Disseminated BCG infection revealing X-linked severe combined immunodeficiency
被引:9
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作者:
Marchand, I.
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机构:
Hop Ambroise Pare, AP HP, Serv Pediat, F-92100 Boulogne, FranceHop Ambroise Pare, AP HP, Serv Dermatol, F-92100 Boulogne, France
Marchand, I.
[2
]
Mahe, E.
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机构:
Hop Ambroise Pare, AP HP, Serv Dermatol, F-92100 Boulogne, FranceHop Ambroise Pare, AP HP, Serv Dermatol, F-92100 Boulogne, France
Mahe, E.
[1
]
Clerici, T.
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机构:
Hop Ambroise Pare, AP HP, Serv Anatomopathol, F-92100 Boulogne, FranceHop Ambroise Pare, AP HP, Serv Dermatol, F-92100 Boulogne, France
Clerici, T.
[3
]
Saiag, P.
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机构:
Hop Ambroise Pare, AP HP, Serv Dermatol, F-92100 Boulogne, FranceHop Ambroise Pare, AP HP, Serv Dermatol, F-92100 Boulogne, France
Saiag, P.
[1
]
Chevallier, B.
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机构:
Hop Ambroise Pare, AP HP, Serv Pediat, F-92100 Boulogne, FranceHop Ambroise Pare, AP HP, Serv Dermatol, F-92100 Boulogne, France
Chevallier, B.
[2
]
机构:
[1] Hop Ambroise Pare, AP HP, Serv Dermatol, F-92100 Boulogne, France
[2] Hop Ambroise Pare, AP HP, Serv Pediat, F-92100 Boulogne, France
[3] Hop Ambroise Pare, AP HP, Serv Anatomopathol, F-92100 Boulogne, France
来源:
ANNALES DE DERMATOLOGIE ET DE VENEREOLOGIE
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2008年
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135卷
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8-9期
关键词:
BCG;
immunodeficiency;
D O I:
10.1016/j.annder.2008.02.023
中图分类号:
R75 [皮肤病学与性病学];
学科分类号:
100206 ;
摘要:
Background. - Live attenuated Bacillus Calmette-Guerin (BCG) vaccine is rarely responsible for disseminated infection. We report a case of X-linked severe combined immunodeficiency (SCID) revealed by a disseminated skin infection. Patients and methods. - A 4-month-old baby was hospitalized for prolonged gastroenteritis. He was in poor general condition, with prolonged fever, oral and gluteal candidiasis and purple nodules associated with ulceration of the BCG scar. The absence of a thymus, T-cells and NK-cells, and the presence of nonfunctional B-Lymphocytes led to a diagnosis of SCID. Biopsies of nodules revealed a dermal infiltrate without necrosis. A Ziehl-Neelson stain was highly positive and the culture grew Mycobacterium bovis. Treatment consisted of a four-drug antibiotic regimen directed against M. bovis combined with gamma interferon, immunoglobulins and antibiotic prophylaxis by cotrimoxazole and was followed by a haploid-identical bone marrow transplant without rejection at six months. The early death of the child's maternal uncle from sepsis suggested X-linked transmission, which was subsequently confirmed by genetic analysis. Discussion. - BCG vaccination can cause serious infections in immunocompromised subjects. Skin involvement is extremely rare but may be the first sign of SCID, of which the X-linked form is the most common and corresponds to a variety of mutations in the gene coding for the gamma chain common to several interleukin receptors. Genetic counselling is essential to identify female carriers and allow early antenatal diagnosis. Bone marrow transplantation is the only treatment. (C) 2008 Elsevier Masson SAS. Tous droits reserves.
机构:
Tokyo Med & Dent Univ, Grad Sch, Dept Dermatol, Bunkyo Ku, Tokyo 1138519, JapanTokyo Med & Dent Univ, Grad Sch, Dept Dermatol, Bunkyo Ku, Tokyo 1138519, Japan
Yu, Rie
Satoh, Takahiro
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机构:
Tokyo Med & Dent Univ, Grad Sch, Dept Dermatol, Bunkyo Ku, Tokyo 1138519, JapanTokyo Med & Dent Univ, Grad Sch, Dept Dermatol, Bunkyo Ku, Tokyo 1138519, Japan
Satoh, Takahiro
Wakabayashi, Tomoe
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机构:
Tokyo Med & Dent Univ, Grad Sch, Dept Dermatol, Bunkyo Ku, Tokyo 1138519, JapanTokyo Med & Dent Univ, Grad Sch, Dept Dermatol, Bunkyo Ku, Tokyo 1138519, Japan
Wakabayashi, Tomoe
Ueda, Nobuhiko
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机构:
Tokyo Med & Dent Univ, Grad Sch, Dept Dermatol, Bunkyo Ku, Tokyo 1138519, JapanTokyo Med & Dent Univ, Grad Sch, Dept Dermatol, Bunkyo Ku, Tokyo 1138519, Japan
Ueda, Nobuhiko
Yokozeki, Hiroo
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机构:
Tokyo Med & Dent Univ, Grad Sch, Dept Dermatol, Bunkyo Ku, Tokyo 1138519, JapanTokyo Med & Dent Univ, Grad Sch, Dept Dermatol, Bunkyo Ku, Tokyo 1138519, Japan
机构:
Hosp Civil Guadalajara Dr Juan I Menchaca, Dept Allergy & Clin Immunol, Guadalajara, Jalisco, MexicoHosp Civil Guadalajara Dr Juan I Menchaca, Dept Allergy & Clin Immunol, Guadalajara, Jalisco, Mexico
Nunez-Nunez, M. E.
Lona-Reyes, J. C.
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机构:
Hosp Civil Guadalajara Dr Juan I Menchaca, Dept Allergy & Clin Immunol, Guadalajara, Jalisco, MexicoHosp Civil Guadalajara Dr Juan I Menchaca, Dept Allergy & Clin Immunol, Guadalajara, Jalisco, Mexico
Lona-Reyes, J. C.
Pedroza-Flores, K. P.
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机构:
Hosp Civil Guadalajara Dr Juan I Menchaca, Dept Allergy & Clin Immunol, Guadalajara, Jalisco, MexicoHosp Civil Guadalajara Dr Juan I Menchaca, Dept Allergy & Clin Immunol, Guadalajara, Jalisco, Mexico
Pedroza-Flores, K. P.
Rios-Aguirre, A.
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机构:
Hosp Civil Guadalajara Dr Juan I Menchaca, Dept Allergy & Clin Immunol, Guadalajara, Jalisco, MexicoHosp Civil Guadalajara Dr Juan I Menchaca, Dept Allergy & Clin Immunol, Guadalajara, Jalisco, Mexico
Rios-Aguirre, A.
Torres-Lozano, C.
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h-index: 0
机构:
Hosp Civil Guadalajara Dr Juan I Menchaca, Dept Allergy & Clin Immunol, Guadalajara, Jalisco, MexicoHosp Civil Guadalajara Dr Juan I Menchaca, Dept Allergy & Clin Immunol, Guadalajara, Jalisco, Mexico