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Cover page of the Journal of Health Sciences


 
 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 15  |  Issue : 3  |  Page : 298-301

Rhinoscleroma – still a predominant entity managed by rubber tube recanalization


Department of ENT and Head and Neck Surgery, Kaher's J. N. Medical College, Belagavi, Karnataka, India

Date of Submission18-Feb-2022
Date of Acceptance27-Apr-2022
Date of Web Publication17-Sep-2022

Correspondence Address:
Dr. Yashita Singh
MIG26 Bandhavgarh Colony, Satna - 485 005, Madhya Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/kleuhsj.kleuhsj_136_22

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  Abstract 


Rhinoscleroma is a chronic granulomatous infection affecting the nasal cavity and nasopharynx primarily. It results in functional and esthetic sequelae in patients. A patient presented to us with chronic extensive vestibular stenosis with external deformity of the nose. The patient underwent multistaged diagnostic nasal endoscopy with biopsy and recanalization using a sterile rubber catheter which showed significant clinical improvement. This case demonstrates that medical advancements have enabled a quicker diagnosis of rhinoscleroma and recanalization still remains an essential entity in managing the sequelae of rhinoscleroma.

Keywords: Anosmia, narrowed nasal cavities, recanalization, rhinoscleroma, rubber tube, saddle nose


How to cite this article:
Harugop A S, Nayak S P, Singh Y, Gulaganji R. Rhinoscleroma – still a predominant entity managed by rubber tube recanalization. Indian J Health Sci Biomed Res 2022;15:298-301

How to cite this URL:
Harugop A S, Nayak S P, Singh Y, Gulaganji R. Rhinoscleroma – still a predominant entity managed by rubber tube recanalization. Indian J Health Sci Biomed Res [serial online] 2022 [cited 2022 Sep 25];15:298-301. Available from: https://www.ijournalhs.org/text.asp?2022/15/3/298/356268




  Introduction Top


Rhinoscleroma is a rare, chronic, slowly progressive, granulomatous infection caused by Klebsiella rhinoscleromatis, rod-shaped Gram-negative bacilli. It was first described by the dermatologist Ferdinando Von Hebra in 1870.[1] It commonly affects the nasal cavity and nasopharynx, but it can also involve the larynx, trachea, bronchi, middle ear, and orbit.[2] Nasal involvement is initial and almost constant. After the nasal cavity, other common sites affected in the respiratory tract are the nasopharynx (18%–43%), paranasal sinus (22%), and larynx (15%–40%). Rhinoscleroma is common in the rural areas where socioeconomic status is very low. It is more common in females, the female-to-male ratio being (13:1).[3] Usually encountered in the second and third decades of life. The disease is endemic in Central America, Indonesia, Poland, Russia, Hungary, and African countries. Despite the diverse presentations in patients across the world, the epidemiological data are unsubstantial.[4],[5]


  Case Report Top


A 30-year-old male presented to the ENT outpatient department with chief complaints of bilateral progressive nasal obstruction for 1½ years. It was associated with complaints of epistaxis for 4 months which was intermittent and resolved spontaneously. History of anosmia and mouth breathing were also present. On examination, external deformity of the nose was observed. Depressed dorsum, pinched up ala, and drooping tip were observed [Figure 1] and [Figure 2]. A written informed consent was taken and patient was managed by performing diagnostic nasal endoscopy and biopsy. On rhinoscopy, the bilateral nasal cavity showed circumferential stenosis at the nasal valve area along with crusting. The patient underwent diagnostic nasal endoscopy with biopsy for similar complaints previously which was conclusive of rhinoscleroma on histopathological examination [Figure 3]. Ear, throat, and neck examination was normal. Routine blood investigations were normal except for elevated bilirubin levels. Computed tomography [CT] scan of paranasal sinuses was normal.
Figure 1: Right lateral view

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Figure 2: Basal view

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Figure 3: Showing marked valvular stenosis in the nasal cavity

