Case Report
Sellar Toxoplasmosis and Nonfunctioning Pituitary Adenoma Sven Berkmann1, Ingeborg Fischer2, Beat Sonderegger3, Stefan Fischli4, Javier Fandino1
Key words Pituitary - Prolactin - Toxoplasmosis -
Abbreviations and Acronyms MRI: Magnetic resonance imaging PRL: Prolactin From the Departments of 1Neurosurgery and 2Pathology, Kantonsspital Aarau, Aarau; and Departments of 3Infectious Diseases and Hospital Epidemiology and 4Internal Medicine, Kantonsspital Lucerne, Lucerne, Switzerland To whom correspondence should be addressed: Sven Berkmann, M.D. [E-mail: [email protected]] Citation: World Neurosurg. (2015). http://dx.doi.org/10.1016/j.wneu.2015.05.029 Journal homepage: www.WORLDNEUROSURGERY.org Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2015 Elsevier Inc. All rights reserved.
INTRODUCTION We present a rare case of a healthy woman with a pituitary tumor, who was medically treated for hyperprolactinemia; on transsphenoidal resection of the sellar mass, Toxoplasma gondii bradycysts within a nonfunctioning pituitary adenoma were found. T gondii, an obligate intracellular parasite of the phylum Apicomplexa, causes chronic infections in one third of the human population (7). It may infect a great variety of cell types including epithelial cells and blood leukocytes (10). Intracranial infections with T gondii are mainly diagnosed after congenital transmission and in patients with immunodeficiencies (11). The cooccurrence of sellar toxoplasmosis with a pituitary adenoma and hyperprolactinemia may be extremely rare; nevertheless, this report may raise awareness of clinicians to reconsider the differential diagnoses of hyperprolactinemia before long-lasting drug therapies are indicated. CASE DESCRIPTION Informed consent for submission of this case report was obtained from the patient. A 27year-old woman presented with secondary
- BACKGROUND:
Sellar toxoplasmosis is associated with congenital infections or immunodeficiency. The finding of Toxoplasma bradycysts in a pituitary adenoma is very unusual.
- CASE
DESCRIPTION: An otherwise healthy 27-year-old woman presented with secondary amenorrhea and moderately elevated prolactin levels. A macroprolactinoma was suspected on magnetic resonance imaging, and cabergoline was initiated. Although dopamine levels decreased, the tumor did not show significant shrinkage; after 2 years, transsphenoidal resection was indicated to clarify the diagnosis and to cure hyperprolactinemia. Histology showed an inactive pituitary adenoma and Toxoplasma bradycysts. Seropositivity for Toxoplasma gondii, but neither immunodeficiency nor intracerebral spread, was found. During a postoperative follow-up period of 15 months, the patient did not show any recurrence.
- CONCLUSIONS:
Sellar toxoplasmosis in conjunction with pituitary adenoma is extremely rare. Nonfunctioning lesions should be suspected in cases of sellar masses and moderate hyperprolactinemia.
