Flower cells in patients with infective dermatitis associated with HTLV-1

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  • Journal of Clinical Virology 48 (2010) 288290

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    Short communication

    Flower cells in patients with infective dermatitis

    Maria de b, JEdgar M. hila Department o alvadb Department o ador,c Department o hia, Sad Laboratory of lvadoe Laboratory of alvado

    a r t i c l

    Article history:Received 22 MReceived in reAccepted 15 M

    Keywords:HTLV-1 infectionInfective dermatitis associated with HTLV-1Adult T-cell leukemia/lymphomaFlower cellsSkin diseases caused by HTLV-1


    ths, ttions, routine laboratory exams, full blood count and blood smears, and to six-monthly blood samplingfor HTLV-1 proviral load determination. HTLV-1 proviral load was quantied using real-time TaqManPCR assay.Results: Abnormal cells (Ably) were found in the peripheral blood smears of nine patients (30%), owercells being detected in ve of these cases (16.6%). The presence of Ably and ower cellswas not associatedwith a higher proviral load in those patients.

    1. Backgro

    Manyclition and adevelop sev(ATL), HTLV(HAM/TSP)occurs in veinfected forandgeneraleruption invease respontreatment i

    ATL is afty years aclassied in

    Corresponversitrio ProViana, s/n Canfax: +55 71 32

    E-mail add

    1386-6532/$ doi:10.1016/j.Conclusions: This is the rst report on the presence of ower cells in HTLV-1-infected children and adoles-cents. Furthermore, these cells have not previously been reported in IDH patients. The cases with owercells probably represent precursory ATL cases, these patients being at a greater risk of developing ATL.

    2010 Elsevier B.V. All rights reserved.


    nical disorders havebeenassociatedwithHTLV-1 infec-small percentage of HTLV-1-infected subjects may

    ere diseases including adult T-cell leukemia/lymphoma-1 associated myelopathy/tropical spastic paraparesisand HTLV-1-associated infective dermatitis (IDH).1 IDHrtically-infected children.2 It is a severe, recurrent andm of eczema that usually begins at 18 months of agely disappears in adolescence. It presents as an exudativeolving mainly the scalp, external ear and neck. The dis-ds to antibiotic therapy but relapses immediately onces withdrawn.2,3

    n aggressive T-cell malignancy that generally occursfter vertically acquired HTLV-1 infection.4 It has beento ve clinical types: acute, chronic, lymphomatous,

    ding author at: Laboratrio de Patologia, Complexo Hospitalar Uni-f. Edgard Santos, Universidade Federal da Bahia, Rua Dr. Augustoela, CEP: 40110-060 Salvador, Bahia, Brazil. Tel.: +55 71 3283 8016;83 8016.ress: achilea@uol.com.br (A.L. Bittencourt).

    smoldering and primary cutaneous tumoral.4,5 The acute andchronic types present lymphocytosis, which is not observed in theother types of ATL.5 In acuteATL andoccasionally in the chronic andsmoldering types of ATL, lymphocytes showing markedly polylob-ulated nuclei with homogeneous and condensed chromatin, smallor absent nucleoli and basophilic cytoplasm, referred to as owercells, may appear in peripheral blood.5 In addition to these cells,abnormal lymphocytes (Ably) with other morphologies are alsoobserved.6

    The relationship between IDH and ATL appears to be close, atleast in the population living in the Brazilian state of Bahia, where37.5% of patients with ATL affecting the skin were found to havehad a history of severe eczema in childhood with characteristicssimilar to those of IDH.7 Moreover, two cases of ATL in adolescencewere diagnosed in Bahia and in both cases the patients had had IDHin childhood.1,8

    2. Objectives

    The objective of this study was to evaluate the presence of clin-ical and laboratory parameters suggestive of ATL in a cohort of 30patients with IDH.

    see front matter 2010 Elsevier B.V. All rights reserved.jcv.2010.05.005Ftima Paim de Oliveiraa, Maria das Gracas VieiraCarvalhod, Lourdes Farre, Priscila Lima Fatala, Ac

    f Dermatology, Professor Edgar Santos Teaching Hospital, Federal University of Bahia, Sf Pathology, Professor Edgar Santos Teaching Hospital, Federal University of Bahia, Salvf Internal Medicine, Professor Edgar Santos Teaching Hospital, Federal University of BaImmunology, Professor Edgar Santos Teaching Hospital, Federal University of Bahia, SaExperimental Pathology, Goncalo Muniz Research Center, Oswaldo Cruz Foundation, S

    e i n f o

    arch 2010vised form 11 May 2010ay 2010

    a b s t r a c t

    Background: Infective dermatitis assocmay progress to adult T-cell leukemiaObjective: In this study, thepresenceofin a cohort of 30 patients with IDH.Study design: Over a period of 33 monom/ locate / j cv

    associated with HTLV-1

    aneusa Primoc, Isadora C. Siqueirad,a L. Bittencourtb,

    or, Bahia, BrazilBahia, Brazillvador, Bahia, Brazilr, Bahia, Brazilr, Bahia, Brazil

    with HTLV-1 (IDH) is a severe childhood form of eczema thatphoma (ATL).cal and laboratoryparameters suggestiveofATLwasevaluated

    he patients were submitted to three-monthly clinical evalua-

  • M.d.F.P. de Oliveira et al. / Journal of Clinical Virology 48 (2010) 288290 289

