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Actinomycetoma of the head—a clinical and therapeutic challenge
P2302
P2304
Regional cutaneous BCGosis in a host with normal interferon gamma cellular responses Rishika Sinha, MBBCh, University College Hospital, London, United Kingdom; Emma Edmonds, MBBS, Department Of Dermatology, London, United Kingdom; Francisco Vega-Lopez, MD, MBBS, PhD, Department Of Dermatology, London, United Kingdom; Patricia Gorak-Stolinkska, MBBS, Immunology Unit, London, United Kingdom It has been estimated that approximately one third of the worldwide population is infected with Mycobacterium tuberculosis. From this pool, nine million cases of active tuberculosis emerge every year resulting in approximately two million deaths per year. Early last century, researchers developed the Bacillus-Calmette-Gue´rin vaccine (BCG) from a weakened strain of M bovis. This vaccine confers variable degrees of protection against tuberculosis and other mycobacterial infections. Eventhough vaccination with BCG can be frequently associated with local cutaneous reactions, disseminated BCG infection following vaccination is rare and largely seen in patients with inherited or acquired immunodeficiency syndromes. In the context of effective cell mediated immunity, interferon gamma (IFN-g) is a pivotal cytokine in the protective response to M tuberculosis via macrophage activation. A 16-year-old patient was referred to the dermatology department for assessment of three nonhealing nodular lesions on her left upper arm that had persisted following a BCG vaccination at that site 4 years earlier. Routine serologic markers and a chest radiograph were normal. Microscopic appearances of an incisional skin biopsy taken from the scar site and adjacent nodule were consistent with a BCG vaccination granuloma and scar. Special stains for acid-fast bacilli, fungi and bacteria failed to detect microorganisms. Bacterial culture of biopsy tissue revealed no growth. Investigations were carried out to assess her cytokine profile before and during therapeutic intervention. An enzyme-linked immunosorbant assay (ELISA) demonstrated an appropriate decrease in the number of spot forming cells during treatment. A cytokine release assay was performed showing a reduction in the magnitude of response to BCG antigen both before and during treatment. A functional assay confirmed our patient’s ability to produce tumor necrosis factorealfa in response to stimulation with recombinant IFN-g and lipopolysaccharide. On the basis of the clinical presentation and positive ELISA testing, the patient was treated for local and regional cutaneous BCGosis and commenced on rifampicin, isoniazid, and ethambutol. Pyrizinamide was not offered as chemotherapy because of the known resistance of BCG to this drug. Clinical and immunologic resolution of the infection was seen after completion of triple therapy.
Actinomycetoma of the head—a clinical and therapeutic challenge Amelia Morales-Toquero, MD, Hospital Universitario Dr Jose Eleuterio Gonzalez, Monterrey, Nuevo Leon, Mexico; Jorge Ocampo Candiani, MD, Hospital Universitario Dr Jose Eleuterio Gonzalez, Monterrey, Nuevo Leon, Mexico; Lucio Vera-Cabrera, DDSc, Hospital Universitario Dr Jose Eleuterio Gonzalez, Monterrey, Nuevo Leon, Mexico; Minerva Gomez-Flores, MD, Hospital Universitario Dr Jose Eleuterio Gonzalez, Monterrey, Nuevo Leon, Mexico; Oliverio Welsh, MD, DDSc, Hospital Universitario Dr Jose Eleuterio Gonzalez, Monterrey, Nuevo Leon, Mexico Treatment of cranial actinomycetoma is difficult. It requires rapid diagnosis and effective treatment to prevent spreading to the brain. We report a case of a 44-yearold woman with diabetes with actinomycetoma of the head cured with a combination of trimethroprim-sulfamethoxazole (SXT) and amikacin. She had an automobile accident 7 years earlier that caused multiple injuries of the head and face that were surgically repaired. Six months before presenting at our service, a neoformation of the left temporoparietal region appeared. A plastic surgeon performed a fineneedle biopsy before that showed a lymphohystiocytotic infiltrate. Imaging studies