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| Year : 2001 | Volume
: 49
| Issue : 3 | Page : 314-6 |
Suprasellar tuberculoma presenting with diabetes insipidus and hypothyroidism--a case report.
Jain R, Kumar R
Department of Radiodiagnosis, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow-226014, India.
Correspondence Address:
Department of Radiodiagnosis, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow-226014, India.
Suprasellar tuberculomas are exceptionally rare and are even rarer in children. A case of suprasellar tuberculoma in a five year old male child who presented with diabetes insipidus, hypothyroidism and visual field defect is described. Deranged CSF parameters, imaging findings of a suprasellar rounded, peripherally enhancing lesion with thickening of the pituitary stalk, led to an accurate diagnosis of tuberculosis. The patient responded well to antituberculous treatment and an unnecessary surgery was avoided.
How to cite this article:
Jain R, Kumar R. Suprasellar tuberculoma presenting with diabetes insipidus and hypothyroidism--a case report. Neurol India 2001;49:314 |
Tuberculomas of central nervous system (CNS) can occur at any site and in any age group, though they are more frequent in children than in adults. In developing countries, tuberculomas constituted approximately 30% of space-occupying intracranial lesions before the advent of antitubercular chemotherapy. They still constitute a major health hazard and may account for 0.15-4% of intracranial space occupying lesions.[1] Tuberculomas commonly involve cerebrum and cerebellum, but can rarely occur in brain stem, basal ganglia, thalamus and even sellar or suprasellar regions. In the pre-CT era, their clinical diagnosis could only be presumptive. The imaging appearance of tuberculomas is non-specific, however, their diagnosis in clinically suspected cases is still possible with a reasonable degree of certainty, particularly in endemic areas. Sellar and suprasellar tuberculomas, as isolated manifestations of the disease, are rare though not uncommon at post-mortem examinations.[2],[3] Very few cases of sellar tuberculomas presenting as nonsecreting masses or mimicking secreting adenomas have been reported in the literature. We discuss here a case of suprasellar tuberculoma presenting mainly with early diabetes insipidus and associated hypothyroidism.
A 5 years old male child presented with polydipsia, polyuria and irritability of 10 months duration. The child had headache for 5 months, decreased appetite and decreased tolerance to cold for 3 months and diarrhoea and diffuse abdominal pain for 2 months. The child also had fever for one month and vomiting of 2 days duration. On examination, the child was conscious, well oriented, febrile and mildly dehydrated. He had right temporal hemianopia with visual acquity of 6/6 on both the sides. Fundi were normal. Systemic examination revealed no extraneural tubercular focus. There was neither any past or family history of tuberculosis nor any known contact with the disease. Skull skiagrams showed a normal sized sella turcica. CT scan (axial and coronal sections) showed a small, peripherally ring enhancing, well defined, rounded, supra-sellar mass along with evidence of thickening of the pituitary stalk [Figure - 1]. No other lesion or hydrocephalus was seen. Routine haematological and biochemical investigations were unremarkable, except for a raised erythrocyte sedimentation rate of 85 mm in the first hour. CSF examination revealed pleocytosis (200 cells/mm3 with a lymphocytic predominance (95%), reduced glucose level (18 mg/dl) and raised proteins (84 mg/dl). CSF culture was, however, negative. Reaction to intra-dermal tuberculin was significantly positive.
The endocrinological assessment revealed hypothyroidism (T3-1.5 µg/L, T4-5.0 µg/L and thyroid-stimulating hormone-0.9 µU/ml). Diabetes insipidus of central origin was confirmed by a water deprivation test. All other pituitary hormone levels were within normal limits. The patient was put on a four drug antitubercular chemotherapy regimen and thyroid hormone supplements. Polydipsia and polyuria showed marked improvement within a week of starting ATT. Irritability and fever improved within two weeks. The child had no visual field defects and headache at 8 weeks follow up. Repeat CT scan examination at 3 months interval revealed a small, dot like enhancing lesion at the same site in suprasellar region, however another tuberculoma appeared in right sylvian fissure region along with appearance of enhancing exudates in the suprasellar cistern [Figure - 2]. Subsequent follow up examination while on ATT, showed progressive clinical improvement with normal hypophysial function.
