Neurol Sci (2015) 36:167–169 DOI 10.1007/s10072-014-1861-y
LETTER TO THE EDITOR
Tuberculous ventriculitis as a relapse following central nervous system tuberculoma Selda Keskin Guler • Nalan Gu¨nes¸ • Burcu Gokce Cokal • Tahir Yoldas
Received: 2 April 2014 / Accepted: 18 June 2014 / Published online: 2 July 2014 Ó Springer-Verlag Italia 2014
A 74-year-old female patient was admitted with complaints of memory loss, nausea, loss of balance, and urinary incontinence, all of which started approximately 2 months earlier and increased gradually. These symptoms were so severe that she could no longer take care of herself, and her children had begun to assist her. Her medical history included a diagnosis of central nervous system (CNS) tuberculoma and tuberculous lymphadenitis 3 years prior, which were diagnosed upon complaints of severe headache and treated with isoniazid (300 mg/day), rifampicin (600 mg/day), ethambutol (1,500 mg/day) and pyrazinamide (2,000 mg/day) for 1 year. Cranial MR imaging (MRI) at diagnosis of CNS tuberculoma revealed an intense gadolinium-enhanced mass lesion with lobulated contour in the left basal ganglia (Fig 1), and thoracic computed tomography (TCT) identified paratracheal and subcarinal lymphadenopathies. The diagnosis of tuberculosis was confirmed by biopsy of the mediastinum. Following anti-tuberculosis treatment applied during that period, the patient’s ability to walk and her cognitive capacities recovered, and MRI revealed complete elimination of the tuberculoma. Due to the complaints mentioned above, the patient was hospitalized
S. K. Guler (&) N. Gu¨nes¸ B. G. Cokal T. Yoldas Department of Neurology, Ankara Training and Research Hospital, S¸ u¨kriye district, Ulucanlar Avenue, No: 89, 06340 Ankara, Altındag˘, Turkey e-mail: [email protected] N. Gu¨nes¸ e-mail: [email protected] B. G. Cokal e-mail: [email protected] T. Yoldas e-mail: [email protected]
in the neurology service, where her vital signs and systemic physical examination remained normal. Neurological examination revealed an increased tendency to sleep and limited cooperation, even though she could comply with verbal directions, and her visual orientation was intact. No signs of motor deficit, cranial nerve palsy, or meningeal irritation were observed. DTRs and fundus examination were normal. Laboratory analysis indicated normal complete blood counts, biochemical parameters, thyroid function, and vitamin B12 levels, and an erythrocyte sedimentation rate of 25 mm/h. MRI identified a large ependymal contrast enhancement on the ventricular walls of the third, fourth, and lateral ventricles (Fig 2a–c). Lumbar puncture led to the following findings: protein 2,165 mg/L (150–450), glucose 43 mg/dL (simultaneous blood glucose level was 110 mg/dL), cell count 70 leukocyte/mm3 (all lymphocytes) and a positive Pandy’s test. A cerebrospinal fluid (CSF) smear showed large numbers of lymphocytes, few plasmocytes, degenerated cells and giant cells similar to Langhans type 1–2, and areas resembling eosinophilic small necroses, which led to a diagnosis of tuberculous meningitis (TBM). The CSF culture was negative. We evaluated albumin and IgG in serum and CSF, and found the patient’s IgG index to be 0.70, indicative of intrathecal IgG synthesis. TCT showed no evidence of tuberculous infection. After initiation of fourdrug antituberculosis treatment (isoniazid 300 mg/day, rifampicin 600 mg/day, ethambutol 1,500 mg/day, and pyrazinamide 2,000 mg/day and dexamethasone 2 mg/ day), she was referred to the Infectious Diseases Clinic where she was treated for 2 months. Upon discharge, the patient had full cognitive function; she could easily follow directions and was well-oriented to her surroundings. Control cranial MRI revealed a decrease in ependymal contrast uptake (Fig 2d–f).
