A rare cause of hypopituitarism: pituitary tuberculosis / Hipopituitarizmin Nadir Bir Nedeni: Hipofiz Tuberkulozu.
Pituitary tuberculosis is a rare condition that can present with hypopituitarism even without any evidence of systemic tuberculosis and is easily confused with pituitary adenomas. Headache and hypopituitarism are the most common presenting symptoms. We report the case of pituitary tuberculosis in a 39-year-old male patient who presented with panhypopituitarism. Although it is rare and difficult to diagnose, pituitary tuberculosis should be considered in every nonfunctional sellar masses, especially in fairly small ones with unexpected hypopituitarism. Turk Jem 2012; 16: 26-8
Key words: Hypopituitarism, pituitary, pituitary tumors, tuberculosis
Hipofiz tuberkulozu, sistemik tuberkulozun herhangi bir kaniti olmaksizin hipopituitarizm ile prezante olabilen ve hipofiz adenomlari ile kolaylikla karisabilen nadir bir durumdur. Bas agrisi ve hipopituitarizm en sik gorulen baslangic semptomlaridir. Bu yazida panhipopituitarizm nedeniyle basvurmus, 39 yasinda bir erkek hastada saptanan hipofiz tuberkulozu olgusu sunulmustur. Hipofiz tuberkulozu, ozellikle beklenmeyen hipopituitarizme neden olmus, kucuk boyutlu, non-fonksiyone sellar kitlelerde goz onunde bulundurulmasi gereken nadir ve tanisi zor bir hastaliktir. Turk Jem 2012; 16: 26-8
Anahtar kelimeler: Hipopituitarizm, hipofiz, hipofiz tumorleri, tuberkuloz
Granulomatous hypophysitis, which is histologically characterized by histiocytes, plasma cells and giant cells, is a rare condition that can be caused by tuberculosis, fungal infections, sarcoidosis, and autoimmune diseases (1). While granulomatous hypophysitis due to tuberculosis is extremely rare overall, it is relatively common in developing countries. Tuberculosis has been relatively well controlled following the development of efficient therapies and with global improvements in socioeconomic status (2,3). The incidence of pituitary tuberculosis has markedly decreased in recent decades. Since pituitary tuberculosis is very rare, clinicians seldom consider it in cases of hypopituitarism. If the condition is recognized preoperatively, it can be effectively managed with anti-tuberculosis drug therapy without requiring surgery. In this case report, we present a patient with pituitary tuberculosis who presented with hypopituitarism and was successfully treated with drug therapy without requiring surgery or pathologic examination.
A 39-year-old male presented with headache, generalized weakness and decreased libido two weeks after the diagnosis of skin tuberculosis. His neurological findings and visual field examination results were normal. Except for erythrocyte sedimentation rate of 88mm/h and low levels of anterior pituitary hormones, his biochemical test results were unremarkable. Magnetic resonance imaging (MRI) of the sellar area revealed a large intrasellar mass with a dimension of 15x14x12mm that extended to the suprasellar cisterns, with thickening of the infundibular stalk and elevating optic chiasma. After injection of gadolinium, the pituitary lesion was markedly enhanced except the central cystic or necrotic portion (Figure 1). Endocrine evaluation showed panhypopituitarism, and commencement of pituitary hormones replacement therapy significantly resolved the patient's complaints (Table 1). Surgical intervention was discussed with the patient and was pending until the response to the medical treatment of tuberculosis obtained. After four months of anti-tuberculosis treatment with isoniaside, rifampicin, pyrazinamide and streptomycin, the dimensions of the lesion were regressed to 9x7x5mm and it was confined to the pituitary gland. The lesion was totally invisible after eight months of therapy (Figure 2). However, the patient remained in need of hormone replacement treatment.
[FIGURE 1 OMITTED]
[FIGURE 2 OMITTED]
Table 1. Results of the hormonal parameters at presentation Patient's Serum Normal Range Level Free T3 (pg/ml) 1,55 1,71-3,71 Free T4 (ng/dl) 0,49 0,7-1,48 TSH ([micro]IU/ml) 0,012 0,35-4,94 FSH ([micro]IU /ml) 0,86 1,1-13,9 LH (mIU/ml) 0,12 0,67-23,5 Total testosterone 0,08 2,8-11 (ng/ml) Prolactin (ng/ml) 2,84 2,58-18,12 GH (ng/ml) 0,17 0,06-5 IGF-1 (ng/ml) 86 94-358 ACTH (pg/ml) 5 0-46 Cortisol ([micro]g/dl) 1 6-19
Pituitary tuberculoma was first described in 1940 by Coleman and Meredit, and many cases have been reported in subsequent years (4). Tuberculosis bacillus may usually reach the hypophysis via the hematogenous pathway but might also spread directly from the meninges or paranasal sinuses (3-5). On the other hand, in more than 70% of pituitary tuberculosis patients, primary focus could not be identified (5).
