Thyrotropin-secreting pituitary adenoma in an 11-year-old boy with type 1 autoimmune polyglandular syndrome

Abstract

Thyrotropinomas (TSHomas) are rare pituitary adenomas, particularly in childhood. We present here the case of an 11-year-old boy with type 1 autoimmune polyglandular syndrome (APS1) and TSHoma which was diagnosed by elevated thyroid - stimulating hormone and thyroid hormones levels without evident clinical signs of hyperthyroidism. He was underwent partial resection of the tumor via transsphenoidal approach and subsequently radiation therapy. Consequently, 1 year after radiotherapy, the patient developed growth hormone deficiency, three and half years after radiation became euthyroid, and five and half years after treatment - hypothyroid. This is the first case of the coexistence of these two rare endocrine diseases in one patient.

Long-Term Follow-Up in a Male Patient with Micro-TSH-Oma Diagnosed at 8-Yr-Old

“…TSH-omas are rare in adults and to our knowledge our patient is one of the youngest children with hyperthyroidism due to TSH-oma ever reported. Other 13 children or adolescents described were 8-yr. or older (8 to 16yr) and had macroadenoma except a 13 yr-old girl who had microadenoma and a 15 yr-old girl whose tumor size was not decribed [7][8][9][10][11][12][13][14][15][16][17][18][19].…”

“…*Basal cortisol = 3.43 mg/dL(6)(7)(8)(9)(10)(11)(12)(13)(14)(15)(16)(17)(18)(19); urine density = 1005; ** Total testosterone = 106 ng/dL (<100); peak LH and FSH after GnRH = 1.1 and…”

Clinical and morphological characteristics, diagnostic criteria, and outcomes of surgical treatment of TSH-secreting pituitary adenomas

“…ТТГ-АГ выявляются у пациентов различного возраста от 8 до 84 лет [8][9][10]. Однако большинство описанных в литературе пациентов принадлежат к воз-растной категории 50-60 лет [1].…”

Challenges in the Management of a 7-Year-Old Child with Thyrotropin-Secreting Pituitary Adenoma and the Review of the Literature

“…Long-term follow-up and disease outcome are reported in only a few patients, the longest being 8 years. Final height has been reported in only 3 patients (two with GH replacement after surgery) [7, 8, 18]. In paediatric TSHomas, long-term outcomes, progression of the disease, and bone age (BA) progression under treatment are not well described due to the rarity of the disease and heterogenous hormone expression of the tumour.…”

“…TSHomas usually secrete alpha-subunit of the glycoprotein hormones (α-SU) and less frequently growth hormone (GH) and/or prolactin (PRL), ACTH, and FSH/LH [5]. Until now, only nineteen paediatric cases have been proven to have pituitary TSHoma in the literature [6][7][8][9][10][11][12][13][14][15][16][17][18][19][20][21][22][23][24]. Almost all of them had macroadenoma and presented with symptoms of hyperthyroidism and/or goitre [6,8,[10][11][12][13][14][15][16][17][18][19][20][21][22]24].…”