III I
l
I
I 9
I
I
III
I
Unilateral laterothoracic exanthem in children: A new disease? Christine Bodemer, MD, and Yves de Prost, MD Paris, France
Background: We have examined 18 children with a similar laterothoracic exanthem that appears to represent a distinct entity. Objective: Our purpose was to describe the characteristic signs and clinical course of this eruption and its epidemiology data. Methods: We observed the clinical course of the eruption in each child. Results: The eruption has characteristic features. It occurs in a homogeneous age group (mean 23.3 months). It is initially unilateral and localized close to the axilla. The basic lesion is eczematous or scarlatiniform. The eruption evolves in two phases: it spreads centrifugally during the first 8 days and becomes more widespread on the tenth to fifteenth days, with predominant involvement on the half of the body initially affected. The lesions resolve spontaneously within 4 weeks. The long-term course is uneventful. Conclusion: The similarity of the cases suggests the existence of a new clinical entity. Many features favor a viral origin. (J AM ACAD DERMATOL1992;27:693-6.) In 3 years we have observed 18 children with a predominantly laterothoracic exanthem with a characteristic clinical course. It probably represents a new clinical entity. To our knowledge, this eruption has been reported only once before. 1 This disease probably has an infectious cause, but no bacterial or viral agent has been demonstrated. CASE REPORTS The features of these cases are summarized in Table I. The series consisted of 14 girls (77.8%) and 4 boys (22.2%) between the ages of 14 months and 4 years (mean 23.3 months), with an eruption that lasted 8 to 20 days. These children had no significant medical history. Most (77.8%) lived in the Ile-de-France region or in its immediate vicinity and had not recently traveled out of this region. The eruption had characteristic features. It was initially unilateral and localized (axilla, 38.9%; laterothoracic close to the axilla; 44.4%; flank, 16.7%), on the left in 12 cases and on the right in 6 cases. It consisted of a scarlatiniform or eczematous eruption that was poorly demarcated in 88.9% of cases. In two cases the upper part of the lesion was well demarcated and more erythematous. Pruritus was reported by 61.1% of patients, but this From the Department of Dermatology, Hospital Necker Enfants malades. Accepted for publication April 24, 1992. No reprints available.
16/1/38872
symptom is difficult to interpret because of the age of these children. Excoriations were observed in only one case. A purpufic spot, suggestive of an inoculation site, but without any history of a bite, was detected in two children (patients 2 and 13). The clinical course of the eruption consisted of two phases. (1) There was centrifugal extension of the initial lesion occasionally separated by intervals of normal skin. On the eighth day, all patients had a morbilliform or eczematous exanthem with a hemicorporeal distribution (Figs. 1-3). (2) There were varying degrees of dissemination between the eighth and fifteenth days (83.3% of cases). The face, palms, and soleswere always spared and the bilateral exanthem retained a markedly asymmetric appearance with predominant involvement of the half of the body initially affected. The lesions resolved spontaneously during the third week. Only residual dryness of the skin was often present during the fourth week. All patients were completely clear by 1 month. Seven children were treated with antibiotics, during the second week of the eruption in four cases (1, 2, 10, and 16) and several days before the eruption (because of fever or upper respiratory tract infection) in three other cases (5, 9, and 12). In six cases treatment with a topical corticosteroid was given after the first week of the eruption, and in two of these cases hydroxyzinewas added because of pruritus. None of these treatments appeared to alter the appearance or duration of the eruption (see Table I). Accompanying signs were inconstant and variable: regional adenopathy draining the initial lesion (axillary or
693
Journal of the American Academy of Dermatology
Bodemer and de Prost
694
T a b l e I. Patient characteristics
PentI IAep
Onset (site)
I' t~ i ization
aden~
No.