did not reveal bone affection. She was referred to our department for evaluation before surgery. On physical examination, we observed a 6- 3 5-cm red friable granulomatous vascular tumor on the left temporoparietal region with 10cm of perilesional swelling and a 5-mm nodule on the external cantus of the left eye. Direct mycologic examination showed multiple granules of Nocardia. Biopsy and culture confirmed the diagnosis of actinomycetoma caused by N brasiliensis. Antibodies were positive for N brasiliensis with an initial absorbance titer of 0.8935. After normal renal and audiometric study, she was treated with amikacin 15mg/kg/day IM for 12 weeks together with oral SXT 8/40mg/kg/day (8 months). After therapy, there was total remission of the lesion. Antinocardia antibodies returned to normal levels (0.317). Actinomycetoma of the head is rare presentation. This case represents a diagnostic challenge because it mimics a vascular neoplasm, sarcoma, and other tumors as well as fungal and mycobacterial infection. In cases with a risk of spreading infection to underlying organs or in those unresponsive to previous treatment, amikacin together with SXT can be used until cure is achieved. Treatment can be changed if antimicrobial resistance or side effects develop. N brasiliensis is the most frequent agent causing actinomycetoma in Mexico and generally responds well to SXT. In a case such as this, SXT-amikacin is the best choice because it achieves a rapid cure and prevents dissemination. Commercial support: None identified.
Commercial support: None identified.
P2303
P2305
Reactive arthritis with psoriasiform skin lesions and the importance of using DNA amplification tests for not missing Chlamydia trachomatis genital infections: Report of a case Joao Costa, Servic¸o de Dermatologia do Hospital de Santa Maria, Lisboa, Portugal; Filomena Martins, Unidade de DST do Instituto de Higiene e Medicina Tropical, Lisboa, Portugal; Luı´s Soares de Almeida, Servic¸o de Dermatologia do Hospital de Santa Maria, Lisboa, Portugal; Manuel Marques Gomes, Servic¸o de Dermatologia do Hospital de Santa Maria, Lisboa, Portugal Reactive arthritis, also known as Reiter syndrome, is defined by the classical triad of arthritis, urethritis, and conjunctivitis following a gastrointestinal or a nongonococcal genitourinary infection. We describe a 23-year-old man with recurrent episodes, after engagement in new relationships, of mucous urethral discharge and dysuria usually followed 3 weeks later by a mild bilateral conjunctivitis, severe joint pain, and articular swelling in an asymmetrical distribution, cutaneous lesions of keratoderma blennorrhagicum, balanitis circinata, and nail dystrophy. An etiologic agent was never found, and after this admittance he developed also psoriasiform skin lesions in a disseminated pattern. Besides elevation of the laboratory parameters of inflammation he had a positive a skin biopsy compatible with psoriasis and tested positive for HLA-B27. Serology for HIV and syphilis was negative as also were the laboratory tests for autoimmunity and antirheumatoid factor. We treated him and his asymptomatic female partner with 1g per os of azithromycin after collection of urine and exsudates for testing with a polymerase chain reaction for Chlamydia trachomatis with a technique targeting the cryptic plasmid of these bacteria. The patient and his partner had both positive reactions to C trachomatis with the visualization of a 241 pb band in the gel electrophoresis. As recommend by the Center of Disease Control and also with epidemiologic purpose, the tests were repeated with a different target in the genome of C trachomatis. These tests were also positive and the bacteria had genotype E in both the patient and his partner. The patient was then informed of the association of C trachomatis infection with his reactive arthritis, the absence of protective immunity to new infections with this bacteria and the use of condoms was promoted. The genitourinary complaints remitted with the antibiotic but control and regression of skin lesions and articular complains were only achieved after the initiation of IM methotrexate.