In many ways, pituitary tuberculosis resembles a nonfunctional pituitary adenoma. Usual presenting symptoms are headache, visual disturbances or typical symptoms of hypopituitarism (2,5). When a mass compresses on the dopaminergic axis, symptoms due to hyperprolactinemia may occur. Although panhypopituitarism is a common laboratory finding, the majority of patients do not clearly manifest clinical features of impaired pituitary function. There is no definitive symptom or laboratory finding that can clearly differentiate nonfunctional adenoma from pituitary tuberculosis (3,5). Increased erythrocyte sedimentation rate (ESR), as seen in systemic tuberculosis, is a distinctive clue towards pituitary tuberculosis in a patient with pituitary mass lesion. Our patient had been diagnosed with skin tuberculosis prior to experiencing complaints due to hypopituitarism.
Due to the distinguishing image capabilities of MRI, it is the preferred imaging modality for pituitary lesions. Nevertheless, there is no radiological finding that is specific to pituitary tuberculoma (3-5). Pituitary tuberculous lesions are usually hypodense and homogenous lesions, frequently extending to the suprasellar area. Various case series reported differing rates of suprasellar extension (1-3). Although thickening and nodularity of the pituitary stalk are considered indications of pituitary tuberculoma, it should be kept in mind that these can also be seen in other inflammatory diseases such as sarcoidosis, syphilis, and in some neoplastic diseases (2,5). Thickening of sphenoid sinus is another non-specific finding associated with pituitary tuberculosis. In our patient, T1-weighted MRI showed an isointense intrasellar mass with slightly deviated stalk. After administration of gadolinium, the lesion was well defined, homogenous and obviously enhanced with thickened infundibular stalk. We were not able to perform peripheral ring leptomeningeal enhancement or other parenchymatous brain tuberculomas (Figure 1).
Surgical therapy for pituitary tuberculoma is indicated when tissue diagnosis or tumor debulking is essential (5). It has been reported that the response to bactericidal drug combination therapies such as rifampicin, isoniaside, pyrazinamide or streptomycin is extremely positive, as these drugs can effectively penetrate the blood-brain barrier. Even though there are no guidelines on duration of therapy, it is recommended to continue combined therapy with isoniaside and rifampicin for at least nine months (2,5). Long-term chemotherapy could lead to resolution of the lesion and also reversal of the hypopituitarism. Our patient was already receiving anti-tuberculosis therapy for skin tuberculosis. Four-agent therapy was given for three months, and then a two-agent therapy with isoniaside and rifampicin was continued for an additional nine months. While this report was being prepared, the patient was at the eighth month of therapy. His pituitary lesion was totally resolved visually and his disease was following a stable course, but recovery of the endocrine functions has not yet been achieved.
In conclusion, although it is a rare condition and difficult to diagnose, pituitary tuberculosis should be considered in all non-functional sellar masses, especially in relatively small masses with unexpected hypopituitarism. Thickening of the stalk and leptomeningeal enhancement may be radiologic clues to the diagnosis of tuberculosis. Although most patients have no history of tuberculosis, this is the most critical evidence for diagnosis without surgery.
(1.) Sinha S, Singh AK, Tatke M, Singh D. Hypophyseal tuberculoma: direct radiosurgery is contraindicated for a lesion with a thickened pituitary stalk: case report. Neurosurgery 2000;46:735-8.
(2.) Pereira J, Vaz R, Carvalho D, Cruz C. Thickening of the pituitary stalk. A finding suggestive of intrasellar tuberculoma? Case report. Neurosurgery 1995;36:1013-6.
(3.) Esposito V, Fraioli B, Ferrante L, Palma L. Intrasellar tuberculoma: case report. Neurosurgery 1987;21:721-3.
(4.) Coleman CC, Meredith JM. Diffuse tuberculoma of the pituitary gland simulating tumor with postoperative recovery. Arch Neurol Psychiatry 1940;44:1076-85.
(5.) Sharma MC, Arora R, Mahapatra AK, et al. Intrasellar tuberculoma-an enigmatic pituitary infection: a series of 18 cases. Clin Neurol Neurosurg 2000;102:72-7.
Address for Correspondence/Yazisma Adresi: Ozen Oz Gul MD, Uludag University, Faculty of Medicine, Department of Endocrinology and Metabolism, Bursa, Turkey Phone: +90 224 295 11 64 E-mail: email@example.com Recevied/Gelis Tarihi: 03.02.2012 Accepted/Kabul Tarihi: 02.04.2012
Turkish Journal of Endocrinology and Metabolism, published by Galenos Publishing.
Ozen Oz Gul, Erdinc Erturk, Soner Cander, Oguz Kaan Unal, Bahattin Hakyemez *, Sazi Imamoglu
Uludag University, Faculty of Medicine, Department of Endocrinology and Metabolism, Bursa, Turkey
* Uludag University, Faculty of Medicine, Department of Radiology, Bursa, Turkey
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|Title Annotation:||Case Report Olgu Sunumu|
|Author:||Gul, Ozen Oz; Erturk, Erdinc; Cander, Soner; Unal, Oguz Kaan; Hakyemez, Bahattin; Imamoglu, Sazi|
|Publication:||Turkish Journal of Endocrinology and Metabolism|
|Date:||Mar 1, 2012|
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