Parenchymal neuro-tuberculosis can occur with or without meningitis and usually presents as either solitary or multiple tuberculomas. Suprasellar tuberculomas are very rare. They are believed to spread via haematogenous dissemination from a source outside the CNS, such as the lung or gastro-intestinal tract or can result directly from an infection of the cranial base, though no known explanation for this atypical location of the disease exists.[4] Clinically, they can present as non-secreting masses or mimic secreting adenomas. Tuberculous meningitis has also been recognized to cause hypothalamic-pituitary dysfunction. Patients may have pituitary or
hypothalamic abnormalities such as short stature, hypogonadism and diabetes insipidus. But these may only become evident months or years after recovery, apparently due to progressive scarring of either the hypothalamus itself or the adjacent basal cisterns.[5]
Ashkan et al[6] could find only 13 patients,[2],[3],[4],[6],[7],[8],[9],[10],[11],[12] including their own, of sellar and suprasellar tuberculomas. Suprasellar extension of sellar tuberculomas was present in 86% of cases, whereas visual field defects were found in 46.7% cases. Associated pituitary dysfunction was seen in 78.6% cases. All the patients were adults. In addition to these, we could find only three cases[13],[14],[15] of suprasellar tuberculomas in children and only one of these had presented mainly with endocrinologic disturbances in the form of pituitary failure.[13]
The child, in the present case, mainly presented with diabetes insipidus and hypothyroidism. Visual field examination revealed right temporal hemianopia suggesting a sellar and suprasellar lesion with compression of optic pathways. Imaging findings of a round, suprasellar, peripherally enhancing mass with thickening of the pituitary stalk in view of abnormal CSF findings led to a diagnosis of suprasellar tuberculoma. The patient responded quickly to antituberculous chemotherapy. Pereira et al had also suggested that in the presence of thickened pituitary stalk in association with a sellar/suprasellar mass, it is wise to consider the possibility of tuberculosis, particularly in light of the increasing incidence of tuberculosis all over the world.[11] These findings can occur in other neoplastic or chronic inflammatory diseases as well such as sarcoidosis, syphilis and giant cell hypophysitis.[16] However, each one has its exclusive clinical findings. Hence it is probably not difficult to differentiate these from tuberculosis and the patient can be spared of an unnecessary surgery.
Most intracranial tuberculomas resolve completely if treated adequately with ATT. Although these can even develop or enlarge during supervised antituberculous chemotherapy. Infrequently, the appearance of a tuberculoma may be delayed as happened in the present case. Repeat CT examination after three months on ATT, revealed resolution of the suprasellar tuberculoma. Appearance of enhancing exudates in the suprasellar cistern, further strengthens the diagnosis.
Thus summarizing, though suprasellar tuberculomas presenting with pituitary-hypothalamic dysfunction in children are exceptionally rare, yet can be differentiated from other neoplastic or inflammatory conditions on the basis of serial imaging findings, clinical profile and response to ATT. Hence, unnecessary surgery can be avoided if there is no real threat to the life or vision of the patient.
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| 13. | Altunbasak S, Baytok V, Alhan E et al : Suprasellar tuberculoma causing endocrinologic disorders and imitating craniopharyngioma. Pediatr Neurosurg1995; 23 : 328-331. |
| 14. | Gucuyener K, Baykaner MK, Keskil IS et al : Tuberculoma in the suprasellar cistern : Possible CT misinterpretation as aneurysm . Pediatr Radiol 1993; 23 : 153-154. |
| 15. | Poon WS, Ahuja A, Li AKC : Optochiasmatic tuberculoma causing progressive visual failure : When was medical treatment failed. Postgrad Med J 1993; 69 : 147-149. |
| 16. | Kumar R : Atypical response of antitubercular chemotherapy in neurotuberculosis. Br J Neurosurg 1998; 12 : 344-347. |
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