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ABSTRACT Rhinoscleroma (more appropriately 'scleroma') is a chronic, disfiguring inflammatory lesion that is rarely encountered in the present-day otolaryngology practice. The diagnosis often becomes difficult especially when it recurs and presents late with complications. This report illustrates the clinical and histologic features of rhinoscleroma in a defaulter patient who represents advanced-stage disease with orbital and intracranial extensions. Special emphasis has been provided on the computed tomography impressions such that the evolution of scleroma with time can be studied.

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El vestíbulo nasal corresponde a la primera porción de la fosa nasal, éste se encuentra delimitado lateralmente por los cartílagos alares y medialmente por el borde caudal del septum nasal y la columela. Las enfermedades infecciosas del vestíbulo nasal son patologías frecuentes en la práctica clínica; su diagnóstico se realiza en base a sospecha clínica y examen físico, requiriendo habitualmente solo manejo médico ambulatorio. Desde el punto de vista etiológico pueden ser virales, bacterianas y fúngicas. Las principales especies bacterianas involucradas corresponden a Staphylococcus coagulasa negativa, S. epidermidis, S. hominis y S. haemolyticus, difteroides spp y S. aureus. Su manejo es esencialmente médico con casos excepcionales requiriendo manejo quirúrgico. En la actualidad existe escasa información epidemiológica al respecto, lo que dificultad la clasificación de los dichos cuadros clínicos. Se realizó una revisión de la literatura sobre cuadros infecciosos que afectan el vestíbulo nasal para lograr sistematizar y clarificar las distintas patologías y sus tratamientos.

The nasal vestibule corresponds to the first portion of the nasal passage, limited laterally by the lateral crus and medially by the caudal edge of the nasal septum and columella. Infectious diseases of the nasal vestibule are frequent in clinical practice, diagnosis is made based on clinical suspicion and physical examination, usually requiring only ambulatory medical management. In terms of etiology, they can be viral, bacterial and fungal. The main bacterial species involved correspond: Coagulase-negative Staphylococcus, S. epidermidis, S. hominis and S. haemolyticus, difteroides spp and S. aureus. Management is essentially medical and only exceptionally requires surgery. Currently, there is a lack of epidemiological information in this regard, which makes it difficult to classify these clinical conditions. A review of the literature on infectious conditions that affect the nasal vestibule was performed, to systematize and clarify the different pathologies and their management.

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OBJECTIVES: To evaluate clinical and pathologically cases of respiratory scleroma diagnosed in a 30-year period in Guatemala. MATERIAL AND METHODS: Fifty-one cases of respiratory scleroma diagnosed from 1988 to 2018 in a single pathology service in Guatemala were confirmed using Warthin-Starry staining. Immunohistochemical reactions against CD68, LCA, CD20, CD3, and CD138 were performed to illustrate the inflammatory infiltrate. Scanning electron microscopy (SEM) was performed to illustrate bacteria morphology. RESULTS: All 51 cases affected patients from poor areas of Guatemala, particularly women (66.7%), with a mean age of 31 years (range 7-66 years). Nose was affected in most cases (96.1%). Other sites involved included pharynx, larynx, palate, maxillary sinuses, and upper lip. Depending on the stage, the disease manifested as ulcerations, nasal deformities, or laryngeal stenosis. Nasal obstruction, epistaxis, dysphonia, fetid discharge, and pain were the main symptoms. Mikulicz cells (CD68+) in a plasma cell-rich inflammatory background (CD138+, CD20+, CD3+/-) were the typical microscopic presentation. In SEM, each macrophagic vacuole contained few to dozens of Klebsiella rhinoscleromatis diplobacilli. Treatment consisted of long-term trimethoprim and sulfamethoxazole, with adequate control of disease. CONCLUSION: Respiratory scleroma is a rare infectious disease affecting the upper respiratory tract, in poor regions of the world, including Guatemala.

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Rhinoscleroma is a chronic, infectious and granulomatous disease of the respiratory tract. There is often a delay in diagnosis due to unfamiliarity with the disease and also because culture is not always positive. We report a case in a 26-year-old woman with granular mass obstructing bilateral nasal cavities and causing breathing difficulty. Histopathological examination showed characteristic Mikulicz histiocytes containing numerous Gram-negative intracellular rod-shaped bacilli consistent with the diagnosis of rhinoscleroma. The patient was treated with gemifloxacin and tetracycline and remains asymptomatic over a year follow-up period. It is important to consider rhinoscleroma in cases of chronic nasal obstruction. As culture is not always positive, histopathological examination may be crucial to the diagnosis.

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Rhinoscleroma is a chronic, infectious and granulomatous disease of the respiratory tract. There is often a delay in diagnosis due to unfamiliarity with the disease and also because culture is not always positive. We report a case in a 26-year-old woman with granular mass obstructing bilateral nasal cavities and causing breathing difficulty. Histopathological examination showed characteristic Mikulicz histiocytes containing numerous Gram-negative intracellular rod-shaped bacilli consistent with the diagnosis of rhinoscleroma. The patient was treated with gemifloxacin and tetracycline and remains asymptomatic over a year follow-up period. It is important to consider rhinoscleroma in cases of chronic nasal obstruction. As culture is not always positive, histopathological examination may be crucial to the diagnosis.

