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LETTER TO EDITOR
Year : 2012  |  Volume : 60  |  Issue : 5  |  Page : 530--532

Spontaneous disappearance of the pituitary macroadenoma after apoplexy: A case report and review of the literature

Shuang Liu, Xiang Wang, Yan-hui Liu, Qing Mao 
 Department of Neurosurgery, West China Hospital of Sichuan University, Chengdu, P. R. China

Correspondence Address:
Shuang Liu
Department of Neurosurgery, West China Hospital of Sichuan University, Chengdu, P. R. China




How to cite this article:
Liu S, Wang X, Liu Yh, Mao Q. Spontaneous disappearance of the pituitary macroadenoma after apoplexy: A case report and review of the literature.Neurol India 2012;60:530-532


How to cite this URL:
Liu S, Wang X, Liu Yh, Mao Q. Spontaneous disappearance of the pituitary macroadenoma after apoplexy: A case report and review of the literature. Neurol India [serial online] 2012 [cited 2020 Apr 9 ];60:530-532
Available from: http://www.neurologyindia.com/text.asp?2012/60/5/530/103211


Full Text

Sir,

Pituitary apoplexy is a rare but potentially life-threatening clinical syndrome resulting from the acute hemorrhage, infarction, or hemorrhagic infarction of a pituitary tumor. Spontaneous disappearance of pituitary adenoma following apoplexy is very rare; therefore, this report has been presented.

A 66-year-old man, with history of occasional headaches of 2 months duration, was referred for sudden-onset severe headache, vomiting, and right ptosis. Neurological examination revealed a confused individual, right ocular nerve palsy, decreased visual acuity, and signs of meningeal irritation. He was diagnosed as a case of pituitary tumor with hypopituitarism based on clinical and computed tomography (CT) scan [Figure 1] a 20 days before this admission. Emergent magnetic resonance imaging (MRI) scan done at this admission revealed an intra-sellar mass of size 1.8 Χ 1.4 Χ 1.7cm [Figure 1]b-e. The lesion was hyperintense on both T1- and T2-weighted images (T1WI and T2WI, respectively) suggestive of hemorrhage. MR-angiography excluded any intracranial vascular lesion [Figue 1]f. Pituitary hormone profile revealed the following: serum free T4 6.51 pmol/l (normal 12-22 pmol/l), free T3 2.56 pmol/l (normal: 3.60- -7.50 pmol/l), and thyroid stimulating hormone 0.254 mIU/l (normal: 0.27-4.2 mIU/l), prolactin 1.43 ng/ ml (normal: 4.6-21.4 ng/ml), testosterone 0.03 ng/ml (normal: 1.93-7.40 ng/ml), follicular stimulating hormone - 0.7 mIU/ml (normal:1.7-8.6 mIU/ml), and luteinizing hormone 4.3 mIU/ml (normal:1.5-12.4 mIU/ml. Plasma cortisol at 8 a.m. was 47.17 nmol/l (normal: 147.3-609.3 ng/l)and, adeno-corticotrophic hormone was 2.79 ng/l (normal:5.0-78 ng/l), and growth hormone was normal. Diagnosis of pituitary apoplexy was considered and he was put on hormonal replacement therapy with oral prednisone (5 mg daily) and l-thyroxine (2 μg, daily). He had relief of headache and improvement of eyesight in the next 2 days. Considering these clinical improvement, He was discharged 7 days later. Repeat MRI done at 3-months follow-up revealed complete disappearance of the sellar lesion [Figure 2]. Physical examination was essentially normal with normal vision. Pituitary hormonal functions were normal and hormone replacement therapy was discontinued.{Figure 1}{Figure 2}

Spontaneous disappearance of pituitary adenoma following apoplexy is reported rarely; however, remission of hormonal effects (e.g., acromegaly, Cushing's disease) has occasionally been described. [1],[2] Functioning pituitary adenomas may exhibit spontaneous resolution of the symptoms by reduced production of pituitary hormones following pituitary apoplexy and may result in hypopituitarism. [3],[4] We reviewed the relevant literature on the spontaneous partial or complete radiological disappearance of adenoma following pituitary apoplexy and the cases treated with dopaminergic agonist cabergoline were excluded from the review. Dopaminergic agonist therapy can result in the resolution of macroprolactinomas. [5] The review included nine patients including the present patient. [1],[4],[6],[7],[8],[9],[10] The average age of the patients was 48 years, with male gender predominance (78%). Eight patients had macroadenoma. The average length of follow-up was 16 months, and 7 patients had a compete disappearance of the tumor. Six patients underwent supplemental hormonal therapy, while 3 patients were managed conservatively, and all the patients had a good recovery.

