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Twenty patients with medically treated rhinoscleroma and residual ethmoidal manifestations were examined using CT scanning. Cases with positive radiological data of the ethmoids underwent diagnostic and therapeutic nasal endoscopy. It has been found that ethmoidal scleroma is not as rare a condition as was previously thought. The diagnostic and therapeutic value of nasal endoscopy in ethmoidal scleroma is discussed.
Twenty-two cases of rhinoscleroma were studied to determine the involvement of the larynx. The study revealed that scleroma affected the larynx in 40 per cent of cases. In the larynx, scleromatous lesions were found in the subglottic region and the disease was observed in stages i.e., atrophic, granulomatous (proliferative i.e., nodular) and a fibrotic or sclerotic (scarring) stage as found in the nose. The role of laryngological examination is established in the diagnosis of asymptomatic and early laryngeal lesions and in assessing response to initial treatment. Extensive granulomatous lesions were treated by open excision by the laryngofissure approach which was found to be the best method resulting in a quick recovery without any evidence of subglottic stenosis. The disease as a whole is discussed in the light of available literature.
Two cases of rhinoscleroma in patients infected with the human immunodeficiency virus (HIV) who had stayed in an area of endemic Klebsiella rhinoscleromatis are reported. One of the patients presented with oropharyngeal lesions, an unusual clinical picture. Both patients suffered from a major cellular immune deficiency. The importance of Klebsiella rhinoscleromatis infection in AIDS-related oropharyngeal pathology and the possible treatment of such infection in HIV-positive patients are not yet clearly established.
Two patients presented with almost total obliteration of the pharynx. In one, a membrane developed after corrosive poisoning; in the other, the oropharynx was filled with a dense cicatrix in the sclerosing phase of rhinoscleroma. In both patients, a single opening in the membrane provided access to both the larynx and oesophagus. Fibreoptic intubation allowed both a thorough assessment of the pathology and subsequently the passage of a cuffed tracheal tube to secure the airway. To overcome the problem of respiratory obstruction while the fibrescope passed through the opening in the membrane, either rapid intubation, or a technique using pre-oxygenation and voluntary hyperventilation followed by breath-holding during bronchoscopy, was used. The thin calibre and manoeuvrability of the flexible fibreoptic bronchoscope makes fibreoptic intubation an excellent technique of airway management in cicatricial membranes of the pharynx.
We tested in vitro the activities of streptomycin and tetracycline--antibiotics that have long been used to treat rhinoscleroma--as well as several newer oral agents by using 23 isolates of the causative organism Klebsiella rhinoscleromatis. All isolates were inhibited by clinically achievable concentrations of trimethoprim-sulfamethoxazole, amoxicillin-clavulanate, chloramphenicol, ciprofloxacin, cephalexin, cefuroxime, and cefpodoxime.
The distribution of rhinoscleroma in Asia is reviewed and two cases from Iran are added, a region in which the disease seems not to have been previously reported. The pathogenesis of the disease is reviewed.
Nonneoplastic diseases of the central airways are uncommon but can be categorized as either focal or diffuse, although there is some overlap. Focal diseases include postintubation stenosis, postinfectious stenosis, posttransplantation stenosis, and various systemic diseases that may involve the airways and lead to focal stenosis (eg, Crohn disease, sarcoidosis, Behçet syndrome). Diffuse diseases of the central airways include Wegener granulomatosis, relapsing polychondritis, tracheobronchopathia osteochondroplastica, amyloidosis, papillomatosis, and rhinoscleroma. Conventional radiography is often the first step in the evaluation of suspected central airway disease and may be adequate in itself to identify the abnormality. However, computed tomography (CT) improves both the detection and characterization of central airway disease. Bronchoscopy remains the primary procedure for the diagnostic work-up of these disease entities. Nevertheless, a thorough radiologic evaluation with radiography and CT may demonstrate specific imaging findings (eg, calcification) that can help narrow the differential diagnosis and aid in the planning of bronchoscopy or therapeutic intervention.
Three cases of rhinoscleroma in the granulomatous stage were examined under the electrom microscope. The rough endoplasmic reticulum of plasma cells in scleroma displayed cisternal dilatations forming Russell bodies and cytoplasmic vacuolations containing viable and degenerated bacilli forming Mikulicz cells. Thus, they play a double role, producing antibodies and providing protection for the intracellular bacilli. New ultrastructural findings of Klebsiella rhinoscleromatic bacilli were described.
Rhinoscleroma is a chronic, slowly progressive, infectious disease of the respiratory tract that can produce disability and death, if untreated. Once considered an anomaly in the United States, the disease is now seen more frequently in this country, owing to increased travel from endemic areas. This report presents a comprehensive review of the several types of therapy that have been published in the literature. The results of a clinical experiment are also presented.
