Sphingobacterium multivorum Meningitis in an Immunocompetent Patient with Pituitary Macroadenoma Apoplexy: A Case Report and Review of the Literature
Correspondence Address :
Dr. Mitra Kar,
Senior Resident, Department of Microbiology, 2nd Floor, C-Block, SGPGIMS, Lucknow-226014, Uttar Pradesh, India.
E-mail: mitrakar25@gmail.com
Sphingobacterium multivorum is a Gram-negative bacterium previously classified as a Flavibacterium. It produces non lactose fermenting colonies and is capable of producing oxidase and catalase enzymes. It is found ubiquitously in the environment and has been isolated from food, plants, soil, and aquatic environments, including hospital water supplies. Only a few cases of clinical infections caused by Sphingobacterium multivorum have been reported. Most cases of infection have been demonstrated in immunosuppressed patients. This case report presents the case of a 23-year-old immunocompetent woman with pituitary macroadenoma haemorrhage who developed Sphingobacterium meningitis following neurosurgery and subsequently died from cardiac arrest.
Aquatic milieu, Gram-negative bacteria, Non lactose fermenting colonies, Patients, Post neurosurgery meningitis
A 23-year-old woman presented to the emergency Outpatient Department (OPD) with the chief complaints of bilateral diminished vision for the past four years, headache for the past 2.5 years, and altered sensorium for one day. On examination, she was drowsy and unresponsive to commands. Her higher mental functions could not be assessed, and speech, memory, olfaction, and comprehension could not be evaluated. Fundus examination revealed normal findings bilaterally. No facial asymmetry was observed, and assessment of other cranial nerves was not possible due to her altered sensorium. Sensory and motor examination of all four limbs showed normal nutrition, power, and tone, assessed using various physical tests commonly used in clinical examinations. The abdominal reflex was present, and the plantar reflex was bilaterally flexor. No spinal deformity was observed, but assessment of lobar signs or cerebellar signs was not possible due to the absence of neck rigidity. Upon consultation with a neuromedicine specialist, she was advised to undergo Contrast Enhanced Magnetic Resonance Imaging (CEMRI), which revealed a large, lobulated, heterogeneously enhancing soft tissue lesion in the sellar region measuring 2.7×2.4×4.1 cm3, along with associated haemorrhage suggestive of pituitary macroadenoma with apoplexy (Table/Fig 1).
She was referred to the Department of Neurosurgery, where she was suggested to undergo endoscopic endonasal trans-sphenoidal complete excision of the tumour. In the preoperative period, the patient experienced severe headache. After a thorough work-up and consultations with the endomedicine team, the patient was scheduled for emergency surgery. However, before being shifted to the operation theatre, the patient’s condition deteriorated, and she was immediately transferred to the Intensive Care Unit (ICU). She received resuscitation measures, including intubation, ventilation, and ionotropic support, in order to stabilise her. Due to life-saving priorities, an External Ventricular Drainage (EVD) was inserted, but there was no improvement in the patient’s Glasgow Coma Scale (GCS), which remained at E1V1M1, and her pupils were non-reactive, indicating a GCS score of three. Given the critical condition, the patient was promptly taken to the operation theatre for life-saving surgery.
Postoperatively, the patient arrived in the ICU in an unreversed and intubated state, with haemodynamic instability and a GCS score of three, similar to the preoperative state. Her general condition continued to deteriorate, and inotropic support was gradually increased. Despite all efforts, her condition worsened. She developed fever with chills, and her total leukocyte count was elevated to 20,800 cells/cubic mm. Cerebrospinal Fluid (CSF) obtained from the EVD was sent for body fluid analysis, which revealed 25 pus cells/cubic mm. CSF glucose was measured at 20 mg/dL, and a routine bacterial culture was sent to the bacteriology section of the Department of Microbiology due to the deteriorating condition. The CSF sample was inoculated on MacConkey agar, blood agar, and Robertson Cooked Meat broth (RCM broth). After 48 hours of incubation, growth of non lactose fermenting colonies was observed on MacConkey agar, and a smear prepared from the culture showed Gram-negative bacilli (Table/Fig 2).
The causative organism was identified as Sphingobacterium multivorum using Matrix-Assisted Laser Desorption/Ionisation-Time Of Flight-Mass Spectrometry (MALDI-TOF-MS). Antibiotic Sensitivity Testing (AST) was performed using the Kirby-Bauer Disc diffusion method on cation-adjusted Muller Hinton Agar (MHA), and the isolate was found susceptible to ceftazidime, ceftriaxone, levofloxacin, and Trimethoprim-Sulfamethoxazole (TMP/SMX), but resistant to aminoglycosides. Another CSF sample was sent after 72 hours of the first sample, which showed growth of the same microorganism. The patient was empirically started on intravenous ceftriaxone 4 g every 24 hours. Unfortunately, as haemorrhage from the pituitary gland was uncontrollable, the patient experienced sudden cardiopulmonary arrest and could not be revived despite multiple cycles of Cardiopulmonary Resuscitation (CPR). She was declared deceased.
