Journal of Neuro-Ophthalmology, September 1982, Volume 2, Issue 3

Carotid artery aneurysm simulating pituitary adenoma.

We describe a case of a giant carotid artery aneurysm presenting as an intrasellar mass with suprasellar extension, with radiographic and endocrinologic findings suggestive of pituitary tumor. This case emphasizes that elevated prolactin is not specific for prolactinoma. This case also supports consideration of angiography before proceeding with transsphenoidal surgery for presumed pituitary adenoma.

f. Clin. Neuro-ophthalmol. 2: 1b9-174, 1982. Carotid Artery Aneurysm Simulating Pituitary Adenoma HOWARD R. KRAUSS, M.D. THOMAS l. SLAMOVITS, M.D. PATRICK A. SIBONY, M.D. JOSEPH G. VERBALlS, M.D. PAUL B. NELSON, M.D. Abstract We describe a case of a giant carotid artery aneurysm presenting as an inlrasellar mass with suprasellar ex­tension, with radiographic and endocrinologic find­ings suggestive of pituitary tumor. This case empha­sizes that elevated prolactin is not specific for prolacli­noma. This case also supports consideration of an­giography before proceeding with transsphenoidal surgery for presumed pituitary adenoma. It has been said that "obvious enlargement or distortion of the pituitary fossa on x-ray in a hyperprolactinemic patient clearly allows a diag­nosis of prolactinoma."1 Others have confirmed this sentiment.2 Within recent years, however, el­evated serum prolactin has been reported in a number of hypothalamo-pituitary disorders be­sides pituitary adenomas, including craniopharyn­gioma, 3--'l meningioma;l. x metastatic tumor/ 7 ec­topic pinealoma,7.8 neurinoma,:} empty sella, I. :J. 12. 13 sarcoid,4. 9. If! reticulum cell sarcoma,'! malignant chordoma;l benign pituitary cyst,lI third vertricle tumor,x lymphoid hypophysitis,I4 and aneurysm.H In the one previous reported case of hyperprolac­tinemia in association with aneurysm, the patient had bilateral intracavernous carotid aneurysms projecting into the sella, with a serum prolactin of 50 nglml. Several author.>, recognizing that prolactin ele­vations are typically lower in nonprolactinomas, have attempted to issue numeric guidelines. Lund­berg et al.3 noted a maximum prolactin of 114 ngl ml in their nonadenoma population; in their series of 50 patients with expansive pituitary adenomas, 40% had normal serum prolactin, 34% had values in the 15-100 nglml range, and 26% had serum prolactins greater than 100 ng/ml; in their series of From the Departments of Ophthalmology (HRK, TLS, PAS), Neurology (TLS), Medicine (JGV), and Neurological Surgery (PBN), University of Pittsburgh, Pittsburgh, Pennsylvania. September 1982 15 patients with invasive, bone destroying, pitui­tary adenomas all showed serum prolactins greater than 1,000 nglml. Tindall et aLI" suggest that, "whenever the fasting level of prolactin exceeds 150 ng/m!, it almost certainly indicates ... a pitui­tary adenoma." We report a case of a giant carotid artery aneu­rysm, presenting with ipsilateral visual loss, with polytomography and computerized tomography suggestive of pituitary adenoma, with serum pro­lactin levels ranging 147-172 nglml and suppres­sion of other pituitary functions. Case Report A 59-year-old woman was evaluated by her family physician in September 1979 for a com­plaint of severe, intermittent suboccipital head­aches, of increasing severity for 1 year. Her phys­ical exam was unremarkable. An electroencepha­logram was normal; cervical spine x-rays showed early degenerative changes. Skull series revealed an enlarged sella; further evaluation was done to rule out a pituitary tumo!". Visual acuity and visual fields were normal. Polytomography revealed thin­ning of the sellar floor and demineralization of the posterior clinoids. An unenhanced computerized tomographic scan was normal. Basal endocrine