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Successful treatment of nodular lymphangitis due to in two immunosuppressed patients

  • Author(s): Schneider, Pierre
  • Monsel, Gentiane
  • Veziris, Nicolas
  • Roujeau, Jean-Claude
  • Bricaire, François
  • Caumes, Eric
  • et al.
Main Content

Successful treatment of nodular lymphangitis due to Mycobacterium chelonae in two immunosuppressed patients
Pierre Schneider1, Gentiane Monsel1, Nicolas Veziris2, Jean-Claude Roujeau3, François Bricaire1, Eric Caumes1
Dermatology Online Journal 17 (3): 8

1. Tropical and Infectious Diseases Department, Pitié-Salpétrière Hospital, Paris, France
2. Bacteriology department, Pitié-Salpétrière Hospital, Paris, France
3. Department of Dermatology, Henri-Mondor Hospital, Paris, France


Abstract

M. chelonae is a classical but uncommon etiology of nodular lymphangitis. We report 2 cases of nodular lymphangitis caused by M. chelonae occurring in immunocompromised hosts; both were cleared with antibiotic therapy.



Introduction

Nodular lymphangitis is a lymphocutaneous syndrome with multiple etiologies ranging from leishmaniasis to sporothrichosis. It may also be caused by atypical mycobacteria, mainly Mycobacterium marinum, but other mycobacterial species may be involved [1].

We describe two immunosuppressed patients with nodular lymphangitis related to M. chelonae were cleared with antibiotic therapy.


Case reports

Case 1. A 55-year-old woman was referred to our department for multiple erythematous-violaceous nodules located on her left arm. She had been treated 36 months previously for a right breast cancer by mastectomy and removal of the axillary lymph nodes. At admission she was receiving prednisone (40 mg/day), exemestane (5 mg/day), levetiracetam (500 mg twice/day), navelbine, and herceptine. The cutaneous eruption had started three months previously with an erythematous nodule on the left forearm, which subsequently opened to the skin with a fistula from which pus was discharging. Then other cutaneous lesions appeared on the same arm. There were no recent records of travel, aquarium cleaning, trauma, or any recent medical or surgical procedures (lymph node removal was on the other side). However she had undergone mesotherapy injections more than 15 years previously.


Figure 1
Figure 1. Patient 1 with multiple nodules of her left arm following a sporotrichoid pattern

Clinical examination at admission showed twenty nodules or papules located on the left arm and following a linear distribution with a sporotrichoid pattern (Figure 1). Some of these lesions were inflammatory, ulcerated, or opened to the skin, whereas other nodules were non inflammatory and only palpable. There was neither axillary lymphadenopathy nor a noticeable cutaneous portal of entry.

Laboratory results showed minor lymphopenia (1100/mm³) and cholestasis caused by hepatic metastasis. HIV serology was negative. A 6 mm punch biopsy specimen was obtained from an erythematous nodule. The histology examination showed a lymphohistiocytic infiltrate. Acid fast staining was negative. The culture of the biopsy specimen on Löwenstein-Jensen medium was positive for M. chelonae. It was only sensitive to tobramycin, tigecycline, and clarithromycin. The bone scintigraphy revealed intense fixation on the seventh dorsal vertebral body and on the right hip-bone related to bone metastasis.

Treatment was initiated with oral clarithromycin (500 mg twice a day), intravenous tigecycline (100 mg on day 1 followed by 50 mg x 2), and intravenous tobramycin (3 mg/kg/day) for 5 days. Clarithromycin and tigecycline were administered for 10 more days. Then she received only clarithromycin for four months. The skin lesions improved after 2 weeks of treatment and had cleared at 12 weeks. She had not relapsed at examination 10 months after treatment.


Figure 2
Figure 2. Patient 2 with multiple nodules of her left arm following a sporotrichoid pattern

Case 2. A 90-year-old man was admitted to our institution with a 1-month history of localized skin lesions on the right forearm. He was undergoing hemodialysis for chronic renal failure complicating nephroangiosclerosis. He also had a history of tertiary hyperparathyroidism, arterial hypertension, auriculoventricular block treated with a pacemaker, obliterative arteriopathy, and Horton disease. Physical examination showed six painless, erythematous papules and nodules distributed on the right forearm and following a sporotrichoid pattern (Figure 2). Some lesions had opened to the skin. The arteriovenous fistula was also located on this arm. M. chelonae infection was diagnosed by the culture of a skin biopsy. The histology examination was not specific. Acid fast staining was negative. The same treatment as in case 1 was started but adapted to the patient’s creatinine clearance. The nodules healed after 3 months and the antibiotics were stopped after 6 months. The patient had not relapsed 10 months after treatment.


