Retrospective Study of the Rhinoscleroma About 14 Cases in ENT Departments of University Hospitals (Côte D’Ivoire)

European Annals of Otorhinolaryngology, Head and Neck diseases (2011) 128, 7—10

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ORIGINAL CLINICAL RESEARCH Retrospective study of the rhinoscleroma about 14 cases in ENT departments of university hospitals (Côte d’Ivoire)

K.V. N’gattia a,∗, N. Kacouchia a, L. Kofﬁ-N’guessan b, N.M. Mobio c, J. Kouassi-Ndjeundo a, M. Kouassi d,M.Yodad, T.S. Vroh Bi a

a Service ORL et chirurgie cervico-faciale, CHU de Bouaké, 22 BP 1438 Abidjan 22, Côte d’Ivoire b Service ORL et chirurgie cervico-faciale, CHU de Cocody, Abidjan, Côte d’Ivoire c Service ORL et chirurgie cervico-faciale, CHU de Treichville, Abidjan, Côte d’Ivoire d Service ORL et chirurgie cervico-faciale, CHU de Yopougon, Abidjan, Côte d’Ivoire

Available online 20 January 2011

KEYWORDS Summary Rhinoscleroma; Purpose of study: To report the epidemiological, clinical, therapeutic and evolutionary aspects Nasal tumor; of rhinoscleroma in ENT departments of university hospitals (Côte d’Ivoire). Medical and surgical Material and method: Retrospective study of rhinoscleroma conducted in the ENT and head treatment and neck surgery departments in Côte d’Ivoire from January 1980 to December 2008 including the cases of confirmed rhinoscleroma and the treated cases. Results: Fourteen cases of rhinoscleroma were found in 28 years. The early manifestations were not specific enough so the patients were seen with clinical status with obvious disorders or physical discomfort. Treatment was medical and surgical. Medical therapy was based on streptomycin, thiopenicol or ciprofloxacin administration. Surgery consisted in removing the fibrous adhesions to correct the functional and aesthetic disorders. Conclusion: Rhinoscleroma has become a more and more rare disease because of the sen- sitivity to the new molecules. Diagnosis can be difficult and delayed because of its clinical polymorphism. © 2010 Elsevier Masson SAS. All rights reserved.

Introduction

Scleroma or rhinoscleroma is a chronic pathology of the

∗ respiratory tract caused by Klebsiella rhinoscleromatis.It Corresponding author. Tel.: +00 225 07 85 99 55; most commonly affects the nasal cavity in more than 95% fax: +00 225 22 44 63 53. of the cases [1—2] (hence the term of rhinoscleroma). It is E-mail address: [email protected] (K.V. N’gattia).

cases among which one where the nasal signs had appeared Table 1 Distribution of patients according to the year. a long time after the laryngeal signs), trachea (one case) Number of patients Percentage % and to oral cavity (one case). Physical examination evidenced a morphological alte- 1980 — 1989 9 64.3 ration of the nose: flattened nose and radix with an aspect 1990 — 2000 3 21.4 of saddle nose (five cases), bumpy, pinched nose or broad 2001 — 2008 2 14.3 based nose (five cases), upper lip invasion (one case). Pal- Total amount 14 100 pation showed ligneous hardness of nasal ala in six cases. In anterior rhinoscopy, the external nasal valve was imper- 64.3% of the patients were seen between 1980 and 1990. meable (seven cases) with partial nasal obstruction in four cases with plaques of inflamed tissue, nodules or sclero- characterized by a specific spreading granulomapous condi- sis. In three cases, rhinoscopic examination evidenced a tion with pseudotumoral process then fibrosis evolution. The thickening of the nasal mucosa with dry (two cases) or oo- result is a progressive obstruction of the respiratory tract zing (one case) granulomatous-crusted secretions. Posterior that can lead to aesthetic and functional prejudices. This rhinoscopy demonstrated hypertrophy of the torus tubarius pathology is rarely encountered in our ENT departments. Its with subtotal choanae obstruction in three cases. Examina- early diagnosis is difficult to establish and long-term treat- tion of oral cavity and nasopharynx revealed right endolabial ment is necessary. We report our experience management granulations (one case) on the uvula (four cases), the pil- of this pathology about 14 cases. lars of soft palate and palatine tonsils (two cases), the right hemipalate (one case) and the larynx (three cases Material and method among which two in the glottic region and one in the supra and subglottic region). The diagnosis of rhinoscleroma was established after the histological examination of biopsy sam- This retrospective study was conducted in the ENT and ples showing Mikulicz cells (13 cases) and Russel bodies (one head and neck surgery departments in Treichville, Cocody, case). Yopougon and Bouaké from January 1980 to December Evaluation of extension of the disease with air sinus 2008. All the patients treated for rhinoscleroma with com- radiography (Waters’ view) revealed an opacity of at least plete medical records (clinical examination, histological one sinus in nine cases. On posterior-anterior and lateral confirmation and treatment) were included in the study. views, a tracheal and laryngeal intraluminal opacity sug- The non-included patients had incomplete medical records. gested spreading of scleroma to trachea (one case). Nasal Thus, we retained 14 complete medical records out of 97 650 and paranasal sinus CT Scan realized in three cases, evi- consultations over this period of time that is 0.01% of the denced a bulky expanding firm soft process in the nasal fossa patients. (three cases) with erosion and harmonious deformity of the From 2000 onwards, Medical treatment was antibio- medial wall of the right and left maxillary sinuses causing therapy associated with corticoids: streptomycin (1 g/d) or bilateral collapse of the maxillary sinus due to the tumor thiophenicol (1 g/d) between 1980 and 1999 associated with mass effect (one case) (Fig. 1). betamethasone (1 mg/g per day) and ciprofloxacin/250 mg Medical treatment was based on the association of antibi- one tablet twice daily) surgical treatment was required otics and corticoids: streptomycin (10 cases) in patients in cases of obstructive granuloma and/or sequelae (unaes- showing no inner ear impairment with regular follow-up thetic deformities of the nasal pyramid). through pure-tone audiometry until the stabilization of Results

