Cystic Mesenteric Inflammatory Pseudotumor Associated to Multidrug-Resistant Meningeal Tuberculosis and a Cerebrospinal Fluid Shunt: An Unusual Case

Abstract

Abdominal masses in childhood have a very large spectrum of benign and malignant causes. Inflammatory pseudotumor (IP) arising from intra-abdominal sites has only rarely been designated previously in children. We report the case of a 21-year-old HIV-negative man, in whom a cystic mesenteric IP appeared after 46 months of diagnosis of tuberculous meningitis (TBM) complicated with brain infarcts, tuberculoma, hydrocephalus and multiple neurosurgical procedures including ventriculoperitoneal (VP) cerebrospinal fluid (CSF) shunt placement, shunt exchanges and endoscopic ventricular septostomies. At diagnosis, primary antituberculous drugs were administered and mycobacterial CSF cultures were persistently negative; nevertheless, follow-up elevated adenosine deaminase levels in CSF suggested multidrug-resistant (MDR) tuberculosis and moxifloxacin and aminoglycoside were added with improvement. Repeated formation of abdominal cystic masses with shunt obstructions, abdominal distention and pain lead us to conclude that the peritoneum was no longer useful for CSF derivation and a ventriculo-atrial shunt was placed. After laparoscopic excision of an abdominal mass, microscopic examination showed a cystic tumor formed by proliferation of fibroblasts, myofibroblasts and inflammatory cells. Focally a granuloma with multinucleated giant cells (Langerhans cells) was observed and Ziehl-Neelsen staining showed acid-fast bacilli. Diagnosis of IP associated to MDR tuberculosis was made. This complication is probably under-recognized since histopathological study of abdominal masses related with CSF VP shunts is not performed systematically. Our case illustrates the importance of microscopic tissue examination to detect MDR and extensively drug-resistant (XDR) tuberculosis and the difficult treatment of these cases.