Acta Tropica 134 (2014) 1316

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Acta Tropica
journal hoepa!e" ###$elsevier$co%locate%actatropica
&'phe(ea in a )*'ear*ol( bo' in+ecte( #ith ,uchereria bancro+ti
in Sierra &eone" A case report
-ac.en/ie Doe a01 0 1ashi( Ansuana b0c0201 0 An(rea &$ Covin!ton b 0 -aria 3$ 1ebollo c 0
Santi!ie Sesa' ( 0 4athr'n 5$ 6acobsen e 0 D/ie(/o 4$ (e Sou/a + 0 7enjain 8$ 4ou(ou c 0
9(#in -ichael ! 0 -oses 6$ 7oc.arie c
a
9c. :nstitute +or 8lobal 5ealth0 ;niversit' o+ <otre Dae0 <otre Dae0 :<0 ;SA
-erc' 5ospital 1esearch &aborator'0 7o0 Sierra &eone
cCentre +or <e!lecte( Tropical Diseases0 &iverpool School o+ Tropical -e(icine0 &iverpool0 ;4
(-inistr' o+ 5ealth an( Sanitation0 =reeto#n0 Sierra &eone
eDepartent o+ 8lobal an( Counit' 5ealth0 8eor!e -ason ;niversit'0 =air+a>0 ?A0 ;SA
+<o!uchi -eorial :nstitute +or -e(ical 1esearch0 ;niversit' o+ 8hana0 &e!on0 8hana
!Departent o+ 7iolo!ical Sciences0 ;niversit' o+ <otre Dae0 <otre Dae0 ;SA
b
a r t i c l e i n + o a b s t r a c t
,e present a case o+ con!enital l'phe(ea in a )*'ear*ol( bo' in Sierra &eone #ith active @larial
in+ection an( penile e(ea$ The !enital e(ea #ith onset at 6 onths o+ a!e a' have been (ue to a
con!enital abnoralit' in l'phatic (raina!e$ Ather possible causes o+ chil(hoo( l'phe(ea0 inclu(in!
-ilro'Bs (isease0 are (iscusse($
C 2014 The Authors$ 3ublishe( b' 9lsevier 7$?$ This is an open access article un(er the CC 7D*<C*SA
license (http"%%creativecoons$or!%licenses%b'*nc*sa%3$0%)$
Article histor'"
1eceive( 23 Actober 2013
1eceive( in revise( +or 2 =ebruar' 2014
Accepte( 10 =ebruar' 2014
Available online 1E =ebruar' 2014
4e'#or(s"
&'phatic @lariasis
=ilarial elephantiasis
&'phe(ea
-ilro'Bs (isease
Chil(
Coplications
1$ :ntro(uction
&'phatic @lariasis (&=) is a ajor cause o+ acute an( chronic
orbi(it' ani+este( as l'phe(ea an( h'(rocele$ The clinical
si!ns an( s'ptos o+ &= aFect ore than 40 illion people !lob*
all'0 a.in! the (isease the secon(*lar!est cause o+ peranent an(
lon!*ter (isabilit' #orl(#i(e (Sheno' an( 7oc.arie0 2011G Chu
et al$0 2010)$ &= is a ajor obstacle to socioeconoic (evelopent in
the )3 countries across A+rica0 Southeast Asia0 the Aericas0 an( the
3aci@c re!ion that are .no#n to be en(eic +or the osHuito*borne
(isease (,5A0 2013G A((iss0 2010)$
&= in+ection is o+ten acHuire( in chil(hoo( in areas o+ hi!h trans*
ission intensit'0 but in+ecte( in(ivi(uals usuall' (o not (evelop
clinical (isease until 'ears later #hen the' are a(ults$ -ore than
2I0 illion chil(ren are e>pose( to &= #orl(#i(e0 an( in hi!h
en(eic areas up to 10J o+ chil(ren un(er the a!e o+ 1I 'ears are
in+ecte( (7oc.arie et al$0 2002)$ :n 20000 the ,orl( 5ealth Ar!ani/a*
tion launche( the 8lobal 3ro!ra to 9liinate &= (839&=) usin! the
pro*poor strate!' o+ preventive cheotherap' an( transission
control (3CT) to!ether #ith vector control0 orbi(it' ana!eent0
an( (isabilit' prevention (,5A0 2013)$ The ass (ru! a(inistra*
tion (-DA) strate!' use( +or 3CT cobines alben(a/ole #ith either
iverectin or (ieth'lcarbaa/ine citrate (D9C)$ 5o#ever0 not all
at*ris. chil(ren in hi!h*ris. areas are able to access -DA$
&'phatic @lariasis cause( b' ,uchereria bancro+ti is hi!hl'
en(eic in Sierra &eone0 #here ore than K0J o+ the 6$6 illion