Sex
(too)
1 2 3 4
F F F F
36 24 I8 21
Axillary Axillary Axillary Axillary
+ + + +
0 + + +
5 6 7 8 9
F F F F M
21 24 15 14 18
Axillary Axillary Flank Flank Laterothoracic
+ 0 0 + +
0 + + 0 0
10 11 12
F F NI
48 26 24
Laterothoracic Axillary Laterothoracic
+ + +
0 0 +
13 14 15 16
F M F F
24 14 16 25
Laterothoracic Laterothoracic Laterothoracic Flank
+ + + 0
+ 0 0 0
17
F
22
Laterothoracic
+
0
18
M
30
Laterothoracic
+
0
I
Accompanying signs None None None 38 ~ C, conjunctivitis 2 days before onset 38 ~ C 2 days before onset None Rhinopharyngitis Rhinopharyngitis 39 ~ C, sore throat, otitis 8 days before onset 38 ~ C, rhinopharyngitis None 39 ~ to 40 ~ C 9 days before onset None None None 39 ~ C, sore throat 10 days after onset Vomiting and diarrhea 5 days before onset 39 ~ C, 5 days after onset with diarrhea
J
Treatments
Duration
Penicillin V Penicillin V None None
3 3 3 4
wk wk wk wk
Penicillin A None None None Penicillin A
3 wk 2 wk 2 wk 1 mo 3 wk
Penicillin A None Penicillin A
3 wk 3 wk 3 wk
None None None Penicillin A
4 wk 3 wk 3 wk 3 wk
None
4 wk
None
4 wk
T a b l e I I . L a b o r a t o r y investigations
No. Blood cell count AST, A L T Serologic tests Spiroplasma groups IV, XIII, XXII, X HBs Borreliosis Mycoplasma EBV CMV Toxoplasmosis Parvovirus B19 Coxsackie virus Rickettsia HIV Throat-anus swabs
]
Findings/Results
9/9 5/5
Normal Normal
5/5 4/4 5/ 5 3/3 10/10 10/10 5/5 4/4 5/5 3/ 3 1/ 1 15/15
Negative Negative Negative Negative Negative Negative Negative Negative Negative Negative Negative Negative
.4LT, Alanineaminotransferase;AST, aspartateaminotransferase;CMV, cytomegalovirusIgG and [gM;EBF, ENtein-Barrvirus [gG and IgM;HBs, HBs antigen and anti-HBs antibody; HIV, human immunodeficiencyvirus; Parvovirus B19, [gG and IgM.
inguinal) was detected in seven cases; in seven children fever developed that was associated with an ear infection or diarrhea in five cases (see Table I); an enanthem was never observed. None of the children had a relevant medical history and had not taken any drugs other than the antibiotics. None
of them had a history of inoculation via a wound, scratch, or bite. Similar cases in the child's family or friends were found in two patients; patients 4 and 5 were twin sisters in whom the eruption developed at an interval of 10 days. Patient 17 was a child whose aunt (25 years old) was said to have developed the same exanthem at the same time.
Volume 27 Number 5, Part 1 November 1992
Unilateral laterothoracic exanthem
695
Fig. 1. Case 1. Unilateral exanthem with hemicorporal distribution (eighth day). Fig. 2. Case 1. Eczematous eruption. Fig. 3. Case 2. Unilateral exanthem with peripheral extension and purpuric spot near axilia.
An analysis of environmental factors revealed that 72.2% of children had a garden and/or had walked in the country during the 48 hours preceding the eruption. Sixty-one percent had a pet dog. The eruption occurred m spring in eight cases, in winter in four cases, in autumn in three cases, and in summer in three cases. Laboratory investigations were performed but not according to any defined protocol (Table II). None revealed any abnormalities. A skin biopsy was performed in two cases. In the first case (No. 11), the exanthem had been present for 4 days.
