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Malaysian J Pathol 2012; 34(2) : 153 – 156

Klebsiella ozaenae sepsis in a young healthy male

Yared Wondmikun ENDAILALU MD, PhD, Peter SEALY MD, Miriam MICHAEL MD, Kauter AL KHALLOUFI MD and Hasan NABHANI MD*

Departments of Internal Medicine and *Radiology, Howard University Hospital, Washington, DC, USA

Abstract

Introduction: K. ozaenae is a weak pathogenic organism known to cause primary atrophic rhinitis or ozena. There are few reports that the bacteria could cause serious invasive infection in debilitated patients. This is fi rst report of K. ozaenae in a young previously healthy adult. Case presentation: A 34-year-old Filipino male with no signifi cant previous medical history presented with severe frontal headache of two days duration with fever and chills. Blood and serum work-up showed leukocytosis, mild thrombocytopenia, hypomagnesemia, hypokalemia, and hypophopatemia. Liver function test indicated elevated bilirubin and transaminases. CT of the head indicated sinus disease including mucoperiosteal-like thickening of the right maxillary sinus, left mastoid hypoaeration and sclerosis characteristics of chronic infl ammation. Blood culture grew gram negative rods identifi ed as Klebsiella ozaenae species. Conclusion: Klebisella ozaenae sepsis is rarely reported in medical literature. There are about 12 case reports all of which identifi ed with one or more chronic conditions causing decline in patient immunity resulting in invasive infection by the weak pathogen. Our patient is a young physically active adult male with no identifi able risk factors except chronic ozena-like infection that might serve as a source for haematologic seeding.

Keywords: Klebsiella ozaenae, sepsis, ozena

Address for correspondence and reprint requests: Yared W. Endailalu, Howard University Hospital, Department of Internal Medicine, 2041 Georgia Avenue NW, Washington, DC 20060, USA. Tel: 001 202 865 1920; Fax: 001 202 865 7199. Email: yared.endailalu@howard.edu

CASE REPORT

INTRODUCTION

Klebsiella ozaenae is a gram negative aerobic bacillus, a member of a family Enterobacteriaceae. Members of Klebsiella genus typically express lipopolysaccaride (O) and capsular polysaccharide (K) antigens which are the base of their serotype variability and virulence. K. ozaenae is a weakly pathogenic organism known to be a causative agent of the rare disease primary atrophic rhinitis or ozena.1 Patients with ozena have progressive chronic infl ammatory disease of the upper respiratory tract with a distinctive feature of fetid endonasal crusting and discharge with mucosal atrophy and bone resorption.2 Patients may present with chronic nasal discharge and malodor, anosmia, epistaxis, nasal obstruction or headache. Mucoperiosteal wall thickening of the maxillary and ethmoid sinus is a characteristic radiological fi nding in chronic infection with Klebsiella ozaenae.3

There are only few literature reports that this bacterium is associated with serious invasive infection such as meningitis,4 sepsis5 and

intracranial abscesses.6 Here we report a case of K. ozaenae sepsis in a patient with asymptomatic sinusoidal and mucosal changes.

CASE REPORT

A 34-year-old Filipino male who is working in a Sushi Bar with no signifi cant previous medical history presented with severe frontal headache of two days duration with fever, chills, loss of appetite, nausea and one episode of vomiting. The patient reports no abdominal pain, diarrhoea, cough, skin rash, recent throat infection, neck stiffness, blurring of vision, nasal discharge, tinnitus or urinary symptoms. The patient denied contact with similarly ill patients, recent travel or head trauma. He stated that he drinks once to twice weekly about 4-6 beers in a session. At admission his axillary temperature was 101.50F, blood pressure 110/60 mmHg, respiratory rate 18/min, pulse 108 beats/min and SaO

2 99% in

ambient air. He is a well-built athletic young male with slightly dry buccal and glossal surfaces. The remaining physical examination

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of all body systems was none rewarding except mild tenderness to pressure over maxillary sinus and epigastric tenderness without guarding or rigidity. Initial blood and serum work up showed leukocytosis [15,300cells per mm3 with neurophils of 95% and bands 21%], mild thrombocytopenia (111,000 cells per mm3), hypomagnesemia (1.4 mg/dl), hypokalemia (3.4 meq/l), and hypophopatemia (1 mg/dl). His random blood glucose level was 180mg/dl.