people are at ris. o+ acHuirin! the in+ection$ (5o(!es et al$0 2012G
4oroa et al$0 2012) =ilarial (isease is coon in all the en(eic
(istricts in the countr'0 but there is no recor( previousl' o+
l'phe(ea in a chil( less than 10 'ears ol($ 5ere #e present a
case o+ l'phe(ea in a )*'ear*ol( bo' #ith patent &=$
2$ Case report
Durin! a transission assessent surve' to (eterine the suc*
cess o+ +our roun(s o+ -DA on re(ucin! &= transission in north
2 Correspon(in! author at" -erc' 5ospital 1esearch &aborator'0 4ulan(a To#n0
7o0 Sierra &eone$ Tel$" L232 )6 6E3E32$
9*ail a((ress" r$ansuanaMliverpool$ac$u. (1$ Ansuana)$
1These authors contribute( eHuall' to this stu('$
http"%%(>$(oi$or!%10$1016%j$actatropica$2014$02$00)
0001*)06N%C 2014 The Authors$ 3ublishe( b' 9lsevier 7$?$ This is an open access article un(er the CC 7D*<C*SA license (http"%%creativecoons$or!%licenses%b'*nc*sa%3$0%)$
14 -$ Doe et al$ % Acta Tropica 134 (2014) 1316
=i!$ 1$ &'phe(ea o+ 1i!ht &o#er 9>treit'$
=i!$ 2$ De+ore( 3repuce$
central 3ujehun District0 Sierra &eone0 a )*'ear*ol( bo' +ro a
rural villa!e presente( #ith chronic pro!ressive l'phe(ea o+
the ri!ht lo#er e>treit'$ The overl'in! tissue o+ the (istal e>tre*
it' #as in(urate(0 possibl' (ue to secon(ar' in+ection an( tissue
traua0 an( there #ere (eratolo!ic chan!es o+ the ri!ht +oot con*
sistent #ith elephantiasis (=i!$ 1)$ The teperature o+ the s#ollen
areas #as noral0 #ith no si!ns o+ no(ular or #art' e>crescences$
The bo'Bs +ather reporte( that as'etrical non*pittin! e(ea ha(
been present since birth0 an( ha( increase( in severit' over tie$
4eratosis spots #ere present over the entire bo('0 an( the pre*
puce o+ the penis sho#e( a si!ni@cant (e+orit' that the +ather
sai( ha( been present since the chil( #as si> onths o+ a!e (=i!$ 2)$
The +ather !ave perission +or photo!raphs o+ the chil( an( case
in+oration to be publishe($
The bo' teste( positive +or circulatin! @larial anti!eneia usin!
a 7ina><A, @lariasis :uno Chroato!raphic Test (:CT) (:nver*
ness -e(ical 3ro+essional Dia!nostics0 3reehol(0 <60 ;SA)0 an( a
ni!ht bloo( sear e>aination #ith 8iesa stain con@re( 7an*
cro+tian @lariasis #ith 313 ,$ bancro+ti +%l o+ bloo( prior to
treatent$ The parasite #as sheathe( #ith nuclei in the tail en( an(
#as orpholo!icall' (istin!uishable +ro the -ansonella species
#hich can also be +oun( in huan bloo( in A+rica$ These observa*
tions an( the species*speci@c :CT positive result con@re( that the
icro@laria observe( in the bloo( #ere ,$ bancro+ti$ :nitial treat*
ent prescribe( +or the active @larial in+ection #as a 6 ! (ose o+
iverectin an( a 400 ! (ose o+ alben(a/ole ever' three onths
#ith +ollo#*up$ The +ather reporte( that the bo' ha( not been pre*
viousl' treate( +or &= in+ection$
The bo' ha( live( #ith his parents in rural 3ujehun District +or
the past +our 'ears0 an( the' previousl' resi(e( in 4ono District
(=i!$ 3)$ 7e+ore her pre!nanc' #ith the bo' an( (urin! the en( o+
her @rst triester0 his other ha( resi(e( in 4ono District #ith her
!ran(other0 a resi(ent o+ 7obali District0 #ho ha( elephantiasis$
7oth 7obali an( 4ono Districts #ere en(eic +or &= prior to the
-DA an( the birth o+ the bo' (4oroa et al$0 2013)$
As part o+ the investi!ation o+ the chil(Bs (isease0 +ail' e*