This biopsy specimen revealed areas of epidermal spongiosis accompanied by exocytosis of mononuclear ce/ls. No necrotic lesions were observed. A moderate perivascular and periappendageal lympho-histiocytic infiltrate was observed in the dermis. Warthin-Starry stain was negative. In the second case (No. 15), the lesions had been present for 15 days. The biopsy specimen revealed only a mild, nonspecific superficial dermatitis. The long-term course of these patients was uneventful. For the first six cases, the follow-up is now more than 1 year (follow-up of 3 years in one case). No unexplained
Journal of the American Academy of Dermatology
696 Bodemerand de Prost
clinical manifestations (neurologic, articular, cardiac) have occurred. DISCUSSION Many articles on exanthems in children have been published, 2-4 but only one described five children with a "localized erythema" similar to that observed in our patients.1 These authors suggested an inoculation disease, possibly related to Spiroplasma infection, as they found weakly positive serologic resuits for group XIII Spiroplasma in one of their patients. 5 Our investigations did not confirm this hypothesis because Spiroplasma serology was negative in five cases (see Table II). This exanthem of young children is probably more frequent than suggested by the literature. An infectious cause, most likely viral, is the most probable explanation. Features that favor a viral origin include the age of the children, the presence of two familial cases, the frequency of associated rhinopharyngitis, the lack of efficacy of broad-spectrum antibiotics, and the possibly seasonal nature of the eruption. However, an inoculation dermatosis
cannot be excluded as suggested by the initially localized nature of the eruption followed by centrifugal spread, the presence of regional lymphadenopathy in some cases, and a purpuric spot suggestive of an arthropod bite in two patients. We thank Dr. Vincent Tranie, Dr. Claude Chastel (Virology Laboratory, Faculty of Medicine, Brest, France), and Dr. Sylvie Fraitag (Anatomopathology Laboratory, H6pital Necker Enfants-Malades, Paris) for technical assistance. REFERENCES
1. Taieb A, Megraud F, LeRoy JM, et al. Eryth~me localis~ avec ad~nopathie r6gionale de l'enfant: une maladie d'inoculation? Ann Dermatol V6n6rb,ol 1986;I 13:1023-4. 2. Lerner AM, Klein JO, Cherry JD, et al. New viral exant.hems. N Engl J Med 1963;269:678-85. 3. GoodyearH.M, Laidler PW, Price EH, et al. Acute infectious erythema in children: a clinico-microbiologicalstudy. Br J Dermatol 1991;124:433.8. 4. Grossman KL, RasmussenJE. Recent advances in pediatric infectious disease and their impact on dermatology. J AM ACADDERMATOL1991;24:379-89. 5. ChastelC, Abalain-CoUocMZ, Gilot B, et al. Spiroplasmes et arthropodes h6matophages: perspectives en pathologic tropic,ale. Bull Soc Pathol Exot Filiales 1985;78:769-79.
l
I
I 9
I
I
III
I
Unilateral laterothoracic exanthem in children: A new disease? Christine Bodemer, MD, and Yves de Prost, MD Paris, France
Background: We have examined 18 children with a similar laterothoracic exanthem that appears to represent a distinct entity. Objective: Our purpose was to describe the characteristic signs and clinical course of this eruption and its epidemiology data. Methods: We observed the clinical course of the eruption in each child. Results: The eruption has characteristic features. It occurs in a homogeneous age group (mean 23.3 months). It is initially unilateral and localized close to the axilla. The basic lesion is eczematous or scarlatiniform. The eruption evolves in two phases: it spreads centrifugally during the first 8 days and becomes more widespread on the tenth to fifteenth days, with predominant involvement on the half of the body initially affected. The lesions resolve spontaneously within 4 weeks. The long-term course is uneventful. Conclusion: The similarity of the cases suggests the existence of a new clinical entity. Many features favor a viral origin. (J AM ACAD DERMATOL1992;27:693-6.) In 3 years we have observed 18 children with a predominantly laterothoracic exanthem with a characteristic clinical course. It probably represents a new clinical entity. To our knowledge, this eruption has been reported only once before. 1 This disease probably has an infectious cause, but no bacterial or viral agent has been demonstrated. CASE REPORTS The features of these cases are summarized in Table I. The series consisted of 14 girls (77.8%) and 4 boys (22.2%) between the ages of 14 months and 4 years (mean 23.3 months), with an eruption that lasted 8 to 20 days. These children had no significant medical history. Most (77.8%) lived in the Ile-de-France region or in its immediate vicinity and had not recently traveled out of this region. The eruption had characteristic features. It was initially unilateral and localized (axilla, 38.9%; laterothoracic close to the axilla; 44.4%; flank, 16.7%), on the left in 12 cases and on the right in 6 cases. It consisted of a scarlatiniform or eczematous eruption that was poorly demarcated in 88.9% of cases. In two cases the upper part of the lesion was well demarcated and more erythematous. Pruritus was reported by 61.1% of patients, but this From the Department of Dermatology, Hospital Necker Enfants malades. Accepted for publication April 24, 1992. No reprints available.
