ABSTRACTUrticaria is one of the most common dermatoallergic conditions occurring mainly in children and adolescents and in atopic individuals. The term of chronic idiopathic urticaria (CIU) is used when the pathophysiological mechanisms remain unclear. Chronic infections and parasitic infestations have been suggested to play an important role in the etiology of CIU. In the present paper, we describe a case of chronic, apparently idiopathic urticaria in an adult woman where Dirofilaria has been recognized to play a pathogenic role in the determination of the cutaneous disease

Alessandra Orsi

Alessandra Vultaggio

Andrea Matucci

Berlow

Enrico Maggi

Gaia Deleonardi

Greaves

Kuna

Liutu

Muro

Oliviero Rossi

Paola Parronchi

Romagnani

Sergio Romagnani

Allergology International

English

Matucci, Andrea

Parronchi, Paola

Rossi, Oliviero

Vultaggio, Alessandra

Deleonardi, Gaia

Orsi, Alessandra

Maggi, Enrico

Romagnani, Sergio

Open Access Repository

 Allergology International  (2004)  53 : 383–385 Case Report A case of chronic urticaria due to  Dirofilaria  infestation Andrea Matucci, 1  Paola Parronchi, 1  Oliviero Rossi, 1  Alessandra Vultaggio, 1  Gaia Deleonardi, 1  Alessandra Orsi, 2  Enrico Maggi 1  and Sergio Romagnani 1 1 Department of Internal Medicine, Section of Immunoallergology and Respiratory Diseases, University of Florence and  2 Laboratory of Microbiology, Virology and Parasitology, Azienda Ospedaliera Careggi, Florence, Italy A BSTRACT Urticaria is one of the most common dermatoallergicconditions occurring mainly in children and adoles-cents and in atopic individuals. The term of chronicidiopathic urticaria (CIU) is used when the pathophysio-logical mechanisms remain unclear. Chronic infec-tions and parasitic infestations have been suggested toplay an important role in the etiology of CIU. In thepresent paper, we describe a case of chronic, appar-ently idiopathic urticaria in an adult woman where Dirofilaria  has been recognized to play a pathogenicrole in the determination of the cutaneous disease. Key words: chronic urticaria,  Dirofilaria . I NTRODUCTION Urticaria is one of the most common dermatoallergicconditions, mainly occurring in children and adoles-cents, as well as in atopic individuals. Urticaria isdefined by wheals, with or without angioedema, appear-ing within minutes to hours, usually disappearing within24 h and accompained by pruritus. Urticaria is causedby cutaneous mast cell and/or basophil degranulationdue to immunological and non-immunological causes.Following degranulation, potent vasoactive mediatorsthat induce vasodilatation are released, resulting inclassical cutaneous manifestations. 1  Among the mediators,histamine plays an essential role, but other substances,such as eicosanoids, kinins, serotonin and neuropeptides,may also be involved. 2  Moreover, mononuclear cells,lymphocytes, eosinophils, platelets or endothelial cellsare also involved in the late-phase reaction, in whichcytokines, chemokines and adhesion molecules play amajor role. 3,4  Persistence of urticaria beyond 6 weeks isdefined as chronic urticaria. The term ‘chronic idiopathicurticaria’ (CIU) is used when the pathophysiologicalmechanisms remain unclear. The etiopathophysiology ofchronic urticaria is usually more difficult to determinethan that of acute urticaria. 5  Chronic infections andparasitic infestations have been suggested to play animportant role in the etiology of CIU and it has beensuggested that viral, bacterial, parasitic or fungal anti-gens may form immune complexes together with theirspecific antibodies, then bind to the vascular walls andactivate complement. 6  In any case, it is quite unusualto recognize a parasite infestation as the only causeof CIU.In the present paper, we describe a case of chronicapparently idiopathic urticaria in an adult woman inwhom  Dirofilaria  has been recognized to play a patho-genic role in the determination of the cutaneous disease. C ASE   REPORT A 63-year-old woman presented with CIU that hadstarted 1 year previously. The patient showed gianturticaria with intense pruritus resistant to common anti-histamine treatment and only partially and temporarilyresponsive to corticosteroid therapy. The cutaneouslesions were classical pale red wheals, more than 10 cmin diameter, diffuse to the entire body but prevalentlylocalized at the trunk and legs, showing less than 24 hduration and diseappearing without chromatic altera-tions. Allergological investigations performed in different Correspondence: Dr Andrea Matucci, Department of InternalMedicine, Section of Immunoallergology and Respiraty Diseases,Policlinico di Careggi/University of Florence, Viale Morgagni85, 50134 Firenze, Italy. Email: a.matucci@dmi.unifi.itReceived 19 December 2003. Accepted for publication18 May 2004.384 A MATUCCI ET AL.departments in the absence of therapy were completelynegative. In particular, skin prick tests for inhalant andfood allergens were negative, as