Rhinoscleroma and malignancy; two cases of rhinoscleroma associated with carcinoma.
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Carcinoma↗
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Identification of rhinoscleroma by immunoperoxidase technique.
Rhinoscleroma is a chronic disease of the respiratory system. Its onset is insidious and its symptoms are often nonspecific. Yet rhinoscleroma is a progressive disease that can lead to significant disability. The proper diagnosis is difficult because of the nonspecific presentation and rarity of rhinoscleroma. Biopsy tissue often is not properly cultured. Diagnostic histologic features may not be readily apparent or may be overlooked by the unsuspecting pathologist. A case of rhinoscleroma is reported in which an immunoperoxidase technique was applied to retrospectively confirm the diagnosis. The usefulness of this new technique in the diagnosis of rhinoscleroma is discussed.
Adult↗
Modification of lymphocyte subsets in patients with rhinoscleroma.
PURPOSE: Rhinoscleroma is a rare, chronic, granulomatous disorder of the upper airways. This disease presents some etiopathogenetic aspects that are not yet clear. Infection by Klebsiella rhinoscleromatis is fundamental for the onset of the disease, but it is impossible to reproduce rhinoscleroma experimentally only via infection with the bacteria both in man and in animals. Furthermore, this disease mainly affects blood-related people and occurs in certain geographic areas. In this context, we present a study that brings to light some of the quantitative abnormalities of the lymphocyte subsets. MATERIALS AND METHODS: The study group consisted of 5 patients with rhinoscleroma. The following parameters were studied for each patient: clinical manifestations, histologic examinations, number of leukocytes, lymphocytes, and lymphocyte subsets. RESULTS: In all patients, we noted the following: There was a relative reduction of the CD4+ cells, an absolute increase of the CD8+ cells, and an inversion of the CD4+/CD8+ ratio. There was an absolute increase of the CD56+ cells and cytotoxic cells that coexpress CD8+CD56+ antigens. There was a relative reduction of the CD3+ cells, and the CD19+ cells tended to show an ambiguous behavioral pattern. CONCLUSION: We believe that K. rhinoscleromatis does not play a major role in the etiopathogenesis of rhinoscleroma. However, we do believe that the anomalous behavior of the immune system can favor rhinoscleroma.
Adult↗
Rhinoscleroma: a growing concern in the United States? Mayo Clinic experience.
Rhinoscleroma is a chronic, progressive, granulomatous infection of the upper airways caused by the bacterium Klebsiella rhinoscleromatis. Although most cases occur in developing countries, recent immigration patterns have led to an increasing number of patients with rhinoscleroma in the United States. Rhinoscleroma may mimic various inflammatory and neoplastic processes, including leprosy, paracoccidioidomycosis, sarcoidosis, basal cell carcinoma, and Wegener's granulomatosis. Current therapy consists of a combination of surgical débridement and prolonged antimicrobial therapy. Rhinoscleroma should be added to the list of opportunistic infections that can occur in patients with human immunodeficiency virus.
Adult↗
A case of rhinoscleroma treated with ciprofloxacin.
Respiratory scleroma (often termed 'rhinoscleroma') is a chronic inflammatory condition in which deforming masses of tissue distend the nasal cavity. Klebsiella rhinoscleromatis is the causative agent of this infection and the Mikulicz cell is specific to the lesion being a large macrophage with clear cytoplasm containing the bacilli. Antibiotic therapy has traditionally consisted of streptomycin and tetracycline long-term but this presents problems with adverse side-effects and poor patient compliance. We report on a young patient with nasal rhinoscleroma who achieved resolution after treatment with oral ciprofloxacin. As mentioned in a review of patients with rhinoscleroma at the Mayo clinic in 1993, the fluoroquinolones deserve further study as potentially highly effective agents for this condition. Ciprofloxacin is convenient for oral administration and has few adverse effects. It achieves good tissue penetration, is concentrated in macrophages and may prove to be useful in the therapy of rhinoscleroma.
Adolescent↗
Rhinoscleroma treated with ciprofloxacin: a case report.
