This article has Open Peer Review reports available.
Human T cell lymphotropic virus type 1- associated infective dermatitis in KwaZulu Natal, South Africa
© Hlela et al.; licensee BioMed Central Ltd. 2013
Received: 8 February 2013
Accepted: 11 October 2013
Published: 23 October 2013
The Human T cell lymphotropic virus type 1 (HTLV-1)-associated infective dermatitis (IDH), is a chronic relapsing dermatitis which usually presents in children older than 2 years. A total of 300 cases have been reported worldwide (Latin America, the Caribbean and only 5 from Senegal). Neither IDH, nor its complications have been reported from the rest of Africa. We aimed to examine the clinical and aetiological characteristics of IDH in a cohort of South African children.
Attendees at the dermatology clinic at King Edward VIII Hospital, Durban underwent clinical examination. After obtaining consent those suspected of IDH had specimens taken for blood counts, immunoglobulins, serum protein electrophoresis, viral studies (including genotyping), skin swabs and stool examinations.
Nineteen of 60 suspected cases recruited over 3 years met the diagnostic criteria for IDH. The male-to-female ratio was 1:2; mean age 8 years (range 0.7 to 15). Dermatitis mostly affected the scalp (78.9%) and axilla (73.7%); fewer children had nasal crusting (47.4%). Mean Ig A, IgG and IgM were raised, at 3.52 g/l, 22.6 g/l and 1.38 g/l, respectively. The median CD4 cell count was 1958 cells/mm3. Viral genotyping of all tested samples were positive for the Cosmopolitan, Subtype A (HTLV-1a).
IDH is a distinct entity which also affects South Africans. Our patients were older at presentation and the majority did not present with nasal crusting as has been described in other countries.
The human T-cell lymphotropic virus type 1 (HTLV-1)-associated infective dermatitis (IDH) was first reported in Carribean children; the incidence and pathogenesis are unknown. Somewhat reminescent of seborhoeic dermatitis, the clinical presentation of IDH is that of severe exudative dermatitis with crusting of the scalp, neck, axillae, groin, external ear, and retro-auricular areas; watery nasal discharge, and/or crusting of the anterior nares, from about 2 years of age. We identified fewer than 30 publications which report less than 300 cases of IDH worldwide; reviewed in  The largest series reported 50 patients from Jamaica . There has only been one report of five African cases with IDH from Senegal . IDH has been linked with the development of adult T cell leukemia/lymphoma (ATLL) and HTLV-1-associated myelopathy/paraparesis (HAM/TSP) [4–7]. We aimed to examine the clinical and aetiological characteristics of IDH in a cohort of South African children.
Clinical criteria for IDH diagnosis
1. Eczema of scalp, axillae and groin external ear and retro-auricular areas, eyelid margins, paranasal skin and/or neck
1. Positive cultures for Staphylococcus aureus and/or Beta-haemolytic streptococcus from the skin or anterior nares
2. Chronic watery nasal discharge without other signs of rhinitis and/or crusting of the anterior nares
2. Generalized fine papular rash (in most severe cases)
3. Chronic relapsing dermatitis with prompt response to appropriate therapy but prompt recurrence on withdrawal of use of antibiotic
3. Generalized lymphadenopathy with dermatopathic lymphadenitis
4. Usual onset in early childhood
5. Human T cell lymphotropic virus type 1 antibody seropositivity
5. Elevated erythrocyte sedimentation rate
6. Hyperimmunoglobulinaemia (IgD and IgE )
7. Elevated CD4 count, CD8 count, and CD4/CD8 ratio
Blood counts, measurement of immunoglobulin levels, serum protein electrophoresis, viral studies, skin swabs for bacterial culture and examination of stool samples for parasites were done. All enrolled participants (n = 60) had HTLV-1 and HIV-1 serological testing done. HIV-1 serology was performed using the Vironostika HIV-1 IMPVD Microelisa system (Biomerieux, Durham, NC) with all positives samples confirmed by a second HIV ELISA test. Serology for HTLV-1 was performed on plasma using an enzyme-immune assay (EIA) that detects anti-HTLV-1 and anti-HTLV-2 antibodies (Ab-Capture EIA Test System – Ortho-Clinical Diagnostics, Inc., Rarita, New Jersey). HTLV-1 Western blots were not available so HTLV-1 infection was confirmed by HTLV PCR. DNA was extracted from peripheral blood mononuclear cells (PBMC) of seropositive patients using the QIAamp Blood kit (Qiagen Inc., Chatsworth, CA, U.S.A). Detection of a 318-bp product or a 161-bp product confirmed the presence of HTLV-1 or HTLV-2 pol respectively. To sub-type HTLV-1 a fragment of LTR was amplified by nested PCR using primers 12P1/SK111 and 12P5/1P1/2P3 and sequenced (n = 6) as previously described . HTLV-1 proviral copy number and beta-globin gene copy number were quantified using real-time quantitative PCR monitored by SYBR Green I dye incorporation in a Roche LightCycler 1.5, using the Tax sequence-specific primers SK43 and SK44. HTLV-1 copy number was estimated by interpolation from standard curves and expressed as a percentage of infected PBMCs.