function was within normal range, including morn­ing and evening cortisol, thyroxine, thyroid stim­ulating hormone, lutenizing hormone, follicle stim­ulating hormone, and growth hormone. A diagno­sis of cervical osteoarthritis with tension headaches was made, and she was treated with indomethacin which produced limited symptomatic relief. In December 1979, the patient complained of sudden onset of right eye pain and visual disturb­ance, and a vesicular rash developed over the right forehead. Ophthalmologic evaluation, elsewhere, noted diminished vision, a right relative afferent pupillary defect and iritis with elevated intraocular pressure; examination of the left eye was normal. Visual field testing revealed a central scotoma in 169 Carotid Aneurysm Figure 1. Visual field testing revealed a cecocentral scotoma, right eye, and temporal loss of 1-1. left eye. the right eye; the left eye was normal. A diagnosis of herpes zoster ophthalmicus was made. The pa­tient's vision did not improve and a repeat com­puterized tomographic brain scan was performed, this time with contrast, revealing an area of abnor­mal enhancement in the sella and extending 3 cm above the sella. The patient was referred to the University of Pittsburgh-Presbyterian Hospital in April 1980. Ophthalmologic consultation recorded: visual acu­ity, right eye: count fingers, left eye: 20/25, right relative afferent pupillary defect, right dendritic keratitis, and right iritis with keratic precipitates. Goldmann visual field testing (Fig. 1), revealed a cecocentral scotoma, right eye, and displayed a temporal field cut to I-I only, left eye. Radiographic evaluation was repeated, including skull films and polytomography of the sella (Fig. 2), and comput­erized tomography of the brain (Fig. 3), confirming the presence of a large intra- and suprasellar mass. Repeat endocrinologic evaluation revealed: normal basal serum growth hormone, but a blunted re­sponse to a maximum of 6 nglml with arginine­insulin stimulation, low follicle stimulating hor­mone 11.4 miu/ml (normal postmenopausal range 30-200), low luteinizing hormone 12.6 miulml (normal postmenopausal range 35-200), low thy­roxine 2.8 mcgldl (normal range 4.9-10.1), low morning cortisol 1.25 mcg/dl (normal range 7-25), with a blunted response to only 3 after insulin, and elevated serum prolactin levels of 147, 165, and 172 ng/ml (normal female range below 30). So far, the radiologic and endocrinologic evaluation was suggestive of a pituitary adenoma, but cerebral angiography revealed a giant right carotid artery aneurysm arising near the origin of the ophthalmic artery and projecting medially and inferiorly into the sella turcica and suprasellar cistern (Fig. 4). The patient was begun on replacement therapy of hydrocortisone and thyroxine and readmitted 2 weeks later when she was endocrinologically stable for surgery. A frontotemporal craniotomy was per­formed; subfrontal retraction revealed a very stretched and attenuated right optic nerve and a bulging giant aneurysm, which was subsequently clipped. Postoperatively the patient did well; serum prolactin rapidly normalized (Fig. 5), hydrocorti­sone and thyroxine were gradually discontinued without adverse effect, and 2 months postopera­tively tests confirmed complete normalization of all pituitary function. Subsequent ophthalmologic evaluations revealed no significant improvement in right eye visual acuity or field; the keratouveitis has resolved. Discussion Carotid artery aneurysms mimicking pituitary tumors are uncommon but well described.1 6-22 In 1912, Cushing17 noted than an aneurysm could, "by its compression effects cause outspoken hy­popituitarism." The presumed mechanism of hy­perprolactinemia in our case, and in other non­adenomatous lesions, is hypothalamic andlor pi­tuitary