16/1/38872
symptom is difficult to interpret because of the age of these children. Excoriations were observed in only one case. A purpufic spot, suggestive of an inoculation site, but without any history of a bite, was detected in two children (patients 2 and 13). The clinical course of the eruption consisted of two phases. (1) There was centrifugal extension of the initial lesion occasionally separated by intervals of normal skin. On the eighth day, all patients had a morbilliform or eczematous exanthem with a hemicorporeal distribution (Figs. 1-3). (2) There were varying degrees of dissemination between the eighth and fifteenth days (83.3% of cases). The face, palms, and soleswere always spared and the bilateral exanthem retained a markedly asymmetric appearance with predominant involvement of the half of the body initially affected. The lesions resolved spontaneously during the third week. Only residual dryness of the skin was often present during the fourth week. All patients were completely clear by 1 month. Seven children were treated with antibiotics, during the second week of the eruption in four cases (1, 2, 10, and 16) and several days before the eruption (because of fever or upper respiratory tract infection) in three other cases (5, 9, and 12). In six cases treatment with a topical corticosteroid was given after the first week of the eruption, and in two of these cases hydroxyzinewas added because of pruritus. None of these treatments appeared to alter the appearance or duration of the eruption (see Table I). Accompanying signs were inconstant and variable: regional adenopathy draining the initial lesion (axillary or
693
Journal of the American Academy of Dermatology
Bodemer and de Prost
694
T a b l e I. Patient characteristics
PentI IAep
Onset (site)
I' t~ i ization
aden~
No.
Sex
(too)
1 2 3 4
F F F F
36 24 I8 21
Axillary Axillary Axillary Axillary
+ + + +
0 + + +
5 6 7 8 9
F F F F M
21 24 15 14 18
Axillary Axillary Flank Flank Laterothoracic
+ 0 0 + +
0 + + 0 0
10 11 12
F F NI
48 26 24
Laterothoracic Axillary Laterothoracic
+ + +
0 0 +
13 14 15 16
F M F F
24 14 16 25
Laterothoracic Laterothoracic Laterothoracic Flank
+ + + 0
+ 0 0 0
17
F
22
Laterothoracic
+
0
18
M
30
Laterothoracic
+
0
I
Accompanying signs None None None 38 ~ C, conjunctivitis 2 days before onset 38 ~ C 2 days before onset None Rhinopharyngitis Rhinopharyngitis 39 ~ C, sore throat, otitis 8 days before onset 38 ~ C, rhinopharyngitis None 39 ~ to 40 ~ C 9 days before onset None None None 39 ~ C, sore throat 10 days after onset Vomiting and diarrhea 5 days before onset 39 ~ C, 5 days after onset with diarrhea
J
Treatments
Duration
Penicillin V Penicillin V None None
3 3 3 4
wk wk wk wk
Penicillin A None None None Penicillin A
3 wk 2 wk 2 wk 1 mo 3 wk
Penicillin A None Penicillin A
3 wk 3 wk 3 wk
None None None Penicillin A
4 wk 3 wk 3 wk 3 wk
None
4 wk
None
4 wk
T a b l e I I . L a b o r a t o r y investigations
No. Blood cell count AST, A L T Serologic tests Spiroplasma groups IV, XIII, XXII, X HBs Borreliosis Mycoplasma EBV CMV Toxoplasmosis Parvovirus B19 Coxsackie virus Rickettsia HIV Throat-anus swabs
]
Findings/Results
9/9 5/5
Normal Normal
5/5 4/4 5/ 5 3/3 10/10 10/10 5/5 4/4 5/5 3/ 3 1/ 1 15/15
Negative Negative Negative Negative Negative Negative Negative Negative Negative Negative Negative Negative
.4LT, Alanineaminotransferase;AST, aspartateaminotransferase;CMV, cytomegalovirusIgG and [gM;EBF, ENtein-Barrvirus [gG and IgM;HBs, HBs antigen and anti-HBs antibody; HIV, human immunodeficiencyvirus; Parvovirus B19, [gG and IgM.
inguinal) was detected in seven cases; in seven children fever developed that was associated with an ear infection or diarrhea in five cases (see Table I); an enanthem was never observed. None of the children had a relevant medical history and had not taken any drugs other than the antibiotics. None
of them had a history of inoculation via a wound, scratch, or bite. Similar cases in the child's family or friends were found in two patients; patients 4 and 5 were twin sisters in whom the eruption developed at an interval of 10 days. Patient 17 was a child whose aunt (25 years old) was said to have developed the same exanthem at the same time.