were patch tests forcommon haptens. Specific and total IgE had beendetected with normal results. The history of the patientwas negative for allergic manifestations at the ocular orrespiratory level. Moreover, the patient denied anyhypersensitivity reactions to foods or the occurrence ofany adverse reaction including urticaria/angioedemaafter drug intake.At clinical examination, in addition to classical wheals,some nodules were detected at the right axillary regionand these nodules were characterized echographically ascystic nodules showing peculiar features.Laboratory tests showed normal levels of γ-globulins(IgG 1500 mg/dL, IgM 89 mg/dL and IgA 154 mg/dL),aspartate aminotransferase, alanine aminotransferaseand glutamyl transpeptidase and a normal number ofwhite cells in the peripheral blood. In particular, the eosi-nophils ranged between 2 and 4%. Analysis of the phe-notypic profile of circulating CD3+ cells showed a normalnumber of CD4+ and CD8+ T cells. The complement (C3and C4) fractions were within the normal range. Cultureanalysis of repeated fecal specimens was negative forparasitic or mycotic infestations. The prick test for inha-lant and food allergens was negative. The detection ofspecific serum IgE antibodies (CAP RAST FEIA; Pharma-cia, Upsalla, Sweden) failed to show any sensitizationtowards foods or inhalant allergens, the total serum IgElevel being 75 kU/L.An excisional biopsy of one of the cutaneous nodulesrevealed inflammatory tissue containing a high numberof eosinophils and structures consistent with parasitic sec-tions. The specimen was then analyzed in the Laboratoryof Microbiology, Parasitology and Virology of FlorenceUniversity Hospital, where the parasites were identified.The parasites showed characteristic morphological fea-tures of Dirofilaria repens (Fig. 1). Radical surgicalexcision of the nodules was performed and the clinicalcondition of the patient improved rapidly, with completedisappearence of urticaria within 2 weeks.DISCUSSIONIn this paper, we present the first case, to our know-ledge, of human dirofilariasis associated with CIU. Dirofilaria species are nematode parasites that arewidely distributed in southern Europe, mostly infectingdogs and cats but, more rarely, also humans.7In humans, these parasites seldom or never completetheir life cycle and do not usually reach the adult stage,so that filaremia is exceptional. The parasites are trans-mitted to humans by zooanthropophilic blood-suckinginsects. Two types of clinical presentation have beendescribed: pulmonary and subcutaneous dirofilariasis.7In this second form, subcutaneous nodules can bedistributed widely all over the body resulting from thecolonization of filarids. Definitive diagnosis and treat-ment most often result from surgical excision of thenodules, which also allows microscopic examination.The segregation of worms into the tissues is probably thecause of the usual absence of circulating anti-Dirofilariaantibodies in patients. Thus, excisional biopsy is bothdiagnostic and therapeutic. In the present patient, diag-nosis and resolution of the clinical manifestations wereachieved by exisional biopsy.Thus, it is recommended that dirofilariasis should betaken into account by both clinicians and pathologists inthe differential diagnosis of patients with CIU and sub-cutaneous nodules.Fig. 1 Transverse section of a female Dirofilaria repens,identified at the histological examination of one of the cuta-neous nodules, showing the presence of oocytes in the uterinesections. The digestive system is a unique tube that can vary interms of its position in the body cavity, diameter and contents.In the present case, amorphous material is present. Sections ofthe female reproductive organs show the existence of two tubes,as shown in the present histological specimen. The lateralspicula can be seen in the section because it is close to thecaudal end. CHRONIC URTICARIA AND PARASITES 385 R EFERENCES 1 Greaves MW. Chronic urticaria.  N. Engl. J. Med.  1995; 332 : 1767–72.2 Berlow RJ, Ross EL, MacDonald DM, Black AK,Greaves MW. Mast cells and T lymphocytes in chronicurticaria.  Clin. Exp. Allergy  1995;  25 : 317–22.3 Kuna P, Reddigari ST, Schall TJ, Rucinski D, Viksman MY,Kaplan AP. RANTES, a monocyte and T lymphocyte chemo-tactic cytokine releases histamine from human basophils. J. Immunol.  1992;  146 : 636–42.4 Romagnani P, De Paulis A, Beltrame C  et al.  Tryptase-chymase double-positive human mast cells express theeotaxin receptor CCR3 and are attracted by CCR3-bibding chemokines.  Am. J. Pathol.  1999;  155 :1195–204.5 Liutu M, Kalimo K, Uksila J, Kalimo H. Etiological aspectsof chronic urticaria.  Int. J. Dermatol.  1998;  37 : 515–19.6 Greaves MW. Chronic idiopatic urticaria (CIU) and Helicobacter pylori : Not directly causative, but could therebe a link?  Aci. Int.  2001;  13 : 23–6.7 Muro A, Genchi C, Cordero M, Simon F. Human diro-filariasis in the European Union.  Parasitol. Today  1998; 15 : 386–9.