IDH study summary data: summary of the analysis of 19 IDH patients
n = 60*
n = 33
n = 19
n = 9
(of n = 33)
all black african
8 months −15 years (IQR 7–11)
scalp (78.9%) and axilla (73.7%) commonest sites of involvement
no evidence for HAM/TSP
corneal opacities 3(15.6%)
mean (normals) g/l
mean (normals) g/l
Alpha 1 globulin
Alpha 2 globulin
12.3 (11.5-13.5) g/dl
40 (3–9 mm/hr)
1958 (500–1500) cells/mm3
1150 (436–2278) cells/mm3
CD4 : CD8 ratio
HTLV-1 proviral load
HTLV-1 is endemic in KZN with a reported seroprevalence of 2.6% in the Ngwelezana  and of 3.35 in Ubombo (Tarin, 1997) both districts within the same province as Durban. The mean age of our cohort (8 years) was much older than reported in the Caribbean where IDH usually presents between the ages of 2 and 4 years. Perhaps the subtropical climate of South Africa could explain these differences or the disease may be less severe in the early stages resulting in late presentation. The clinical features of IDH in South Africa are similar to that described elsewhere. However, chronic nasal discharge and/or nasal crusting was much less common that expected, (a mandatory criteria for diagnosis). These findings support the suggestion by de Oliveira  and Suite et al.  who have contested the inclusion of nasal crusting as a major criterion for the diagnosis of IDH. Farre et al., in their evalution of 42 cases from Brazil, records the fact that not all but 30 of their 42 cases, from Brazil exhibited crusting of the nares , raising the same issue that crusting around the nostrils should not be mandatory in cases of IDH. There appears to be a major difference between the disease in South Africa and that in Senegal, Brazil and Jamaica.
Except for the absence of anemia in our study population in contrast to the previous reports, other laboratory parameters were in keeping with what has been documented in the previous descriptions.
The data show that cases of IDH had a significantly high HTLV-1 proviral load (mean levels of 10.5%). It has been suggested that IDH act as a cofactor for the development of HTLV-1- associated diseases, such as HAM/TSP and ATLL but the mechanisms are still unclear. HTLV-1 proviral load levels is regarded as important predictor of ATLL and HAM/TSP. It would be interesting to follow all the identified cases with IDH over time and monitor the clinical evolution of the disease to determine if they develop any of the HTLV-1 associated conditions (ATLL, HAM/TSP).
IDH is a distinct entity that affects the African population of KwaZulu Natal, South Africa. It is predominantly a disease of childhood. The clinical features were in keeping with other series worldwide. Some differences in presentation are noted, particularly later age of onset and less nasal discharge. Interplay between genetic factors and environmental (including socioeconomy and climate) factors, may determine the age of presentation, severity of lesions and therefore explain the variation of IDH presentations between countries. The currect IDH criteria, needs revision. Crusting of the anterior nares should not be mandatory. The revised criteria should take into consideration the heterogeity this disease might have between different geographical locations.
I wish to thank Dr Luiz Alcantara and his team, from Bahia School of Medicine, Brazil for their contribution in PCR sequencing. Drs Ncoza Dlova from the Department of Dermatology, University of KwaZulu Natal. and Nilesh Morar from Westminister and Chelsea Hospital, UK for their support.
