• View in gallery

    A, Lesion on the ear of the patient before any treatment had been administered. B, After two courses of meglumine antimoniate and several courses of dicloxacillin and pristinamycin but before the administration of ciprofloxacin. C, After administration of ciprofloxacin at the end of follow-up. D, Controlateral ear as a control. This figure appears in color at www.ajtmh.org.

  • View in gallery

    A, Typical extracellular (arrows) and intracellular (star) amastigotes on a Giemsa-stained smear from the initial ulceration of the patient (magnification × 2,000). B, Histopathologic examination of an ear biopsy specimen at the time of abscess maximal development showed a dense dermal monocellular and polymorphonuclear infiltrate invading the superficial layers of the cartilage, without amastigotes (magnification × 100). This figure appears in color at www.ajtmh.org.

  • 1

    Andrade-Narvaez FJ, Vargas-Gonzalez A, Canto-Lara SB, Damian-Centeno AG, 2001. Clinical picture of cutaneous leishmaniases due to Leishmania (Leishmania) mexicana in the Yucatan peninsula, Mexico. Mem Inst Oswaldo Cruz 96 :163–167.

    • Search Google Scholar
    • Export Citation
  • 2

    Vargas-Gonzalez A, Canto-Lara SB, Damian-Centeno AG, Andrade-Narvaez FJ, 1999. Response of cutaneous leishmaniasis (chiclero’s ulcer) to treatment with meglumine antimoniate in southeast Mexico. Am J Trop Med Hyg 61 :960–963.

    • Search Google Scholar
    • Export Citation
  • 3

    Edrissian GH, Mohammadi M, Kanani A, Afshar A, Hafezi R, Ghorbani M, Gharagozloo AR, 1990. Bacterial infections in suspected cutaneous leishmaniasis lesions. Bull World Health Organ 68 :473–477.

    • Search Google Scholar
    • Export Citation
  • 4

    Vera LA, Santos JB, Macedo VO, de Magalhaes AV, Ciuffo IA, Santos CG, 2001. Evaluation of secondary bacterial infection’s influence on the course of cutaneous leishmaniasis in Corte de Pedra. Bahia. Rev Soc Bras Med Trop 34 :233–237.

    • Search Google Scholar
    • Export Citation
  • 5

    O’Meara SM, Cullum NA, Majid M, Sheldon TA, 2001. Systematic review of antimicrobial agents used for chronic wounds. Br J Surg 88 :4–21.

    • Search Google Scholar
    • Export Citation
  • 6

    Margulis A, Bauer BS, Alizadeh K, 2003. Ear reconstruction after auricular chondritis secondary to ear piercing. Plast Reconstr Surg 111 :891–897.

    • Search Google Scholar
    • Export Citation
  • 7

    Mills DC II, Roberts LW, Mason AD Jr, McManus WF, Pruitt BA Jr, 1988. Suppurative chondritis: its incidence, prevention, and treatment in burn patients. Plast Reconstr Surg 82 :267–276.

    • Search Google Scholar
    • Export Citation
  • 8

    Bentrem DJ, Bill TJ, Himel HN, Edlich RF, 1996. Chondritis of the ear: a late sequela of deep partial thickness burns of the face. J Emerg Med 14 :469–471.

    • Search Google Scholar
    • Export Citation
  • 9

    More DR, Seidel JS, Bryan PA, 1999. Ear-piercing techniques as a cause of auricular chondritis. Pediatr Emerg Care 15 :189–192.

  • 10

    Doroghazi RM, Nadol JB Jr, Hyslop NE Jr, Baker AS, Axelrod L, 1981. Invasive external otitis. Report of 21 cases and review of the literature. Am J Med 71 :603–614.

    • Search Google Scholar
    • Export Citation
  • 11

    Keene WE, Markum AC, Samadpour M, 2004. Outbreak of Pseudomonas aeruginosa infections caused by commercial piercing of upper ear cartilage. JAMA 291 :981–985.

    • Search Google Scholar
    • Export Citation
  • 12

    Caksen H, Odabas D, 2002. Perforation of the nasal septum and palatum durum in a severely malnourished infant. Pediatr Dermatol 19 :89–90.

