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Review
. 2022 Dec 21;22(1):325.
doi: 10.1186/s12902-022-01220-2.

Pleiomorphism plurihormonal Pit-1-positive macroadenoma with central hyperthyroidism: a rare case report and literature review

Guiliang Peng  1 , Chuanhong Guo  2 , Yangfan Lv  2 , Dandan Li  3 , Ling Zhou  1 , Rufei Shen  1 , Yong Chen  4 , Xin Zheng  4 , Zheng Sun  5 , Hongting Zheng  6 , Min Long  7
Affiliations
Review

Pleiomorphism plurihormonal Pit-1-positive macroadenoma with central hyperthyroidism: a rare case report and literature review

Guiliang Peng et al. BMC Endocr Disord. .

Abstract

Background: Thyrotropin-secreting pituitary neuroendocrine tumors (PitNETs) are rare pituitary adenomas that are occasionally accompanied by hypersecretion of other anterior pituitary hormones, such as growth hormone (GH) and prolactin (PRL). The clinical, biochemical, and pathological characteristics may represent diverse circumstances.

Case presentation: In this report, a 33-year-old female diagnosed with a TSH PitNET co-secreting GH presented no obvious clinical symptoms. The main characteristics were elevated thyroid-stimulating hormone (TSH), free tri-iodothyronine (FT3), and free thyroxine (FT4) levels accompanied by slightly elevated GH and insulin-like growth factor-1 (IGF-1) levels. Magnetic resonance imaging (MRI) detected a pituitary macroadenoma (18 × 16 × 16 mm) with cavernous sinus and suprasellar invasion. Immunohistochemistry revealed diffuse positivity for TSH, strong immunoreactivity for GH, and sporadic positivity for PRL. The electron microscope and double immunofluorescence staining confirmed a plurimorphous plurihormonal adenoma producing TSH, GH, and PRL. After preoperative somatostatin receptor ligand (SRL) treatment and transsphenoidal surgery, the patient achieved temporary clinical and biochemical remission. However, 3 months after surgery, the patient was suspected of having Hashimoto's thyroiditis due to higher thyroglobulin antibody (TGAb), thyroid peroxidase antibody (TPOAb), and thyroid receptor antibody (TRAb) and an enlarged thyroid nodule. During follow-up, thyroid function and TSH slowly transformed from transient hyperthyroidism to hypothyroidism. They were maintained in the normal range by L-T4.

Conclusion: In the TSH PitNET, the positive immunohistochemistry for TSH, GH, and PRL translated into hormonal overproduction with TSH and GH.

Keywords: Growth hormone (GH); Immunohistochemistry; Pituitary neuroendocrine tumor (PitNET); Plurihormonal pit-1-positive adenoma; Thyroid-stimulating hormone (TSH).

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Conflict of interest statement

The authors declare that they have no competing interests.

Figures

Fig. 1
Fig. 1
The suppression ratio of TSH and GH during 24 hours octreotide tests. The maximum suppression rate of GH was 94.4% at 2 hours; The maximum suppression ratio of TSH was 81.2% at 24 hours
Fig. 2
Fig. 2
Brain magnetic resonance imaging. There was a pituitary macroadenoma (18 × 16 × 16 mm) with a quasi-circular equal signal shadow in the sellar region. A. gadolinium-enhanced sagittal T1WI, B. gadolinium-enhanced coronal T1WI, C. coronal T2WI
Fig. 3
Fig. 3
Histopathological findings: a IHC for Pit-1 revealed positive staining in adenoma cell nuclei. b IHC for GH. c IHC for TSH. d IHC for PRL. e IHC for KI-67. f IHC for p53. Original magnification × 200
Fig. 4
Fig. 4
Electron microscopy revealed the adenoma cells were Plurimorphous. Ultrastructural examination identified that some adenoma cells were densely granulated, and secretory granules were spherical or ovoid and measured 300–450 nm (black arrow). The thyrotropin cells were a predominantly spherical or ovoid nucleus, Scattered lysosomes and mitochondria, prominent Golgi apparatus, and numerous secretory granules measuring 100 to 200 nm (red arrow)
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