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Ectopic TSH-secreting pituitary tumor: a case report and review of prior cases.

Song M, Wang H, Song L, Tian H, Ge Q, Li J, Zhu Y, Li J, Zhao R, Ji HL - BMC Cancer (2014)

Bottom Line: TRH stimulating test was negative, whereas octreotide inhibition test showed a reduction in TSH by 30.8%.Furthermore, a large space-occupying lesion located at the nasopharynx was found by computed tomography and magnetic resonance imaging (MRI).The characteristics and successful interventions summarized in this report provide a guideline for clinicians.

View Article: PubMed Central - PubMed

Affiliation: Department of Endocrinology, Weihai Municipal Hospital, 70 Heping Road, Weihai, Shandong 264200, China. whsmq1201@hotmail.com.

ABSTRACT

Background: Ectopic TSH-secreting pituitary adenoma (TSH-oma) is a very unusual disorder. To date, there are only four cases reported. It is difficult to distinguish ectopic cases from both regular TSH-omas and resistance to thyroid hormone (RTH).

Case presentation: A newly identified case of ectopic TSH-oma arising from the nasal pharynx was described, and reports of four prior cases were reviewed. The patient was a 41-year-old male who developed what appeared to be typical hyperthyroidism and atrial fibrillation in 2009. Thyroid function tests showed elevated basal levels of free T3 (FT3, 24.08 pmol/L), free T4 (FT4, 75.73 pmol/L), and serum TSH (7.26 μIU/ml). Both TSH-oma and resistance to thyroid hormone syndrome were considered. TRH stimulating test was negative, whereas octreotide inhibition test showed a reduction in TSH by 30.8%. Furthermore, a large space-occupying lesion located at the nasopharynx was found by computed tomography and magnetic resonance imaging (MRI). A normal pituitary was visualized. Ectopic TSH-oma was preliminarily established. Using an endoscopic endonasal approach, the tumor was resected. Histological features and immunophenotypes were consistent with those of TSH-secreting tumor. The levels of both free thyroxine and TSH returned to normal ranges the day after surgery and remained within normal range for 48 months.

Conclusions: Although exceedingly rare, ectopic TSH-oma should be considered for patients with inappropriate secretion of TSH with hyperthyroidism and pituitary tumor undetectable by computed tomography and MRI. To our knowledge, this is the first case followed up more than 4 years. The characteristics and successful interventions summarized in this report provide a guideline for clinicians.

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Related in: MedlinePlus

MRI and CT images. A &B, MRI of the pituitary showing a normal pituitary gland and an ectopic pituitary tumor in the nasopharynx (white arrow). C &D, CT scan showing a 1.9 × 1.7 cm mass in the nasopharyngeal cavity (white arrows). E &F, CT scan 48 months post surgery.
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Fig1: MRI and CT images. A &B, MRI of the pituitary showing a normal pituitary gland and an ectopic pituitary tumor in the nasopharynx (white arrow). C &D, CT scan showing a 1.9 × 1.7 cm mass in the nasopharyngeal cavity (white arrows). E &F, CT scan 48 months post surgery.

Mentions: A 41-year-old male suffering from palpitations, dyspnea, weight loss, and fatigue for one year was referred to Weihai Municipal Hospital in June 2009. He also had atrial fibrillation. Thyroid functional tests showed increased FT3 (24.08, normal 2.8-7.1 pmol/L), FT4 (75.73, normal 12-22 pmol/L), and TSH (7.26, normal 0.27-4.2 μIU/ml). He was diagnosed with hyperthyroidism and given propylthiouracil (300 mg daily) together with either propranolol or propafenone. The patient's electrocardiogram displayed sinus rhythm. The levels of FT3 and FT4 (FT3 11.54 pmol/L, FT4 27.09 pmol/L) but not TSH (14.08 μIU/ml) were reduced after six months of treatment. However, the concentration of free thyroid hormones were still not normal. In sharp contrast, the TSH level was further elevated after intensive treatment. Pituitary MRI examination was therefore performed to rule out TSH-oma. The MRI image indicated a normal pituitary gland (Figure 1A and B). Thus, resistance to thyroid hormone syndrome was diagnosed, and triiodothyroacetic acid was prescribed. The plasma levels of FT3, FT4, and TSH transiently decreased and then rebounded.