The possible mechanism underlying the spontaneous disappearance of pituitary adenoma following apoplexy is poorly understood. Pituitary apoplexy may occur when the large tumor compresses or outgrows the nutrient vessels from the superior and inferior hypophyseal arteries. This may result in ischemic necrosis and hemorrhage into the necrotic tumor. Histology of the surgically removed lesion demonstrated hemorrhagic lesions, ischemic necrosis, or combinaton both. [8] The complex and variable imaging features seen on MRI may be related to these histological findings. [9] Maccagnan et al. analyzed the follow-up MRI/CT scans of pituitary apoplexy and suggested that the presence of a large hypodense area within the tumor is probably due to infarction and may predict subsequent tumor resolution. [2],[3] Wu et al. suggested that ischemic necrosis of pituitary adenoma after apoplexy may be liquefied and absorbed, ultimately resulting in spontaneous resolution of adenoma. [5] In our patient, MRI showed hemorrhagic infarction of the pituitary adenoma. We propose that there might have been inflammatory infiltrates of the ischemic necrotic area with subsequent liquefaction and absorption, and hemorrhage-induced inflammation might have promoted hematoma absorption. In addition, the leakage of blood or necrotic material into the subarachnoid space might have also contributed to the complete disappearance of the adenoma.

The optimal treatment for the acute pituitary apoplexy is still controversial. Some authors believe that surgical intervention is required when there is an acute deterioration in conscious state or visual acuity. A potential advantage of surgical management is a lower recurrence rates of apoplexy. Some authors advocate conservative management in some carefully selected patients who have no impaired vision or consciousess impairment. [9] Our patient underwent a conservative therapy and had a good outcome. A spontaneous radiological disappearance of the lesion in this process may result in the improvement of clinical features and may also result in endocrine remission. This exceptional curative process should be considered when we opt for the conservative therapy. This case showed spontaneous recovery with satisfactory results with conservative treatment. Though the MRI showed apparent complete disappearance of the adenoma, it is not likely that all the tumor cells have been fully destroyed, and thus there may be a risk recurrence. [1],[10] Thus So, these patients require careful follow-up study.

References

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2Wichers M, Kristof RA, Springer W, Schramm J, Klingmüller D. Pituitary apoplexy with spontaneous cure of acromegaly and its Possible Relation to Gd-DTPA-Administratio. Acta Neurochir (Wien). 1997;39:992-994.
3Maccagnan P, Macedo CL, Kayath MJ, Nogueira RG, Abucham J. Conservative management of pituitary apoplexy: A prospective study.J Clin Endocrinol Metab. 1995;80:2190-7.
4Schatz NJ, Job OM, Glaser JS. Spontaneous resolution of pituitary adenoma after apoplexy. J Neurophthalmol. 2000;20:42-44.
5Wu ZB, Su ZP, Wu JS. Spontaneous remission of pituitary macroadenomas in women: Report of two cases. Chin Med J (Engl). 2007;120:2062-4.
6Armstrong MR, Douek M, Schellinger D, Patronas NJ. Regression of pituitary macroadenoma after pituitary apoplexy: CT and MR studies J Comput Assist Tomogr. 1991;15:832-4.
7Yoshino A, Katayama Y, Watanabe T, Hirota H. Vanishing pituitary mass revealed by timely magnetic resonance imaging: Examples of spontaneous resolution of nonfunctioning pituitary adenoma. Acta Neurochir (Wien). 2005;147:253-7.
8Piotin M, Tampieri D, Rüfenacht DA, Mohr G, Garant M, Del Carpio R, et al. The various MRI patterns of pituitay apoplexy. Eur. Radiol. 1999;9:918-23.
9Turgut M, Ozsunar Y, Baþak S, Güney E, Kir E, Meteoðlu I. Pituitary apoplexy: An overview of 186 cases published during the last century. Acta Neurochir (Wien). 2010;152:749-61.
10Kamiya Y, Jin-No Y, Tomita K, Suzuki T, Ban K, Sugiyama N, et al. Recurrence of cushing's disease after long-term remission due to Pituitary Apoplexy. Endocr J 2000;47:793-797.