Abdominal Tuberculosis in a Child With Neuro Tuberculosis: Is it Due to Ventriculoperitoneal Shunt?

To the Editors: The management of hydrocephalus can include medical therapy with dehydrating agents and steroids for patients with good grades of the Vellore grading system and those with communicating hydrocephalus.1 Surgery in the form of Ventriculoperitoneal (VP) shunt or endoscopic third ventriculostomy is required for those with poor Vellore grade or obstructive hydrocephalus.1 We present a patient of multidrug-resistant tuberculosis (TB) peritonitis due to seeding of tuberculous bacilli through VP shunt from multidrug-resistant tuberculous meningitis. A 4.5-year-old male presented in December 2018 with fever and vomiting since 1 week. On examination, weight was 9.8 kg (<3rd SD as per World Health Organization growth charts), height of 103 cm (Median to 1 SD as per World Health Organization). He was vitally stable, with meningeal signs present on examination. Central nervous system (CNS) examination showed brisk lower limb reflexes with plantar’s sign positive. Hemogram showed hemoglobin 9.2 mg/dL, total leucocyte count of 9900 cells/cumm (neutrophils 52%, lymphocytes 33%), platelet count of 3.44 lacs/cumm. Erythrocyte sedimentation rate was 5 mm at the end of the first hour. Chest radiograph and ultrasound (USG) abdomen were normal. There was no history of TB contact. Xpert MTB/Rif assay on gastric aspirate was negative. Magnetic resonance imaging (MRI) brain showed tuberculoma (2.6 × 1.7 cm) in left occipital region and right frontal region with meningeal enhancement along falx cerebri. Cerebrospinal fluid (CSF) analysis showed proteins 219 mg/dL, glucose 13 mg%, leucocytes 160 cells/cumm (99% lymphocytes). CSF Xpert MTB/Rif assay revealed very low Mycobacterium tuberculosis (MTB) with rifampicin resistance. CSF culture after 6 weeks grew MTB with phenotypic drug susceptibility testing showing sensitivity to linezolid (Lzd), sodium aminosalicylate (PAS), amikacin, moxifloxacin high dose, clofazimine (Cfz), kanamycin and resistance to pyrazinamide, ethambutol, ethionamide, moxifloxacin low dose, isoniazid, rifampicin, ofloxacin suggestive of pre-extremely drug resistant pattern. Patient was started on anti-TB therapy (ATT) consisting of kanamycin, moxifloxacin high dose, Lzd, Cfz and cycloserine along with oral prednisolone (2 mg/kg/d). A month later, the child developed fever and difficulty in walking. CT brain showed moderate hydrocephalus noted with periventricular ooze (which was not seen in previous scan), enhancement noted along the interpeduncular cistern and cerebropontine cistern which was suggestive of meningitis with 2 small ring-enhancing lesions noted in the right frontal region. An external ventricular drain was placed followed by conversion into a VP shunt. At 18 months, MRI brain and spine was suggestive of multiple enhancing granulomas (maximum size 4 mm), with resolution of right thalamic infarct with diffuse thick spinal meningeal enhancement. At 20 months, MRI brain showed enhancing conglomerate granuloma in the left crus of the left middle cerebral peduncle with perilesional edema extension into the interpeduncular cistern with diffuse enhancement along basal cisterns involving prepontine, perimesencephalic, bilateral sylvian cisterns with meningeal enhancement along bilateral frontal-parietal region suggestive of meningitis, multiple enhancing granuloma in calcarine fissure, parietal lobes and right middle frontal gyrus. Child received multiple courses of oral prednisolone during these 36 months in view of the paradoxical reaction. CSF analysis at 36 months of ATT revealed proteins of 275 mg/dL, glucose 40 mg/dL, leucocyte counts 3 cells/cumm (100 % lymphocytes) with TB MGIT culture negative after incubation for 6 weeks. MRI brain at 24 months showed a mild decrease in meningeal enhancement with decrease in size of tuberculomas in the right crus, right foramen of Luschka (left crus 11 × 7 mm, right foramen of Luschka 8 × 7 mm). Hence, in view of clinical and laboratory improvement, ATT was stopped after 36 months in December 2021. Three months after stopping ATT, the child presented with altered sensorium. MRI brain showed increase in size of tuberculomas with leptomeningeal enhancement and perilesional edema. CSF analysis was done as show in investigation Table 1. He was suspected to have treatment failure and was started on ATT containing bedaquiline, delamanid, Cfz, cycloserine, Lzd and PAS. TABLE 1. - Serial Cerebrospinal Fluid Analysis of the Patient December 2018 December 2021 March 2022 Cells (/cumm) 160 3 0 Polymorphs (%) 01 0 N/A Lymphocytes (%) 99 100 N/A Proteins (gm/dL) 219 275 290 Sugar (mg%) 13 40 38 Xpert Mtb/Rif Positive Negative Negative TB MGIT Positive Negative Negative Six months later, the child developed ascites. USG abdomen showed mild peritoneal and minimal serosal thickening with peritoneal thickness 4.5 mm with moderate free fluid in the abdomen with few internal thick septations. Ascitic fluid tap showed protein 1.1 gm%, total leucocyte counts 1000 cells/cumm (99% lymphocytes). Ascitic fluid Xpert MTB/Rif assay was negative while TB MGIT culture grew MTB with the same sensitivity pattern of pre-extremely drug resistant. CSF is depicted in Table 1. VP shunt was removed. Subsequently a ventriculo-atrial shunt was inserted in September 2022. A VP shunt, utilized for hydrocephalus, comprises a ventricular catheter, valve and distal catheter. Complications include infection, misplacement, overdrainage, nephritis, disconnection, obstruction, abdominal fluid collections and shunt breakage.2 Our patient had several unusual features in form of recurrence of TB despite 36 months of ATT and there was presence of extensive CNS and spinal disease, but no extra CNS site was involved which signifies VP shunt as the only link for transmission to abdomen. We believe that the VP shunt likely provided a conduit for the elusive mycobacteria to spread down to the tip of the catheter in the abdomen as there was no evidence of extra CNS disease elsewhere. Abdominal complications following VP shunt have been reported in the literature with a variable incidence of 5%–47%.3 Ascites and abdominal pseudocysts are 2 complications that can occur following placement of a VP shunt.4 In our patient ascitic fluid TB MGIT culture grew MTB. Tuberculous meningitis itself can theoretically be considered the source of infection spread and shunt complications in the peritoneal cavity including pseudocyst formation.5 But the literature surrounding this is controversial, with no definite reported case of meningitis extension following VP shunt. Our child required almost 36 months of ATT. At the time of stopping ATT, tuberculomas had remained stable and CSF TB MGIT culture was negative. However, within 3 months of stopping ATT, there was worsening of tuberculomas which represent treatment failure. Inspite of starting the ATT, there was a possible transmission of MTB to the abdomen from the VP shunt in September 2022 highlighting that in patients with tuberculous meningitis with treatment failures and VP shunt, there may be a risk of transmission of MTB to other parts of the body through the shunt.