In 1870, F. Hebra described a slowly progressing granulomatous disease of the nasal mucous membrane he called rhinoscleroma. Later, N. M. Volkovich and A. Frisch isolated from patients suffering from this disease a microorganism they termed Klebsiella rhinoscleromatis. In 1932, S. Belinov proposed to name the disease Scleroma respiratorium because the pathological process developing in rhinosclerosis may involve not only upper but also lower airways. In 1961, T. Steffen and J. Smith demonstrated that Klebsiella rhinoscleromatis conformed to Koch's postulates and was an etiological factor of inflammatory changes typical of scleroma. This paper presents results of ultrastructural examinations of Klebsiella rhinoscleromatis isolated from two patients with Scleroma respiratorium. It was shown that as the degenerative process developed, the amount of electron-dense material around the bacterial cell wall decreased progressively and only fragments of the cellular membrane remained.
Granulomatous lesions of the cranio-facial area are frequent and various in their nature: lymphohistiocytic with or without eosinophils, tuberculoid-like with epithelioid and giant cells, or sometimes made essentially of giant cells. Their etiology can be known or easy to find: foreign body granuloma, sarcoidosis, leprosy, rhinoscleroma, fungal diseases especially zygomycosis and rhinosporidiosis, parasitic diseases. The lethal midline granuloma is a clinical entity characterized by its necrotic and relentlessly progressive destructive presentation. After elimination of a malignant process, especially lymphoid, and of a Wegener's granulomatosis the diagnosis will be "idiopathic midline non-healing granuloma". Some of them will stay located at the facial area; others will disseminate as a malignant disease. Central giant cell granuloma and histiocytosis X, especially eosinophilic granuloma, are two other varieties of granuloma, different of the former granulomatous infiltrates by their clinical presentation and their evolution.
A case of rhinoscleroma with laryngeal localization is reported. Geographic distribution of this granulomatous disease is discussed. Clinical and paraclinical features are explained. Its tumor-like appearance and local spread arouses suspicion of malignancy but differential diagnosis also includes fungal infections and numerous granulomatous diseases. The classic histopathology consists of large Mikulicz's cells. Treatment is essentially based upon antibiotics.
In a female aged 83 who had been living in Prague for years, a bioptic diagnosis of rhinoscleroma was made. The disease responded well to Septrin treatment. Electron microscopical examination confirmed the histiocytic origin of Mikulicz cells, but cast doubt upon the belief that the large phagocytic cysts in such cells developed by confluence of smaller vacuoles. The examination revealed that the large cystic formations only contained Klebsiellae and a dense finely granular material identical with that of the bacterial capsiles. The small vacuoles found in the vicinity of the large ones never contained bacteria and their contents was either coarsely granular of finely filamentous. Therefore it rather corresponded to degradation products of the original content of the larger phagocytic cysts. The final picture of the Mikulicz cell are macrophages with only small light rounded vacuoles containing mucoid but no Klebsiellae.
A case of scarring stenosis of the nasopharynx occurring in the fifth decade without previous medical history is presented. Transpalatal resection with a prolonged stent, produced patent nasal cavities, but the different studies performed did not establish the etiological diagnosis. A final stage of rhinoscleroma was suspected.
The frozen section diagnosis of scleroma (rhinoscleroma) may pose some difficulty to the unsuspecting pathologist. Intraoperative cytology is a helpful technique that, when combined with frozen section, enhances the diagnostic accuracy. Imprint smears obtained from a tissue sample of an obstructive nasal mucosal mass demonstrated the characteristic Mikulicz cells, thus confirming the frozen section diagnosis of scleroma. The importance of intraoperative cytology in the evaluation of surgical specimens is emphasized.
The Author reviews the cases diagnosed in the Republic of Panama of Mycetoma, Paracoccidioidosis, Lobo's disease, Chromomycosis, Histoplasmosis, Rhinosporidiosis, Sporotrichosis, Lepra, Rhinoscleroma, and cutaneous and mucocutaneous Leishmaniasis, and mentions the observed clinical manifestations in order to familiarize young physicians with the tropical dermatopathology which occurs in the rural areas of the country.
A case of rhinoscleroma in an elderly Danish woman is presented. The characteristics of the disease are described. An 84-year old female presented with serous rhinorrhoea, swelling of the columella and the upper lip and granulomatous masses in the right inferior concha and the right maxillary sinus. The diagnosis was confirmed by histological studies. Until now no serological or bacteriological cultures have been positive. The patient recovered clinically after surgical debridement and three months of orally administered ciprofloxacin 500 mg twice daily. There remains, however, a sclerotic swelling in the columella and the upper lip. No obvious explanation as to how this patient had caught the disease could be found.
Scleroma, more commonly known as rhinoscleroma, is a progressive, chronic, granulomatous disease of the respiratory tract and adjacent tissues. Early diagnosis and prolonged therapy are critical in avoiding its late sequelae. We describe an unusual case of scleroma presenting as worsening chronic lung disease. The variety of clinical presentations and treatment modalities is reviewed.