A Case of Chronic Urticaria Due to Dirofharia Infestation

Crossref

Elsevier - Publisher Connector 

A Case of Chronic Urticaria Due to Dirofharia Infestation 

 Allergology International
 
 (2004) 
 
53
 
: 383–385
 
Case Report
 
A case of chronic urticaria due to 
 
Dirofilaria
 
 infestation
 
Andrea Matucci,
 
1
 
 Paola Parronchi,
 
1
 
 Oliviero Rossi,
 
1
 
 Alessandra Vultaggio,
 
1
 
 
Gaia Deleonardi,
 
1
 
 Alessandra Orsi,
 
2
 
 Enrico Maggi
 
1
 
 and Sergio Romagnani
 
1
 
1
 
Department of Internal Medicine, Section of Immunoallergology and Respiratory Diseases, University of 
Florence and 
 
2
 
Laboratory of Microbiology, Virology and Parasitology, Azienda Ospedaliera Careggi, 
Florence, Italy
 
A
 
BSTRACT
 
Urticaria is one of the most common dermatoallergic
conditions occurring mainly in children and adoles-
cents and in atopic individuals. The term of chronic
idiopathic urticaria (CIU) is used when the pathophysio-
logical mechanisms remain unclear. Chronic infec-
tions and parasitic infestations have been suggested to
play an important role in the etiology of CIU. In the
present paper, we describe a case of chronic, appar-
ently idiopathic urticaria in an adult woman where
 
Dirofilaria
 
 has been recognized to play a pathogenic
role in the determination of the cutaneous disease.
 
Key words:
 
chronic urticaria, 
 
Dirofilaria
 
.
 
I
 
NTRODUCTION
 
Urticaria is one of the most common dermatoallergic
conditions, mainly occurring in children and adoles-
cents, as well as in atopic individuals. Urticaria is
defined by wheals, with or without angioedema, appear-
ing within minutes to hours, usually disappearing within
24 h and accompained by pruritus. Urticaria is caused
by cutaneous mast cell and/or basophil degranulation
due to immunological and non-immunological causes.
Following degranulation, potent vasoactive mediators
that induce vasodilatation are released, resulting in
classical cutaneous manifestations.
 