bers #ere e>aine( +or &=$ The other teste( &= positive base(
on both :CT an( ni!ht bloo( sear e>aination0 an( reporte( ta.*
in! onl' one roun( o+ -DA (urin! her li+etie$ The bo'Bs +ather
an( the bo'Bs onl' siblin! both teste( ne!ative$ <one o+ the ie*
(iate +ail' ebers ha( anatoical abnoralities relate( to &=$
5o#ever0 a +ail' histor' su!!este( that the bo'Bs (ecease( ater*
nal !ran(other li.el' also ha( an a(vance( @larial in+ection0 as
she ha( pro!ressive l'phe(ea o+ the lo#er ri!ht e>treit' that
(evelope( over the last @ve 'ears o+ her li+e$
T#o onths a+ter the bo'Bs initial (ia!nosis0 +ollo#in! pre*
scribe( treatent #ith iverectin an( alben(a/ole0 a ni!ht bloo(
sear test sho#e( no evi(ence o+ circulatin! icro@laria$ A co*
plete bloo( count0 coprehensive etabolic panel0 an( urinal'sis
#ere all +oun( to be #ithin noral liits$ A ph'sical e>aination
reveale( no evi(ence o+ another incitin! con(ition$ 7oth the chil(
an( his +ather #ere also teste( at this tie +or several other in+ec*
tious (iseases0 inclu(in! huan iuno(e@cienc' virus (5:?)0
(en!ue0 leptospirosis0 chi.un!un'a0 alaria0 s'philis0 onchocerci*
asis0 an( hepatitis A0 70 an( C$ 7oth teste( positive +or alaria0 #ith
all other tests ne!ative$
The patient #as prescribe( a (ail' (ose o+ 200 ! o+ (o>'*
c'cline0 #hich has been sho#n to cause si!ni@cant re(uctions
in l'phe(ea in patients #ith 8ra(e ::::: &= +ollo#in! 1224
onths o+ treatent0 re!ar(less o+ circulatin! @larial status (-an(
et al$0 2012)$ Althou!h soe ris. o+ a(verse eFects has been
reporte( in a(inistration o+ (o>'c'cline to chil(ren un(er 10
'ears ol(0 the treatin! ph'sician conclu(e( that #ith appropriate
sa+et' onitorin! the bene@ts o+ (o>'c'cline +or this patient out*
#ei!he( the potential ris.s$ A((itionall'0 the balloonin! +ores.in
o+ the bo' #as reove( throu!h circucision$ The biopsie( tissue
#as unrear.able$
-$ Doe et al$ % Acta Tropica 134 (2014) 1316 1I
=i!$ 3$ -ap o+ Sierra &eone Sho#in! &ocation o+ Case 3atient an( =ailial 1elations$
3$ Discussion
&'phatic @lariasis is rarel' s'ptoatic in chil(ren0 espe*
ciall' those #ho are less than 10 'ears ol(0 an( #hen chil(ren (o
have s'ptos &= rarel' presents as l'phe(ea (7oc.arie et al$0
200K)$ A coprehensive eta*anal'sis o+ &= in chil(ren b' ,itt an(
Attesen (2001) i(enti@e( onl' I cases o+ l'phe(ea or elephan*
tiasis o+ a lib in chil(ren less than 10 'ears ol( in the publishe(
literature0 ost o+ #hich occurre( in the i(*20th centur'$ A stu('
+ro the i(*1KK0s o+ 441 :n(ian chil(ren (a!es 114 'ears) #ith
&= +oun( onl' 12 cases o+ l'phe(ea (5arinath et al$0 2000)$ Thus0
the case patient appears to have an unusual presentation o+ lo#er
lib enlar!eent in a )*'ear ol( bo' in+ecte( #ith ,$ bancro+ti$
Ane possible cause o+ this con!enital (e+orit' is -ilro'Bs (is*
ease0 an autosoal (oinant !enetic (isor(er characteri/e( b'
l'phe(ea0 especiall' l'phe(ea o+ the le!s0 that is present
at birth or (evelops in in+anc' (-a.houl et al$0 2002)$ A 2011 case
report +ro an &= non*en(eic area (escribe( a health' +ull*ter
ne#born #ith no coplications (urin! pre!nanc' or (eliver' #ho
(evelope( s#ellin! o+ his ri!ht le! soon a+ter birth (3ere/*Crespo
et al$0 2011)$ As observe( in our case0 the lo#er lib s#ellin!