Ectopic TSH-secreting pituitary tumor: a case report and review of prior cases.

Song M, Wang H, Song L, Tian H, Ge Q, Li J, Zhu Y, Li J, Zhao R, Ji HL - BMC Cancer (2014)

MRI and CT images. A &B, MRI of the pituitary showing a normal pituitary gland and an ectopic pituitary tumor in the nasopharynx (white arrow). C &D, CT scan showing a 1.9 × 1.7 cm mass in the nasopharyngeal cavity (white arrows). E &F, CT scan 48 months post surgery.
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC4125694&req=5

Fig1: MRI and CT images. A &B, MRI of the pituitary showing a normal pituitary gland and an ectopic pituitary tumor in the nasopharynx (white arrow). C &D, CT scan showing a 1.9 × 1.7 cm mass in the nasopharyngeal cavity (white arrows). E &F, CT scan 48 months post surgery.
Mentions: A 41-year-old male suffering from palpitations, dyspnea, weight loss, and fatigue for one year was referred to Weihai Municipal Hospital in June 2009. He also had atrial fibrillation. Thyroid functional tests showed increased FT3 (24.08, normal 2.8-7.1 pmol/L), FT4 (75.73, normal 12-22 pmol/L), and TSH (7.26, normal 0.27-4.2 μIU/ml). He was diagnosed with hyperthyroidism and given propylthiouracil (300 mg daily) together with either propranolol or propafenone. The patient's electrocardiogram displayed sinus rhythm. The levels of FT3 and FT4 (FT3 11.54 pmol/L, FT4 27.09 pmol/L) but not TSH (14.08 μIU/ml) were reduced after six months of treatment. However, the concentration of free thyroid hormones were still not normal. In sharp contrast, the TSH level was further elevated after intensive treatment. Pituitary MRI examination was therefore performed to rule out TSH-oma. The MRI image indicated a normal pituitary gland (Figure 1A and B). Thus, resistance to thyroid hormone syndrome was diagnosed, and triiodothyroacetic acid was prescribed. The plasma levels of FT3, FT4, and TSH transiently decreased and then rebounded.

Bottom Line: TRH stimulating test was negative, whereas octreotide inhibition test showed a reduction in TSH by 30.8%.Furthermore, a large space-occupying lesion located at the nasopharynx was found by computed tomography and magnetic resonance imaging (MRI).The characteristics and successful interventions summarized in this report provide a guideline for clinicians.

View Article: PubMed Central - PubMed

Affiliation: Department of Endocrinology, Weihai Municipal Hospital, 70 Heping Road, Weihai, Shandong 264200, China. whsmq1201@hotmail.com.

ABSTRACT

Background: Ectopic TSH-secreting pituitary adenoma (TSH-oma) is a very unusual disorder. To date, there are only four cases reported. It is difficult to distinguish ectopic cases from both regular TSH-omas and resistance to thyroid hormone (RTH).

Case presentation: A newly identified case of ectopic TSH-oma arising from the nasal pharynx was described, and reports of four prior cases were reviewed. The patient was a 41-year-old male who developed what appeared to be typical hyperthyroidism and atrial fibrillation in 2009. Thyroid function tests showed elevated basal levels of free T3 (FT3, 24.08 pmol/L), free T4 (FT4, 75.73 pmol/L), and serum TSH (7.26 μIU/ml). Both TSH-oma and resistance to thyroid hormone syndrome were considered. TRH stimulating test was negative, whereas octreotide inhibition test showed a reduction in TSH by 30.8%. Furthermore, a large space-occupying lesion located at the nasopharynx was found by computed tomography and magnetic resonance imaging (MRI). A normal pituitary was visualized. Ectopic TSH-oma was preliminarily established. Using an endoscopic endonasal approach, the tumor was resected. Histological features and immunophenotypes were consistent with those of TSH-secreting tumor. The levels of both free thyroxine and TSH returned to normal ranges the day after surgery and remained within normal range for 48 months.

Conclusions: Although exceedingly rare, ectopic TSH-oma should be considered for patients with inappropriate secretion of TSH with hyperthyroidism and pituitary tumor undetectable by computed tomography and MRI. To our knowledge, this is the first case followed up more than 4 years. The characteristics and successful interventions summarized in this report provide a guideline for clinicians.

Show MeSH
Related in: MedlinePlus