Rhinoscleroma, a chronic progressive infection of the nose and associated structures caused by Klebsiella rhinoscleromatis, has posed a therapeutic dilemma since its identification in the late 1800s. Although a number of antibiotics have been found to be effective in this relapsing disorder, the lengthy duration of treatment can lead to problems with adverse effects and compliance, especially with the traditional therapies of streptomycin and tetracycline. We report on a patient with extensive nasal rhinoscleroma who achieved pathologic and bacteriologic resolution during treatment with oral ciprofloxacin after previous courses of tetracycline and trimethoprim-sulfamethoxazole. Ciprofloxacin may prove to be useful in the therapy of rhinoscleroma because it is convenient for oral administration, achieves good tissue levels, is concentrated in macrophages, and is generally well tolerated as long-term therapy. As mentioned in a recent review of patients with rhinoscleroma at the Mayo Clinic, the fluoroquinolones deserve further study as potentially highly effective agents for this uncommon but significant infectious condition.
Adult↗
MR appearance of rhinoscleroma.
BACKGROUND AND PURPOSE: We describe the MR imaging appearance of rhinoscleroma, an endemic, chronic, granulomatous disease whose causative agent is Klebsiella rhinoscleromatis. METHODS: The study included 15 patients (nine males and six females; mean age, 25 years; range, 13-36 years) with rhinoscleroma. MR imaging was performed in all patients. The signal intensity of the nasal masses was compared with that of fat, muscle, and CSF on both T1- and T2-weighted images. All cases were proved by histopathologic examination. RESULTS: The nasal masses were bilateral and symmetrical (n = 6), asymmetrical (n = 4), or unilateral (n = 5). They extended through the anterior nares (n = 9) or posterior choana into the nasopharynx (n = 3). They obstructed the ostiomeatal units with retained secretions in the related sinuses (n = 10). On T1-weighted images, rhinoscleroma showed striking (n = 9) or mild (n = 6) high signal intensity relative to muscle and CSF, but less hyperintensity than fat. On T2-weighted images, the nasal masses showed homogeneous high signal intensity (n = 10) or heterogeneous high signal intensity associated with hypointense foci (n = 5). They were hyperintense relative to fat and muscle, but less hyperintense than CSF. CONCLUSION: The hypertrophic stage of rhinoscleroma has characteristic mild to marked high signal intensity on both T1- and T2-weighted MR images.
Adipose Tissue↗
Antroscopy in rhinoscleroma.
Rhinoscleroma is a chronic specific granulomatous inflammatory condition that has an affinity to the mucosa of the upper respiratory tract. Involvement of maxillary antrum is said to be very uncommon. In the present study antroscopy was performed in 20 patients with rhinoscleroma to find out the type, nature and site of the lesion in the maxillary antrum. The maxillary antrum was involved in 60 per cent of cases with rhinoscleroma. The lesions occurred in the form of atrophic changes, granuloma, and the fibrotic thick healed stage. The anterior-inferior part and medial wall of the antrum are found to be more commonly affected. It is suggested that involvement of maxillary antrum in scleroma may act as a reservoir of infection and such patients may therefore take a longer time to respond to antibiotic therapy.
Adult↗
[Rhinoscleroma: a case report].
BACKGROUND: Rhinoscleroma is a rare infectious disease of the upper respiratory tract caused by Klebsiella rhinoscleromatis. The nasal mucosa represents the primary region of occurrence which is most likely a result of respiratory transmission. Without adequate++ treatment, the disease can potentially spread to the rest of the upper and middle respiratory tract within a period of several years. Healing often occurs with extensive scarring and adhesions of the nose, palate and larynx. A life threatening late stage manifestation includes subglottic stenosis requiring immediate surgical intervention. Medical treatment primarily consists of a long-term course of antibiotics. Ciprofloxacin (Fluoroquinolon) has proved to be one of the most effective drugs. CASE REPORT: We report about a 33 year-old Egyptian male, who presented in our department with a 10 year history of a previously, only temporarily successfully treated rhinoscleroma. His main symptoms were nasal obstruction, epistaxis and inspiratory stridor. FINDINGS: We began treatment with Ciprofloxacin over a four week period, which lead to an improvement of his symptoms. The treatment was considered complete after several biopsies and smears were negative for live specimens. We then chose to reduce the scar tissue that was causing obstruction in the larynx and the nose. CONCLUSION: The rhinoscleroma is a rare disease in geographic areas with poor hygiene and can mimic several other infectious and malignant diseases. Treatment should include a long-term antimicrobial therapy and surgical intervention in cases with symptomatic obstruction.
Adult↗
Upper airway obstruction due to rhinoscleroma: case report.