Liver function test indicated elevated bilirubin (3.1mg/dl) and transaminases (AST 64 mu/ml, ALT 79 mu/ml). Chest x-ray showed clear lungs. Urinalysis, renal functional state tests and coagulation profi le were normal. CT of the head indicated sinus disease including mucoperiosteal-like thickening and possible air-fl uid level in the right maxillary sinus, mild soft tissue density of the left maxillary and ethmoid sinus and left mastoid hypoaeration and sclerosis (Fig.1 and Fig. 2).

FIG. 1: CT scan of the head showing maxillary sinuses mucoperiosteal-like thickening and a possible air-fl uid level in the right maxillary sinus.

FIG. 2: CT scan of the head showing pneumatization of left maxillary sinus with sclerosis consistent with chronic infectious process.

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KLEBSIELLA OZAENAE SEPSIS

Patient was primarily managed with a working diagnosis of Systemic Infl ammatory Response Syndrome to rule out meningitis. Patient did not consent for LP. He was started empirically with vancomycin and ceftriaxone. In the mean time blood culture grew gram negative rods identifi ed as Klebsiella ozaenae species. The working primary diagnosis was Klebsiella ozaenae sepsis. The bacterium was sensitive to ampicillin/sulbactam, ciprofl oxacin, cefazolin, piperacillin, imipenem and timentin. The antibiotic treatment was changed to piperacillin. The ENT team evaluated the patient and took scrapings from the nasal mucosa and turbinates. No bacteria grew from these specimens. Imaging studies of the abdomen and pelvis structures were carried out with sonography. It revealed a 4.6 cm mixed ecogenic mass in the medial aspect of the right lobe of the liver without fl ow on color Doppler imaging. Further characterization of the mass using IV contrast CT has indicated that the mass is an incidental benign hemangioma. A stool culture grew normal fl ora. No ova, parasites or WBC were found in the stool. Hepatitis B panel showed no vaccination or previous encounter with the virus. Patient was negative for hepatitis A IgM. With the continuation of piperacillin therapy, patient’s headache and fever disappeared. Two sets of blood cultures showed no new growth. Patient’s low platelet count and abnormal liver function started trending to normality. Patient became apyrexial for more than 48 hours and is discharged home with oral moxyfl oxacin after 7 days of hospital stay. Subsequent follow-ups at 3 weeks after discharge have revealed no change in the size and architecture of the hepatic mass and sinus fi ndings. He remained symptom free and was well.

DISCUSSION

K. ozaenae is weakly pathogenic and is part of the normal fl ora of the upper respiratory passageways. The bacterium is the putative cause of a rare ENT disease; chronic atrophic rhinitis. However, there are few literature reports that it could also cause life threatening serious infections. Tang and Chen4 reported two cases of K. ozaenae meningitis and reviewed other two similar cases of meningitis previously reported in literature. Multiorgan infection and formation of abscesses in brain, lung, prostate and around the kidney was reported by Ng et al. from Malaysia.7 Case reports of abscess formation at various anatomical sites including