Mycobacterium chelonae: A rare but important cause of leg ulceration Kusuma R Narayana, MBBS, Addenbrooke’s Hospital, Cambridge, Cambridgeshire, United Kingdom; Jane Sterling, MBBCh, Addenbrooke’s Hospital, Cambridge, cambridgeshire, United Kingdom; Jonathan Batchelor, MBBS, Addenbrooke’s Hospital, Cambridge, Cambridgeshire, United Kingdom Mycobacterium chelonae is a rare pathogen that causes infection among humans. It has been reported among both immunocompetent and immunosuppressed patients. We describe a case of leg ulcers caused by M chelonae. A 86-year-old man presented with painful red areas on his legs, which appeared spontaneously. These rapidly changed to multiple, shallow, superficial ulcerated areas of erythema with overlying eschar. There was no history of trauma or vascular disease. He had been on long term oral corticosteroids for polymyalgia rheumatica and had a long-term indwelling catheter for urethral stricture related to bladder carcinoma. A skin biopsy specimen revealed a normal epidermis with multiple microabscesses and faintly granulomatous inflammation in the dermis. There were vacuoles containing elongated and rod-shaped organisms, which stained strongly positive with both Zeil-Neilson (ZN) and modified ZN stain, suggesting atypical mycobacteria. Liquid culture and subcultures identified M chelonae. The patient was treated with clarithromycin and moxifloxacin but discontinued treatment because of side effects. Topical fucibet and regular dressings has made significant improvement. M chelonae is a Gram-positive, opportunistic, fast growing atypical mycobacterium, which is rarely pathogenic. The organism is found in sewage, soil, and is especially common in tap water. Infection with M chelonae can be caused by trauma to the skin, cosmetic procedures (eg, pedicure, Botox injections) and invasive procedures (eg, central venous catheters, implants, and subcutaneous injections). The infection is more frequent in males, the elderly, and those with autoimmune disorders. More than 90% of cases occur in patients on long-term corticosteroids. There is no evidence of human-to-human transmission. M chelonae can also cause serious lung disease, osteomyelitis, corneal ulceration, and keratitis. Local wound care and antibiotics are reported to be the mainstay of medical management. Surgical debridement is required for extensive lesions. Standard antituberculous drugs have been shown to have no role in treatment. Dermatologists should be aware that M chelonae is a rare but important cause of skin ulceration.
Commercial support: None identified.
Commercial support: None identified.
MARCH 2010
J AM ACAD DERMATOL
AB81
P2304
Regional cutaneous BCGosis in a host with normal interferon gamma cellular responses Rishika Sinha, MBBCh, University College Hospital, London, United Kingdom; Emma Edmonds, MBBS, Department Of Dermatology, London, United Kingdom; Francisco Vega-Lopez, MD, MBBS, PhD, Department Of Dermatology, London, United Kingdom; Patricia Gorak-Stolinkska, MBBS, Immunology Unit, London, United Kingdom It has been estimated that approximately one third of the worldwide population is infected with Mycobacterium tuberculosis. From this pool, nine million cases of active tuberculosis emerge every year resulting in approximately two million deaths per year. Early last century, researchers developed the Bacillus-Calmette-Gue´rin vaccine (BCG) from a weakened strain of M bovis. This vaccine confers variable degrees of protection against tuberculosis and other mycobacterial infections. Eventhough vaccination with BCG can be frequently associated with local cutaneous reactions, disseminated BCG infection following vaccination is rare and largely seen in patients with inherited or acquired immunodeficiency syndromes. In the context of effective cell mediated immunity, interferon gamma (IFN-g) is a pivotal cytokine in the protective response to M tuberculosis via macrophage activation. A 16-year-old patient was referred to the dermatology department for assessment of three nonhealing nodular lesions on her left upper arm that had persisted following a BCG vaccination at that site 4 years earlier. Routine serologic markers and a chest radiograph were normal. Microscopic appearances of an incisional skin biopsy taken from the scar site and adjacent nodule were consistent with a BCG vaccination granuloma and scar. Special stains for acid-fast bacilli, fungi and bacteria failed to detect microorganisms. Bacterial culture of biopsy tissue revealed no growth. Investigations were carried out to assess her cytokine profile before and during therapeutic intervention. An enzyme-linked immunosorbant assay (ELISA) demonstrated an appropriate decrease in the number of spot forming cells during treatment. A cytokine release assay was performed showing a reduction in the magnitude of response to BCG antigen both before and during treatment. A functional assay confirmed our patient’s ability to produce tumor necrosis factorealfa in response to stimulation with recombinant IFN-g and lipopolysaccharide. On the basis of the clinical presentation and positive ELISA testing, the patient was treated for local and regional cutaneous BCGosis and commenced on rifampicin, isoniazid, and ethambutol. Pyrizinamide was not offered as chemotherapy because of the known resistance of BCG to this drug. Clinical and immunologic resolution of the infection was seen after completion of triple therapy.