    • Search Google Scholar
    • Export Citation
  • 13

    Marsden PD, 1986. Mucosal leishmaniasis (“espundia” Escomel, 1911). Trans R Soc Trop Med Hyg 80 :859–876.

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

PSEUDOMONAS AERUGINOSA OTOCHONDRITIS COMPLICATING LOCALIZED CUTANEOUS LEISHMANIASIS: PREVENTION OF MUTILATION BY EARLY ANTIBIOTIC THERAPY

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  • 1 Institut Pasteur Medical Center, Paris, France, Department of Pathology, Dermatology Department, and Infectious Diseases Department, Necker Hospital, Assistance-Publique, Hôpitaux de Paris, Paris, France

A patient with an ulcerated cutaneous leishmaniasis of the pinna had suppurative otochondritis after a first unsuccessful course of treatment with meglumine antimoniate. Although the Leishmania ulceration healed after a second course of meglumine antimoniate, and despite three oral dicloxacillin or pristinamycin courses, the otochondritis extended and an abscess developed. Pus from the abscess revealed a pure culture of Pseudomonas aeruginosa. Five days of oral ciprofloxacin plus rifampin led to a marked improvement. The P. aeruginosa isolate was sensitive to ciprofloxacin but fully resistant to rifampin. Healing with minimal mutilation was obtained at the end of a six-week course of multiple antibiotic therapy. Pseudomonas aeruginosa otochondritis was a co-factor of cartilage mutilation in this patient. Thus, infection with P. aeruginosa should be promptly treated when present in tender cutaneous or mucosal leishmaniasis lesions near cartilaginous areas.

Localized cutaneous leishmaniasis (LCL) of the ear is characterized by a single ulceration without mucosal or lymphatic involvement.1 When progressing on the ear, LCL often involves cartilagenous structures, spontaneously lasts for months to years, and may cause severe mutilation.2 This case report suggests that Pseudomonas aeruginosa secondary infection of the cartilaginous structures may be a co-factor of mutilation.

CASE REPORT

A 28-year-old immunocompetent man returning from Brazil and Bolivia sought medical treatment for ulcerated cutaneous lesions of the right forearm and the pinna of the right ear (Figure 1A). Smears showed typical amastigotes (Figure 2A). Culture was not performed. Intramuscular meglumine antimoniate (Glucantime), 20 mg of pentavalent antimony/ kg/day for 13 days and two intralesional injections in the lesion in the forearm resulted in complete reepithelialization on the forearm but no significant improvement of the pinna lesion.

Two months later, pain, erythema, and swelling occurred on the pinna. A second intramuscular treatment (20 mg of pentavalent antimony/kg/day for 20 days) and two weekly intralesional injections of meglumine antimoniate led to complete reepithelialization of the ulcer on the pinna. However, three 8-day oral courses of dicloxacillin and pristinamycin failed at to control a local abscess that had developed on the pinna (before any intralesional injection had been performed, Figure 1B).

A full-thickness biopsy specimen showed otochondritis without amastigotes (Figure 2B). Two drainages did not control the abscess. Culture of the material drained from the abscess showed a pure culture of P. aeruginosa. We prescribed oral rifampin plus ciprofloxacin. One week later, suppuration had stopped and the pain had been consistently attenuated. Since an antibiogram showed a P. aeruginosa strain fully resistant to rifampin, but sensitive to ceftazidim, tobramycin, and fluoroquinolones, a course of intravenous ceftazidime and tobramycin plus oral ciprofloxacin was administered. After three weeks, residual inflammation was moderate but failed to resolve completely, and the administration of ciprofloxacine alone for three weeks was required to achieve complete healing. The final clinical evaluation six months later showed a minor ear deformity (Figure 1C and D).