amenorrhea, which was noted 9 months after discontinuing oral contraceptives. Blood tests revealed an elevation of prolactin (PRL) to 121 mg/L (normal range, 4.8e23.3 mg/L) and insufficiency of the gonadotroph and corticotroph axes. A “hook effect” (i.e., false low levels (3, 16)) was excluded. Magnetic resonance imaging (MRI) showed an intrasellar, partially cystic tumor of 14 mm 12 mm 10 mm (Figure 1). A macroprolactinoma was suspected, and medical therapy with cabergoline (Cabaser; Pfizer AG, Zurich, Switzerland) 2 0.25 mg/week was initiated. Hydrocortisone (Hydrocortone; Pro Concepta Zug AG, Baar, Switzerland) 2 10 mg/day additionally was prescribed based on morning cortisol levels of 152 nmol/L (normal range, 170e540 nmol/L); dynamic testing with tetracosactide (Synacthen, Mallinckrodt Schweiz AG, Switzerland) led to an insufficient increase after 1 hour (426 nmol/ L; normal level, >550 nmol/L). During therapy, PRL levels normalized, and menorrhea started again. Hydrocortisone could be discontinued after 3 months. Cabergoline was reduced stepwise and could be stopped after 2 years. Thereafter, PRL levels increased slightly to
WORLD NEUROSURGERY - [-]: ---, MONTH 2015
25 mg/L; however, the patient remained asymptomatic. MRI showed a slight decrease of the total tumor volume; nevertheless, hemorrhage into the cyst was suspected. Restarting dopamine agonist therapy was discussed; however, doubts about the correct diagnosis were raised, and the patient was not compliant with the prospect of continuing medication for an uncertain duration. Selective transsphenoidal adenomectomy was indicated to clarify the diagnosis and possibly to cure the condition. Intraoperatively, the tumor manifested as a fibrous mass, which could be easily dissected from the adenohypophysis. Its texture was not suggestive of a pituitary adenoma. posthemorrhagic changes could be found within the tumor. Intraoperative MRI did not suggest any remnants. There were no adverse events during the postoperative course, and PRL levels decreased below the normal range. On histologic analysis, a pituitary adenoma not staining for any of the tested hormones of the adenohypophysis (PRL, growth hormone, luteinizing hormone, follicle-stimulating hormone, adrenocorticotropic hormone, thyroid-stimulating
www.WORLDNEUROSURGERY.org
1.E1
CASE REPORT SVEN BERKMANN ET AL.
SELLAR TOXOPLASMOSIS AND NONFUNCTIONING PITUITARY ADENOMA
Figure 1. Magnetic resonance imaging scans before therapy with dopamine agonists and before surgery. Sagittal and coronal T1-weighted contrast-enhanced magnetic resonance images show an intrasellar mass with maximum diameters of 14 mm 12 mm 10 mm, dislocation of the pituitary gland and stalk to the right side, and intratumoral cyst (arrowhead).
hormone) was detected (Figure 2). The proliferation index was 10 mm in diameter and PRL levels
Sellar Toxoplasmosis and Nonfunctioning Pituitary Adenoma Sven Berkmann1, Ingeborg Fischer2, Beat Sonderegger3, Stefan Fischli4, Javier Fandino1
Key words Pituitary - Prolactin - Toxoplasmosis -
Abbreviations and Acronyms MRI: Magnetic resonance imaging PRL: Prolactin From the Departments of 1Neurosurgery and 2Pathology, Kantonsspital Aarau, Aarau; and Departments of 3Infectious Diseases and Hospital Epidemiology and 4Internal Medicine, Kantonsspital Lucerne, Lucerne, Switzerland To whom correspondence should be addressed: Sven Berkmann, M.D. [E-mail: [email protected]] Citation: World Neurosurg. (2015). http://dx.doi.org/10.1016/j.wneu.2015.05.029 Journal homepage: www.WORLDNEUROSURGERY.org Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2015 Elsevier Inc. All rights reserved.
INTRODUCTION We present a rare case of a healthy woman with a pituitary tumor, who was medically treated for hyperprolactinemia; on transsphenoidal resection of the sellar mass, Toxoplasma gondii bradycysts within a nonfunctioning pituitary adenoma were found. T gondii, an obligate intracellular parasite of the phylum Apicomplexa, causes chronic infections in one third of the human population (7). It may infect a great variety of cell types including epithelial cells and blood leukocytes (10). Intracranial infections with T gondii are mainly diagnosed after congenital transmission and in patients with immunodeficiencies (11). The cooccurrence of sellar toxoplasmosis with a pituitary adenoma and hyperprolactinemia may be extremely rare; nevertheless, this report may raise awareness of clinicians to reconsider the differential diagnoses of hyperprolactinemia before long-lasting drug therapies are indicated. CASE DESCRIPTION Informed consent for submission of this case report was obtained from the patient. A 27year-old woman presented with secondary
- BACKGROUND:
Sellar toxoplasmosis is associated with congenital infections or immunodeficiency. The finding of Toxoplasma bradycysts in a pituitary adenoma is very unusual.