Actinomycetoma of the head—a clinical and therapeutic challenge Amelia Morales-Toquero, MD, Hospital Universitario Dr Jose Eleuterio Gonzalez, Monterrey, Nuevo Leon, Mexico; Jorge Ocampo Candiani, MD, Hospital Universitario Dr Jose Eleuterio Gonzalez, Monterrey, Nuevo Leon, Mexico; Lucio Vera-Cabrera, DDSc, Hospital Universitario Dr Jose Eleuterio Gonzalez, Monterrey, Nuevo Leon, Mexico; Minerva Gomez-Flores, MD, Hospital Universitario Dr Jose Eleuterio Gonzalez, Monterrey, Nuevo Leon, Mexico; Oliverio Welsh, MD, DDSc, Hospital Universitario Dr Jose Eleuterio Gonzalez, Monterrey, Nuevo Leon, Mexico Treatment of cranial actinomycetoma is difficult. It requires rapid diagnosis and effective treatment to prevent spreading to the brain. We report a case of a 44-yearold woman with diabetes with actinomycetoma of the head cured with a combination of trimethroprim-sulfamethoxazole (SXT) and amikacin. She had an automobile accident 7 years earlier that caused multiple injuries of the head and face that were surgically repaired. Six months before presenting at our service, a neoformation of the left temporoparietal region appeared. A plastic surgeon performed a fineneedle biopsy before that showed a lymphohystiocytotic infiltrate. Imaging studies did not reveal bone affection. She was referred to our department for evaluation before surgery. On physical examination, we observed a 6- 3 5-cm red friable granulomatous vascular tumor on the left temporoparietal region with 10cm of perilesional swelling and a 5-mm nodule on the external cantus of the left eye. Direct mycologic examination showed multiple granules of Nocardia. Biopsy and culture confirmed the diagnosis of actinomycetoma caused by N brasiliensis. Antibodies were positive for N brasiliensis with an initial absorbance titer of 0.8935. After normal renal and audiometric study, she was treated with amikacin 15mg/kg/day IM for 12 weeks together with oral SXT 8/40mg/kg/day (8 months). After therapy, there was total remission of the lesion. Antinocardia antibodies returned to normal levels (0.317). Actinomycetoma of the head is rare presentation. This case represents a diagnostic challenge because it mimics a vascular neoplasm, sarcoma, and other tumors as well as fungal and mycobacterial infection. In cases with a risk of spreading infection to underlying organs or in those unresponsive to previous treatment, amikacin together with SXT can be used until cure is achieved. Treatment can be changed if antimicrobial resistance or side effects develop. N brasiliensis is the most frequent agent causing actinomycetoma in Mexico and generally responds well to SXT. In a case such as this, SXT-amikacin is the best choice because it achieves a rapid cure and prevents dissemination. Commercial support: None identified.
Commercial support: None identified.