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The patient was managed by performing a diagnostic nasal endoscopy with biopsy and recanalization procedure using a sterile rubber catheter under general anesthesia. Marked circumferential valvular stenosis was noted in bilateral nasal cavities which were excised and sent for histopathological examination. There was a circumferential narrowing at the mucocutaneous junction of the vestibule, and no space was observed. Concentric narrowing of choana was also observed bilaterally. Before the recanalization procedure, multiple biopsies were taken from middle turbinate mucosa and sent for histopathological examination which was reported as mucosa lined by stratified squamous epithelium. Subepithelial tissue showed mixed inflammatory infiltrate composed of lymphocytes, plasma cells, Mikulicz cells, and Russell bodies suggestive of rhinoscleroma [Figure 4]. The patient was treated with antibiotics consisting of piperacillin + tazobactam, rifampicin, and saline nose drops. The circumferential stenosed mucosa was excised endoscopically, following which the patient underwent recanalization by the placement of a sterile rubber catheter in both nasal cavities. Through-and-through sutures were passed through the catheter and septum on both sides to secure the rubber catheter in both nasal cavities [Figure 5], [Figure 6], [Figure 7] which were removed after 6 weeks. The recanalization was done by the conventional method without the use of LASER technology. The patient was followed up for 6 months and showed significant improvement clinically.
Figure 4: Showing H- and E-stained histopathology slide showing Mikulicz cells (black arrows) and Plasma cells (red arrows)

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Figure 5: Demonstrating the recanalization procedure using sterile rubber

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Figure 6: Sterile rubber catheter being inserted in the nasal cavity

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Figure 7: Sterile rubber catheter being secured by suture in the nasal cavity

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  Discussion Top


Rhinoscleroma is a chronic granulomatous disease which can involve any area of the respiratory tract from the nose to the trachea. The causative agent Klebsiella rhinoscleromatis, which belongs to the Enterobacteriaceae family, is nonmotile, encapsulated, facultative, and glucose-fermenting. The bacteria is contracted through direct inhalation or dissemination of respiratory droplets. The pathophysiology indicates that the disease predominantly occurs in transition zones of two epithelia-stratified squamous epithelium of the vestibule and respiratory epithelium of the nose. It is hypothesized that decreased iron in the body may lead to this disease. The scarcity of iron alters the epithelial regeneration causing squamous metaplasia. The disease progression can be staged into stage 1 or catarrhal stage which comprises features similar to nonspecific rhinitis which evolves into foul-smelling purulent nasal discharge, crusting, and nasal obstruction. Stage 2 or stage of granuloma formation includes granulation tissue formation, widening of the nasal pyramid causing external deformity, subdermal infiltration causing “woody hard” feel of nose, and nasal septum cartilage destruction. Finally, stage 3 or stage of sclerosis is characterized by extensive scarring and laryngeal and nasal vestibular stenosis in severe cases. The histological picture of rhinoscleroma is characterized by the presence of Mikulicz cells and Russel bodies seen in granulomatous stage. Mikulicz cells are pale vacuolated cells with clear cytoplasm possessing the bacteria. Russel bodies originate from the disintegration of plasma cells. Intracytoplasmic bacilli can be demonstrated by special stains such as Gram stain, periodic acid–Schiff, Giemsa stain, Warthin–Starry, and Grocott's silver methenamine stains.[6],[7],[8] A study done by Abdel Razek concluded that rhinoscleroma of the nose in the hypertrophic stage appeared as a well-defined mass on CT, and involvement of the inferior and middle turbinate was more common. On magnetic resonance imaging, granulomas can be identified in the T1-weighted images owing to the high protein content within Mikulicz cells and Russell bodies.[9]