- CASE
DESCRIPTION: An otherwise healthy 27-year-old woman presented with secondary amenorrhea and moderately elevated prolactin levels. A macroprolactinoma was suspected on magnetic resonance imaging, and cabergoline was initiated. Although dopamine levels decreased, the tumor did not show significant shrinkage; after 2 years, transsphenoidal resection was indicated to clarify the diagnosis and to cure hyperprolactinemia. Histology showed an inactive pituitary adenoma and Toxoplasma bradycysts. Seropositivity for Toxoplasma gondii, but neither immunodeficiency nor intracerebral spread, was found. During a postoperative follow-up period of 15 months, the patient did not show any recurrence.
- CONCLUSIONS:
Sellar toxoplasmosis in conjunction with pituitary adenoma is extremely rare. Nonfunctioning lesions should be suspected in cases of sellar masses and moderate hyperprolactinemia.
amenorrhea, which was noted 9 months after discontinuing oral contraceptives. Blood tests revealed an elevation of prolactin (PRL) to 121 mg/L (normal range, 4.8e23.3 mg/L) and insufficiency of the gonadotroph and corticotroph axes. A “hook effect” (i.e., false low levels (3, 16)) was excluded. Magnetic resonance imaging (MRI) showed an intrasellar, partially cystic tumor of 14 mm 12 mm 10 mm (Figure 1). A macroprolactinoma was suspected, and medical therapy with cabergoline (Cabaser; Pfizer AG, Zurich, Switzerland) 2 0.25 mg/week was initiated. Hydrocortisone (Hydrocortone; Pro Concepta Zug AG, Baar, Switzerland) 2 10 mg/day additionally was prescribed based on morning cortisol levels of 152 nmol/L (normal range, 170e540 nmol/L); dynamic testing with tetracosactide (Synacthen, Mallinckrodt Schweiz AG, Switzerland) led to an insufficient increase after 1 hour (426 nmol/ L; normal level, >550 nmol/L). During therapy, PRL levels normalized, and menorrhea started again. Hydrocortisone could be discontinued after 3 months. Cabergoline was reduced stepwise and could be stopped after 2 years. Thereafter, PRL levels increased slightly to
WORLD NEUROSURGERY - [-]: ---, MONTH 2015
25 mg/L; however, the patient remained asymptomatic. MRI showed a slight decrease of the total tumor volume; nevertheless, hemorrhage into the cyst was suspected. Restarting dopamine agonist therapy was discussed; however, doubts about the correct diagnosis were raised, and the patient was not compliant with the prospect of continuing medication for an uncertain duration. Selective transsphenoidal adenomectomy was indicated to clarify the diagnosis and possibly to cure the condition. Intraoperatively, the tumor manifested as a fibrous mass, which could be easily dissected from the adenohypophysis. Its texture was not suggestive of a pituitary adenoma. posthemorrhagic changes could be found within the tumor. Intraoperative MRI did not suggest any remnants. There were no adverse events during the postoperative course, and PRL levels decreased below the normal range. On histologic analysis, a pituitary adenoma not staining for any of the tested hormones of the adenohypophysis (PRL, growth hormone, luteinizing hormone, follicle-stimulating hormone, adrenocorticotropic hormone, thyroid-stimulating
www.WORLDNEUROSURGERY.org
1.E1
CASE REPORT SVEN BERKMANN ET AL.
SELLAR TOXOPLASMOSIS AND NONFUNCTIONING PITUITARY ADENOMA
Figure 1. Magnetic resonance imaging scans before therapy with dopamine agonists and before surgery. Sagittal and coronal T1-weighted contrast-enhanced magnetic resonance images show an intrasellar mass with maximum diameters of 14 mm 12 mm 10 mm, dislocation of the pituitary gland and stalk to the right side, and intratumoral cyst (arrowhead).
hormone) was detected (Figure 2). The proliferation index was 10 mm in diameter and PRL levels
