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Pediatric adrenocortical tumors: morphological diagnostic criteria and immunohistochemical expression of matrix metalloproteinase type 2 and human leucocyte-associated antigen (HLA) class II antigens. Results from the Italian Pediatric Rare Tumor (TREP) Study project
Authors:Magro Gaetano  Esposito Giovanni  Cecchetto Giovanni  Dall'Igna Patrizia  Marcato Raffaella  Gambini Claudio  Boldrini Renata  Collini Paola  D'Onofrio Vittoria  Salfi Nunzio  d'Amore Emanuele  Ferrari Andrea  Bisogno Gianni  Alaggio Rita
Affiliation:
  • a Department G.F. Ingrassia, Anatomia Patologica, Università di Catania, 95123 Catania, Italy
  • b Istituto Oncologico Veneto—IRCCS, 35128 Padova, Italy
  • c Department of Pediatrics, Section of Pediatric Surgery, Università, 35121 Padova, Italy
  • d Servizio di Anatomia ed Istologia Patologica, Istituto Giannina Gaslini, 16147 Genova, Italy
  • e Servizio di Anatomia Patologica, Ospedale Pediatrico Bambin Gesù, 00165 Roma, Italy
  • f Unità Operativa di Anatomia Patologica, Istituto Nazionale per lo Studio e la Cura dei Tumori (INT), 20133 Milano, Italy
  • g Servizio di Anatomia e Istologia Patologica, Azienda Ospedaliera di Rilievo Nazionale “Santobono Pausillipon,” 80129 Napoli, Italy
  • h Unità Operativa di Anatomia e Istologia Patologica, Ospedale Sant'Orsola-Malpighi, 40138 Bologna, Italy
  • i Unità Operativa di Anatomia e Cito-Istopatologia, Presidio Ospedaliero San Bortolo, 36100 Vicenza, Italy
  • j Oncologia Pediatrica, Istituto Nazionale per lo Studio e la Cura dei Tumori (INT), 20133 Milano, Italy
  • k Divisione di Onco-Ematologia, Dipartimento di Pediatria, Università di Padova, 35121 Padova, Italy
  • l Department of Pathology, Azienda Ospedaliera/Università, 35121 Padova, Italy
  • Abstract:Pediatric adrenocortical tumors are neoplasms that only rarely occur in pediatric patients. Their clinical behavior is often unpredictable, and the histologic criteria of malignancy used in adults are not always useful in children. The aim of this study was to validate the prognostic value of the pathologic criteria of Wieneke et al and to evaluate the potential prognostic expression of matrix metalloproteinase 2 and human leucocyte-associated antigen (HLA) class II antigens in a series of 20 pediatric patients affected by adrenocortical tumors, who were enrolled in the Italian Pediatric Rare Tumor (TREP) Study between 2000 and 2007. The age range was 0 to 17.5 years (mean, 7.28 years) with a male-female ratio of 1:2. The mean follow-up was 64.4 months. The histologic diagnoses were reviewed, and the cases were classified using the criteria for malignancy proposed by Wieneke et al. The immunohistochemical expression of matrix metalloproteinase 2 and HLA class II antigens was scored by semiquantitative analysis and compared with the clinicopathologic parameters and outcome. Based on the scoring system of Wieneke et al, 7 tumors were classified as malignant; 12 tumors, as benign; and only 1 tumor, with "unpredictable behavior." In all cases, the clinical behavior was consistent with the pathologic criteria of Wieneke et al. Notably, areas of regressive myxoid changes, not included among the criteria of Wieneke et al, were observed in all but 1 case of malignant tumors and only in 2 cases of benign tumors. Matrix metalloproteinase 2 was focally to diffusely expressed in all malignant and in most benign tumors. HLA class II antigens immunoreactivity was absent in all benign tumors and restricted to rare isolated cells in most malignant tumors. Our findings confirm that the pathologic scoring system of Wieneke et al is a simple and reproducible diagnostic tool to predict prognosis in pediatric adrenocortical tumors. Unlike in their adult counterpart, the expression of matrix metalloproteinase 2 or the loss of HLA class II antigens does not discriminate between benign and malignant tumors in children. Although pediatric adrenocortical tumors seem to be similar histologically to their adult counterparts, it is likely that they have distinctive molecular features.
    Keywords:Adrenocortical tumors   Pediatric   Histologic features   Matrix metalloproteinases   HLA class II antigens
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