Download Treatment of human lagochilascariasis with

TRANSACTIONS OF THE ROYAL SOCIETY OF TROPICAL MEDICINE AND HYGIENE (1998) 92, 223-224
I Short Report I
Treatment of human lagochilascariasis
with ivermectin: first case report from
Ecuador
Manuel Calvopiiia1, Angel G. Guevaral, Marcelo
Herreral, Miguel Serrano2 and Ronald H. Guderi­
an 1
1 Departamento de Investigaciones Clinicas, Hospi­
tal Vozandes, Quito, Ecuador; 2Servicio de Otorrinolaring­
ologia, Hospital 'Eugenio Espejo', Quito, Ecuador
Keywords: lagochilascariasis, Lagochilascaris minor, case re­
port, chemotherapy, ivermectin, Ecuador
Lagochilascariasis is a rare parasitic infection in hu­
mans. During the past decade, human infections by
Lagochilascaris minor LEIPER, 1909 have been docu­
mented in Brazil, Colombia, Venezuela, Bolivia, Trini­
dad and Tobago, Suriname, Costa Rica and Mexico
(VOLCAN et al., 1982; VELOSO et al., 1992; OLLI-GOIG
et al., 1996; VARGAS-OCAMPO & ALVARADO-ALEMAN,
1997). A human infection usually presents as a tumour­
al lesion in the cervical region, involving nodules, pseu­
docysts or abscesses with draining sinuses. However,
aggressive invasion of the cranial fossae, temporal bone,
paranasal sinuses, dental alveoli, tonsils, ear, and mas­
toid region can occur (CAMPOS et al., 1988). Prognosis
is poor if these essential structures are compromised,
and several fatalities have been reported (ROSEMBERG et
al., 1986; ORIHUELA et al., 1987).
Therapeutic difficulties are still encountered when
managing L. minor infections, especially since recrudes­
cence frequently occurs. Many drugs with variable effi­
cacy have been employed. Relapse of infection has
occurred even after the use of drug combinations (BoT­
ERO & LITTLE, 1984). However, the combination of
prolonged use of oral levamisole and surgical extirpation
of the mass seemed to have a favourable outcome
(AGUILAR-NASCIMENTO et al., 1993). The efficacy of
ivermectin for human use in Lagochilascaris infection is
unknown. Reported here is the first human case ofLago­
chilascaris infection in Ecuador and its response to iver­
mectin treatment.
Case report
A 30 years old female agricultural worker from the
coastal province of Esmeraldas in Ecuador presented
with a large left cervical mass, which had progressively
increased in size during the previous 18 months. Her
chief complaint was related to the continuous discharge
containing small white worms that drained from the
mass. Six months previously she had undergone surgical
removal of the mass, but its rapid recurrence at the orig­
inal site in the following weeks prompted the patient to
seek further medical assistance. The patient lived in a
tropical forested area and had close contact with domes­
tic animals such as cats and dogs. She had ingested wild
animal meat, such as Cuniculus paca (guanta), Dasyproc­
ta agouti (guatusa), wild cats, wild pigs, Dasypus novem­
cinctus (armadillo) and Pelis pardalis (tigrillo). No similar
symptom was noticed in any of her relatives or neigh­
bours.
Physical examination found her in good health, but
with a large, hard, non-tender palpable mass with indef­
inite borders measuring approximately 6-7 cm in diam-
223
eter in the left lateral region of the neck. From 2
prominent draining fistulae in the tumoural mass, a pu­
rulent discharge containing small white worms was ob­
served.
The
exudate contained
Gram-positive
organisms, but no acid-fast bacillus or parasite egg was
seen. Ultrasonography of the neck area disclosed a het­
erogeneous mass with indefinite limits but with no inva­
sion of deep structures. Computerized tomography
showed a mass with various compartments in the subcu­
taneous and dermal tissue level. There was no invasion
of any bone or cartilage structure. The isolated white
worms, both females and males, measured 8-16 mm in
length and were identified as L. minor according to the
description given by SPRENT (1971).
The patient was treated weekly with ivermectin (Mec­
tizan®, 6 mg tablets), 300 µg/kg of body weight. There
was a slow, continuous reduction in the volume of pu­
rulent discharge, with a corresponding decrease in the
number of emerging parasites. After 4 weeks of treat­
ment, closure of both fistulae occurred. With 6 weeks of
treatment, the size of the mass had decreased to a diam­
eter of 2 cm, and it had become hard with limited bor­
ders. Because of the persistence of the tumoural mass
even after 2 more weeks of treatment, surgical resection
was done under general anaesthesia. Histopathological
examination revealed fibrotic tissue with a granuloma­
tous reaction still surrounding viable nematodes. Iver­
mectin was administered for 4 more weeks after surgery.
Currently, after 8 months of follow-up, the patient is do­
ing well with no evidence of disease recurrence.
Discussion
The ideal drug for the treatment of lagochilascariasis
would have efficacy against the adult parasite as well as
against the different larval stages and eggs, since all the
different stages of the parasite can be seen in the lesions.
Existing evidence indicates that ivermectin may be ef­
fective against L. minor. In vitro, ivermectin impedes the
embryogenesis of L. minor eggs (CAMPOS et al., 1988).
A human infection in Brazil was treated using a prepa­
ration for oral use in sheep, and resolved successfully af­
ter 9 months (14 doses) of treatment (BENTO et al.,
1993). In the present study, ivermectin for human use
produced an acceptable clinical evolution. No adverse
effect was observed. Ivermectin has a definite advantage
over the formerly employed benzimidazoles, use of
which has been abandoned due to associated clinical
side effects and intolerance (BENTO et al., 1993). Iver­
mectin was effective in killing all exposed parasites, as
shown by the reduction of associated inflammation and
the elimination of purulent discharge containing the
parasites.
However, even after 8 weeks of chemotherapy, viable
parasites were still present. This was primarily due to
the nature of the mass, which is formed of multiple
small deep abscesses containing parasites. It is surmised
that, due to poor drug penetration and absence of me­
chanical drainage, killing of all the existing worms was
impeded. W hether administering the drug for a longer
period would have resulted in complete resolution of the
infection is unknown. However, to assure complete
cure, the remaining reduced mass was surgically resect­
ed. The reduction in size of the mass and the develop­
ment of definite borders after ivermectin treatment
facilitated its resection. Ivermectin has a long half-life
and is widely disseminated throughout the body (FINK
& PORRAS, 1989), potentiating the destruction of any L.
minor present. It was therefore given for 4 weeks after
surgery to kill any parasite that might have escaped due
to the manipulation of the mass during the surgical pro­
cedure.
Acknowledgements
Address for correspondence: Manuel Calvopifia H, Departa­
mento de Investigaciones Clinicas, Hospital Vozandes, HCJB,
Casilla 17-17-691, Quito, Ecuador.
Donation of the ivermectin tablets and the professional sup­
port of Dr Philippe Gaxotte, Medical Director of Merck Sharp
& Dohme lnterpharma, La Celle Saint Cloud, France is ac-
MANUEL CALVOPINA ETAL.
224
knowledged. T he surgical intervention and medical supplies
provided by the AXXIS Medical Center were greatly appreciat­
ed.
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Received 13 October 1997; revised 11 December 1997; ac­
cepted for publication 11 December 1997
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