363Neurology India | September 2005 | Vol 53 | Issue 3

CMYK363

with bromocriptine and prepared for surgery. A right frontal

craniotomy using subfrontal approach was performed. Upon

incising the diaphragma sellae 5–7 ml of necrotic material

was excised. Postoperatively, the patient developed frank dia-

betes insipidus that needed aqueous vasopressin treatment.

His visual acuity did not improve.

Haematoxylin and eosin-stained sections from the biopsy

showed complete effacement of the pituitary structure by a

dense lymphoplasmacytic infiltrate and foci of neutrophilic

infiltration, necrosis, and fibrosis [Figure 3]. No tumor could

be identified. Stains for bacteria, fungi, Acid Fast Bacilli and

spirochaetes were negative. Immunohistochemistry revealed

a polyclonal lymphoid population with a mixture of T and B

cells. A diagnosis of lymphocytic hypophysitis was considered.

The patient was discharged on a maintenance dose of steroids

and is currently on regular follow up.

Lymphocytic hypophysitis closely mimics a pituitary ad-

enoma both clinically and radiologically. MR features sugges-

tive of an inflammatory pituitary process include loss of

Figure 2: T2W sagittal MR image showing a sellar mass withthickened stalk and suprasellar extension

Figure 3: Photomicrograph of pituitary showing a focus of denselymphoplasmacytic infiltrate, H&E x 400

Lymphocytic hypophysitispresenting as pituitaryapoplexy in a male

Sir,

Inflammatory lesions of the pituitary are rare with an esti-

mated incidence of one case in ten million population.[1] Lym-

phocytic or autoimmune hypophysitis was first described by

Goudie and Pickerton[2] in 1962, and since then approximately

379 cases have been reported.[3] Most cases occur in women,

mostly during late pregnancy or early postpartum period. Pi-

tuitary apoplexy, a clinical syndrome of sudden onset of se-

vere neurological dysfunction due to hemorrhage or infarc-

tion of pituitary, usually occurs in the setting of a pre-existing

adenoma. Association of pituitary apoplexy and lymphocytic

hypophysitis has been reported only twice earlier, both in fe-

male patients.[4],[5]

A 42-year male presented with progressive deterioration of

vision in both eyes over a 1-year period. A contrast enhanced

CT scan revealed the presence of a sellar mass suggestive of

an adenoma. The patient was advised surgery, but he refused

to accept this advice. He was brought in an unconscious state

to our institution. A magnetic resonance (MR) image showed

a sellar mass with suprasellar extension [Figures 1, 2]. He

was treated managed with high-dose intravenous steroids, in-

travenous fluids, and eltroxine. He progressively regained con-

sciousness within 72 h. Physical examination at this time

showed a pale sallow complexion. He was blind in the right

eye with temporal hemianopia in the left eye. Visual acuity of

left eye was 6/60. Fundus examination showed primary optic

atrophy in both eyes. Hormonal profile revealed

hyperprolactinemia (4,000 IU/l). The patient also developed

polyuria with a urinary volume of 9–12 l/day. He was treated

Figure 1: T2W coronal MR image showing a large sellar mass withheterogenous signal intensity

Letter to Editor

364 Neurology India | September 2005 | Vol 53 | Issue 3

364 CMYK

hyperintense bright spot signal of the normal neurohypophy-

sis, thickening of pituitary stalk and enlargement of neurohy-

pophysis in cases where it is involved.[6] A review of literature

revealed only two case reports of this association with lym-

phocytic hypophysitis (both in female patients)[3],[4], and one

case with granulomatous hypophysitis (intrasellar tubercu-

loma).[7] Surgical intervention is required to establish the di-

agnosis and to reduce the size of the lesion to relieve the mass

effect on adjacent structures. Prasad et al.[8] proposed a man-

agement paradigm for suspected cases of lymphocytic

hypophysitis and advocated a trans sphenoidal stereotactic

biopsy to achieve a tissue diagnosis, which might avoid the

need for an open exploration of the sella.

