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PEDOT-7494; No. of Pages 3 International Journal of Pediatric Otorhinolaryngology xxx (2015) xxx–xxx

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Case report

Pediatric atrophic rhinosinusitis: What can we do? Clara Magalha˜es a,*, Miguel Viana a, Valquı´ria Alves b, Roberto Nakamura a, Delfim Duarte a a b

Department of Otolaryngology, Pedro Hispano Hospital, Matosinhos, Portugal Department of Pathology, Pedro Hispano Hospital, Matosinhos, Portugal

A R T I C L E I N F O

A B S T R A C T

Article history: Received 8 December 2014 Received in revised form 7 February 2015 Accepted 25 February 2015 Available online xxx

A 5-year-old female had history of chronic foul smelling nasal discharge. Rhinoscopy showed greenish crusts lining the nasal cavities and inferior turbinates were shriveled significantly. Nasal cavity cultures of crusts by swab revealed Klebsiella ozaenae making the diagnosis of primary atrophic rhinosinusitis. After several unsuccessful treatment, we have decided to try sulfamethoxazole–trimethoprim prophylaxis and 1 year later there was a complete clinical improvement. There are many medical therapies and surgical options described, but none of them showed effective at long term. We present antibiotic prophylaxis as a viable alternative for long term control of the disease. ß 2015 Elsevier Ireland Ltd. All rights reserved.

Keywords: Atrophic rhinosinusitis Chronic disease Child

Case report The authors present the case of a 5 years old girl, with complaints of nasal obstruction and chronic, purulent and fetid rhinorrhea. Anterior rhinoscopy showed greenish crusts filling up the nasal cavities (Fig. 1), and atrophy of the inferior turbinate and enlargement of the nasal cavity after removing crusts (Fig. 2). The swab of nasal mucosa and the culture of crusts showed colonization for Klebsiella ozaenae (Fig. 3) and the biopsy of inferior turbinate squamous metaplasia. Enlargement of the nasal cavities, atrophy of the middle and inferior turbinates, hypoplasia of the maxillary sinus and ethmoid-maxillary sinusitis was found in sinuses CT scan (Fig. 4). The remaining complementary study was negative and included: analytical study [CBC to study the iron (total iron, ferritin, transferrin), biochemical (glucose, renal function, liver function and ionogram), immunological study (ANCA/ANA and IgE, IgA, IgM, C3 and C4), vitamins A and D, serological study (VDRL, HIV, HCV, HBV, HAV), sweat test and chest X-ray. Medical therapy started with saline nasal washes and the application of wet gel (5 drops per day), frequent cleaning of nasal crusts, daily nebulization of gentamicin (60 mg/dose diluted with 4 mL) for three weeks, amoxicillin with acid clavulanic (50 mg/kg/ day 2 id) for twelve weeks and oral multivitamin supplements.

* Corresponding author. Tel.: +00 351 229391000. E-mail address: [email protected] (C. Magalha˜es).

The nasal crusts decreased significantly during treatment, as well as rhinorrhea and foul odor. Nasal crusting and foul odor relapsed three weeks after discontinuation of therapy. After this cycle we established a new treatment with nebulization of gentamicin (60 mg/dose diluted with 4 mL) for 15 days, amoxicillin–clavulanic acid (50 mg/kg/day 2 id) for one month, and nasal irrigation with sodium bicarbonate solution and application of nasal drops, with solution of glucose with glycerin. The sodium bicarbonate solution was obtained by dissolving 28.4 g of sodium bicarbonate, 28.4 g of sodium diborato and 56.7 g of sodium chloride in 280 ml of water. This solution was administered three times a day, using 10 cm3 syringe. After this cycle was started prophylaxis with trimethoprim–sulfamethoxazole (150 mg/m2/ day trimethoprim and 750 mg/m2/day sulfamethoxazole) for 3 days per week. After 6 months of prophylaxis and 1 year of follow-up only with irrigations of saline water the fetid odor and rhinorrhea disappeared, and no acute infection occur. In anterior rhinoscopy the nasal crusts was cleared. Discussion The diagnostic evaluation of primary atrophic rhinosinusitis results of clinical suspicion in a patient with chronic, purulent and fetid rhinorrhea associated with nasal obstruction and crusts filling nasal cavities, refractory to medical therapy. This suspicion is confirmed by culture of biopsy of the nasal mucosa or picking scabs insulated Klebsiella ozenae. Several articles on microbiology in atrophic rhinosinusitis indicate that the presence of Klebsiella

http://dx.doi.org/10.1016/j.ijporl.2015.02.035 0165-5876/ß 2015 Elsevier Ireland Ltd. All rights reserved.

Please cite this article in press as: C. Magalha˜es, et al., Pediatric atrophic rhinosinusitis: What can we do? Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.02.035

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Fig. 1. Nasal endoscopy showed greenish crusts filling up the nasal cavities.

