 Université de Paris, Paris, France
  • | 4 Hôpital Larrey, Service d’Endocrinologie, Toulouse, France
  • | 5 Assistance Publique-Hôpitaux de Paris, Hôpital Henri Mondor, Service de Médecine Interne et Endocrinologie, Créteil, France
  • | 6 Département de Chirurgie Viscérale, Métabolique et Cancérologique Unité Médico-chirurgicale de chirurgie Métabolique, Endocrinienne et Thyroïdienne (UMET), Université de Lorraine, CHU de Nancy, Hôpital Brabois Adultes, Nancy, France
  • | 7 Université Côte d’Azur, Service d’Endocrinologie, Diabétologie et Reproduction, CHU de Nice, Hôpital de l’Archet 2, Nice, France
  • | 8 Assistance Publique-Hôpitaux de Paris, Hôpital Saint-Antoine, Service d’Endocrinologie et Maladies de la Reproduction, Paris, France
  • | 9 Centre Hospitalier Universitaire de Reims, Service d’Endocrinologie-Diabète-Nutrition, Reims, France
  • | 10 Département d’Endocrinologie, CUB-Hôpital Erasme, Bruxelles, Belgique
  • | 11 Centre Hospitalier Universitaire de Nantes, Hôpital Nord Laënnec, Service d’Endocrinologie, Diabétologie et Maladies Métaboliques, L’institut du Thorax, Nantes, France
  • | 12 CHU de Nîmes, Service des Maladies Métaboliques et Endocriniennes, Nîmes, France
  • | 13 CHU de Dijon, Chirurgie Endocrinienne et Métabolique, Dijon, France
  • | 14 Centre de Référence des Maladies Rares de la Thyroïde et des Récepteurs Hormonaux, Département d’Endocrinologie Diabétologie Nutrition, Angers, France
  • | 15 Assistance Publique-Hôpitaux de Paris, Hôpital Pitié-Salpêtrière, Service d’Endocrinologie-Métabolisme, Institut E3M, Paris, France
  • | 16 Université de Rouen Normandie, UNIROUEN, INSERM U1239, CHU de Rouen, Service d’Endocrinologie, Diabète et Maladies Métaboliques et CIC-CRB 1404, Rouen, France
  • | 17 Sorbonne Université, Hôpital Pitié-Salpêtrière, Service de Médecine Nucléaire, Paris, France
  • | 18 Assistance Publique-Hôpitaux de Paris, Hôpital Européen Georges-Pompidou, Laboratoire d’Anatomo-Pathologie, Paris, France
  • | 19 Assistance Publique-Hôpitaux de Paris, CHU Ambroise-Paré, Service d’Endocrinologie et Nutrition, Boulogne-Billancourt, France
  • | 20 CHRU de Montpellier, Service d’Endocrinologie, Diabète, Maladies Métaboliques, Montpellier, France
  • | 21 Centre Hospitalier Universitaire de Tours, Département d’Endocrinologie, Tours, France
  • | 22 Assistance Publique-Hôpitaux de Paris, Hôpital Universitaire Robert Debré, Service d’Endocrinologie Diabétologie Pédiatrique, Paris, France
  • | 23 CHU de Bordeaux, Service d’Endocrinologie, Diabétologie et Maladies Métaboliques, Bordeaux, France
  • | 24 Centre Hospitalier Régional Universitaire de Lille, Service d’Endocrinologie et Maladies Métaboliques, Lille, France
  • | 25 Assistance Publique-Hôpitaux de Paris, Hôpital Bichat, Service de Physiologie, Paris, France
  • | 26 Institut Gustave Roussy, Service de Médecine Nucléaire et de Cancérologie Endocrinienne, Villejuif, France
  • | 27 Université Paris Descartes, Hôpital Cochin, Centre de Référence des Maladies Rares de la Surrénale, Service d’Endocrinologie, Paris, France
  • | 28 Sorbonne Université, Médecine Paris, France, undefined
  • | 29 UMR970, Paris-Cardiovascular research Center, INSERM, F-75015, Paris, France

Correspondence should be addressed to L Amar Email Laurence.Amar@aphp.fr
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Objective

Adrenal ganglioneuromas are rare, differentiated, neuroblastic tumors that originate from the peripheral sympathetic nervous system. Because of their rarity, information is limited, derived from small cases series. Our objective was to characterize this tumor and provide help for its management.

Methods

A retrospective multicenter analysis of adrenal ganglioneuromas from 20 French centers belonging to the COMETE network and one Belgian center.

Results

Among the 104 cases identified, 59.6% were women (n = 62/104), median age at diagnosis was 29 years, with 24 pediatric cases. 60.6% (n = 63/104) were incidentalomas. Ganglioneuromas were non-secreting tumors in 90.8% of cases (n = 89/98), whereas the preoperative hormonal evaluation was indeterminate for 9.2% of patients (n = 9/98). CT imaging, performed on 96 patients, revealed large tumors (median diameter of 50 mm) with a non-contrast density > 10 Hounsfield units in 98.1% (n = 52/53) and calcifications in 64.6% of cases (n = 31/48). Increased uptake on 123I-MIBG scintigraphy and 18F-FDG-PET/CT was observed in 26.7% (n = 8/30) and 42.2% (n = 19/45) of the tumors, respectively. All 104 patients underwent surgery. No recurrence was observed among the 42 patients who had an imaging follow-up (mean 29.6 months, median 18 months (4–156)).

Conclusion

Adrenal ganglioneuromas are large tumors, mostly nonfunctioning, without benign imaging features. Although the duration of follow-up was limited in our series, no recurrence was identified. A review of the literature confirms the absence of postoperative recurrence. Based on all available data, in the absence of special circumstances (genetic form, uncertain histological diagnosis), long-term follow-up is not necessary after complete surgery for patients with an adrenal ganglioneuroma.

 

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