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Monomorphous Plurihormonal Pituitary Adenoma of Pit-1 Lineage in a Giant Adolescent with Central Hyperthyroidism

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Abstract

Thyrotropin (TSH)-secreting pituitary adenomas are exceedingly rare at the pediatric age and no cases of co-secretion with other pituitary hormones in these tumors have been described in this age range. We present a case of a monomorphous plurihormonal pituitary adenoma that co-secreted TSH and GH in a pediatric patient. A 13-year-old male presented with increasing height velocity (17.75 cm/year, 9.55SD), weight loss, and visual impairment. Initial biochemical evaluations revealed secondary hyperthyroidism. A giant pituitary tumor compressing the surrounding structures was detected by magnetic resonance, and a transsphenoidal surgery was initially performed. Pathological examinations revealed an atypical, monomorphous plurihormonal Pit-1 lineage tumor with mixed features of silent subtype 3 adenoma and acidophil stem cell adenoma. In the postoperative period, secondary hyperthyroidism recurred with high levels of both GH and IGF1. In addition, due to tumor re-growth, a multimodality treatment plan was undertaken including surgery, somatostatin analogs, and radiotherapy. We report the first pediatric case of a plurihormonal TSH- and GH-secreting pituitary adenoma, further expanding the clinical manifestations of pediatric pituitary tumors. Comprehensive pathological evaluation and close follow-up surveillance are crucial to the prompt delivery of the best therapeutic options in the context of this particularly aggressive pituitary tumor.

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Acknowledgments

The authors acknowledge Manuel Lemos, MD, PhD, Eduarda Coutinho, MD, and Marina Santos, MD (Centro de Investigação em Ciências da Saúde, Universidade da Beira Interior) for their contribution in the genetic analysis of the AIP gene.

Susana Prazeres, MD, PhD (Laboratório de Endocrinologia, Instituto Português de Oncologia de Lisboa, Francisco Gentil) for her valuable comments to the manuscript.

Pedro Martins, Ar. (Câmara Municipal de Lagoa, Açores) for his valuable contribution to the Artwork.

Conflict of Interest

The authors declare that they have no competing interests.

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Authors and Affiliations

  1. Serviço de Endocrinologia e Diabetes, piso 8, Hospital Garcia de Orta, E.P.E, 2801-951, Almada-Setúbal, Portugal

    Bernardo Dias Pereira, Luísa Raimundo & Jorge Portugal

  2. Department of Pathology, University Health Network, 200 Elizabeth Street, 11th Floor, Toronto, ON, Canada

    Ozgur Mete & Sylvia L. Asa

  3. Serviço de Anatomia Patológica, piso 0, Hospital Garcia de Orta, E.P.E, 2801-951, Almada-Setúbal, Portugal

    Ana Oliveira

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  1. Bernardo Dias Pereira
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  2. Luísa Raimundo
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Correspondence to Bernardo Dias Pereira.

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Pereira, B.D., Raimundo, L., Mete, O. et al. Monomorphous Plurihormonal Pituitary Adenoma of Pit-1 Lineage in a Giant Adolescent with Central Hyperthyroidism. Endocr Pathol 27, 25–33 (2016). https://doi.org/10.1007/s12022-015-9395-2

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