In rhinoscleroma, owing to the high relapse rate, a prolonged antibiotic therapy is usually recommended. It is inclusive of doxycycline, rifampicin, streptomycin, second- and third-generation cephalosporins, and ciprofloxacin. Traditionally, streptomycin was the drug of choice for the treatment of rhinoscleroma. However, tetracycline soon became the preferred antibiotic, owing to its oral administration and avoidance of the vestibulotoxic side effects of streptomycin. Fluoroquinolones have been researched to be a potent and economical treatment option. The advantage being that quinolones are concentrated within macrophages, less toxic and are comparatively less expensive.[10] Surgical intervention is indicated in patients with external nasal deformity or airway obstruction.[11] Surgical excision and recanalization are usually done for the tertiary stage of sclerosis.[12] Silastic sheet stents or polyethylene tubes can be used for nasal dilatation and recanalization. It is inserted in each nostril and fixed in place by silk sutures. There are very limited studies that highlight the recanalization technique in advanced cases of rhinoscleroma. It ensures adequate clinical improvement by relieving the nasal obstruction and nasal dilatation.


  Conclusion Top


Rhinoscleroma is a chronic disfiguring disease. The advancements in diagnosis and medical and surgical management have made its early recognition possible. Therefore, a planned complete medical management along with endoscopic surgical procedures with recanalization can aid in managing advanced sequelae of rhinoscleroma such as nasal stenosis and yield apt results as well as provide symptomatic relief to the distressed patients.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Belaldavar BP, Havaldar RR, Sindhu N, Das SS. Dacryoscleroma - A Pandora's box. Clinical Case Reports: Open Access [Internet]. 2020;3. Available from: http://dx.doi.org/10.46527/2582-5038.150. [Last accessed on 2022 Aug 25].  Back to cited text no. 1
    
2.
Santosh S, Priyanka D, Smarita L, Smrutipragnya S, Anurag D, Somalee M, Swain SK. Rhinoscleroma-A rare granulomatous disease in a child. Indian J Forensic Med Toxicology 2020;14;8858-61.  Back to cited text no. 2
    
3.
Bhagyalaksmi A, Rao CV, Krishna Kishore T, Kartheek BV. Rhinoscleroma: A case report. Int J Case Rep Imag [Internet] 2014;5:868. Available from: http://dx.doi.org/10.5348/ijcri-201457-ci-10014. [Last accessed on 2022 Aug 25].  Back to cited text no. 3
    
4.
Elwany S, Fattah HA, Mandour Z, Ismail AS, Abdelnabi M. A myriad of scleroma presentations: The usual and unusual. Head Neck Pathol 2020;14:588-92.  Back to cited text no. 4
    
5.
Chmiel A. Kinetic studies on citric acid production by Aspergillus niger. II. The two-stage process. Acta Microbiol Pol B 1975;7:237-42.  Back to cited text no. 5
    
6.
Chan TV, Spiegel JH. Klebsiella rhinoscleromatis of the membranous nasal septum. J Laryngol Otol 2007;121:998-1002.  Back to cited text no. 6
    
7.
Montone KT. Infectious diseases of the head and neck: A review. Am J Clin Pathol. 2007;128:35-67.  Back to cited text no. 7
    
8.
Andraca R, Edson RS, Kern EB. Rhinoscleroma: A growing concern in the United States? Mayo Clinic experience. Mayo Clin Proc 1993;68:1151-7.  Back to cited text no. 8
    
9.
Abdel Razek AA. Imaging of scleroma in the head and neck. Br J Radiol 2012;85:1551-5.  Back to cited text no. 9
    
10.
Badia L, Lund VJ. A case of rhinoscleroma treated with ciprofloxacin. J Laryngol Otol [Internet]. 2001;115:220-2. Available from: http://dx.doi.org/10.1258/0022215011907028. [Last accessed on 2022 Aug 25].  Back to cited text no. 10
    
11.
Zhong Q, Huang Z, Guo W, Zhang S, Ge W. Rhinoscleroma: Case report. Am J Otolaryngol 2010;31:381-3.  Back to cited text no. 11
    
12.
Pathak RD. Rhinoscleroma- surgical management-A case report. Indian J Otolaryngol Head Neck Surg 1999;52:64-6.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]



 

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