    3. Study design

    The cohort consisted of 19 girls and 11 boys with ages rang-ing from 2 to 18 years (mean 12.734.96 years), followed-up atthe dermatology and pediatric neurology outpatient clinics of theProfessor Edgard Santos Teaching Hospital, Federal University ofBahia, Brazil. All patients were HTLV-1 positive (as shown by ELISAand conrmed by Western Blot) and HIV-negative. Ten patientshad associated HAM/TSP. Diagnosis of IDH and HAM/TSP was per-formed according to previously established criteria.3,9 BetweenJune 2006 and March 2009, the patients were submitted to three-monthly clinical evaluations, routine laboratory exams and bloodsampling for full blood count and blood smears. In cases in whichower cells were present in blood smears, blood levels of lac-tate dehydrogenase (LDH) and calcium were investigated. ChestX-rays and abdominal ultrasonography scanswere also performed.Evaluation of lymphocytosis was based on age-specic normalvalues using previously established criteria.10 Mild lymphocyto-sis detected at one single test was not taken into consideration.The percentage of Ably was determined in 100 lymphocytes bymicroscopic analysis of a Wrights stained peripheral blood smear.Ably were iral load ana2.7 samplesMan polympatients vicalculated aload for eac4.03 (San Dysis. The M

  • 290 M.d.F.P. de Oliveira et al. / Journal of Clinical Virology 48 (2010) 288290

    5. Discussion

    The nding of ower cells in HTLV-1-infected children and ado-lescents is reported here for the rst time. In fact, ower cells werereported for the rst time in IDH. As previously mentioned owercells are commonly found in the acute type of ATL and occasionallyin the chronic and smoldering types. The presence of Ably withoutreference to ower cells has been reported in symptomless HTLV-1carrier children.12

    Ably have been found in frequencies ranging from 10% to 43% inthe peripheral blood of adult HTLV-1 carriers considered to be at ahigh risk of developing ATL1315 In addition, ower cells have beenfound in 7% of adult HTLV-1 carriers.16 These ndings are quitesimilar to those observed in the present study.

    The presence of ower cells in children and adolescents withIDH, adiseasewithahighproviral load,17 maybeconsidered indica-tive of a greater risk for the development of ATL compared to theoccurrencethermore, inwas short, i

    Accordinof Ably areindividual,be considerstudy the cered leukemdetected on

    Ably havers with a hload.13,18 Inof IDH patiecompared tnot found.nicant, faismall numba larger nuchild/adolerelationship

    Althoughsidered as claboratorystudies ove

    Conict of



    This stu(CNPq) and(FAPESB). A

    are grateful to Dr. Vitria Regina Rego, head of the DermatologyClinic at the Federal University of Bahia, who permitted access tothe clinic facilities.


    1. Farr L, de Oliveira MF, Primo J, Vandamme AM, Van Weyenbergh J, Bit-tencourt AL. Early sequential development of infective dermatitis, humanT cell lymphotropic virus type 1-associated myelopathy, and adult T cellleukemia/lymphoma. Clin Infect Dis 2008;46:4402.

    2. LaGrenadeL,MannsA, FletcherV,DermD,CarberryC,HanchardB, et al. Clinical,pathologic, and immunologic features of human T-lymphotrophic virus type I associated infective dermatitis in children. Arch Dermatol 1998;134:43944.

    3. Oliveira MF, Brites C, Ferraz N, Magalhes P, Almeida F, Bittencourt AL. Infec-tive dermatitis associated with the human T cell lymphotropic virus type I inSalvador, Bahia, Brazil. Clin Infect Dis 2005;40:906.

    4. Bittencourt AL, Vieira MG, Brites CR, Farr L, Barbosa HS. Adult T-cellleukemia/lymphoma (ATL) in Bahia, Brazil: analysis of prognostic factors ina group of 70 patients. Am J Clin Pathol 2007;128:87582.

    5. ShimoyamaM,Membersof theLymphomaStudyGroup.Diagnostic criteria andsication of clinical subtypes of adult T-cell leukaemia-lymphoma. A reportthe

    kasakersitys, aber9;105encouL) pretures oencouphotr006;8o JR

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    oulderoshitaleukemhumaher RAceofs typo J,

    hHTLstic paed wiada Ml. Premphoof these cells in asymptomatic and adult carriers. Fur-the cases included in this study the period of infection

    ncluding a child of less than 3 years of age.g to Shimoyama et al.,5 cases in which 5% or morefound in the peripheral blood of an HTLV-1-infected

    even when no other manifestations are present, shoulded leukemic smoldering ATL. However, in the presentases in which 5% of Ably were found were not consid-ic smoldering ATL since this percentage of Ably wasone single o