In 28 years, 14 cases of rhinoscleroma were studied in the ENT departments of the four university hospitals in the country (Table 1) among which nine cases (64.3%) between 1980 and 1990. The study included nine male and five female patients aged from 16 to 81. Mean age was 33.4. The population included nine natives of Côte d’Ivoire and five foreigners among whom two natives of Burkina Faso, two Malians and one Guinean. Among the natives of Côte d’Ivoire, five were originally from the north of the country. Patients’ monthly income was below guaranteed minimum income (GMI). The delay in consultation varied between 1 month and 8 years with an average of 2 years. The patients consulted for nasal tumefaction (11 cases), permanent nasal obstruction (seven cases), endobuccal tumefaction (one case), dysphonia (three cases), mouth breathing (three cases). These signs were associated in some patients. Figure 1 Computed tomography of paranasal sinuses eviden- In 11 cases, the disease which first involved the nasal cing nasal fossa tumor (rhinoscleroma) with bilateral collapse septum spread to the pharynx (six cases), the larynx (three of maxillary sinuses. Retrospective study of the rhinoscleroma about 14 cases 9 the lesions; thiophenicol in one 81-year-old patient pre- tissues...). Bacillus is isolated in 98% of the cases by granu- senting with sensorineural hearing loss. As early as 2000, lation tissue biopsy and, in 60% of the cases by smears from ciprofloxacine was used in three patients. Treatment dura- lesions [1]. Bacteriological study with culture for antibio- tion was from 6 months to 1 year. Surgical management therapy was realized in several series [8,10] and only 50 to completed medical treatment in two cases. In the first case, 60% of the cultures are positive for klebsiella rhinosclero- we performed the removal of residual scar tissue lesions of matis [2]. The confirmation in our series was made by the the upper lip, desobstruction of nasal fossa and calibrating histopathological examination. Spread and repercussion of with tubes after nasal septum plasty with bone graft taken rhinoscleroma on paranasal sinuses and osteocartilaginous from a rib in order to lift up the nasal dorsum. Six-month skeleton will be at best demonstrated by CT-Scan and/or MRI follow-up was satisfactory; then, the patient was lost to or at least by Waters view [7,11,12]. Initial exploration with follow-up. Removal of nasal fibrosis with nasal fossa cali- tomography, flexible fiber-optics and/or endoscopy must brating was realized in the second case. The patient was look for other localizations as well (especially laryngeal and reviewed 2 years after surgery and there was no recurrence. thoracic) often unnoticed [13,14]. Differential clinical diagnosis at early stage is common rhinitis. At tumoral stage diagnoses of leprosy, tertiary Comments syphilis, cutaneous tuberculosis, sarcoidosis and wegener granulomatosis must be ruled out; when scar forma- Rhinoscleroma endemic regions are well-known: Tropical tion process occurs, destructive mycosis, mucocutaneous Africa, the Maghreb, South East Asia, Mexico, Central and leishmaniasis on scar tissues and ozena must be evoked South America, Eastern and Central Europe [1—2]. Migra- [1,3]. Histopathological diagnosis of rhinoscleroma must tion of population currently causes more and more sporadic be distinguished from the diagnosis of cutaneous lep- cases in regions spared before then by this disease [3—4]. rosy, syphilis, tuberculosis, mucocutaneous leishmaniasis, Mean age of occurrence is between 15 and 35 years [5] paracoccidioidomycosis, rhinosporidiosis and wegener gran- as in our study: 33.4 years. Rhinoscleroma is rare in Côte ulomatosis [1]. For Abalkhail et al., histologic differential d’Ivoire: 14 cases in 28 years that is an average of one case diagnosis of rhinoscleroma at proliferative stage includes every 2 years. It is important to note that we observe less cutaneous leprosy, Malakoplakia, tumors