Sphingobacterium multivorum is a Gram-negative bacterium previously classified as a Flavibacterium. It produces non lactose fermenting colonies and is capable of producing oxidase and catalase enzymes. The high concentration of sphingolipids in its cell wall is mainly responsible for its name. Common species within the Sphingobacterium genus include S. thalophilum, S. multivorum, S. mizutae, and S. spiritorum (1). Initially, Sphingobacterium multivorum was known as CDC IIK biotype-2 strains and was found ubiquitously in the environment, isolated from food, plants, soil, and aquatic environments, including hospital water supplies (2). Only a few cases of clinical infections caused by Sphingobacterium multivorum have been reported. Recent reports have observed infections in immunocompromised patients with cancer undergoing chemotherapy (3), patients with End-Stage Renal Disease (ESRD) undergoing haemodialysis, patients with Human Immunodeficiency Virus (HIV) and cystic fibrosis (4),(5), and patients with diabetes mellitus (6). The most common infections caused by this ubiquitous microorganism include bactaeremia, meningitis, spontaneous bacterial peritonitis, and lung infections.
This case involves an immunocompetent patient who presented with progressive bilateral vision loss, along with a headache and altered sensorium. She was diagnosed with pituitary macroadenoma with haemorrhage on CEMRI. She underwent endoscopic endonasal trans-sphenoidal complete excision of the tumour, after which she developed a headache. Due to a lowering GCS, an EVD was inserted, but there was no improvement. The patient experienced fever with chills, and her CSF sample from the EVD showed growth of a rare organism, Sphingobacterium multivorum, which was susceptible to ceftazidime, ceftriaxone, levofloxacin, and TMP/SMX. Unfortunately, she succumbed to pituitary haemorrhage and fever, and despite four cycles of CPR, she could not be revived.
To date, most isolates of Sphingobacterium multivorum have been identified in immunosuppressed individuals, including those with type 2 diabetes mellitus, chronic lung diseases, and underlying co-morbidities such as cystic fibrosis and chronic kidney disease. Infections caused by this bacterium can be deadly if not recognised early (7). A case report by Abro AH et al., showed confirmed infections in immunocompetent individuals, such as the young patient in this case report with a diagnosis of pituitary macroadenoma (8).
The antibiotic susceptibility of Sphingobacterium multivorum consistently shows resistance to aminoglycosides, as evident in this case report and previous studies by Lambiase A et al., Abro AH et al., and Tronel H et al., (4),(8),(9). Sphingobacterium multivorum is known to exhibit properties of Extended-Spectrum Beta-Lactamase (ESBL) resistance, Metallo-Beta-Lactamase (MBL) resistance, and resistance to third-generation cephalosporins (9). However, the isolate identified in this case was found to be sensitive to ceftazidime, ceftriaxone, levofloxacin, and TMP/SMX, which are extensively used at this centre for suspected opportunistic infections among immunosuppressed individuals. Previous studies have also shown that most isolates of Sphingobacterium multivorum are susceptible to TMP/SMX (4),(6),(10),(11),(12),(13), while a study by Verma RK et al., suggested sensitivity to only gatifloxacin (14). Gatifloxacin was not tested for the isolate in this study. Other cases reported in the literature have involved immunocompromised individuals, including patients with cystic fibrosis (13), patients with End-Stage Renal Disease (ESRD) with benign prostatic hyperplasia (14), patients with multiorgan dysfunction (15), and patients with multiple myeloma (16). This study presents a case of Sphingobacterium multivorum meningitis in an immunocompetent patient, which is rare. (Table/Fig 3) represents a review of infections caused by Sphingobacterium multivorum in recent years (3),(4),(6),(7),(8),(10),(11),(12),(14),(15),(16),(17),(18).
To the best of our knowledge, this is one of the rare cases of Sphingobacterium multivorum reported as a causative pathogen among immunocompetent individuals. It is the fourth case of Sphingobacterium multivorum meningitis reported worldwide and the second case reported from India. The exact source of infection could not be determined in this study, but the repeated isolation of the microorganism suggests it as a probable cause of meningitis. The suggested source of infection could be nosocomial, as the patient developed fever after surgery and placement of the External Ventricular Drainage (EVD). Improved implementation of infection control practices may be necessary to prevent such cases in the future.
This study provides an insight into the isolation of meningitis caused by Sphingobacterium multivorum, along with its specific antibiotic sensitivity pattern. This information can help clinicians in selecting appropriate antibiotic therapy. Additionally, it emphasises the importance of implementing strict infection control practices to prevent the nosocomial spread of infections caused by rare pathogens, which have the potential to acquire drug resistance from multidrug-resistant pathogens present in the hospital environment.
DOI: 10.7860/JCDR/2023/64118.18584
Date of Submission: Mar 17, 2023
Date of Peer Review: Jun 30, 2023
Date of Acceptance: Jul 26, 2023
Date of Publishing: Oct 01, 2023
AUTHOR DECLARATION:
• Financial or Other Competing Interests: None
• Was informed consent obtained from the subjects involved in the study? Yes
• For any images presented appropriate consent has been obtained from the subjects. No
PLAGIARISM CHECKING METHODS:
• Plagiarism X-checker: Mar 25, 2023
• Manual Googling: Jul 06, 2023
• iThenticate Software: Oct 18, 2023 (9%)
ETYMOLOGY: Author Origin
EMENDATIONS: 5
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