Primary Cutaneous Mycobacterium kansasiiInfection in a Child

Antonio Chaves, M.D.,* Antonio Torrelo, M.D.,* Imelda G. Mediero, M.D.,†Mercedes Menendez-Rivas, M.D.,‡ Arturo Ortega-Calderon, M.D.,§ and

Antonio Zambrano, M.D.*

Departments of *Dermatology, †Pathology, and ‡Microbiology, Hospital del Nin˜o Jesus, and §Department ofMicrobiology, Hospital Carlos III, Madrid, Spain

Abstract: A 6-year-old girl with a primary cutaneous form of Mycobac-terium kansasii infection is presented. Disseminated infection and im-mune deficiency were excluded in our patient. She was successfullytreated with surgical excision and oral erythromycin. Primary cutaneousM. kansasii is an exceedingly rare infection in children.

Mycobacterium kansasiiis a slow-growing, photo-chromogenic mycobacterium (Runyon group I), first iso-lated in 1953 by Buhler and Pollak (1,2). It grows opti-mally at 37°C on Lowenstein–Jensen agar, and producesa yellow pigment when exposed to light. Water is thenatural habitat ofM. kansasii, including water supplies,swimming pools, and sewage. It has also been isolated incattle. M. kansasii is prevalent in the central UnitedStates, the United Kingdom, France, and Belgium. Inmost countries,M. kansasiirepresents less than 4% ofmycobacteria isolated, and it is considered the most com-mon atypical mycobacterium in AIDS afterM. aviumcomplex (3,4).M. kansasiihas been isolated in 2.9% and7% of U.S. and French AIDS patients, respectively (3,5).

CutaneousM. kansasiiinfections are rare. The firstcase was described by Mayberry et al. (6) in 1965, and anadditional 44 cases have been reported since then (7–33).Furthermore, none of these patients with cutaneousM.kansasiiinfection were children. We present a 6-year-oldgirl with primary skin infection byM. kansasii.

CASE REPORT

A 6-year-old girl developed a slow-growing lesion onher left elbow in August 1998. Shortly after, new lesions

appeared surrounding the first one. There was no clearantecedent trauma, wound, or exposure to raw milk. Shehad no other medical antecedents or risk factors for HIVinfection, and was previously in good health. Oral treat-ment with two antibiotics (amoxicillin and cloxacillin)was of no benefit. In 1999 she was sent to our departmentfor evaluation. On examination, the girl was afebrile, andwas in excellent general condition. A crop of erythema-tous, infiltrated, ill-defined nodules was seen on the leftelbow (Fig. 1). The surface of the lesions was rough andcovered by yellowish crusts, with some keratotic areas.The lymph nodes and liver were not enlarged, and theremainder of the clinical examination was normal.

A skin biopsy specimen from one nodule revealed ahyperplastic epidermis with irregular acanthosis. Thedermoepidermal junction was blurred, and an inflamma-tory infiltrate occupied the upper and middermis. Thisinfiltrate was granulomatous and consisted of numerousepithelioid cells, giant cells Langhans type, and lympho-cytes (Fig. 2). A Ziehl–Neelsen stain revealed the pres-ence of acid-fast bacilli (AFB). Cultures of fresh tissueon Lowenstein–Jensen medium were negative at first,and a PCR-based technique failed to identify mycobac-terial DNA. Subsequently the whole lesion was excised,and the histopathologic specimen showed results similar

Address correspondence to Antonio Torrelo, M.D., Department ofDermatology, Hospital del Nin˜o Jesu´s, Menendez Pelayo 65, 28009-Madrid, Spain, or e-mail: atorrelof@meditex.es.

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to those of the first specimen. Again, AFB were seen.Culture on Lowenstein–Jensen medium grew AFB,which were identified asM. kansasii.

A complete examination including hemogram, eryth-rocyte sedimentation rate, serum biochemical profile, se-rum immunoglobulins, blood lymphocyte populations,chest radiograph, HIV testing, tuberculin test, and cul-tures from sputum and gastric juice were all normal or

negative. Thus a generalized infection or immune defi-ciency was excluded. Although the surgical excision wascomplete, additional oral treatment with erythromycin 50mg/kg/day was begun. This therapy was stopped after 5months of treatment were completed. During this period,drug tolerance was good, and the skin lesions did notreappear.

DISCUSSION

Mycobacterium kansasiiinfections are sporadic, with noevidence of person-to-person transmission (1). They usu-ally take the form of a granulomatous pulmonary infec-tion, mainly in middle-aged men with underlying lungdisease such as chronic obstructive disease or pneumo-coniosis (1).M. kansasiimay also cause localized infec-tions such as cervical lymphadenopathy, meningitis, ten-donitis, synovitis, arthritis, osteomyelitis, carpal tunnelsyndrome, hepatic or splenic infection, and skin lesions.Furthermore, a disseminated infection may occur, espe-cially in immunocompromised hosts, involving many or-gans.

Cutaneous infection byM. kansasii is rare. To ourknowledge, 45 patients have been described, with agesranging from 16 to 84 years (6–33). In 72% of thosecases, an immunologic disorder resulting from chemo-therapy, autoimmune disease, AIDS, renal or cardiactransplantation, or other predisposing factors (14) wasidentified. In 28% of cases, the skin infection appears inimmunocompetent patients. In such instances, the modeof infection is exogenous inoculation following minimalcutaneous trauma, which has been demonstrated innearly two-thirds of the patients (32). Furthermore, asporotrichoid pattern of lymphatic dissemination hasbeen described (7,8,33). Finally, skin infection may ap-pear as a consequence of hematogenous dissemination ofM. kansasii, mainly in immunocompromised hosts. Inour patient there was no obvious portal of entry. How-ever, since the lesion appeared during summer, wespeculate that minimal skin trauma incurred at a swim-ming pool might have produced a route of entry forM.kansasiiinto the skin.

The clinical appearance of these skin lesions variesand has been reported as papules, nodules, pustules,crusted ulcers, cellulitis, sporotrichoid lesions, rhino-phyma-like lesions (17), and oral or perianal ulcers(10,31). The histologic pattern depends on the stage ofthe infection and the immune status of the host (15,21).Immunocompromised patients show neutrophilic inflam-matory infiltrates with occasional abscess formation. Inimmunocompetent hosts, a granulomatous infiltrate, withor without necrosis, is seen in more than 90% of cases.

The treatment of primary skin infections is controver-

Figure 1. A crop of erythematous, crusted papules andnodules on the left elbow.

Figure 2. Histology shows an epithelioid granulomatousdermal infiltrate, with giant cells. (Hematoxylin and eosin;original magnification 100×.)

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sial. The American Thoracic Society suggests that inadults a regimen similar to that for pulmonary diseaseshould be used: isoniazid (300 mg), rifampin (600 mg),and ethambutol (25 mg/kg for 2 months and then 15mg/kg) for 18 months, or at least 12 months after clear-ance of lesions (34). Addition of a fourth drug such asclarithromycin or azithromycin has been proposed (35).However, other authors have had success with singletherapy using minocycline (7) or erythromycin (13) forseveral months in immunocompetent patients. Surgi-cal excision has been performed in a single patient (20),with recurrence of the lesion. Most authors suggest thatantibiotic sensitivity tests be done, since resistance toantituberculous drugs, mainly isoniazid, is fairly com-mon (2).

Our case highlights the usual problems of manage-ment of atypical mycobacterial infections of the skin.Surgical and oral treatment with erythromycin has beensuccessful. Furthermore, difficulties are common inreaching a microbiologic diagnosis, and more than onesurgical specimen may be needed.

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