Acta Medica Mediterranea, 2019, 35: 1245 ROTHIA MUCILAGINOSA MENINGITIS AFTER SINUS SURGERY: A CASE REPORT AND LITER- ATURE REVIEW BAHAR AKGÜN KARAPıNAR1, Cömert Şen2, Bora BaŞaran2, DERYA AYDıN3 1Department of Medical Microbiology, İstanbul Faculty of Medicine, İstanbul University, İstanbul, Turkey - 2Department of Otorhinolaryngology, İstanbul Faculty of Medicine, İstanbul University, İstanbul, Turkey - 3Department of Medical Microbiology, Faculty of Medicine, Biruni University, İstanbul, Turkey ABSTRACT Introduction: Rothia mucilaginosa, a member of the family Micrococcaceae, which is a normal flora member of the upper res- piratory tract. Infections are very rare, but often causes opportunistic infections in immunosuppressed patients. We report the first case of Rothia mucilaginosa meningitis due to sinus surgery in a healty adult in Turkey. A literature research was done for the meningitis caused by Rothia mucilaginosa until recently. Materials and methods: Pubmed and Google Scholar databases were investigated between January 1990 and March 2018 using the keywords Stomatococcus mucilaginosus, Rothia mucilaginosa and meningitis, respectively. Results: A total of 25 patients, including this report, were reached. The mean age was 18 years (range: 28th gestational week-46 years) and 34,7% female, 65,2% were male. The underlying diseases were acute myeloid leukemia (n:9), acute lymphoblastic leukemia (n:8), chronic myeloid leukemia (n:2), non-Hodgkin lymphoma (n:1), multiple myeloma (n:1) and neuroectodermal cancer (n:1). No neutropenia was detected in the three patients. Totally 13 patients were revovered, 11 patients died and the outcome of one patient was not reported. Conclusion: Rothia mucilaginosa infections, which are mostly caused by underlying factors, are rarely seen in the literature. This report has been presented to attract attention especially to this bacteria which can easily be overlooked in conventional identifi - cation. Keywords: Rothia mucilaginosa, Stomatococcus mucilaginosa, meningitis, sinus surgery, antibiotic susceptibility. DOI: 10.19193/0393-6384_2019_3_190 Received November 30, 2018; Accepted February 20, 2019 Introduction non-hemolytic. R.mucilaginosa is differentiated from Staphylococci and Micrococci with the presence of Rothia mucilaginosa, which is a member of the capsule and no growth in a 5% NaCl containing me- Micrococcaceae family that has been previously re- dium(4,5), and generally leads to opportunistic infec- ferred as Micrococcus mucilaginosus, Staphylococ- tions such as pneumonia in immunosuppressed pa- cus salivarius, and Stomatococcus mucilaginosus, is tients(6,7). Furthermore, it may cause infections such as a member of the normal upper respiratory system flo- bacteremia, endocarditis, meningitis, peritonitis etc. ra(1-3). It is a catalase positive (although some species although they are more frequently encountered in the are catalase negative), oxidase negative, fermentative presence of risk factors(5,8,9). This study presents a case and nonmotile microorganism in a Gram positive of meningitis in a healthy adult following endocopic coccus structure that presents in pairs or clusters (2). It sinus surgery caused by R.mucilaginosa, which has grows easily in a non-selective medium and its col- never before been reported as a cause of meningitis in onies are mucoid and sticky, white to a greyish and Turkey to the date. 1246 Bahar Akgün Karapınar, Cömert Şen et Al The study makes a literature review of cases of the results were evaluated considering the Staphylo- meningitis caused by this same bacteria. coccus breakpoints, since no standard breakpoints were defined. According to this, penicillin (10 unit), Case Presentation erythromycin (15 µg), clindamycin (2 µg), gentamicin (10 µg), tetracycline (30 µg), ciprofloxacin (5 µg), A 45-year-old male patient with no known ad- levofloxacin (5 µg), norfloxacin (10 µg), rifampicin ditional disease, and who had previously been oper - (5 µg) and linezolid (30 µg) were evaluated using the ated on for the same reason 5 and 3 years ago, was disk diffusion method, and vancomycin and ceftriax- operated for nasal polyposis in December 2017. The one minimum inhibitor concentrations was evaluated patient, who had been administered routine preopera- using a gradient test (E TEST, bioMérieux, France). tive 1 g ampicillin-sulbactam IM once for prophylaxis with minimum inhibitor concentrations. The bacteria and who had no intraoperative complications, had a was found to be sensitive to penicillin, erythromycin, clear nasal discharge on postoperative day 1 that was linezolid, levofloxacin, tetracycline, rifampicin, van- considered to be cerebrospinal fluid (CSF) rhinor - comycin (1.5 mg/L) and ceftriaxone (0.50 mg/L), and rhea, as a complication