Unique Co-Existence Of Rhinolithiasis And Nasal Diphtheria: A Case Report
A rhinolith is a rare pathology which can affect a variety of people in different age groups. Rhinolith forms around a nidus of a small foreign body, blood clots or secretions by slow deposition of minerals salts. Over a period of time, it can grow to form very large irregular mass that can even extend to the other side by destroying the septum or infiltrate the sinuses. Although toxigenic strains of Corynebacterium diphtheria cause the majority of respiratory diphtheria cases, non-toxigenic strains of diphtheria can also cause diseases, and are becoming more common. Infection that is limited to the anterior nares (nasal diphtheria) is a well-described but rare condition, We report a case involving chronic carriage of nasal diphtheria complicated by a rhinolith in a 21 years old female patient of low socioeconomic standards with full vaccination history complained of unilateral nasal obstruction and discharge, caused by non-toxigenic C. diphtheria. Mild or asymptomatic nasal which can be the source of transmission, may be under-recognized. Our case highlights the importance of rhinoliasis as a cause of unilateral nasal obstruction and the importance of taking a culture of the nasal secretions commonly found with rhinoliasis as a growth of unexpected dangerous organisms can coexist and simple removal of the stone may not be enough.
Introduction
A rhinolith is a rare pathology which usually presents by nasal obstruction and malodorous nasal discharge. Rhinolith formation starts with a nidus, which could be endogenous in origin, such as ectopic teeth, bone fragments and epithelial debris or exogenous in origin, such as, plastic material, paper fragments or fruit seeds. Diphtheria manifests as either an upper respiratory tract or cutaneous infection and is caused by the aerobic gram positive bacteria. Overcrowding, poor health, substandard living conditions, incomplete immunization, and immunocompromised states increases risk of diphtheria infection. Although human carriers are the main reservoir, some case reports have linked it to livestock, Immunity wanes over time, so inadequate boosting of vaccinated individuals may result in increased risk of infection. Additionally, since the advent of widespread vaccination, cases of non-toxigenic strains causing invasive disease have increased. A diphtheria carrier is someone whose cultures are positive but does not have signs and symptoms of diphtheria. The aim of this report is to present a unique case of nasal diphtheria complicated by a rhinolith and to emphasize the importance of nasal microbiota in rhinolithiasis cases.
Case Report
A 21 years old female patient with low socioeconomic standards presented to our outpatient clinic with left sided nasal congestion, obstruction and unilateral foul-smelling nasal discharge for 2 years. Her family and past history was insignificant and her vaccinations were complete. Her general physical examination and vital signs were within normal levels. Endoscopic examination showed a left sided purulent nasal discharge and a hard irregular mass filling the left nasal cavity. Nasal swab culture showed a growth of C. diphtheria. After consulting the results to the infectious disease department, amoxicillin+clavulonate (3X1000mg daily) was recommended for a period of 3 weeks. The computerized tomography (CT) of the paranasal sinuses also showed a left calcified mass in the left nasal cavity which was interpreted as a rhinolith. The patient was operated and the rhinolith was sent to microbiological examination. The material showed a growth of C. diphtheria. The patient’s complaints improved following surgery, postoperative control cultures at 2nd and 4th months were negative and her family screening was negative.
Microbiology
Tissue specimen was taken into ESwab™ Liquid Amies Collection and Transport System (COPAN Diagnostics, USA) and was sent to Central Medical Microbiology Laboratory. The tissue was grinded, Gram stained and qualitatively plated on Columbia Nalidicsic acid agar/MacConkey Agar (CNA/MCA) biplates and on Chocolate agar. CNA/MCA biplates were incubated at 37 0C for 24-48 hours in ambient air conditions, and chocolate agar plates were incubated at 37 oC for 24-48 hours in 10% CO2 atmosphere. Pre-reduced Brucella blood agar (Oxoid, USA) was also streaked for anaerobic culture and incubated at 37 oC for 72 hours under anaerobic conditions. For fungal growth, the sample was plated onto Sabauraud Dextrose Agar (SDA) (Becton Dickinson, USA). The Gram stain revealed 10-24 epithelial cells and 1-9 polymorphonuclear leukocytes per low power field magnification (LPF). Under 1000X magnification, microscopic examination revealed gram positive bacilli and gram positive coccus/high power field (HPF).
No fungal and anaerobic microorganisms were grown. The growing bacterium on CNA was identified as Corynebcaterium diphteriae by Matrix Assisted Laser Desorption Ionisation-Time of Flight Mass Spectrometry (MALDI-TOF MS, Bruker Daltonics, Bremen, Germany).
For further identification and tox gene evaluation the isolate was sent to the National Reference Laboratory for Respiratory Pathogens, Public Health Institution of Turkey, where the isolate was identified as Corynebacterium diphteriae biotype mitis with API Coryne (Bio-Mérieux, France). The presence of tox gene was evaluated PCR amplification (BioRad T100 Thermal Cycler / Bio-Rad) and toxin production was evaluated by the ELEK test. The isolate was found to be non-toxigenic by both methods.
Discussion
Nasal diphtheria is an extremely rare pathology of the nasal cavity which has found a place in the literature with only a few case reports.
A 24 years old patient was reported from Japan with no significant family history and a history of complete vaccination. He was diagnosed with the culture of sputum sample. The isolate was identified as C. diphtheria mitis. But unlike our case, he did not have any nasal symptoms. Endoscopic examination showed encrusted lesions in the nasal cavity and the nasal swab culture showed the growth of C. diphtheria. The patient was treated with oral amoxicillin and azithromycin. The control culture was negative after 1. 5 months. The other significant case report was linked to occupational swine contact, although our case did not have any animal contact. In this case, isolation of C. ulcerans from wild-boars have suggested that pigs might serve as reservoir for human infections.
There is a number of rhinolithiasis case series in the literature having the similar evaluation and treatment algorithms. The presenting symptoms were mostly unilateral chronic purulent nasal discharge and obstruction, the choice of radiologic imaging was CT scan and the applied treatment was removal under general anesthesia. However most of the authors did not report a culture of the discharge unlike our case. To the best of our knowledge, this is the first report of a case with a co-incidence of rhinolithiasis and nasal diphtheria. The patient in this report was a 21 years old chronic carrier of nasal diphtheria which was complicated by a rhinolith. She was vaccinated against C. diphtheria in the early childhood, however a non-toxigenic C. diphtheria was diagnosed in the nasal culture.
The limitation of this study was the lack of the nasal cultures of the other members of the patient’s family whom we were unable to contact. This might provide valuable information about the carriage status of the close contacts.
In this patient, it is unclear whether the diphtheria infection led to stasis and rhinolithiasis, or the stasis caused by the rhinolithiasis provided a proper media for diphtherial growth. Nevertheless, the lack of routine culture of nasal discharge especially in cases of rhinolithiasis may have led to under-reporting of such patients.
In sum, we strongly recommend to take a nasal culture in cases with obstructive pathologies such as rhinolithiasis or foreign bodies as the growth of unexpected dangerous organisms can coexist and simple removal of the pathology may not be enough in the treatment.