The present case highlights the fact that apoplectic changes

may complicate the course of inflammatory pituitary lesions,

thus calling for a greater vigil and clinical judgment in man-

agement of these patients.

B. Minakshi, S. Alok, K. P. HillolDepartments of Pathology and Neurosurgery, Dr. Ram ManoharLohia Hospital, New Delhi, India, E-mail: dr_aalok@yahoo.com

References

1. Sautner D, Saeger W, Ludecke D K, Jansen V, Puchner MJA. Hypophysitis in

surgical and autoptical specimens. Acta Neuropathol 1995;90:637-44.

2. Goudie RB, Pickerton PH. Anterior hypophysitis and Hashimoto‘s disease in a

young woman. J Path Bacteriol1962; 83:583-85.

3. Caturegli P, Newschaffer C, Olivi A, Pomper MG, Burger PC, Rose NR. Auto

immune hypophysitis. Endocr Rev 2005 Jan 5; (E pub ahead of print)

4. Dan NG, Feiner RID, Houang MTW, Turner JJ. Pituitary apoplexy in associa-

tion with lymphocytic hypophysitis. J Clin Neurosc 2002; 9:577-80.

5. Lee MS, Pless M. Apoplectic lymphocytic hypophysitis-case report. J Neurosurg;

2003; 98:183-85.

6. Imura H, Nakao K, Shimatsu A,Ogawa Y, Sando T, Fujisawa I, Yamabe H.

Lymphocytic infundibuloneurohypophysitis as a cause of central diabetes in-

sipidus. N Engl J Med 1993; 329:683-89.

7. Arunkumar MJ, Rajshekhar V. Intrasellar tuberculoma presenting as pituitary

apoplexy. Neurol India 2001; 49:407-10.

8. Prasad A, Madan VS, Sethi PK, Prasad ML, Buxi TBS, Kanwar CK. Lym-

phocytic hypophysitis-can open exploration of the sella be avoided. Br J

Neurosurg 1991; 5: 639-42.

Accepted on 24-02-2005

Granulomatous hypophysitis

Sir,

Inflammatory pituitary lesions are extremely rare with an

estimated annual incidence of one case in ten million popula-

tion.[1] Histologically these can be lymphocytic, granuloma-

tous, xanthomatous, and necrotizing hypophysitis. Granulo-

matous hypophysitis is a chronic inflammatory condition of

the pituitary, first described by Simmonds in 1917.[2] Major-

ity of pituitary granulomas represent a specific lesion such as

syphilis, tuberculosis, sarcoidosis, or histiocytosis-X. In ab-

sence of any demonstrable causative agent, the process is

termed idiopathic granulomatous hypophysitis. Isolated in-

volvement of the pituitary by tuberculosis (pituitary tubercu-

loma) is very rare. Coleman and Meredith first described it in

1940, and since only then, few case reports of this entity have

been recorded.[3–5]

A 42-year-old woman presented with diplopia to lateral gaze

on left side and left-frontal headache of 6-month duration.

Her visual acuity was 6/6. A left-temporal hemianopia and

left-sixth nerve peresis were found on neurological examina-

tion. Endocrinological profile revealed mild hyperprolactinemia

(300 IU/l). Magnetic resonance imaging (MRI) showed a

sellar mass suggestive of a pituitary adenoma [Figure 1]. The

tumor was explored via a trans-sphenoidal approach and had

a yellowish gray appearance and leathery consistency. It was

noncurettable, nonsuckable, and relatively avascular.

Postoperatively the vision returned to normal with restora-

tion of left-lateral rectus muscle function and normalization

of left-temporal hemianopia. Hematoxylin and eosin (H&E)

stained sections from the biopsy revealed focal replacement of

pituitary architecture by a lymphocytic infiltrate, noncaseating

eptheloid cell granulomas with giant cells and areas of fibro-

sis [Figures 2 and 3]. Staining for Acid Fast Bacilli,

spirochetes, and fungi was negative. There was no evidence of

extrahypophyseal systemic disease. A provisional diagnosis

idiopathic granulomatous hypophysitis was thus offered. How-

ever, 3 months later, the patient complained of recurrence of

headaches and intermittent diplopia to lateral gaze. The ster-

oid dose was increased with no significant improvement. Eight

months later, she noticed drooping of left eyelid, and exami-

nation showed a left-oculomotor palsy. At this stage, a deci-

sion to start antitubercular therapy (ATT) along with main-

tenance dose of steroids was taken. The treatment regimen

comprised of isoniazid, rifampicin, and pyrizinamide for first

Figure 1: T1-W contrast enhanced MR image showing homogenouslyenhancing pituitary mass with thickened stalk

