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 Table of Contents     
CASE REPORT
Year : 2016  |  Volume : 31  |  Issue : 2  |  Page : 141-143  

Thyrotropinoma with Graves' disease detected by the fusion of indium-111 octreotide scintigraphy and pituitary magnetic resonance imaging


1 Department of Nuclear Medicine, Gulhane Military Medical Academy, Ankara, Turkey
2 Department of Endocrinology, Gulhane Military Medical Academy, Ankara, Turkey
3 Department of Pathology, Gulhane Military Medical Academy, Ankara, Turkey

Date of Web Publication9-Mar-2016

Correspondence Address:
Kursat Okuyucu
Department of Nuclear Medicine, School of Medicine, Gulhane Military Medical Academy, 06018 Etlik, Ankara
Turkey
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-3919.178322

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   Abstract 

Thyroid-stimulating hormone-secreting pituitary adenoma (TSHoma) is a rare benign endocrinological tumor which produces TSH in the pituitary gland. Herein, we presented a female patient having TSHoma with Graves' disease during and just after pregnancy that we found by indium-111 octreotide scintigraphy while investigating the patient for hyperthyroidism symptoms.

Keywords: Graves′ disease, indium-111 octreotide scintigraphy, thyroid-stimulating hormone-secreting pituitary adenoma


How to cite this article:
Okuyucu K, Alagoz E, Arslan N, Taslipinar A, Deveci MS, Bolu E. Thyrotropinoma with Graves' disease detected by the fusion of indium-111 octreotide scintigraphy and pituitary magnetic resonance imaging. Indian J Nucl Med 2016;31:141-3

How to cite this URL:
Okuyucu K, Alagoz E, Arslan N, Taslipinar A, Deveci MS, Bolu E. Thyrotropinoma with Graves' disease detected by the fusion of indium-111 octreotide scintigraphy and pituitary magnetic resonance imaging. Indian J Nucl Med [serial online] 2016 [cited 2019 Sep 23];31:141-3. Available from: http://www.ijnm.in/text.asp?2016/31/2/141/178322


   Introduction Top


Thyroid-stimulating hormone (TSH)-secreting pituitary adenoma (TSHoma), also called thyrotropinoma, comprises 2% of all pituitary adenomas. [1] These tumors provoke central hyperthyroidism through releasing inappropriate TSH secretion. [2] TSHoma displays itself by hyperthyroidism symptoms most usually milder than the ones stemming from the thyroid or mass effects onto its neighbors. [3] Patients with this type of thyrotoxicosis can exhibit thyroid crisis because of stress or prior to surgery that must be intervened. Antithyroid drugs such as thiamazole afford to control hyperthyroidism relevant to TSHomas, but long-term use of these agents may induce expansion of the tumor over a negative feedback mechanism ending in a decrease of thyroid hormone levels. [1] The concomitant mixed pituitary adenoma secreting growth hormone and prolactin at the same time complicate the condition in the existence of a TSHoma accompanying thyroid disease. [4] Treatment modalities are surgery, radiation therapy, and somatostatin analogs. Somatostatin is a normally produced neuropeptide in the body. It acts over its receptors and these receptors are present in many tumors (mainly neuroendocrine tumors) and activated leukocytes of granulomatous and chronic inflammatory diseases. [5]


   Case Report Top


A 37-year-old female was examined with thyroid function tests because of vague exophthalmos at the 20 th week of her pregnancy. Free-T3 was found 8.96 pg/ml (normal range: 2.3-4.2 pg/ml), free-T4 2.79 ng/dl (normal range: 0.89-1.76 ng/dl), TSH 5.54 microIU/ml (normal range: 0.35-5.5 microIU/ml), and alpha-subunit of TSH 32 mIU/ml. Because of this inappropriate TSH secretion, a contrast magnetic resonance (MR) imaging was requested for a possible TSHoma. A macroadenoma of 13 mm × 11 mm in size adjacent to optic chiasm was detected in pituitary gland on MR. Her visual field was normal. She was treated by 3 × 50 mg/day of propylthiouracil orally and followed, for she did not accept the operation. She bore a healthy baby of full term and 3000 g in weight without any complication of this pituitary macroadenoma suspected of secreting TSH during her pregnancy and birth. Thyrotoxicosis and TSH level increased at postpartum 3 rd month. These findings were strongly implying that macroadenoma in pituitary gland was a TSH-secreting tumor causing the hyperthyroidism symptoms of the patient. The patient stopped breastfeeding. A solid thyroid nodule of 11 mm × 10 mm in dimension, hypoactive on Tc-99m pertechnetate thyroid scintigraphy, was detected by ultrasonography and her TSH receptor antibody was 30 U/L (normal range <2 U/L). Fine-needle aspiration biopsy of the nodule revealed oxyphilic changes (Bethesda 4). Indium-111 (In-111) octreotide scintigraphy was performed to confirm the functional status of pituitary adenoma. Focal uptake within sella turcica consistent with the pituitary adenoma on MR [Figure 1] was seen and no other pathologic finding was observed on whole-body screening. She was operated with total thyroidectomy owing to malignancy suspicion. Surgical specimen was confirmed as oxyphilic nodular goiter and Graves' disease pathologically [Figure 2]a. TSH level increased after thyroidectomy. After that, she underwent transsphenoidal microscopic hypophysectomy later on. Immunohistochemical pathology reported the diagnosis as a TSH-secreting adenoma [Figure 2]b.
Figure 1: Indium-111 octreotide single photon emission computed tomography shows focal uptake in sella turcica consistent with thyrotropinoma on coronal single photon emission computed tomography (a), coronal magnetic resonance (b), and fused images of indium-111 octreotide and magnetic resonance (c)