Fig. 2. Nasal endoscopy after removing crusts showed atrophy of the inferior and middle turbinates with enlargement of the nasal cavity.

ozaenae is not required for diagnosis, and other bacteria, and even saprophytic colonization by fungi may have a role in the persistence of symptoms [1]. In our case, the anatomical changes such as hypoplasia of the maxillary sinus and the enlargement of the nasal cavities in physical examination and confirmed by CT determines a predisposition in this entity and a congenital etiology.

Regarding the therapeutic approach, this is empirical and there are no randomized studies to prove the effectiveness and compare the treatments used [1]. Although there exists no generally recommended specific therapy, the irrigation with saline, hypertonic or isotonic solution acting as solvent seems to be safe treatment option. Irrigation with saline solution acts as cleaning the nasal crusts [1]. There are several solutions for nasal application. The bicarbonate solution described by the authors is an ideal mixture, containing sodium bicarbonate, which helps in dissolving crusts, diborato sodium, which acts as an antiseptic, antibacterial and helps in dissolution of the baking, while the sodium chloride isotonic solution turns the solution in isotonic [2]. Nose drops of glycerin and glucose inhibit saprophytic infection: glucose by fermentation produce lactic acid and an acidic pH which inhibits bacterial growth, and glycerin helps lubricate the mucosa and increases vascularisation [2]. The washing solutions containing antibiotics, as well as in the form of nebulization are recommended in cases of acute infection, and appear to provide a significant concentration of antibiotic directly to the nasal cavity [3]. As the use of, oral antibiotics may be used in cases of acute and systemic infection, generally quinolones or rifampin, although no evidence for this practice was found [3]. In the case described antibiotic selection was made on the basis of susceptibility test, fewer side effects and safer (topical) route. Gentamicin intranasally showed to be safe, as it has good tolerance and is not absorbed by the nasal mucosa [4]. Postreatment audiogram showed a normal hearing. Due to the need for a prolonged treatment period, amoxicillin with clavulanic acid was chosen for its susceptibility profile and fewer side effects. In the literature review we did not find reference to the use of antibiotic prophylaxis in primary atrophic rhinosinusitis, having opted for this after discussing the case with Infectious Diseases Department. At this time and after 1 year of follow-up only with saline irrigations and 6 months of prophylaxis with trimethoprim– sulfamethoxazole there is no evidence disease recurrence or new infectious complications. Surgery is only proposed in case of treatment failure and severe symptoms, and is not currently recommended [1]. There are several techniques that allow the partial or complete closure of the nostrils using autogenous or synthetic implants. The first autologous implant used was fat, which has the inconvenience that it can be resorbed. More recently there is reference to surgical techniques using sublabially parietal bone and rib cartilage [5]. There is no experience of these techniques in children, and it is unclear what is the relationship to cranio-facial development, given the importance of nasal breathing in this development.

Fig. 3. The swab of nasal mucosa and the culture of crusts showed colonization for Klebsiella ozaenae.

Please cite this article in press as: C. Magalha˜es, et al., Pediatric atrophic rhinosinusitis: What can we do? Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.02.035

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Fig. 4. Sinuses CT scan showed enlargement of the nasal cavities, atrophy of the middle and inferior turbinates, hypoplasia of the maxillary sinus and ethmoid-maxillary sinusitis.

In our case the antibiotic prophylaxis seems to help in long term control of the disease. We have to think about the susceptibility of Klebsiella ozaenae to sulfamethoxazole–trimethoprim similar to the use of these antibiotics in vesicoureteral reflux. Conclusion We present a case of this rare disease and point antibiotic prophylaxis as a possible alternative for long term control.

References [1] R. De Shazo, S. Stringer, J. Corren, A. Feldweg, Atrophic rhinosinusitis, in: Up to date, 2012. [2] S.N. Dutt, M. Kamesharan, The aetiology and management of atrophic rhinitis, J. Laryngol. Otol. 119 (November) (2005) 843–852. [3] K.A. Elliot, S.P. Stringer, Evidence-based recommendations of antimicrobial nasal washes in rhinossinusitis, Am. J. Rhinol. 20 (2006) 1–6. [4] P. James, M.D. Duddley, Atrophic rhinitis: antibiotic treatment, Am. J. Otolaryngol. 8 (1987) 387–390. [5] Pinto D. Vidigal, M. Paiva, F. Werkema, Surgical treatment of atrophic rhinitis: a new technique, Otolaryngol. Tech. 3 (2008) 7–10.

Please cite this article in press as: C. Magalha˜es, et al., Pediatric atrophic rhinosinusitis: What can we do? Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.02.035

Pediatric atrophic rhinosinusitis: what can we do?

A 5-year-old female had history of chronic foul smelling nasal discharge. Rhinoscopy showed greenish crusts lining the nasal cavities and inferior tur...
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