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Sweet's syndrome and genital infection: Two case reports
Letter
Table 2 Results of immunological analysis in S/9 patients with PR (the bioptic specimens were obtained a fortnight after clinical cure) P Sex Age Biopsy Epidermal Dermal specimen Leu6+ cells’ Leu6+ cellsb AP FD LP GL RM
M M F M F
35 27 20 25 29
HS HS HS HS HS
11 14 10 12 13
0.00 0.00 0.00 0.00 0.00
HS = healthy skin. a Average number of Leu 6+ cells high power per field. This number was obtained in 3 sections (8 high power fields, 40X) (normal: 12 Leu 6+ cells per high power field, 40X). b Average number of Leu 6+ cells high power per field. This number was obtained in 3 sections (8 high power fields, 40X) (normal: no Leu 6 + cells per high power field, 40 X).
tween primary and secondary lesions. Also, all the healthy skin specimens showed a high-grade decrease of epidermal Leu 6 + cells, very similar to the value found in the lesional skin specimens (Table, 1). We would like to emphasize that dermal Leu 6 + cells were found in healthy skin specimens only (2 Leu 6 + cells/8 high power per fields, 40 X ) (Table 1). In the cutaneous specimens obtained on the 15th day following cure the number of epidermal Leu 6 + cells was normal (as mentioned above) and no dermal Leu 6 + cells were found. Our data show a clear decrease in epidermal CDla+ Langerhans cell density in both lesional and non-lesional skin of patients with PR in its active phase compared to the Sweet’s syndrome and genital infection: Two case won To the Editor: Two patients with Sweet’s syndrome and genital infection by Neisseria gonorrhoeae were observed in our department. This association has not been previously reported in the literature. The patients, a 50-year-old Caucasian and a 35 year-old Caucasian gypsy, both women, presented with fever, arthralgia and tender erythematous plaques on the face, neck and arms, with a mamillated surface (Fig. 1). Both patients presented leucocytosis with neutrophilia and the light microscopic
to the editor
clinically normal skin of the same patients after complete cure. The decrease of the number of LC both in lesional and non-lesional skin might suggest the involvement of LC in the presentation of an undefined immunogen to CD4+ cells [3,5,6]. G. Calabro
*,
F. De Natale, A. Schettino
Clinica Demzatologica, Universitir Ii”, 80131 Naples, Italy
degli Shcdi di Napoli
“Federico
111Garsia de Silva L, Gardner PS. Pityriasis rosea: A virological study. Br J Dermato11968,80:514-515.
[21Sechar, L, Weismann K, Kobayasi T. Pitytiasis rosea eruption in secondary syphilis: An isomorphic phenomenon. Cutis 1985;35:403-404. [31 Aiba S, Tagami H. Immunohistologic studies in pityriasis rosea: Evidence for cellular immune reaction in the lesional epidennis.‘Arch Dermatol 1985;121:761-765. [41 Aiba S, Tagami H. Hla-Dr antigen expression on cheratinocytes surface in dermatoses characterized by lymphocytic exocytosis (e.g., pityriasis rosea). Br J Dermatol 19&15111:285-294. 151 Takaki Y, Miyaxaki M. Cytolytic degeneration of keratinocytes adjacent to Langerhans’ cell in pityriasis rosea (Sibert). Acta Derm Venereol (Stockh) 1976;56:99-103. 161Sugiura H, Miysuchi H and Uehara M. Evolutionary changes of immunohistological characteristics of secondary lesions in pityriasis rosea. Arch Dermatol Res 1988;280:405-410. Corresponding author. Tel.: ( + 39)81-7462440; Fax: ( + 39)81-7462442. l
SSDIO926-9959(95)00021-6
examination of the biopsy specimens showed a dense perivascular dermal infiltrate consisting predominantly of neutrophils (Fig. 2). The first patient had a urinary infection and Enterococci was demonstrated in the urine culture; the second one referred a history of tonsilitis two weeks before the onset of the skin lesions. In both cases, Neisseria gonorrhoeae was cultured from an endouterine smear. A good response to antimicrobial and corticosteroid therapy was observed. The second patient relapsed a month after cure (while corticosteroid dosage was being reduced) and Neisseria gonorrhoeae was again isolated from the cervical dis-
/
Letter to the editor
Letter
charge. Therapy with spectinomicin and oral steroids achieved remission. No further relapses were noted in either patient. Sweet’s syndrome was first described by Sweet in 1964 [l]. In spite of the numerous patient series reported in the literature [2-51, the etiopathogenesis of this syndrome remains obscure. Sweet’s syndrome has been reported in association with multiple diseases, including myeloproliferative and malignant disorders [6], connective tissue diseases [7], vaccinations [8] and, most of all, infectious conditions [2-51. Four of the eight patients originally described by Sweet [l] had either a previous or coincidental infectious disorder. Furthermore, there was evidence of an infectious process, usually an upper respiratory tract infection, in about one-third of the patients reported in the larger series [2-51. Sweet’s syndrome has also been occasionally reported in association with urinary infections [9]. However, genital infections were rarely reported. Su and Liu [2] described a patient with Sweet’s syndrome and vaginitis. A review of the literature indicates that Sweet’s syndrome has been preceded by genital tuberculosis [4], bartholinitis [9] and urethroprostatitis caused by Ureoplasma urealitycum [lo]. To the best of our knowledge, no case of associated gonococcal infection has been reported previously. Both our patients had gonococcal cervicitis in association with the typical clinical and histologic features of Sweet’s syndrome. In addition, our first patient had a urinary infection; in the other patient, the clinical picture of acute neutrophilic dermatosis was preceded by an upper respiratory infection. It is interesting to note that this last patient experienced a recurrence of the skin lesions along with a reinfection by Nezkseria gonorrhoeae, suggesting that the association between the two conditions was more than casual.