Letter to Editor

365Neurology India | September 2005 | Vol 53 | Issue 3

CMYK365

Figure 4: Post-treatment T1-W contrast enhanced MR image showingcomplete resolution of pituitary mass

4 months and isoniazid with rifampicin for subsequent

14 months. There was remarkable improvement in 3 weeks

with regression of all symptoms. MRI at 1 year showed com-

Figure 2: Photomicrograph of pituitary biopsy showing granulomatousfocus with a giant cell and chronic inflammation (H&E x 40)

Figure 3: Photomicrograph of pituitary showing an epitheloid cellgranuloma (H&E x 400)

plete resolution of the pituitary mass [Figure 4].

Majority patients with inflammatory pituitary lesions present

with mass effect causing headaches, nausea, vomiting, chiasmal

compression, and many with endocrine abnormalities and many

with both the features, as in the present case. The mild eleva-

tion of prolactin level is attributable to pituitary stalk compres-

sion. Radiological findings mimic those of an adenoma and in-

clude a homogenously enhancing sellar mass that may show

suprasellar extension. Pituitary stalk thickening in an appro-

priate clinical setting is considered a strong predictor of an in-

flammatory pathology.[6] Sarcoidosis is an important differen-

tial diagnosis in this setting and could be suspected on basis of

systemic involvement and appropriate laboratory investigations.

It has been suggested that if suspected at presentation, inflam-

matory lesions of pituitary may be managed conservatively thus

obviating the need for surgery.[6] Trans-sphenoidal surgery is

however both diagnostic and therapeutic and should be per-

formed in cases with progressive compression or those which

show radiological or clinical progression during conservative

management.[7] The present case highlights the importance of

considering a tubercular etiology in the differential diagnosis

of sellar lesions in this part of the world and the efficacy of

ATT in their management.

Minakshi Bhardwaj, Alok Sharma, Hillol K. PalDepartments of Pathology and Neurosurgery, Dr. Ram Manohar

Lohia Hospital, New Delhi, India,E-mail: drminakshi@indiatimes.com

References

1. Sautner D, Saeger W, Ludecke DK, Jansen V, Puchner MJ. Hypophysitis in

surgical and autoptical specimens. Acta Neuropathol 1995;90:637-44.

2. Simmonds M. Uber das vorkommen reisenzellen in der hypophyse.Virchows

Arch Pathol Anat 1917;223:281-90.

3. Coleman CC, Meredith JM. Diffuse tuberculoma of the pituitary gland simu-

lating tumour with postoperative recovery. Arch Neurol Psychiatr 1940;44:1076-

85.

4. Manghani DK, Gaitonde PS, Dastur DK. Pituitary tuberculoma: A case re-

port. Neurol India 2001;49:299-301.

5. Desai KI, Nadkarni TD, Goel A. Tuberculomas of hypophysitis cerebri: A re-

port of five cases. J Clin Neurosc 2003;10:562-66.

6. Flanagan DE, Ibrahim AE, Ellison DW, Armitage M, Gawne-Cain M, Lees

PD. Inflammatory hypophysitis; the spectrum of disease. Acta Neurochir (Wien)

2002;121:152-8.

7. Cheung CC, Ezzat S, Smyth HS, Asa SL. The spectrum and significance of

primary hypophysitis Clin Endocrinol Metab 2001;86:1048-53.

Accepted on 20-02-2005

A solitary cryptococcalgranuloma in animmunocompetent host

Sir,

Cryptococcomas or ‘cryptococcal granulomas’ are rare in

Letter to Editor