stalk compression, interfering with the de­livery of prolactin inhibiting factor to the pituitary. No previous reports have documented pituitary dysfunction which nonnalized following treatment of the aneurysm. The endocrinologic implications of this case have been discussed in greater detail elsewhere.23 Journal of Clinical Neuro-ophthalmology Krauss, et al. Figure 2. wter.!1 polytome showing enlargement of the sella, thinning of the floor, and demineralization 01 the dorsum sellae. Figure 3. Computerized axial tomogram of the brain, with contrast, showing an enh.!ncing suprasellar mass. September 1982 171 Figure 4. (a) Antero-posterior subtracted angiogram. (b I Lateral subtracted angiogram, showing giant rIght carotid artery aneu­rysm projecting into the sella turcica and suprasellar cistern. 200 :surgery • • --L prolactin ng/ml ISO • 100 SO 30 _______~!~Ji~~_!t_~~~~!! _ • 2 mOl. 4 mOl. O........ ...'~I......... ... ... .. _ 3wks. 0371013 Imo. pr••op days post-op Figure 5, Serum prolactin !C'vC'ls prC'- ,lI1d poslopC'ratively. Prolactin fell to one-half the preoperative level the day after surgery and h"ld 'teddy for 10 d.ly'. TWll w(,l'ks ,lJld 2 months postoperatively. the level was normal. There was an unexplained 1,It·v.Ilioll I month po...topl'r.ltlvely. Journal of Clinical Neuro-ophthalmologv With increasing comfort in interpret.ltion of to­day's high resolution computerized tomogr.lIns, along with adjunctive use of polytomogr.lphy and endocrinologic evidence of pituitary adenoma, there has been a move away from cerebral angiog­raph/' to. ~4-~li as p.ut of the ev.llu.ltion prior to transsphenoidal microsurgery. Hold .lngiogr.lphy not been performed in our c.lse.•md .1 tr.lI1ssphe­noidal approach been t,lken, one CJn imdgine the disastrous consequence. White and B.lll.lntine'M de­scribe a case Of.1 61-ye.u-old I.ldy presenting with abnormal fdtigability. whose I.lbor.ltory tests indi­cated severe panhypopituitarism .md whose x-rays were reproduced in a textbook~7 as d typical ex­ample of progressive sellar destruction by a pitui­tary adenoma; she was treated with cortisone and thyroid extract with clinical improvement. Two years later because of suprasellar enlargement, she received radiotherapy. Four years after presenta­tion a dense bitemporal hemianopsia and a left sixth nerve palsy had developed. Transsphenoidal surgery was planned; on entering the eroded floor of the sella a gush of blood was encountered, controlled with difficulty by packing. Subsequent arteriography revealed a right carotid aneurysm filling the sella. The patient had recurrent epistaxis, underwent common carotid ligation, still bled, and following craniotomy with clipping of the aneu­rysm, died the next morning, 3 months after the transsphenoidal approach. Our case emphasizes the need for careful con­sideration of cerebral angiography, before proceed­ing with therapy for what one presumes to be a pituitary adenoma. References 1. Editorial: Hyperprolactinaemia: Pituitary tumor or not? Lancet 1: 517,1980. 2. Keye, W.R., Chang, R.j., Wilson, CB., and joffe, R.B.: Prolactin-secreting pituitary adenomas. III. Fre­quency and diagnosis in am"enorrhea-galactorrhea. f.AM.A 244: 1329, 1980. 3. Lundberg, P.O., Osterman, P.O., and Wide, L.: Se­rum prolactin in patients with hypothalamus and pituitary disorders. ]. Neurosurg. 55: 194, 1981. 4. Kleinberg, D.L, Noel, G.L, and Frantz, A.G.: Galac­torrhea; a study of 235 cases, including 48 with pituitary tumors. N. Engl. ]. Med. 296: 589, 1977. 5. Kapcala, LP., Molitch, M.E., Post, K.D., Biller, B.j., Prager, R.j., jackson, I.M.D., and Reichlin, S.: Galac­torrhea, oligo/amenorrhea, and hyperprolactinemia in patients with craniopharyngiomas. ]. Clin. Endo­crinol. Metab. 51: 798, 1980. 6. Vargas, '.R., Pino, j.A., and Murad, T.M.: Cranio­pharyngioma in two siblings. f.AM.A 246: 1807, 1981. 