1
 
 Among the mediators,
histamine plays an essential role, but other substances,
such as eicosanoids, kinins, serotonin and neuropeptides,
may also be involved.
 
2
 
 Moreover, mononuclear cells,
lymphocytes, eosinophils, platelets or endothelial cells
are also involved in the late-phase reaction, in which
cytokines, chemokines and adhesion molecules play a
major role.
 
3,4
 
 Persistence of urticaria beyond 6 weeks is
defined as chronic urticaria. The term ‘chronic idiopathic
urticaria’ (CIU) is used when the pathophysiological
mechanisms remain unclear. The etiopathophysiology of
chronic urticaria is usually more difficult to determine
than that of acute urticaria.
 
5
 
 Chronic infections and
parasitic infestations have been suggested to play an
important role in the etiology of CIU and it has been
suggested that viral, bacterial, parasitic or fungal anti-
gens may form immune complexes together with their
specific antibodies, then bind to the vascular walls and
activate complement.
 
6
 
 In any case, it is quite unusual
to recognize a parasite infestation as the only cause
of CIU.
In the present paper, we describe a case of chronic
apparently idiopathic urticaria in an adult woman in
whom 
 
Dirofilaria
 
 has been recognized to play a patho-
genic role in the determination of the cutaneous disease.
 
C
 
ASE
 
 
 
REPORT
 
A 63-year-old woman presented with CIU that had
started 1 year previously. The patient showed giant
urticaria with intense pruritus resistant to common anti-
histamine treatment and only partially and temporarily
responsive to corticosteroid therapy. The cutaneous
lesions were classical pale red wheals, more than 10 cm
in diameter, diffuse to the entire body but prevalently
localized at the trunk and legs, showing less than 24 h
duration and diseappearing without chromatic altera-
tions. Allergological investigations performed in different
 
Correspondence: Dr Andrea Matucci, Department of Internal
Medicine, Section of Immunoallergology and Respiraty Diseases,
Policlinico di Careggi/University of Florence, Viale Morgagni
85, 50134 Firenze, Italy. Email: a.matucci@dmi.unifi.it
Received 19 December 2003. Accepted for publication
18 May 2004.
384 A MATUCCI ET AL.
departments in the absence of therapy were completely
negative. In particular, skin prick tests for inhalant and
food allergens were negative, as were patch tests for
common haptens. Specific and total IgE had been
detected with normal results. The history of the patient
was negative for allergic manifestations at the ocular or
respiratory level. Moreover, the patient denied any
hypersensitivity reactions to foods or the occurrence of
any adverse reaction including urticaria/angioedema
after drug intake.
At clinical examination, in addition to classical wheals,
some nodules were detected at the right axillary region
and these nodules were characterized echographically as
cystic nodules showing peculiar features.
Laboratory tests showed normal levels of γ-globulins
(IgG 1500 mg/dL, IgM 89 mg/dL and IgA 154 mg/dL),
aspartate aminotransferase, alanine aminotransferase
and glutamyl transpeptidase and a normal number of
white cells in the peripheral blood. In particular, the eosi-
nophils ranged between 2 and 4%. Analysis of the phe-
notypic profile of circulating CD3+ cells showed a normal
number of CD4+ and CD8+ T cells. The complement (C3
and C4) fractions were within the normal range. Culture
analysis of repeated fecal specimens was negative for
parasitic or mycotic infestations. The prick test for inha-
lant and food allergens was negative. The detection of
specific serum IgE antibodies (CAP RAST FEIA; Pharma-
cia, Upsalla, Sweden) failed to show any sensitization
towards foods or inhalant allergens, the total serum IgE
level being 75 kU/L.
An excisional biopsy of one of the cutaneous nodules
revealed inflammatory tissue containing a high number
of eosinophils and structures consistent with parasitic sec-
tions. The specimen was then analyzed in the Laboratory
of Microbiology, Parasitology and Virology of Florence
University Hospital, where the parasites were identified.
The parasites showed characteristic morphological fea-
tures of Dirofilaria repens (Fig. 1). Radical surgical
excision of the nodules was performed and the clinical
condition of the patient improved rapidly, with complete
disappearence of urticaria within 2 weeks.
DISCUSSION
In this paper, we present the first case, to our know-
ledge, of human dirofilariasis associated with CIU.
 
