Rare manifestation of ocular immune reconstitution inflammatory syndrome from mycobacterium scrofulaceum infection in a patient with AIDS.

Immune reconstitution inflammatory syndrome (IRIS) is a common complication following the initiation of antiretroviral therapy (ART). The most commonly associated pathogens include Mycobacterium tuberculosis and Cryptococcus spp.[1] IRIS following nontuberculosis mycobacteria (NTM) infection is uncommon, particularly, IRIS following NTM conjunctivitis.[2] Herein, we present a case of Mycobacterium scrofulaceum conjunctivitis with peripheral ulcerative keratitis and orbital cellulitis in a 45-year-old patient with AIDS who developed IRIS 1 month after starting ART therapy. A combination of both systemic and topical antibiotics together with corticosteroids were used and resulted in a satisfactory outcome with no early recurrence. This case demonstrated a rare ocular IRIS manifestation involving both the external eye and orbit and to the author's knowledge is the first case in the literature in which M. scrofulaceum has been found to be involved in the eye.

Disseminated Mycobacterium scrofulaceum Infection in a Patient with Anti-Interferon-γ Autoantibodies: A Case Report and Review of the Literature.

Mycobacterium scrofulaceum is an environmental mycobacterial species rarely reported to cause disseminated infection in adults. We report the case of a disseminated M. scrofulaceum infection in a 55-year-old nonhuman immunodeficiency virus-infected Thai man with anti-interferon-γ autoantibodies. The clinical signs of the infection improved after the induction regimen with amikacin, rifampicin, ethambutol, and clarithromycin, followed by the consolidation regimen with ethambutol, clarithromycin, and trimethoprim/sulfamethoxazole. Our review of previous reported cases of this infection indicates its association with immune deficiency, complex treatment, and a high rate of unfavorable outcomes.

Infección por Mycobacterium scrofulaceum en un niño de las Islas Galápagos - Ecuador / Mycobacterium scrofulaceum infection in a pediatric patient from the Galapagos Islands - Ecuador

Resumen Mycobacterium scrofulaceum es una micobacteria atípica de crecimiento lento que tiene como reservorio el ambiente. De forma ocasional causa enfermedad en humanos. Se presenta el caso de un niño de 10 años de edad con fiebre de cuatro meses de evolución, adenopatías cervicales, torácicas, abdominales e inguinales bilaterales, exantema, dolor abdominal y vómitos; con evidencia concomitante de serología positiva para el VEB y cultivo de los ganglios inguinales izquierdos con crecimiento de M. scrofulaceum. Es la primera comunicación de una infección causada por esta micobacteria en Ecuador, y que particularmente se presentó con un patrón de resistencia inusual.

Mycobacterium scrofulaceum is a slow-growing atypical mycobacteria with a reservoir in the environmen, and occasionally it causes disease in humans. The case of a 10-year-old patient with fever of four months of evolution is presented. Symptoms include bilateral adenopathies of neck, thorax, abdomen and inguinal region, rash, abdominal pain and vomiting; in addition, a positive serology for EBV and a culture of the left inguinal ganglia with growth of M. scrofulaceum were evidenced. Thus, this is the first report of human infection caused by the aforementioned mycobacterium in Ecuador, which it was particularly manifested with an unusual pattern of resistance.

Disseminated Mycobacterium scrofulaceum Infection in an Immunocompetent Host.

A 56-year-old woman, without any immunocompromising diseases, was referred to our hospital because of a recurrence of pyogenic spondylitis. Computed tomography revealed multiple osteolytic changes in the whole body. Vertebral magnetic resonance imaging revealed osteomyelitis and spondylitis. Mycobacterium scrofulaceum was detected in sputum cultures, in abscesses from the right knee, and in a subcutaneous forehead abscess. Therefore, the patient was diagnosed with disseminated Mycobacterium scrofulaceum infection. The patient was treated with rifampicin, ethambutol, and clarithromycin, which resulted in symptomatic relief and radiological improvement. We herein report a rare case of disseminated Mycobacterium scrofulaceum infection in an immunocompetent host.