Rhinoscleroma is a very rare cause of upper airway obstruction with only isolated reports in the literature of rhinoscleroma with isolated tracheal obstruction. The course is usually chronic with the presentation most often being non-specific. We report a 54-year-old woman with progressive shortness of breath and wheezing over 7 years' duration. She was diagnosed and treated as bronchial asthma without improvement in her symptoms. At the time of referral to our institution, her flow-volume loop revealed fixed upper airway obstruction. Her chest radiography and other laboratory tests were normal. Bronchoscopy revealed a 70-80% irregular concentric stenosis of the trachea beginning immediately below the vocal cords and extending 4 cm distally. Biopsy showed characteristic Mikulicz histiocytes containing numerous gram-negative intracellular coccobacilli consistent with a diagnosis of rhinoscleroma. The patient was treated with laser resection of the stenosis followed by a course of ciprofloxcin and trimethoprim-sulfamethoxazole. She has remained asymptomatic over a year follow-up period and repeated biopsies have shown no evidence of recurrence.
Airway Obstruction↗
Rhinoscleroma associated with Rosai-Dorfman reaction of regional lymph nodes.
Rhinoscleroma is an uncommon chronic, destructive infection of the respiratory mucosa caused by Klebsiella rhinoscleromatis. This coccobacillus can be found in the typical histiocytes, the Mikulicz cells. Extranasal and nodal involvement in this disease is rare, but documented. Rosai-Dorfman disease or sinus histiocytosis with massive lymphadenopathy is also a rare, non-hereditary disorder. Bilateral cervical lymphadenopathy with emperipolesis, as the main histological characteristic, is the most common presentation. It can also occur extranodally. We report a case of rhinoscleroma occurring in a 62-year-old woman since 1984, who developed parotid gland and lymph node involvement. The changes in the nasal mucosa and the parotid gland showed chronic inflammation with Mikulicz cells. In the lymph nodes, features characteristic of Rosai-Dorfman disease were seen. Taking into consideration the literature dealing with both of these diseases, we discuss that Rosai-Dorfman disease could be a special type of lymph node reaction and is not necessarily an entity of its own. Therefore, it should be known as Rosai-Dorfman lymph node reaction. Furthermore, there seems to be an interconnection between Rosai-Dorfman disease and rhinoscleroma.
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Rhinoscleroma causing upper airway obstruction.
Rhinoscleroma is a chronic granulomatous condition of the respiratory tract, and is not uncommon in tropical regions; particularly, Mexico, Central America and the Middle East. A few cases have been reported in North America, primarily involving immigrants from endemic countries. The causative organism is Klebsiella rhinoscleromatis, a Gram-negative coccobacillus. Diagnosis is made on the basis of culture of the organism and the characteristic pathology of Mikulicz cells on light microscopy. The condition primarily affects the upper airway, and frequently presents with nasal discharge, nasal obstruction or frontal facial pain. Despite the term 'rhinoscleroma', there may be involvement of the entire respiratory tract. Although the condition is slowly progressive, its natural course portends extensive destruction. Laryngotracheal involvement occurs in approximately 15% to 80% of cases, but patients rarely present with isolated laryngotracheal disease. In the present paper, a case of rhinoscleroma presenting with symptoms of upper airway obstruction is described.
Airway Obstruction↗
Laryngotracheal manifestations of rhinoscleroma.
Rhinoscleroma is a rare, chronic granulomatous disease of infective causation. It usually begins in the nose and may progress to involve the larynx and trachea and cause dysphonia, stridor, and airway obstruction. Early rhinoscleroma is usually successfully treated with oral tetracycline, yet laryngotracheal disease may require operative intervention. The disease is rare in the United States, but with an increase in immigration from endemic areas, otolaryngologists should be familiar with the management of this rare disease. Current literature contains only a few reports describing the manifestations of this disease, mostly in the form of case studies. This study is a retrospective review of our institutional experience with the management of 22 patients with rhinoscleroma, 13 of whom had laryngotracheal involvement. The focus of this report is on the clinical manifestations of laryngotracheal scleroma. All of the patients were treated with long-term antibiotics. Nine patients underwent endoscopy with or without dilation and laser excision. Three patients required emergency tracheostomy, all of whom were ultimately decannulated without any sequelae. A rational approach to management of this unusual disease is provided.
Adult↗
Rhinoscleroma of the lower respiratory tract.
Rhinoscleroma is a chronic granulomatous disease of the respiratory tract endemic to Eastern Europe and Central America which is being recognized with increasing frequency in other countries, including the United States. It was initially described as a lesion of the nose and upper respiratory tract, but is now known to involve the larynx, trachea and bronchi as well to cause slowly progressive asphyxia. Eleven cases of rhinoscleroma with varying degrees of involvement of the lower respiratory tract (larynx, trachea and bronchi) are presented. Present day treatment is both medical, consisting primarily of streptomycin and tetracycline, and endoscopic dilatation. Prolonged medication with careful dose control is necessary. Lower respiratory tract involvement should be considered in patients with chronic destructive granulomatous nasal pathology. This should be especially emphasized if they have an Eastern European or Central American background, or have travelled in these areas in which rhinoscleroma is endemic.