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Rhinoscleroma is a chronic, progressive, granulomatous disease resulting from infection with Klebsiella rhinoscleromatis. It is generally uncommon, but endemic in certain regions of the world. It typically affects the nasal cavity, but also other parts of the respiratory system, such as the laryngotracheal region, which may be life-threatening if compromised. We present a 46-year-old male patient with a history of rhinoscleroma diagnosed and treated 15 years ago, with subglottal stenosis as a late sequela of this disease.

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La infección por Klebsiella rinoescleromatis es una condición infrecuente en nuestro país, pero endémica en ciertas regiones del mundo. Se presenta como una entidad crónica, progresiva y de aspecto granulomatoso. Causa típicamente rinoescleroma, pero puede comprometer otras partes del sistema respiratorio. Especial atención requiere el compromiso laríngeo y traqueal pues puede representar un riesgo para la vida del paciente. Su diagnóstico se basa en hallazgos como deformidad nasal u obstrucción de la vía aérea con disnea, disfonía y estridor respiratorio. Se apoya en técnicas de imagen como la tomografía computada y la resonancia magnética, pero el diagnóstico confirmatorio lo entrega la histopatología. Su tratamiento incluye el uso de antibióticos y cirugía, la que se puede realizar por diversas técnicas. En suma, es una enfermedad que requiere un alto grado de sospecha y se debe tener en consideración actualmente ante la inmigración a nuestro medio desde regiones endémicas cercanas.

Klebsiella Rhinoscleromatis infection is a rare condition in our country, but is endemic in certain regions of the world. It is presented as a chronic entity, progressive and granulomatous aspect.Typically cause rhinoscleroma, but may also compromise other parts of the respiratory tract. Special attention requires the laryngeal and tracheal compromise therefore may pose a risk to the patient’s life. Its diagnosis is based on the clinical findings like nasal deformity or obstruction of the airway with dyspnea, dysphonia and stridor. It relies on imaging techniques such as computed tomography and magnetic resonance imaging, but the diagnosis is confirmed by histopathology. Its treatment includes antibiotics and surgery, which can be achieved by various techniques. In short, is a disease that requires a high degree of suspicion and should be taken into consideration now before the immigration to our country from nearly endemic regions.

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Introdução: O rinoscleroma é uma doença nasal granulomatosa rara, causada pela bactéria klebsiella rhinoscleromatis. O nariz representa a região primária de ocorrência. O linfoma não-Hodgkin nasal é uma rara neoplasia maligna, podendo ser do tipo T ou B. O rinoscleroma e o linfoma nasal não-Hodgkin apresentam como principal sintoma a obstrução nasal, e fazem parte do diagnóstico diferencial das granulomatoses nasais. Objetivo: Relatar a associação de rinoscleroma e linfoma não-Hodgkin nasal em um mesmo paciente, destacando a importância do diagnóstico diferencial das granulomatoses nasais. Relato do Caso: Paciente do sexo feminino, 49 anos, atendida com obstrução nasal progressiva há um mês. A rinoscopia evidenciava lesão aspecto papilomatoso em meato médio esquerdo. A imunohistoquímica confirmou rinoscleroma. A paciente foi corretamente tratada com remissão total da lesão. Dez meses após retornou com mesmos sintomas, mas histologia confirmou linfoma não-Hodgkin. Comentários Finais: Ambas as patologias podem causar sintomas nasais graves e complicações. O diagnóstico precoce possibilita a instituição do tratamento adequado e favorece o prognóstico. O estudo imunohistoquímico foi fundamental para a diferenciação diagnóstica.

Introduction: Rhinoscleroma, a rare nasal granulomatous disease, is caused by Klebsiella rhinoscleromatis. The nose is the primary occurrence region. Nasal non-Hodgkin lymphoma is a rare cancer, and could be of T or B type. The rhinoscleroma and the nasal non-Hodgkin lymphoma present with nasal obstruction as the main symptom, and are part of the nasal granulomatosis differential diagnosis. Objective: To report the association of rhinoscleroma and non-Hodgkin lymphoma in the same patient, by remarking the importance of the nasal granulomatosis' differential diagnosis. Case Report: A forty-nine year old female patient that appeared with a one-month progressive nasal obstruction. Rhinoscopy showed papillomatous feature lesion in left middle meatus. The immunohistochemical analysis confirmed rhinoscleroma. The patient was duly treated with total remission of the lesion. Ten months after, she returned with the same symptoms, but the histologic study confirmed non-Hodgkin lymphoma. Final Comments: Both pathologies may cause more severe nasal symptoms and complications. The early diagnostic enables the choice for the right treatment and contributes for the prognosis. The immunohistochemical study was essential for the diagnostic differentiation.

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Se presenta un caso de rinoescleroma de la vía aérea superior e inferior, recidivante, de 20 años de volución, persistente a pesar de múltiples tratamientos. Las calcificaciones del árbol traqueobronquial que presenta plantearon un diagnóstico diferencial con traqueobroncopatía osteocondroplástica. En el artículo se revisan los hallazgos anatomopatológicos y radiológicos relevantes de estas patologías.

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