DISCUSSION

Bacterial colonization (mainly due to staphylococci and streptococci) occurs in 25–55% of patients with open cutaneous leishmaniasis ulcers. In other reports, no abscess was reported in more than 2,250 patients, and bacterial colonization did not impair the healing process.3,4 There is no good guidance on when and how to use antibiotics as adjunct therapy for LCL. Most physicians would add antibiotics (effective primarily on gram-positive bacteria) to the specific antileishmanial therapy only when pain and erythema are present. In chronic leg ulcers, these signs are an indication that the superficial bacterial colonization leads to dermal infection, with the latter requiring systemic antibiotics for cure.5 However, the situation of case we report extends beyond the control of dermal bacterial infection.

Pseudomonas aeruginosa chondritis may result in complete destruction of cartilage of the ear if treatment is delayed.6 This bacteria is the dominant cause of suppurative otochondritis in burn patients7,8 or after ear-piercing.9 The clinical signs of otochondritis may occur several weeks after the initial non-cartilagenous lesion healing.7 Pseudomonas aeruginosa also causes invasive external otitis,10 an additional indication for its associated potential to cause infection of the ear and contiguous cartilaginous structures. In both conditions, early adapted antibiotic therapy is usually efficient.10,11 In LCL lesions of the ear, the prevalence of P. aeruginosa is unknown. Bacterial colonization ≤ 31.6% was reported in 136 cases of LCL caused by Leishmania mexicana,1 including 57 (38.8%) auricular lesions, but bacteria identification was not provided.

The mechanisms of cartilage mutilation during LCL are speculative. In the case reported, P. aeruginosa had a predominant role in the pathogenesis of cartilage mutilation. The ulcer containing Leishmania was cured, but the bacterial abscess had worsened. Improvement was obtained only after the patient received quinolone antibiotics. Histopathologic examination of a cartilage biopsy specimen did not show Leishmania amastigotes. Because P. aeruginosa abscess of cartilagenous structures is a known cause of cartilage mutilation,6 we propose that P. aeruginosa entry and establishment in the cartilage of ears with ulcers caused by Leishmania might be a significant co-factor of cartilage destruction in ear leishmaniasis.

One may speculate that P. aeruginosa is also involved in nasal and palatal mutilations in mucosal leishmaniasis. A recent case report of perforation of the nasal septum and palatum durum caused by P. aeruginosa in a malnourished child12 and the acute inflammatory aspect of mutilating episodes complicating chronic mucosal leishmaniasis lesions support this hypothesis.13 Thus, P. aeruginosa infection should be promptly treated when present in tender cutaneous or mucosal leishmaniasis lesions near cartilaginous areas.

Figure 1.
Figure 1.

A, Lesion on the ear of the patient before any treatment had been administered. B, After two courses of meglumine antimoniate and several courses of dicloxacillin and pristinamycin but before the administration of ciprofloxacin. C, After administration of ciprofloxacin at the end of follow-up. D, Controlateral ear as a control. This figure appears in color at www.ajtmh.org.

Citation: The American Journal of Tropical Medicine and Hygiene Am J Trop Med Hyg 75, 2; 10.4269/ajtmh.2006.75.270

Figure 2.
Figure 2.

A, Typical extracellular (arrows) and intracellular (star) amastigotes on a Giemsa-stained smear from the initial ulceration of the patient (magnification × 2,000). B, Histopathologic examination of an ear biopsy specimen at the time of abscess maximal development showed a dense dermal monocellular and polymorphonuclear infiltrate invading the superficial layers of the cartilage, without amastigotes (magnification × 100). This figure appears in color at www.ajtmh.org.

Citation: The American Journal of Tropical Medicine and Hygiene Am J Trop Med Hyg 75, 2; 10.4269/ajtmh.2006.75.270

*

Address correspondence to Pierre A. Buffet, 211 Rue de Vaugirard, 75015 Paris, France. E-mail: pabuffet@pasteur.fr

Authors’ addresses: Diane van der Vliet, Anne-Sophie Le Guern, Herve Darie, and Pierre A. Buffet, Institut Pasteur Medical Center, Paris, France. Sylvie Freitag, Department of Pathology, Necker Hospital, Assistance-Publique, Hôpitaux de Paris, Paris, France. Nicolas Gounod, Dermatology Department, Necker Hospital, Assistance-Publique, Hôpitaux de Paris, Paris, France. Audrey Therby, Infectious Diseases Department, Necker Hospital, Assistance-Publique, Hôpitaux de Paris, Paris, France.