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Figure 2: Graves' disease and oxyphilic nodular goiter (H and E, ×20) (a) and pituitary thyrotroph adenoma (H and E, ×40) (b)

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   Discussion Top


Octreotide is a somatostatin analog which can be easily labeled with In-111 and is primarily used to detect neuroendocrine tumors. [6] It is used not only to diagnose these tumors but also to treat them. Somatostatin receptors are found in pituitary adenomas too. [7] Coexistence of TSHoma with Graves' disease is very rare and only six cases of such association with histological confirmation, all of whom are female, have been reported previously. [8],[9],[10] Our patient had signs of thyrotoxicosis, goiter, and exophthalmos with elevated thyroid hormone and TSH receptor antibodies, but normal TSH values. She was exhibiting all these typical diagnostic criteria and also had a mass in pituitary gland on MR strongly suggesting a thyrotropinoma. It was necessary to establish the functional status of this mass carrying strong evidence of TSHoma. However, her pregnancy precluded all invasive imaging modality and surgical interventions. It is possible to show TSHoma by In-111 octreotide scintigraphy if it is somatostatin receptor positive. As hyperthyroidism symptoms and abnormality of biochemical markers persisted at postpartum period, we decided to show the functional relation between the abnormal laboratory tests causing patient's complaints and the mass by In-111 octreotide scintigraphy. We used fusion of octreoscan and MR for this purpose. Proving of this mass as a functional tumor with a focal uptake within sella turcica led to an ultimate decision of surgery reinforcing the hand of physician. A different and interesting feature of our case is the concomitant existence of Graves' disease and thyrotropinoma, which is different than the previous 6 cases of thyrotropinoma in the literature. In routine management of such a patient, pituitary surgery for macroadenoma is performed first, total thyroidectomy is performed later. However, in this case, total thyroidectomy was performed first because of malignancy suspicion.


   Conclusion Top


Pituitary lesions causing inappropriate TSH secretion should be confirmed by functional imaging modalities. In-111 octreotide scintigraphy may be useful in illumination of such masses in pituitary gland, particularly in association of TSHoma with Graves' disease especially when TSH values are not compatible with either the clinical history or other thyroid functions tests.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Beck-Peccoz P, Persani L, Mannavola D, Campi I. Pituitary tumours: TSH-secreting adenomas. Best Pract Res Clin Endocrinol Metab 2009;23:597-606.  Back to cited text no. 1
    
2.
Fukuhara N, Horiguchi K, Nishioka H, Suzuki H, Takeshita A, Takeuchi Y, et al. Short-term preoperative octreotide treatment for TSH-secreting pituitary adenoma. Endocr J 2015;62:21-7.  Back to cited text no. 2
    
3.
Losa M, Fortunato M, Molteni L, Peretti E, Mortini P. Thyrotropin-secreting pituitary adenomas: Biological and molecular features, diagnosis and therapy. Minerva Endocrinol 2008;33:329-40.  Back to cited text no. 3
    
4.
Aksoy DY, Gedik A, Cinar N, Soylemezoglu F, Berker M, Gurlek OA. Thyrotropinoma and multinodular goiter: A diagnostic challenge for hyperthyroidism. J Res Med Sci 2013;18:1008-10.  Back to cited text no. 4
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5.
Goel S, Gadiraju R. Diffuse lung uptake of 111In octreotide from chronic emphysema. J Nucl Med Technol 2013;41:117-8, 120.  Back to cited text no. 5
    
6.
Kamoun M, d'Herbomez M, Lemaire C, Fayard A, Desailloud R, Huglo D, et al. Coexistence of thyroid-stimulating hormone-secreting pituitary adenoma and graves' hyperthyroidism. Eur Thyroid J 2014;3:60-4.  Back to cited text no. 6
    
7.
Gatto F, Barbieri F, Castelletti L, Arvigo M, Pattarozzi A, Annunziata F, et al. In vivo and in vitro response to octreotide LAR in a TSH-secreting adenoma: Characterization of somatostatin receptor expression and role of subtype 5. Pituitary 2011;14:141-7.  Back to cited text no. 7
    
8.
Azukizawa M, Morimoto S, Miyai K. TSH-producing pituitary adenoma associated with Graves' disease. In: Stockigt JR, Nagataki S, editors. Thyroid Research. Vol. VII. Canberra: Australian Academy of Science; 1980. p. 645-8.  Back to cited text no. 8
    
9.
Kamoi K, Mitsuma T, Sato H. Hyperthyroidism caused by a pituitary thyrotrophin-secreting tumour with excessive secretion of thyrotrophin-releasing hormone and subsequently followed by Graves' disease in a middle-aged woman. Acta Endocrinol (Copenh) 1985;110:373-82.  Back to cited text no. 9
    
10.
Beck-Peccoz P, Lania A, Beckers A, Chatterjee K, Wemeau JL. 2013 European thyroid association guidelines for the diagnosis and treatment of thyrotropin-secreting pituitary tumors. Eur Thyroid J 2013;2:76-82.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2]



 

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