of the operation. According- was intermediate to clindamycin, while being found ly, a paranasal sinus tomography was performed and to be resistant to gentamicin and norfloxacin. The a defect on the lateral lamella of the cribriform plate ceftriaxone treatment was continued, and the blood was observed on the anterior skull base. Intravenous culture was analyzed according to the BACTEC 9120 ceftriaxone 2 g daily was administered for proph- (Becton Dickinson, USA) system. ylaxis, converted to IV cefuroxime-axetil, 750 mg, twice daily after three days. An anterior skull base CSF rhinorrhea repair was performed after CSF rhi- norrhea continued in spite of conservative therapy for one week. The patient complained of headache, nau- sea and vomiting three days after the CSF rhinorrhea repair. A cranial magnetic resonance imaging (MRI) was carried out with preliminary diagnoses of menin- gitis and intracranial hypotension. A lumbar puncture was performed and IV ceftriaxone was started at 2 g twice daily. The cranial MRI showed no pathology and the CSF cell count of the patient was 2.332 leukocytes/ mm3 and 240 lymphocytes/mm 3, which was inter - preted as meningitis. A Gram stain of the CSF sam- ple revealed polymorphonuclear leukocytes, and no microorganisms were seen. The samples were cul- tured in sheep blood agar (Becton Dickinson, USA), chocolate agar, brain heart infusion broth and tryptic Fig. 1A: Gram positive cocci seen in Gram stain (x1000); soy broth (Oxoid, United Kingdom). The agars were Fig. 1B: Mucoid, greyish colonies on blood agar of Rothia mucilaginosa, which were grown at 35 °C for 48 hours. incubated in a medium at 35°C with 5% CO 2 for 72 hours. Growth was detected at the 48th hour of the cul- An alpha hemolytic streptococcus was isolated ture, and Gram staining was performed on the grayish in the culture of two simultaneously obtained blood sticky colonies and large Gram positive cocci in pairs samples, and so IV vancomycin 1 g twice daily was or clusters were observed (Figure 1A-B). added to the treatment. A viral meningitis panel was The catalase and oxidase test negative, L-Pyrro- applied to the CSF sample using the multiplex PCR lidonyl-β-naphthylamide (PYR) test and esculin hy- method, and the result was negative. Antibiotherapy drolysis positive bacteria was identified as Rothia was stopped after 14 days of treatment in the patient, mucilaginosa using Matrix Assisted Laser Desorption who showed clinical and laboratory improvement. The Ionization Time-of-Flight (MALDI-TOF) (VITEK patient was discharged without complications. MS, France) technology. The antibiotic susceptibility Using the Medline and Google scholar data- test was performed by recommendations of the Clini- bases, a search was carried out using the key words cal and Laboratory Standards Institute (CLSI) (10), and Stomatococcus mucilaginosus, Rothia mucilagino- Rothia mucilaginosa meningitis after sinus surgery: a case report and literature review 1247 Author Gen- Underlying Results of CSF Gram CSF cell count-WBCb CSF cell count- Year Age Bacteremia Treatmentd Outcome [Reference] der diseasea stain; culture (n/mm3) RBCc (n/mm3) Gram (+) cocci in AK,CAZ,RIF, Weinblott 410 1990 Øe F AML clusters; Ø (-)f TOB,IPM,C, TCA,NAF, Ex (11,12,13) (%85 PMNL) S.mucilaginosa VA(IV) Gram stain (-); Clausen 1992 10 M ALL S.mucilaginosa 0-1 0 (-) CAZ,TCA,TOB,RIF,VA(IV) (24) Ex Gram (+) cocci in Souillet FOS,CRO,GN, 1992 11 M ALL clusters; Ø Ø S.mucilaginosa Ex (18) T,VA(IV,IT) S.mucilaginosa McWhinney Gram (+) coccus; ATM,PEN,F, 1992 29 M AML Ø Ø (-) Ex (25) S.mucilaginosa C,VA(IV) Langbaum Gram stain Ø; 360 AMP,GN, 1992 28 wg Ø No neutropenia 8.700 S.mucilaginosa Revovered (26 S.mucilaginosa (%52 PMNL) VA(IV) Gram (+) cocci in Heinwick 60 1993 17 F AML clusters; 10.000 S.mucilaginosa CRO,VA(IV) Revovered (19) (%76 neutrophil) S.mucilaginosa Chanock No cell, Gram (+) coccus; PEN,CAZ, 1993 2.5 F NHL 0 (-) (-) Ex (11,12,13) S.mucilaginosa AMF-B,VA(IV) Ben Salah Ø; 1994 2 M NEC 27 0 S.mucilaginosa CAZ,AK,IPM, VA (IV,IT) Revovered (11,12) S.mucilaginosa Al-Fiar Intra/extracellular S.mucilaginosa, 1995 (27) 14 M ALL Gram (+) coccus; Ø Ø CAZ,AK,CRO,RIF,VA(IV,IT) Ex S.mitis S.mucilaginosa Guarmazi Gram (+) coccus; 1995 14 M AML Ø Ø S.mucilaginosa VA(IV) Revovered (28) S.mucilaginosa Guarmazi Gram (+) coccus; S.mucilaginosa, 1995 46 M AML Ø Ø VA(IV) Revovered (28) S.mucilaginosa Staphylococcus Granlund Ø; 1996 31 M CML Ø Ø S.mucilaginosa CIP,VA,CTX Ex (29) S.mucilaginosa Park Gram (+) coccus; S.mucilaginosa, CRO,AMP, 1997 5 F ALL 1.130 10 Revovered (30) S.mucilaginosa S.mitis VA(IV) Gram (+) cocci in Abraham 1997 48 M MM clusters; 1.318 28 (-) SXT,OX,PEN G, C,VA(IV) Revovered (31) S.mucilaginosa Intracellular CAZ,CTX, Goldman 1998 60 F ALL Gram (+) coccus;