123
168
Neurol Sci (2015) 36:167–169
Fig. 1 Granuloma image having an intense contrast uptake in the left basal ganglia level on contrast-enhanced cranial MR imaging
Fig. 2 a–c Large linear contrast uptake in coronal, axial and sagittal sections on ventricular wall on contrast-enhanced cranial MR imaging. d–f Lessening of contrast uptake on ventricular wall is observed in coronal, axial and sagittal sections on contrast-enhanced cranial MR imaging
123
Neurol Sci (2015) 36:167–169
Tuberculosis is a major health problem worldwide, and tuberculosis of the CNS is relatively common in developing countries. However, tuberculous ventriculitis (TBV) is a rare complication of TBM [1]. Ventriculitis has been reported to develop in 19–40 % of TBM cases based on autopsies; however, reports on the frequency of isolated ventriculitis have been limited to case reports [1, 2]. Our patient represents the fourth isolated TBV case we have found reported in the literature [2, 3], her survival being the most significant distinguishing feature. TBM generally results from the spread of systemic tuberculosis to the CNS and nearly always affects the lung or other organs [4]. In the case reported here, none of the clinical or radiological findings indicated involvement of organs or systems other than the CNS at the time of diagnosis. Together with the patient’s previous case of tuberculous disease, these results suggest that TBM developed from reactivation of latent infection. Intraventricular lesions represent either tumors or evidence of infection. Very few reports of inflammatory lesions of the ependymal or even choroid plexus exist [5]. Ventriculitis in TBV may result from ependymal tuberculous granuloma, rupture of a granuloma in another site, or enlargement of inflammation that leads to ependymitis [2]. In our case, linear contrast in the imaging results likely indicated the spread of inflammation leading to ependymitis.
169
No clear guidelines have been established for treatment of TBV [2]. Apart from oral anti-tuberculosis drugs, hydrocephalus is treated by ventriculoperitoneal shunt or external ventricular drainage [2, 4]. Our patient’s clinical and radiological indications improved upon oral treatment without the need for surgical intervention. TBV should be considered in the differential diagnosis of patients with both altered mental status and intraventricular lesions. Considering that tuberculosis is still a significant public health problem in developing countries, the possibility of TBV should not be overlooked. Conflict of interest
None.
References 1. Lee LV (2000) Neurotuberculosis among Filipino children: an 11 years experience at the Philippine Children’s Medical Center. Brain Dev 22:469–474 2. Kumar S, Kumar R, Radotra BD et al (2013) Tubercular ventriculitis: an uncommon entity. Indian J Pediatr ¨ zaslan A (2004) Hydrocephalus developing related 3. Pakis¸ I, O adhesive aracnoidit: a case report. Turkiye Klinikleri J Foren Med 1(1):39–42 4. Ramachandran TS et al (2013) Tuberculous meningitis. http:// emedicine.medscape.com/article/1166190-overview. Accessed 12 Nov 2013 5. Shah GV (2000) Central nervous system tuberculosis: imaging manifestations. Neuroimaging Clin N Am 10:355–374
123
LETTER TO THE EDITOR
Tuberculous ventriculitis as a relapse following central nervous system tuberculoma Selda Keskin Guler • Nalan Gu¨nes¸ • Burcu Gokce Cokal • Tahir Yoldas
Received: 2 April 2014 / Accepted: 18 June 2014 / Published online: 2 July 2014 Ó Springer-Verlag Italia 2014
A 74-year-old female patient was admitted with complaints of memory loss, nausea, loss of balance, and urinary incontinence, all of which started approximately 2 months earlier and increased gradually. These symptoms were so severe that she could no longer take care of herself, and her children had begun to assist her. Her medical history included a diagnosis of central nervous system (CNS) tuberculoma and tuberculous lymphadenitis 3 years prior, which were diagnosed upon complaints of severe headache and treated with isoniazid (300 mg/day), rifampicin (600 mg/day), ethambutol (1,500 mg/day) and pyrazinamide (2,000 mg/day) for 1 year. Cranial MR imaging (MRI) at diagnosis of CNS tuberculoma revealed an intense gadolinium-enhanced mass lesion with lobulated contour in the left basal ganglia (Fig 1), and thoracic computed tomography (TCT) identified paratracheal and subcarinal lymphadenopathies. The diagnosis of tuberculosis was confirmed by biopsy of the mediastinum. Following anti-tuberculosis treatment applied during that period, the patient’s ability to walk and her cognitive capacities recovered, and MRI revealed complete elimination of the tuberculoma. Due to the complaints mentioned above, the patient was hospitalized
S. K. Guler (&) N. Gu¨nes¸ B. G. Cokal T. Yoldas Department of Neurology, Ankara Training and Research Hospital, S¸ u¨kriye district, Ulucanlar Avenue, No: 89, 06340 Ankara, Altındag˘, Turkey e-mail: [email protected] N. Gu¨nes¸ e-mail: [email protected] B. G. Cokal e-mail: [email protected] T. Yoldas e-mail: [email protected]