with granuloma- and less cases in our departments (nine cases between 1980 tous cells and metastatic cells of renal carcinoma [15]. and 1990, three cases from 1991 to 2000 and two cases Medical treatment must eradicate the germ in focus, sta- from 2001 to December 2008). The majority of our patients bilize or partially reduce the lesions and avoid functional were natives of the northern part of the country bordered by or aesthetic complications. It was based on streptomycin, endemic countries such as Burkina Faso [3]; the patients who rifampicin, sulfamides and clofazimine [1]. Fluoroquinolone are not originally from Côte d’Ivoire are natives of Burkina efficacy was demonstrated in this therapeutical manage- Faso, Mali and Guinea. Socioeconomic level of the patients ment [11,16,17]. This could account for the significant is low (monthly income below GMI). Predisposing factors are decreasing number of cases of rhinoscleroma encountered malnutrition, poor hygiene and crowded living conditions in our consultations over the years. These fluoroquinolones [1]. Rhinoscleroma is the ‘‘pathology of poverty’’ [3]. were successfully administered in three of our patients. Pathology evolution is divided into four stages: catarrhal Surgery combined with antibiotic therapy consisted in rhinitis, atrophic granulomatous rhinitis, infiltrative rhinitis repermeabilization of nasal fossa associated with either sep- and sclerotic rhinitis [1]. Most of these changes contribut- toplasty or rhinoseptoplasty according to the case. These ing to the chronicity of rhinoscleroma occurs in the sube- procedures ensured the improvement of the aesthetic and pithelium tissue during proliferative phase of the disease. functional comfort in these patients. In cases with pala- These critical changes include factors leading to histiocyte tine localization, uvulopalatopharyngoplasty could relieve transformation into Mikulicz cells, the inability of these the nasopharyngeal symptoms [18]. Treatment that requires cells to uniformly destroy Klebsiella, their rupture liberating long duration of antibiotic therapy makes the patients’ viable Klebsiella and the intrinsic resistance of the pathogen follow-up often difficult. The majority of our patients was microbe [6]. Rhinoscleroma evolution is often insidious and lost-to-follow-up sometimes at the onset of stabilization of the patients ask for medical advice only at obstructive gra- the lesions. nulomatous stage with nasal obstruction. Delay in diagnosis In the series of Ouoba et al., out of 51 patients with and treatment promoted the local regional propagation of rhinoscleroma, only 13 were came back to consultation 5 rhinoscleroma in 11 cases. Nasal localization is predominant years after treatment [3]. These authors report two cases in our series as in the literature [2,3,6] but the affection of relapse [3]. can spread to the entire bronchopulmonary tree and to the bronchi [1,8]. After the main site of infection (nasal fossa), rhinoscleroma can involve the nasopharynx (18—43%), the Conclusion larynx (15—40%), the trachea (12%) and the bronchi (from 2 to 7%) [7]. In Côte d’Ivoire, rhinoscleroma has become more and more The clinical presentation is non-specific but any granulo- uncommon in our consultations. Efficacy of fluoroquinolones matous rhinitis in this context must evoke the possibility in the treatment of this disease as well as their frequent use of rhinoscleroma with oriented histological examination certainly accounts for this phenomenon. Its clinical polymor- [9]. Histopathological examination is looking for Mikulicz’s phism makes its diagnosis difficult to establish. Diagnosis cells that are pathognomonic for klebsiella rhinoscleroma- is often delayed and made at tumoral stage, then granulo- tis (examination of smears, nasal secretions, pathological matosis and even nasal fossa carcinoma must also be evoked. 10 K.V. N’gattia et al.

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