Adult↗
Rhinoscleroma: eight Peruvian cases.
Rhinoscleroma is a rare infection in developed countries; although, it is reported with some frequency in poorer regions such as Central Africa, Central and South America, Eastern and Central Europe, Middle East, India and Indonesia. Nowadays, rhinoscleroma may be erroneously diagnosed as mucocutaneos leishmaniasis, leprosy, paracoccidioidomycosis, rhinosporidiasis, late syphilis, neoplasic diseases or other upper airway diseases. From 1996 to 2003, we diagnosed rhinoscleroma in eight patients attended in the Dermatologic and Transmitted Diseases service of "Cayetano Heredia" National Hospital, in Lima, Peru. The patients presented airway structural alterations producing nasopharyngeal, oropharyngeal and, in one patient, laryngeal stenosis. Biopsy samples revealed large vacuolated macrophages (Mikulicz cells) in all patients. Ciprofloxacin 500 mg bid for four to 12 weeks was used in seven patients and oxytetracycline 500 mg qid for six weeks in one patient. After follow-up for six to 12 months the patients did not show active infection or relapse, however, all of them presented some degree of upper airway stenosis. These cases are reported because of the difficulty diagnosing the disease and the success of antibiotic treatment.
Adult↗
Skin affection in rhinoscleroma. A clinical, histological and electron microscopic study on four patients.
Clinical, histological and electron microscopic studies were performed on 4 rhinoscleroma patients with concomitant skin lesions in the upper lip, dorsum of the nose and nasolacrymal sac area. The skin lesions were treated locally. One of the patients was followed up to 16 years. Histologically, the skin lesions showed downward prolongation of the rete pegs. Deep in the dermis, vacuolated Mikulicz cells surrounded by lymphocytes and plasma cells were found. By electron microscopy, numerous small vacuoles containing fine granular material were seen inside Mikulicz cells. A limited number of bacilli were found inside and outside these cells. Two clinical entities of rhinoscleroma are observed in Egypt, an active granulomatous type with possible extranasal extension, and a less active intranasal type with limited mucosal lesions. The skin lesions in rhinoscleroma were found to have an unpredictable course.
Adolescent↗
The Mikulicz cell in rhinoscleroma. Light, fluorescent and electron microscopic studies.
The stages in the development of the Mikulicz cell in human rhinoscleroma were studied in biopsy specimens obtained from 10 patients using light, immunofluorescent and electron microscopy. The Mikulicz cell was identified morphologically as a macrophage, not a plasma cell. Acutely inflamed areas of rhinoscleroma presented abundant bacteria with a slime layer. The microorganism was infrequent and the mucopolysaccharide was scanty in rhinoscleromal tissue, where plasma cells predominated, and in cicatricial fibrous tissue. In the granulomatous stage of rhinoscleroma, the mucopolysaccharide was found within the Mikulicz cells. The vacuoles observed in the Mikulicz cells were considered to be phagosomes containing, principally, bacterial mucopolysaccharide and few bacteria and, to a lesser extent, swollen mitochondria. It was concluded that the slime layer of Klebsiella rhinoscleromatis plays an important role in the pathogenesis of the disease. It is postulated that this material is a nondigestible mucopolysaccharide that resides in the phagosomes of macrophages, increases the osmotic pressure and forms multiple hydropic vacuoles that rupture not only the phagosomes but also the cells, resulting in the liberation of the mucopolysaccharide. This would initiate a cycle that would prolong the disease in the absence of the bacteria.
Biopsy↗
Rhinoscleroma in an octogenarian woman.
Rhinoscleroma, a granulomatous infection caused by Klebsiella rhinoscleromatis and usually afflicting young adults, was diagnosed in an 81-year-old woman. Treatment was started with orally administered antibiotics but was soon halted because of side effects. She was treated instead by local spread of a 3% rifampin ointment. After 6 weeks, the lesion cleared up and she improved remarkably. There was no recurrence during 18 months of follow-up. The current report is the second published case of rhinoscleroma in an elderly person and the first report on its cure by local treatment with a 3% rifampin ointment. This favorable experience contributes to the limited knowledge on rhinoscleroma in the elderly.
Journal Article↗