Historia de la escrófula: de la discrasia humoral a la consunción / History of scrofula: from humoral dyscrasia to consumption

The term “scrofula” was used for a long time to designate a chronic swelling of cervical lymph nodes. This paper outlines the prevalent ideas on the nature, pathogenesis and the treatment of this disorder, from classical Greek medicine up to the 18th century. A Hippocratic treatise regarded scrofula as produced by an accumulation of phlegm, with a consequent imbalance or dyscrasia of the body humors. It was believed that it could heal spontaneously; but it could also soften, open through the skin and have an obstinate course. The treatment consisted mainly on local applications, incision to evacuate the soft content, or extirpation of the abnormal mass. In France and England, crowds of scrofulous patients were touched by the kings who were supposed to have a hereditary miraculous power to cure the disease. A Medieval text mentioned that scrofula could also affect other parts of the body. In the 17th century, scrofula was reputed as a frequent condition and was attributed to blood acrimony which coagulated in spongy organs. It was associated to phthisis or consumption due to the lethal outcome in some patients and to a cheese-like appearance of the pulmonary and the scrofulous lesions.

Characterization of Proinflammatory Responses and Innate Signaling Activation in Macrophages Infected with Mycobacterium scrofulaceum.

Mycobacterium scrofulaceum is an environmental and slow-growing atypical mycobacterium. Emerging evidence suggests that M. scrofulaceum infection is associated with cervical lymphadenitis in children and pulmonary or systemic infections in immunocompromised adults. However, the nature of host innate immune responses to M. scrofulaceum remains unclear. In this study, we examined the innate immune responses in murine bone marrow-derived macrophages (BMDMs) infected with different M. scrofulaceum strains including ATCC type strains and two clinically isolated strains (rough and smooth types). All three strains resulted in the production of proinflammatory cytokines in BMDMs mediated through toll-like receptor-2 and the adaptor MyD88. Activation of MAPKs (extracellular signal-regulated kinase 1/2, and p38, and c-Jun N-terminal kinase) and nuclear receptor (NF)-κB together with intracellular reactive oxygen species generation were required for the expression of proinflammatory cytokines in BMDMs. In addition, the rough morphotypes of M. scrofulaceum clinical strains induced higher levels of proinflammatory cytokines, MAPK and NF-κB activation, and ROS production than other strains. When mice were infected with different M. scrofulaceum strains, those infected with the rough strain showed the greatest hepatosplenomegaly, granulomatous lesions, and immune cell infiltration in the lungs. Notably, the bacterial load was higher in mice infected with rough colonies than in mice infected with ATCC or smooth strains. Collectively, these data indicate that rough M. scrofulaceum induces higher inflammatory responses and virulence than ATCC or smooth strains.

Characterization of Proinflammatory Responses and Innate Signaling Activation in Macrophages Infected with Mycobacterium scrofulaceum

Mycobacterium scrofulaceum is an environmental and slow-growing atypical mycobacterium. Emerging evidence suggests that M. scrofulaceum infection is associated with cervical lymphadenitis in children and pulmonary or systemic infections in immunocompromised adults. However, the nature of host innate immune responses to M. scrofulaceum remains unclear. In this study, we examined the innate immune responses in murine bone marrow-derived macrophages (BMDMs) infected with different M. scrofulaceum strains including ATCC type strains and two clinically isolated strains (rough and smooth types). All three strains resulted in the production of proinflammatory cytokines in BMDMs mediated through toll-like receptor-2 and the adaptor MyD88. Activation of MAPKs (extracellular signal-regulated kinase 1/2, and p38, and c-Jun N-terminal kinase) and nuclear receptor (NF)-kappaB together with intracellular reactive oxygen species generation were required for the expression of proinflammatory cytokines in BMDMs. In addition, the rough morphotypes of M. scrofulaceum clinical strains induced higher levels of proinflammatory cytokines, MAPK and NF-kappaB activation, and ROS production than other strains. When mice were infected with different M. scrofulaceum strains, those infected with the rough strain showed the greatest hepatosplenomegaly, granulomatous lesions, and immune cell infiltration in the lungs. Notably, the bacterial load was higher in mice infected with rough colonies than in mice infected with ATCC or smooth strains. Collectively, these data indicate that rough M. scrofulaceum induces higher inflammatory responses and virulence than ATCC or smooth strains.