Volume 27 Number 5, Part 1 November 1992
Unilateral laterothoracic exanthem
695
Fig. 1. Case 1. Unilateral exanthem with hemicorporal distribution (eighth day). Fig. 2. Case 1. Eczematous eruption. Fig. 3. Case 2. Unilateral exanthem with peripheral extension and purpuric spot near axilia.
An analysis of environmental factors revealed that 72.2% of children had a garden and/or had walked in the country during the 48 hours preceding the eruption. Sixty-one percent had a pet dog. The eruption occurred m spring in eight cases, in winter in four cases, in autumn in three cases, and in summer in three cases. Laboratory investigations were performed but not according to any defined protocol (Table II). None revealed any abnormalities. A skin biopsy was performed in two cases. In the first case (No. 11), the exanthem had been present for 4 days.
This biopsy specimen revealed areas of epidermal spongiosis accompanied by exocytosis of mononuclear ce/ls. No necrotic lesions were observed. A moderate perivascular and periappendageal lympho-histiocytic infiltrate was observed in the dermis. Warthin-Starry stain was negative. In the second case (No. 15), the lesions had been present for 15 days. The biopsy specimen revealed only a mild, nonspecific superficial dermatitis. The long-term course of these patients was uneventful. For the first six cases, the follow-up is now more than 1 year (follow-up of 3 years in one case). No unexplained
Journal of the American Academy of Dermatology
696 Bodemerand de Prost
clinical manifestations (neurologic, articular, cardiac) have occurred. DISCUSSION Many articles on exanthems in children have been published, 2-4 but only one described five children with a "localized erythema" similar to that observed in our patients.1 These authors suggested an inoculation disease, possibly related to Spiroplasma infection, as they found weakly positive serologic resuits for group XIII Spiroplasma in one of their patients. 5 Our investigations did not confirm this hypothesis because Spiroplasma serology was negative in five cases (see Table II). This exanthem of young children is probably more frequent than suggested by the literature. An infectious cause, most likely viral, is the most probable explanation. Features that favor a viral origin include the age of the children, the presence of two familial cases, the frequency of associated rhinopharyngitis, the lack of efficacy of broad-spectrum antibiotics, and the possibly seasonal nature of the eruption. However, an inoculation dermatosis
cannot be excluded as suggested by the initially localized nature of the eruption followed by centrifugal spread, the presence of regional lymphadenopathy in some cases, and a purpuric spot suggestive of an arthropod bite in two patients. We thank Dr. Vincent Tranie, Dr. Claude Chastel (Virology Laboratory, Faculty of Medicine, Brest, France), and Dr. Sylvie Fraitag (Anatomopathology Laboratory, H6pital Necker Enfants-Malades, Paris) for technical assistance. REFERENCES
1. Taieb A, Megraud F, LeRoy JM, et al. Eryth~me localis~ avec ad~nopathie r6gionale de l'enfant: une maladie d'inoculation? Ann Dermatol V6n6rb,ol 1986;I 13:1023-4. 2. Lerner AM, Klein JO, Cherry JD, et al. New viral exant.hems. N Engl J Med 1963;269:678-85. 3. GoodyearH.M, Laidler PW, Price EH, et al. Acute infectious erythema in children: a clinico-microbiologicalstudy. Br J Dermatol 1991;124:433.8. 4. Grossman KL, RasmussenJE. Recent advances in pediatric infectious disease and their impact on dermatology. J AM ACADDERMATOL1991;24:379-89. 5. ChastelC, Abalain-CoUocMZ, Gilot B, et al. Spiroplasmes et arthropodes h6matophages: perspectives en pathologic tropic,ale. Bull Soc Pathol Exot Filiales 1985;78:769-79.