P2303
P2305
Reactive arthritis with psoriasiform skin lesions and the importance of using DNA amplification tests for not missing Chlamydia trachomatis genital infections: Report of a case Joao Costa, Servic¸o de Dermatologia do Hospital de Santa Maria, Lisboa, Portugal; Filomena Martins, Unidade de DST do Instituto de Higiene e Medicina Tropical, Lisboa, Portugal; Luı´s Soares de Almeida, Servic¸o de Dermatologia do Hospital de Santa Maria, Lisboa, Portugal; Manuel Marques Gomes, Servic¸o de Dermatologia do Hospital de Santa Maria, Lisboa, Portugal Reactive arthritis, also known as Reiter syndrome, is defined by the classical triad of arthritis, urethritis, and conjunctivitis following a gastrointestinal or a nongonococcal genitourinary infection. We describe a 23-year-old man with recurrent episodes, after engagement in new relationships, of mucous urethral discharge and dysuria usually followed 3 weeks later by a mild bilateral conjunctivitis, severe joint pain, and articular swelling in an asymmetrical distribution, cutaneous lesions of keratoderma blennorrhagicum, balanitis circinata, and nail dystrophy. An etiologic agent was never found, and after this admittance he developed also psoriasiform skin lesions in a disseminated pattern. Besides elevation of the laboratory parameters of inflammation he had a positive a skin biopsy compatible with psoriasis and tested positive for HLA-B27. Serology for HIV and syphilis was negative as also were the laboratory tests for autoimmunity and antirheumatoid factor. We treated him and his asymptomatic female partner with 1g per os of azithromycin after collection of urine and exsudates for testing with a polymerase chain reaction for Chlamydia trachomatis with a technique targeting the cryptic plasmid of these bacteria. The patient and his partner had both positive reactions to C trachomatis with the visualization of a 241 pb band in the gel electrophoresis. As recommend by the Center of Disease Control and also with epidemiologic purpose, the tests were repeated with a different target in the genome of C trachomatis. These tests were also positive and the bacteria had genotype E in both the patient and his partner. The patient was then informed of the association of C trachomatis infection with his reactive arthritis, the absence of protective immunity to new infections with this bacteria and the use of condoms was promoted. The genitourinary complaints remitted with the antibiotic but control and regression of skin lesions and articular complains were only achieved after the initiation of IM methotrexate.
Mycobacterium chelonae: A rare but important cause of leg ulceration Kusuma R Narayana, MBBS, Addenbrooke’s Hospital, Cambridge, Cambridgeshire, United Kingdom; Jane Sterling, MBBCh, Addenbrooke’s Hospital, Cambridge, cambridgeshire, United Kingdom; Jonathan Batchelor, MBBS, Addenbrooke’s Hospital, Cambridge, Cambridgeshire, United Kingdom Mycobacterium chelonae is a rare pathogen that causes infection among humans. It has been reported among both immunocompetent and immunosuppressed patients. We describe a case of leg ulcers caused by M chelonae. A 86-year-old man presented with painful red areas on his legs, which appeared spontaneously. These rapidly changed to multiple, shallow, superficial ulcerated areas of erythema with overlying eschar. There was no history of trauma or vascular disease. He had been on long term oral corticosteroids for polymyalgia rheumatica and had a long-term indwelling catheter for urethral stricture related to bladder carcinoma. A skin biopsy specimen revealed a normal epidermis with multiple microabscesses and faintly granulomatous inflammation in the dermis. There were vacuoles containing elongated and rod-shaped organisms, which stained strongly positive with both Zeil-Neilson (ZN) and modified ZN stain, suggesting atypical mycobacteria. Liquid culture and subcultures identified M chelonae. The patient was treated with clarithromycin and moxifloxacin but discontinued treatment because of side effects. Topical fucibet and regular dressings has made significant improvement. M chelonae is a Gram-positive, opportunistic, fast growing atypical mycobacterium, which is rarely pathogenic. The organism is found in sewage, soil, and is especially common in tap water. Infection with M chelonae can be caused by trauma to the skin, cosmetic procedures (eg, pedicure, Botox injections) and invasive procedures (eg, central venous catheters, implants, and subcutaneous injections). The infection is more frequent in males, the elderly, and those with autoimmune disorders. More than 90% of cases occur in patients on long-term corticosteroids. There is no evidence of human-to-human transmission. M chelonae can also cause serious lung disease, osteomyelitis, corneal ulceration, and keratitis. Local wound care and antibiotics are reported to be the mainstay of medical management. Surgical debridement is required for extensive lesions. Standard antituberculous drugs have been shown to have no role in treatment. Dermatologists should be aware that M chelonae is a rare but important cause of skin ulceration.
Commercial support: None identified.
Commercial support: None identified.
MARCH 2010
J AM ACAD DERMATOL
AB81