7. Turkington, R.W.: Secretion of prolactin by patients with pituitary and hypothalamic tumors. f. Clin. Endoerinol. Metab. 34: 159, 1972. 8. Balagura, S., Frantz, A.G., Housepian, E.M., and September 1982 Krauss, et .11. Carn)('l, r. W.: The specificity of serum prolactin as a diagnostic indicator of pituitary adenoma. f. Neu­rosurg. 51: 42, 1979. Q. Brust, j.C.M., Rhee, R.5., Plank, C.R., Newmark, M., Felton, c.r., and Lewis, LD.: Sarcoidosis, galactor­rhe, l .lnd .lmenorrhea: 2 autopsy cases, 1 with Chiari-Frommel syndrome. Ann. Neurol. 2: 130, 1<)77. 10. Turkington, R.W., and Maclndoe, j.D.: Hyperpro­l. lCtinemiJ in sJrcoidosis. Ann. Intern. Med. 76: 545, 1972. 1I. robbs, M.E.: Amenorrhea-galactorrhea syndrome c.lUsed by J benign pituitary cyst: Case report. Fertil. Ster;l. 33: 451,1980. 12. Archer, D.F., Maroon, '.C, and DuBois, r.j.: Galac­torrheJ, amenorrhea, hyperprolactinemia, and an empty sella. Obstet. Gynecol. 52(SuppI. 1): 23, 1978. 13. Haney, A.F., Kramer, R.5., Wiebe, R.H., and, Ham­mond, CB.: Hypothalamic-pituitary function and radiographic evaluation of women with hyperpro­lactinemia and an "empty" sella turcica. Am. f. Obstet. Gynecol. 134: 913, 1979. 14. Portocarrero, Cj., Robinson, A.G., Taylor, A.L, and Klein, I.: Lymphoid hypophysitis-An unusual case of hyperprolactinemia and enlarged sella turcica. ].AM.A 246: 1811, 1981. IS. Tindall, G.T., McLanahan, CS., and Christy, j.H.: Transsphenoidal microsurgery for pituitary tumors associated with hyperprolactinemia. f. Neurosurg. 48: 849, 1978. 16. Mitchell, S.W.: Aneurysm of an anomalous artery causing antero-posterior diversion of the chiasm of the optic nerves and producing bitemporal hemi­anopsia. f. Nerv. Ment. Dis. 14: 14, 1889. 17. Cushing, H.: The Pituitary Body and its Disorders: Clinical States Produced by Disorders of the Hypo­physis Cerebri. j.B. Lippincott, Philadelphia, 1912, p. 99. 18. White, j.C, and Ballantine, H.T., jr.: lntrasellar aneurysms simulating hypophyseal tumours. ]. Neu­rosurg. 18: 34, 1961. 19. Gallagher, G.P., Dorsey, j.F., Stefanini, M., and Loo­ney, j.M.: Large intracranial aneurysms producing panhypopituitarism and frontal lobe syndrome. Neurology 12: 829, 1956. 20. Hoff, W.V.T., Hornabrook, R.W., and Marks, V.: Hypopituitarism associated with intracranial aneu­rysms. Br. Med.]. 2: 1190, 1961. 21. Kahana, L., Lebovitz, H., Lusk, W., McPherson, H.T, Davidson, E.T, Oppenheimer, j.H., Engel, F.L. Woodhall, B., and Odom, G.: Endocrine manifesta­tions of intracranial extrasellar lesions. ]. Clin. En­docrin. Metab. 22: 304, 1962. 22. Shantharam, V.V., and Clift, G.V.: Suprasellar aneu­rysm. An unusual case of hypopituitarism. rAM.A 229: 1473, 1974. 23. Verbalis, j.G., Nelson, r.B., and Robinson, A.G.: Reversible panhypopituitarism caused by a supra­sellar aneurysm: The contribution of mass effect to pituitary dysfunction. Neurosurg. 1982. (In press.) 24. Post, K.D., Biller, B.j., Adelman, L.S., Moliteb, M.E., Wolpert, S.M., and Reichlin, S.: Selective transsphe­noidal adenomectomy in women with galactorrhea­amenorrhea. ].AM.A 242: 158, 1979. 25. Richmond, I.L., Newton, TH., and Wilson, CB.: 173 Cuotid Aneurysm Prol'lctin-secreting pituitary adenomas: Correlation of r.ldiogrJphic ,md surgical findings. A.].f\. 134: 707, 1980. 2b. R.lskind, R.: PituitMy Jpoplexy. ].A.M.A. 246: 834, 1°111. 27. Holmes, G. W., .1Ild Robbins, L.L.: Roentgen Inter­pret. ltion Le.l Jnd Ferbiger, PhilJdelphiJ, 1955, p. 141. Acknowledgment This case was presented before the 13th Annual Frank Walsh Society Meeting, February 27, 1981, Houston, Texas. Write fDr reprints to: Thomas L. SJamovits, M.D., Department of Ophthalmology, Eye and Ear Hospital, 230 Lothrop Street, Pittsburgh, Pennsylvania 15213. Journal of Clinical Neuro-ophtha!mo!ogy