Discussion

These two cases of nodular lymphangitis were caused by Mycobacterium chelonae and were cleared with antibiotics despite underlying immunosuppression, even though M. chelonae infection may be difficult to treat [2].

M. chelonae is a rapidly growing mycobacteria, ubiquitous in the environment (water systems, soil, plant material, aerosols) [3]. M. chelonae may give rise to a wide variety of infections including cutaneous abscesses, osteomyelitis and osteitis, primary pulmonary disease, disseminated disease, and prosthetic valve endocarditis [4].

Cutaneous abscesses related to M. chelonae usually occur after surgery procedures such as liposuction [5], breast augmentation, injections, catheter placement, acupuncture [6], and mesotherapy [2]. Less often they occur at the location of recent trauma. In these two cases the mode of contamination was unknown. In the first case, there was no recent history of invasive procedure and specifically no lymph node removal on this side. We initially suspected mesotherapy but the time elapsed between this high-risk medical procedure and the occurrence of the cutaneous lesions was more than fifteen years which does not seem to be compatible. In the second case, we suspected that the portal of entry was the arteriovenous fistula localized on the same arm as the cutaneous nodules. Moreover, his hemodialysis center was the same as that of a patient who had previously died in our department because of a disseminated M. chelonae infection (unpublished data). However, this hypothesis could not be confirmed. In addition, the disease started at the distal part of the forearm and spread towards the fistula and the patient was cured without removal of the fistula.

These two patients were immunosuppressed. One patient had lymphopenia and was treated for breast cancer with third-line chemotherapy and prednisone. The other patient was 90 years old and had been on hemodialysis for 10 years. This immunosuppressed status might explain why a cutaneous portal of entry was not found. Indeed, M. chelonae infections occurring in the absence of a high-risk medical procedure are mostly described in cases involving immunosuppressed patients [7]. Moreover, M. chelonae infection is more often seen in the setting of immunosuppression. As an example, of 100 M. chelonae infections involving the skin, soft tissue, and bones, 62 percent occurred in patients receiving corticosteroids and 72 percent of the patients were conisdered immunosuppressed [8].

M. chelonae is an unusual cause of nodular lymphangitis (“sporotrichoid” infection) [1]. This syndrome may be caused by many fungal, bacterial, mycobacterial, parasitic, and viral pathogens (Table 1). Mycobacterium marinum, Sporothrix schenckii, Leishmania braziliensis, and Nocardia brasiliensis are the most common etiologies. Nevertheless, rapidly growing mycobacteria must also be suspected as in our two cases. To the best of our knowledge, thirteen cases of M. chelonae related nodular lymphangitis have been reported to date. Of these 13 patients, three were renal transplant recipients, three had rheumatoid arthritis or spondylarthropathy and one had several malformations. Eight of these 13 patients were receiving corticosteroids and one had Cushing disease. Thus, nodular lymphangitis caused by M. chelonae is more frequently described in immunocompromised hosts, especially those receiving corticosteroids.

The optimal treatment procedures for skin infections caused by M. chelonae are not well established. No large-scale clinical trials have compared different antibiotic treatment procedures but current guidelines recommend testing antimicrobial susceptibility to better adapt antibiotic therapy. In these two cases, M. chelonae was only susceptible to tigecycline, clarithromycin, and tobramycin as previously reported [2]. However, in vitro susceptibility does not consistently predict treatment effectiveness. Indeed in a series of 14 patients treated with a combination of two or three antibiotics for M. chelonae abscesses, 7 patients had persistent lesions or occurrence of new abscesses, whereas one patient relapsed during the antibiotics course [2]. Clarithromycin was not used alone because resistance may be acquired after monotherapy [9]. Moreover, immunosuppression is usually associated with more severe and sometimes disseminated disease. As a consequence, the two patients received a combination of antibiotics at least during the first weeks of treatment.

Excision and drainage, not antibiotic therapy, may remain the treatment of choice for cutaneous abscesses, whereas antibiotic therapy alone may clear nodular lymphangitis or folliculitis seen after tattooing [10]. The duration of treatment for M. chelonae cutaneous infections is another critical point. Antibiotics are usually recommended for various durations, depending on disease location and severity. Between two to four months of antibiotic therapy is usually recommended in localized infection, with at least six months in disseminated cutaneous disease [11]. Our two patients were treated four and six months respectively, without relapse during a follow-up period lasting 10 months.

In conclusion, M. chelonae is a rare cause of nodular lymphangitis that may occur without a history of medical or surgical procedures, particularly in immunocompromised host. The optimal treatment and its duration are not well established.

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