#as ore pronounce( in the (istal part o+ the le! an( +oot$ The
s.in sho#e( non*pittin! e(ea an( no s#ellin! si!ns in the area$
The +ail' histor' #as also siilar to ours$ The !ran(other an(
soe aternal relatives #ere also +oun( to be aFecte( #ith l'*
phe(ea0 inclu(in! the chil(Bs !ran(other$ 7ase( on the +ail'
histor' an( clinical e>aination alone0 the patient #as (ia!nose(
#ith -ilro'Bs (isease$ Siilar cases (escribe( else#here #ere
also clinicall' (ia!nose( as -ilro'Bs (isease (Connell et al$0 200EG
4itsiou*T/eli et al$0 2010)$ -ilro'Bs (isease ha( also been associ*
ate( #ith s#ellin! o+ the !enitals0 as per a case report +ro 8ue!lio
et al$ (2003) o+ !iant scrotal l'phe(ea (ue to -ilro'Bs (isease$
<o relative o+ the )*'ear ol( bo' #as .no#n to have been born #ith
l'phe(ea0 but up to 10J o+ persons #ith -ilro'Bs (isease a' be
as'ptoatic (7rice et al$0 200I)$ A(vance( olecular (ia!nostics
#oul( be reHuire( to con@r that the l'phe(ea in the )*'ear
ol( observe( in Sierra &eone #as (ue to -ilro'Bs (isease$
This stu(' su!!ests that l'phe(ea (ue to &= in chil(ren a'
be (i+@cult to (ia!nose because o+ the rarit' o+ s'ptoatic &=
in the pe(iatric population$ :n &=*en(eic areas0 the possibilit' o+
other causes o+ l'phe(ea in in+ecte( chil(ren shoul( be care*
+ull' consi(ere( an( -ilro'Bs (isease shoul( not be preaturel'
e>clu(e( as a possible (ia!nosis$
Ac.no#le(!eents
This stu(' #as supporte( b' the Center +or <e!lecte( Tropical
Diseases0 &iverpool School o+ Tropical -e(icine0 throu!h +un(in!
+ro D=:D an( 8S4$ The +un(ers pla'e( no role in the publication
o+ this anuscript$ ,e #oul( li.e to ac.no#le(!e the support o+
the stu(' counities0 in(ivi(uals0 an( research assistants #ho
helpe( in un(erta.in! this stu('$
1e+erences
A((iss0 D$0 2010$ The 6th -eetin! o+ the 8lobal Alliance to 9liinate &'phatic =il*
ariasis" A hal+*tie revie# o+ l'phatic @lariasis eliination an( its inte!ration
#ith the control o+ other ne!lecte( tropical (iseases$ 3arasit$ ?ectors 30 100$
7oc.arie0 -$6$0 Ta'lor0 -$6$0 8'apon!0 6$A$0 200K$ Current practices in the ana!eent
o+ l'phatic @lariasis$ 9>pert 1ev$ Anti :n+ect$ Ther$ )0 IKI60I$
7oc.arie0 -$6$0 Tisch0 D$6$0 4astens0 ,$0 Ale>an(er0 <$D$9$0 Diber0 O$0 7oc.arie0 =$0
:ba0 9$0 Alpers0 -$3$0 4a/ura0 6$,$0 2002$ -ass treatent to eliinate @lariasis
in 3apua <e# 8uinea$ < 9n!l 6 -e($ 34)0 1E411E4E$
7rice0 8$0 Chil(0 A$5$0 9vans0 A$0 7ell0 1$0 -ansour0 S$0 7urnan(0 4$0 Sar+ara/i0 -$0 6eFer'0
S$0 -ortier0 3$0 200I$ -ilro' (isease an( the ?98=1*3 utation phenot'pe$ 6$
-e($ 8enet$ 420 KE102$
Chu0 7$4$0 5ooper0 3$6$0 7ra(le'0 -$5$0 -c=arlan(0 D$A$0 Attesen0 9$A$0 2010$ The eco*
noic bene@ts resultin! +ro the @rst E 'ears o+ the 8lobal 3ro!rae to