Dirofilaria species are nematode parasites that are
widely distributed in southern Europe, mostly infecting
dogs and cats but, more rarely, also humans.7
In humans, these parasites seldom or never complete
their life cycle and do not usually reach the adult stage,
so that filaremia is exceptional. The parasites are trans-
mitted to humans by zooanthropophilic blood-sucking
insects. Two types of clinical presentation have been
described: pulmonary and subcutaneous dirofilariasis.7
In this second form, subcutaneous nodules can be
distributed widely all over the body resulting from the
colonization of filarids. Definitive diagnosis and treat-
ment most often result from surgical excision of the
nodules, which also allows microscopic examination.
The segregation of worms into the tissues is probably the
cause of the usual absence of circulating anti-Dirofilaria
antibodies in patients. Thus, excisional biopsy is both
diagnostic and therapeutic. In the present patient, diag-
nosis and resolution of the clinical manifestations were
achieved by exisional biopsy.
Thus, it is recommended that dirofilariasis should be
taken into account by both clinicians and pathologists in
the differential diagnosis of patients with CIU and sub-
cutaneous nodules.
Fig. 1 Transverse section of a female Dirofilaria repens,
identified at the histological examination of one of the cuta-
neous nodules, showing the presence of oocytes in the uterine
sections. The digestive system is a unique tube that can vary in
terms of its position in the body cavity, diameter and contents.
In the present case, amorphous material is present. Sections of
the female reproductive organs show the existence of two tubes,
as shown in the present histological specimen. The lateral
spicula can be seen in the section because it is close to the
caudal end.
 CHRONIC URTICARIA AND PARASITES 385
 
R
 
EFERENCES
 
1 Greaves MW. Chronic urticaria. 
 
N. Engl. J. Med.
 
 1995;
 
332
 
: 1767–72.
2 Berlow RJ, Ross EL, MacDonald DM, Black AK,
Greaves MW. Mast cells and T lymphocytes in chronic
urticaria. 
 
Clin. Exp. Allergy
 
 1995; 
 
25
 
: 317–22.
3 Kuna P, Reddigari ST, Schall TJ, Rucinski D, Viksman MY,
Kaplan AP. RANTES, a monocyte and T lymphocyte chemo-
tactic cytokine releases histamine from human basophils.
 
J. Immunol.
 
 1992; 
 
146
 
: 636–42.
4 Romagnani P, De Paulis A, Beltrame C 
 
et al.
 
 Tryptase-
chymase double-positive human mast cells express the
eotaxin receptor CCR3 and are attracted by CCR3-
bibding chemokines. 
 
Am. J. Pathol.
 
 1999; 
 
155
 
:
1195–204.
5 Liutu M, Kalimo K, Uksila J, Kalimo H. Etiological aspects
of chronic urticaria. 
 
Int. J. Dermatol.
 
 1998; 
 
37
 
: 515–19.
6 Greaves MW. Chronic idiopatic urticaria (CIU) and
 
Helicobacter pylori
 
: Not directly causative, but could there
be a link? 
 
Aci. Int.
 
 2001; 
 
13
 
: 23–6.
7 Muro A, Genchi C, Cordero M, Simon F. Human diro-
filariasis in the European Union. 
 
Parasitol. Today
 
 1998;
 
15
 
: 386–9.


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A Case of Chronic Urticaria Due to Dirofharia Infestation

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