brain, lungs, eye, pituitary gland, liver and spleen are also reported sporadically.6,8,9 Most of the unusual invasive presentation of klebsiella infection has been reported from South East Asia.4,7,8,10 Ko et al.10 described, of his Taiwan patients who had klebsiella bacteremia, 88% were with liver abscess. In contrast, in the same study, only 12% of cases of K. bacteremia are associated with liver abscess in non-South-East Asian patients. The reason for the geographic preponderance of these severe manifestations is not known, likewise the prevalence of ozena in the same subcontinent. In our Filipino patient, abdominal, pelvic, and head CT scan has been done to rule out both primary abscesses as a source of bacteremia and metastatic abscesses as secondary seeding complications. Septicemia with K. ozaenae is similarly rare and highly fatal. Among the seven patients of K. ozaenae septicemia reported in the literature, fi ve died.5 It is reported that all affected patients have one or more underlying diseases and conditions known to compromise body immunity such as alcoholism, diabetes, malignancy, lepromatous leprosy, leucopenia, immune suppressive treatments or advanced age. In a similar study, Ko et al.10 has also shown that 78% of their patients with Klebsiella bacteremia have underlying predisposing diseases, the most common being diabetes mellitus (40%), liver disease (34%), malignancy (16%) and alcoholism (12%). However, unlike previous reports, the case reported here is a young athletic man in whom no immune compromising chronic diseases could be identifi ed. To our knowledge this is the fi rst report of Klebsiella ozaenae in a previously healthy adult with no apparent chronic illness known to debilitate the patient’s immunity. The purpose of this report is to sensitize practitioners that this unusual organism can cause sepsis in immune competent patients. It is important because it is invariably fatal if treatment is delayed. The source of septicemia could not be defi nitively established in this case. It is not possible, yet, always to identify the primary site of blood invasion. In a large worldwide collaborative study involving 455 episodes of bacteremia caused by Klebsiella pneumoniae, up to 12 % of the studied bacteremia cases have no primary site evident as a source for blood invasion.10 Nonetheless, the CT fi ndings in our patient are very characteristic for chronic atrophic rhinitis and sinusitis of K. ozaenae.3 Clinically, the patient is asymptomatic for rhinitis or sinusitis and unfortunately the organism could

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not be isolated from the nasal mucosal specimen. Those asymptomatic radiological changes of the sinusoids could still be presumed as colonized with K. ozaenae and could be the most likely source for blood infection.

ConsentWritten informed consent was obtained from the patient for publication of this case report.

Competing InterestsThere are no personal or fi nancial competing interests to declare.

REFERENCES

1. Malowany MS, Chester B, Allerhand J. Isolation and microbiologic differentiation of Klebsiella rhinoscleromatis and Klebsiella ozaenae in cases of chronic rhinitis. Am J Clin Pathol. 1972; 58(5): 550-3.

2. Medina L, Benazzo M, Bertino G, et al. Clinical, genetic and immunologic analysis of a family affected by ozena. Eur Arch Otorhinolaryngol. 2003; 260(7): 390-4.

3. Talmi YP, Bar-Ziv J, Cohen D, Finkelstein Y, Kronenberg J. Computed tomography study of sinus involvement in ozena. Am J Rhinol. 1995; 9(5): 281-4.

4. Tang LM, Chen ST. Klebsiella ozaenae meningitis: report of two cases and review of the literature. Infection. 1994; 22(1): 58-61.

5. Murray KA, Clements BH, Keas SE. Klebsiella ozaenae septicemia associated with Hansen’s disease. J Clin Microbiol. 1981; 14(6): 703-5.

6. Danilowicz K, Sanz CF, Manavela M, Gomez RM, Bruno OD. Pituitary abscess: a report of two cases. Pituitary. 2008; 11(1): 89-92.

7. Ng TH, How SH, Kuan YC, Adzura, Aziz AA, Fauzi AR. A mimicry of melioidosis by Klebsiella ozaenae infection. Malays J Pathol. 2009; 31(2): 147-50.

8. Chou FF, Sheen-Chen SM, Chen YS, Chen MC. Single and multiple pyogenic liver abscesses: clinical course, etiology, and results of treatment. World J Surg. 1997; 21(4): 384-8; discussion 388-9.

9. Strampfer MJ, Schoch PE, Cunha BA. Cerebral abscess caused by Klebsiella ozaenae. J Clin Microbiol. 1987; 25(8): 1553-4.

10. Ko WC, Paterson DL, Sagnimeni AJ, et al. Community-acquired Klebsiella pneumoniae bacteremia: global differences in clinical patterns. Emerg Infect Dis. 2002; 8(2): 160-6.