- Lee R, Schwartz RA: Human T-lymphotrophic virus type 1-associated infective dermatitis: a comprehensive review. J Am Acad Dermatol 2011, 64: 52–60.Google Scholar
- La Grenade L, Manns A, Fletcher V: Clinical, pathologic, and immunologicfeatures of human T lymphotropic virus type-1 associated infective dermatitis in children. Arch Dermatol 1998, 134: 439–444. 10.1001/archderm.134.4.439View ArticlePubMedGoogle Scholar
- Mahe A, Meertens L, Ly F, Sow PS, Diop CT, Samb ND, Diop OM, et al.: Human T-cell leukaemia/lymphoma virus type 1-associated infective dermatitis in Africa: a report of five cases from Senegal. Br J Dermatol 2004, 150: 958–965. 10.1111/j.1365-2133.2004.05834.xView ArticlePubMedGoogle Scholar
- La Grenade L, Schwartz RA, Janniger CK: Childhood dermatitis in the tropics:with special emphasis on infective dermatitis, a marker for infection with human T-cell leukemia virus-I. Cutis 1996, 58: 115–118.PubMedGoogle Scholar
- Primo JR, Brites C, Oliveira Mde F, Moreno-Carvalho O, Machado M, Bittencourt AL: Infective dermatitis and human T cell lymphotropic virus type 1-associated myelopathy/tropical spastic paraparesis in childhood and adolescence. Clin Infect Dis 2005, 41: 535–541. 10.1086/432058View ArticlePubMedGoogle Scholar
- Farre L, de Oliveira Mde F, Primo J, Vandamme AM, Van Weyenbergh J, Bittencourt AL: Early sequential development of infective dermatitis, human T cell lymphotropic virus type 1-associated myelopathy, and adult T cell leukemia/lymphoma. Clin Infect Dis 2008, 46: 440–442. 10.1086/524695View ArticlePubMedGoogle Scholar
- Tsukasaki K, Yamada Y, Ikeda S, Tomonaga M: Infective dermatitis among patients with ATL in Japan. Int J Cancer 1994, 57: 293. 10.1002/ijc.2910570227View ArticlePubMedGoogle Scholar
- Osame M, Usuku K, Izumo S, Ijichi N, Amitani H, Igata A, Matsumoto M, et al.: HTLV-I associated myelopathy, a new clinical entity. Lancet 1986, 1: 1031–1032.View ArticlePubMedGoogle Scholar
- Alcantara LCJ, Van Dooren S, Gonçalves MS, Van Dooren S, Gonçalves MS, Kashima S, Costa MCR, et al.: Globin haplotypes of human T-cell lymphotropic virus type I-infected individuals in salvador, bahia, brazil, suggest a post-columbian african origin of this virus. J Acquir Immune Defic Syndr 2003, 33: 536–542. 10.1097/00126334-200308010-00016View ArticleGoogle Scholar
- Bhigjee AI, Vinsen C, Windsor IM, Gouws E, Bill PL, Tait D: Prevalence and transmission of HTLV-I infection in Natal/KwaZulu. S Afr Med J 1993, 83: 665–667.PubMedGoogle Scholar
- Oliveira Mde F, Brites C, Ferraz N, Magalhaes P, Almeida F, Bittencourt AL: Infective dermatitis associated with the human T cell lymphotropic virus type I in Salvador, Bahia, Brazil. Clin Infect Dis 2005, 40: e90-e96. 10.1086/430064View ArticlePubMedGoogle Scholar
- Suite M, Jack N, Basdeo- Maharaj K, et al.: Infective dermatitis in Trinidad and Tobago. AIDS Res Hum Retroviruses 1994, 10: 447.Google Scholar
- de Oliveira Mde F, Fatal PL, Primo JR, da Silva JL, Batista Eda S, Farre L, Bittencourt AL: Infective dermatitis associated with human T-cell lymphotropic virus type 1: evaluation of 42 cases observed in Bahia. Brazil. Clin Infect Dis 2012, 12: 1714–1719.View ArticleGoogle Scholar
- LaGranade L: HTLV-1, infective dermatitis and tropical spastic paraparesis. Mol Neur 1994, 8: 147–148. 10.1007/BF02780665View ArticleGoogle Scholar
- Bhigjee AI, Tarin ML, Bill PL, De Oliveira T, York D: Sequence of the env gene of some KwaZulu-Natal, South African strains of HTLV type I. AIDS Res Hum Retroviruses 1999, 15: 1229–1233. 10.1089/088922299310322View ArticlePubMedGoogle Scholar
- Bittencourt AL, Oliveira Mde F, Ferraz N, Vieira MG, Muniz A, Brites C: Adult-onset infective dermatitis associated with HTLV-I. Clinical and immunopathological aspects of two cases. Eur J Dermatol 2006, 16: 62–66.PubMedGoogle Scholar
- Maragno L, Casseb J, Fukumori LM, Sotto MN, Duarte AJ, Festa-Neto C, Sanches JA: Human T-cell lymphotropic virus type 1 infective dermatitis emerging in adulthood. Int J Dermatol 2009, 48: 723–730. 10.1111/j.1365-4632.2009.04008.xView ArticlePubMedGoogle Scholar
- The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-5945/13/11/prepub
This article is published under license to BioMed Central Ltd. This is an open access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.