REFERENCES

  • 1

    Andrade-Narvaez FJ, Vargas-Gonzalez A, Canto-Lara SB, Damian-Centeno AG, 2001. Clinical picture of cutaneous leishmaniases due to Leishmania (Leishmania) mexicana in the Yucatan peninsula, Mexico. Mem Inst Oswaldo Cruz 96 :163–167.

    • Search Google Scholar
    • Export Citation
  • 2

    Vargas-Gonzalez A, Canto-Lara SB, Damian-Centeno AG, Andrade-Narvaez FJ, 1999. Response of cutaneous leishmaniasis (chiclero’s ulcer) to treatment with meglumine antimoniate in southeast Mexico. Am J Trop Med Hyg 61 :960–963.

    • Search Google Scholar
    • Export Citation
  • 3

    Edrissian GH, Mohammadi M, Kanani A, Afshar A, Hafezi R, Ghorbani M, Gharagozloo AR, 1990. Bacterial infections in suspected cutaneous leishmaniasis lesions. Bull World Health Organ 68 :473–477.

    • Search Google Scholar
    • Export Citation
  • 4

    Vera LA, Santos JB, Macedo VO, de Magalhaes AV, Ciuffo IA, Santos CG, 2001. Evaluation of secondary bacterial infection’s influence on the course of cutaneous leishmaniasis in Corte de Pedra. Bahia. Rev Soc Bras Med Trop 34 :233–237.

    • Search Google Scholar
    • Export Citation
  • 5

    O’Meara SM, Cullum NA, Majid M, Sheldon TA, 2001. Systematic review of antimicrobial agents used for chronic wounds. Br J Surg 88 :4–21.

    • Search Google Scholar
    • Export Citation
  • 6

    Margulis A, Bauer BS, Alizadeh K, 2003. Ear reconstruction after auricular chondritis secondary to ear piercing. Plast Reconstr Surg 111 :891–897.

    • Search Google Scholar
    • Export Citation
  • 7

    Mills DC II, Roberts LW, Mason AD Jr, McManus WF, Pruitt BA Jr, 1988. Suppurative chondritis: its incidence, prevention, and treatment in burn patients. Plast Reconstr Surg 82 :267–276.

    • Search Google Scholar
    • Export Citation
  • 8

    Bentrem DJ, Bill TJ, Himel HN, Edlich RF, 1996. Chondritis of the ear: a late sequela of deep partial thickness burns of the face. J Emerg Med 14 :469–471.

    • Search Google Scholar
    • Export Citation
  • 9

    More DR, Seidel JS, Bryan PA, 1999. Ear-piercing techniques as a cause of auricular chondritis. Pediatr Emerg Care 15 :189–192.

  • 10

    Doroghazi RM, Nadol JB Jr, Hyslop NE Jr, Baker AS, Axelrod L, 1981. Invasive external otitis. Report of 21 cases and review of the literature. Am J Med 71 :603–614.

    • Search Google Scholar
    • Export Citation
  • 11

    Keene WE, Markum AC, Samadpour M, 2004. Outbreak of Pseudomonas aeruginosa infections caused by commercial piercing of upper ear cartilage. JAMA 291 :981–985.

    • Search Google Scholar
    • Export Citation
  • 12

    Caksen H, Odabas D, 2002. Perforation of the nasal septum and palatum durum in a severely malnourished infant. Pediatr Dermatol 19 :89–90.

    • Search Google Scholar
    • Export Citation
  • 13

    Marsden PD, 1986. Mucosal leishmaniasis (“espundia” Escomel, 1911). Trans R Soc Trop Med Hyg 80 :859–876.

Author Notes

Reprint requests: Pierre A. Buffet, 211 Rue de Vaugirard, 75015 Paris, France, Telephone: 33-1-45-68-81-15, Fax: 33-1-40-61-30-19. E-mail: pabuffet@pasteur.fr.
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