A Plurihormonal TSH-Producing Pituitary Tumor of Monoclonal Origin in a Patient with Hypothyroidism

Ma, Wenbin; Ikeda, Hidetoshi; Watabe, N; Kanno, Mitsunobu; Yoshimoto, Takashi

doi:10.1159/000070227

Cited by 19 publications

(16 citation statements)

References 8 publications

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“…TSH-secreting adenomas have been reported in patients ranging from 8 to 84 years of age (most are in the 5th or 6th decade of life), with equal frequency in men and women, 3 which agrees with our findings. Similar to other reports, 21,34 one case was associated with previous Hashimoto's thyroiditis and hypothyroidism and one case was associated with multiple endocrine neoplasia Type 1.…”

Section: Clinical Characteristicssupporting

confidence: 89%

Clinicopathological characteristics and therapeutic outcomes in thyrotropin-secreting pituitary adenomas: a single-center study of 90 cases

Yamada¹,

Fukuhara²,

Horiguchi³

et al. 2014

JNS

126

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Object The aim of this study was to analyze clinicopathological characteristics and treatment outcomes in a large single-center clinical series of cases of thyrotropin (TSH)–secreting pituitary adenomas. Methods The authors retrospectively reviewed clinical, pathological, and treatment characteristics of 90 consecutive cases of TSH-secreting pituitary adenomas treated with transsphenoidal surgery between December 1991 and May 2013. The patient group included 47 females and 43 males (median age 42 years, range 11–74 years). Results Sixteen tumors (18%) were microadenomas and 74 (82%) were macroadenomas. Microadenomas were significantly more frequent in the more recent half of our case series (12 of 45 cases) (p = 0.0274). Cavernous sinus invasion was confirmed in 21 patients (23%). In 67 cases (74%), the tumors were firm elastic or hard in consistency. Acromegaly and hyperprolactinemia were observed, respectively, in 14 (16%) and 11 (12%) of the 90 cases. Euthyroidism was achieved in 40 (83%) of 48 patients and tumor shrinkage was found in 24 (55%) of 44 patients following preoperative somatostatin analog treatment. Conventional transsphenoidal surgery, extended transsphenoidal surgery, and a simultaneous combined supra- and infrasellar approach were performed in 85, 2, and 3 patients, respectively. Total removal with endocrinological remission was achieved in 76 (84%) of 90 patients, including all 16 (100%) patients with microadenomas, 60 (81%) of the 74 with macroadenomas, and 8 (38%) of the 21 with cavernous sinus invasion. None of these 76 patients experienced tumor recurrence during a median follow-up period of 2.8 years. Stratifying by Knosp grade, total removal with endocrinological remission was achieved in 34 of 36 patients with Knosp Grade 0 tumors, all 24 of those with Grade 1 tumors, 12 of the 14 with Grade 2 tumors, 6 of the 8 with Grade 3 tumors, and none of the 8 with Grade 4 tumors. Cavernous sinus invasion and tumor size were significant independent predictors of surgical outcome. Immunoreactivity for growth hormone, prolactin, or both hormones was present in 32, 9, and 24 patients, respectively. The Ki-67 labeling index was less than 3% in 71 (97%) of 73 tumors for which it was obtained and 3% or more in 2. Postsurgery pituitary dysfunction was found in 15 patients (17%) and delayed hyponatremia was seen in 9. Conclusions TSH-secreting adenomas, particularly those in the microadenoma stage, have increased in frequency over the past 5 years. The high surgical success rate achieved in this series is due to relatively early diagnosis and relatively small tumor size. In addition, the surgical strategies used, such as extracapsular removal of hard or solid adenomas, aggressive resction of tumors with cavernous sinus invasion, or extended transsphenoidal surgery or a simultaneous combined approach for large/giant multilobulated adenomas, also may improve remission rate with a minimal incidence of complications.

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Section: Clinical Characteristicssupporting

confidence: 89%

Clinicopathological characteristics and therapeutic outcomes in thyrotropin-secreting pituitary adenomas: a single-center study of 90 cases

Yamada¹,

Fukuhara²,

Horiguchi³

et al. 2014

JNS

126

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“…Autoimmune hypothyroidism may co-exist with TSHoma, but any causality is yet unclear (112,113,114). Pituitary hyperplasia from increased TSH production in hypothyroidism may in some rare cases mimic a macroadenoma (115).…”

Section: Concomitant Diseases In Association With Tshomasmentioning

confidence: 99%

DIAGNOSIS OF ENDOCRINE DISEASE: Diagnostic approach to TSH-producing pituitary adenoma

Tjörnstrand

Nyström

2017

European Journal of Endocrinology

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Thyrotropin (TSH)-secreting adenomas (TSHomas) are the rarest form of pituitary adenomas, and most endocrinologists will see few cases in a lifetime, if any. In most cases, the diagnostic approach is complicated and cases may be referred after being presented as a syndrome of inappropriate TSH secretion or as a pituitary mass. This review aims to cover the past, present and possible future diagnostic approaches to TSHomas, including different clinical presentations, laboratory assessment and imaging advances. The differential diagnoses will be discussed, as well as possible coexisting disorders. By evaluating the existing reports and reviews describing this rare condition, this review aims to present a clinically practical suggestion on the diagnosic workup for TSHomas, Major advances and scientific breakthroughs in the imaging area in recent years, facilitating diagnosis of TSHomas, support the belief that future progress within the imaging field will play an important role in providing methods for a more efficient diagnosis of this rare condition.

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“…It has been demonstrated that TSHomas are monoclonal in origin [15], thus suggesting that the development of TSHomas might be explained by the presence of a transforming event providing gain of proliferative function followed by secondary mutations or alterations favoring tumor progression [16]. However, to date, no mutations either in oncogenes commonly activated in human cancer (e.g.…”

Section: Introductionmentioning

confidence: 99%

2013 European Thyroid Association Guidelines for the Diagnosis and Treatment of Thyrotropin-Secreting Pituitary Tumors

Beck‐Peccoz¹,

Lania²,

Beckers³

et al. 2013

Eur Thyroid J

227

210

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Hyperthyroidism is mainly due to autoimmune thyroid disorders or toxic goiter, and very rarely to the presence of thyrotropin (TSH)-secreting pituitary adenomas (TSHomas). These tumors are characterized by high levels of circulating free thyroid hormones (FT4 and FT3) in the presence of nonsuppressed serum TSH concentrations. Failure to correctly diagnose TSHomas may result in inappropriate thyroid ablation, which results in a significant increase of pituitary tumor mass. The diagnosis is mainly achieved by measuring TSH after T3 suppression and TRH stimulation tests. These dynamic tests, together with pituitary imaging and genetic testing are useful in distinguishing TSHomas from the syndromes of resistance to thyroid hormone action. The treatment of choice is surgery. In cases of surgical failure, somatostatin analogs have been found to be effective in normalizing TSH secretion in more than 90% of patients.

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A Plurihormonal TSH-Producing Pituitary Tumor of Monoclonal Origin in a Patient with Hypothyroidism

Cited by 19 publications

References 8 publications

Clinicopathological characteristics and therapeutic outcomes in thyrotropin-secreting pituitary adenomas: a single-center study of 90 cases

Clinicopathological characteristics and therapeutic outcomes in thyrotropin-secreting pituitary adenomas: a single-center study of 90 cases

DIAGNOSIS OF ENDOCRINE DISEASE: Diagnostic approach to TSH-producing pituitary adenoma

2013 European Thyroid Association Guidelines for the Diagnosis and Treatment of Thyrotropin-Secreting Pituitary Tumors

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