9liinate &'phatic =ilariasis (2000200))$ 3&oS <e!l$ Trop$ Dis$ 40 e)0E$
16 -$ Doe et al$ % Acta Tropica 134 (2014) 1316
4oroa0 6$7$0 Sesa'0 S$0 Sonnie0 -$0 5o(!es0 -$5$0 Sahr0 =$0 Ohan!0 D$0 7oc.arie0 -$6$0
2013$ :pact o+ three roun(s o+ ass (ru! a(inistration on l'phatic @lariasis
in areas previousl' treate( +or onchocerciasis in Sierra &eone$ 3&oS <e!l$ Trop$
Dis$ )0 e22)3$
-an(0 S$0 Debrah0 A$D$0 4larann0 ;$0 7atsa0 &$0 -ar+o*Debre.'ei0 D$0 4#arten!0 A$0
Specht0 S$0 7el(a*Doene0 A$0 =iers0 1$0 Ta'lor0 -$0 A(jei0 A$0 5oerau+0 A$0
2012$ Do>'c'cline iproves @larial l'phe(ea in(epen(ent o+ active @larial
in+ection" a ran(oi/e( controlle( trial$ Clin$ :n+ect$ Dis$ II0 621630$
-a.houl0 :$1$0 Sujov0 3$0 8hane0 <$0 7ronshtein0 -$0 2002$ 3renatal (ia!nosis o+
-ilro'Bs priar' con!enital l'phe(ea$ 3renat$ Dia!n$ 220 E23E26$
3ere/*Crespo0 -$0 7etlloch0 :$0 -artine/*-iravete0 -$T$0 7allester0 :$0 &ucas0 A$0 -atai>0
6$0 2011$ Con!enital lo#er lib enlar!eent in a ne#born$ 3e(iatr$ Deratol$
2E0 I)KIE0$
Sheno'0 1$4$0 7oc.arie0 -$6$0 2011$ &'phatic @lariasis in chil(ren" clinical +ea*
tures0 in+ection bur(ens an( +uture prospects +or eliination$ 3arasitolo!' 13E0
1IIK1I6E$
,itt0 C$0 Attesen0 A$0 2001$ &'phatic @lariasis" an in+ection o+ chil(hoo($ Trop$ -e($
:nt$ 5ealth 60 IE2606$
,orl( 5ealth Ar!ani/ation0 2013$ 8lobal 3ro!rae to 9liinate &'phatic =ilari*
asis" pro!ress report on ass (ru! a(inistration0 2012$ ,.l' 9pi(eiol$ 1ec$
EE0 3EK400$
Connell0 =$0 7rice0 8$0 -ortier0 3$0 200E$ 3henot'pic characteri/ation o+ priar'
l'phe(ea$ Ann$ <$D$ Aca($ Sci$ 11310 140146$
8ue!lio0 8$0 Puija(a0 9$0 Salas0 5$0 Daels0 3$0 Tejeri/o0 6$0 Chernobils.'0 ?$0 8iQ(ice0 C$0
Daia0 A$0 2003$ 8iant scrotal l'phe(ea cause( b' -ilro'Bs (isease$ Arch 9sp
;rol$ I6 (E)0 K4KKI2$
5arinath0 7$C$0 1e(('0 -$?$1$0 7han(ari0 7$7$0 -ehta0 ?$4$0 Chaturve(i0 3$0 3rajapati0
<$C$0 8upta0 1$4$C$0 2000$ =ilaria associate( clinical ani+estations in chil(ren
in an en(eic area an( orbi(it' control b' iunoonitorin! an( optial
D9C therap'" Seva!ra e>perience$ :n(ian 6$ Clin$ 7ioche$ 1I (Suppl$ 1)0 11E
126$
5o(!es0 -$5$0 Sonnie0 -$0 Tura'0 5$0 Conteh0 A$0 -acCarth'0 =$0 Sesa'0 S$0
2012$ -aintainin! eFective ass (ru! a(inistration +or l'phatic @la*
riasis throu!h in*process onitorin! in Sierra &eone$ 3arasit$ ?ectors I0
232$
4itsiou*T/eli0 S$0 ?rettou0 C$0 &e/e0 9$0 -a.r'thanasis0 3$0 4anava.is0 9$0 ,illes0 3$0
2010$ -ilro'Bs priar' con!enital l'phe(ea in a ale in+ant an( revie# o+
the literature$ :n ?ivo 240 30K314$
4oroa0 6$7$0 7an!ura0 -$-$0 5o(!es0 -$5$0 7ah0 -$S$0 Ohan!0 D$0 7oc.arie0 -$6$0 2012$
&'phatic @lariasis appin! b' iunochroato!raphic test car(s an( base*
line icro@laria surve' prior to ass (ru! a(inistration in Sierra &eone$
3arasit$ ?ectors I0 10$