in the neurology service, where her vital signs and systemic physical examination remained normal. Neurological examination revealed an increased tendency to sleep and limited cooperation, even though she could comply with verbal directions, and her visual orientation was intact. No signs of motor deficit, cranial nerve palsy, or meningeal irritation were observed. DTRs and fundus examination were normal. Laboratory analysis indicated normal complete blood counts, biochemical parameters, thyroid function, and vitamin B12 levels, and an erythrocyte sedimentation rate of 25 mm/h. MRI identified a large ependymal contrast enhancement on the ventricular walls of the third, fourth, and lateral ventricles (Fig 2a–c). Lumbar puncture led to the following findings: protein 2,165 mg/L (150–450), glucose 43 mg/dL (simultaneous blood glucose level was 110 mg/dL), cell count 70 leukocyte/mm3 (all lymphocytes) and a positive Pandy’s test. A cerebrospinal fluid (CSF) smear showed large numbers of lymphocytes, few plasmocytes, degenerated cells and giant cells similar to Langhans type 1–2, and areas resembling eosinophilic small necroses, which led to a diagnosis of tuberculous meningitis (TBM). The CSF culture was negative. We evaluated albumin and IgG in serum and CSF, and found the patient’s IgG index to be 0.70, indicative of intrathecal IgG synthesis. TCT showed no evidence of tuberculous infection. After initiation of fourdrug antituberculosis treatment (isoniazid 300 mg/day, rifampicin 600 mg/day, ethambutol 1,500 mg/day, and pyrazinamide 2,000 mg/day and dexamethasone 2 mg/ day), she was referred to the Infectious Diseases Clinic where she was treated for 2 months. Upon discharge, the patient had full cognitive function; she could easily follow directions and was well-oriented to her surroundings. Control cranial MRI revealed a decrease in ependymal contrast uptake (Fig 2d–f).
123
168
Neurol Sci (2015) 36:167–169
Fig. 1 Granuloma image having an intense contrast uptake in the left basal ganglia level on contrast-enhanced cranial MR imaging
Fig. 2 a–c Large linear contrast uptake in coronal, axial and sagittal sections on ventricular wall on contrast-enhanced cranial MR imaging. d–f Lessening of contrast uptake on ventricular wall is observed in coronal, axial and sagittal sections on contrast-enhanced cranial MR imaging
123
Neurol Sci (2015) 36:167–169
Tuberculosis is a major health problem worldwide, and tuberculosis of the CNS is relatively common in developing countries. However, tuberculous ventriculitis (TBV) is a rare complication of TBM [1]. Ventriculitis has been reported to develop in 19–40 % of TBM cases based on autopsies; however, reports on the frequency of isolated ventriculitis have been limited to case reports [1, 2]. Our patient represents the fourth isolated TBV case we have found reported in the literature [2, 3], her survival being the most significant distinguishing feature. TBM generally results from the spread of systemic tuberculosis to the CNS and nearly always affects the lung or other organs [4]. In the case reported here, none of the clinical or radiological findings indicated involvement of organs or systems other than the CNS at the time of diagnosis. Together with the patient’s previous case of tuberculous disease, these results suggest that TBM developed from reactivation of latent infection. Intraventricular lesions represent either tumors or evidence of infection. Very few reports of inflammatory lesions of the ependymal or even choroid plexus exist [5]. Ventriculitis in TBV may result from ependymal tuberculous granuloma, rupture of a granuloma in another site, or enlargement of inflammation that leads to ependymitis [2]. In our case, linear contrast in the imaging results likely indicated the spread of inflammation leading to ependymitis.
169
No clear guidelines have been established for treatment of TBV [2]. Apart from oral anti-tuberculosis drugs, hydrocephalus is treated by ventriculoperitoneal shunt or external ventricular drainage [2, 4]. Our patient’s clinical and radiological indications improved upon oral treatment without the need for surgical intervention. TBV should be considered in the differential diagnosis of patients with both altered mental status and intraventricular lesions. Considering that tuberculosis is still a significant public health problem in developing countries, the possibility of TBV should not be overlooked. Conflict of interest
None.
References 1. Lee LV (2000) Neurotuberculosis among Filipino children: an 11 years experience at the Philippine Children’s Medical Center. Brain Dev 22:469–474 2. Kumar S, Kumar R, Radotra BD et al (2013) Tubercular ventriculitis: an uncommon entity. Indian J Pediatr ¨ zaslan A (2004) Hydrocephalus developing related 3. Pakis¸ I, O adhesive aracnoidit: a case report. Turkiye Klinikleri J Foren Med 1(1):39–42 4. Ramachandran TS et al (2013) Tuberculous meningitis. http:// emedicine.medscape.com/article/1166190-overview. Accessed 12 Nov 2013 5. Shah GV (2000) Central nervous system tuberculosis: imaging manifestations. Neuroimaging Clin N Am 10:355–374
123