Fig. 1. Symmetric Fig. 2. H&E neutrophils.
X 10: Epidermal
multiple
and subepidermal
erythematous
to the editor
We believe that the association of genital infection with Sweet’s syndrome may occur more frequently than previously reported. Therefore, appropriate investigations for gonococcal cervicitis should always be performed in sexually active women with Sweet’s syndrome. Cecilia Mouca *, Cristina Gabciela Marques Pinto
Tapadinhas,
Department of Dermatology, Curry Beneficik?ncia, 1000 Lisbon, Portugal
Cabral
Isabel Conchon, Hospital,
Run
da
ill
Sweet R. An acute febrile neutrophilic dermatosis. Br J Dermatol 1964,76:349-356. 121Su W. Liu H. Diagnostic criteria for Sweet’s syndrome. Cutis 1986;37:167-170. [31 Dias M, Conchon I, Afonso A et al. Sindrome de Sweet-RevisHo dos cases diagnosticados no Hospital Curry Cabral, de 1980 a 1991. Arqui Med 1992;6:253-257. 141 Silva R, Garcia e Silva L, Morais Cardoso J. Sidrome de Sweet - Revisio de 35 cases. Acta Mtdica Portuguesa 1985;6;221-227. neutrophilic dermatosis - A 151 Jordaan H. Acute febrile histopathological study of 37 patients and a review of the literature. Am J Dermatophatol 1989;11:99-111. [61 Cooper P. Imtes D, Greer K Acute febrile neutrophilic dermatosis (Sweet’s syndrome) and myeloproliferative disorders. Cancer 1983;51:1518-1526. E, Gaveau D, Piette F. et al. Acute febrile neu[71 Delaporte trophilic dermatosis (Sweet’s syndrome). Arch Dermatol 1989;125:1101-1104. Bl Maddox R, Motley R. Sweet’s syndrome: A severe complication of pneumococcal vaccination following emergency splenectomy. Br J Surg 1990;70:809-810. [91 Sitjas D, Puig L, Cuatrecasas M et al. Acute febrile neutrophilic dermatosis (Sweet’s syndrome). Int J Dermatol 1993;32:261-268. DOI Stieler W, Schulte C. Localized Sweet syndrome in urethroprostatitis caused by ureoplasma Hautarxt 1988;39:658-661.
Corresponding 7969515. l
author.
Tel.:
351-797494-1;
fax:
351-l-
SSDI 0926-9959(95)00022-4
nodules and plaques on the neck and arms (First patient).
edema, exocytosis
of neutrophils,
and intense dermal
infihrate
consisting
predominantly
of
Table 2 Results of immunological analysis in S/9 patients with PR (the bioptic specimens were obtained a fortnight after clinical cure) P Sex Age Biopsy Epidermal Dermal specimen Leu6+ cells’ Leu6+ cellsb AP FD LP GL RM
M M F M F
35 27 20 25 29
HS HS HS HS HS
11 14 10 12 13
0.00 0.00 0.00 0.00 0.00
HS = healthy skin. a Average number of Leu 6+ cells high power per field. This number was obtained in 3 sections (8 high power fields, 40X) (normal: 12 Leu 6+ cells per high power field, 40X). b Average number of Leu 6+ cells high power per field. This number was obtained in 3 sections (8 high power fields, 40X) (normal: no Leu 6 + cells per high power field, 40 X).
tween primary and secondary lesions. Also, all the healthy skin specimens showed a high-grade decrease of epidermal Leu 6 + cells, very similar to the value found in the lesional skin specimens (Table, 1). We would like to emphasize that dermal Leu 6 + cells were found in healthy skin specimens only (2 Leu 6 + cells/8 high power per fields, 40 X ) (Table 1). In the cutaneous specimens obtained on the 15th day following cure the number of epidermal Leu 6 + cells was normal (as mentioned above) and no dermal Leu 6 + cells were found. Our data show a clear decrease in epidermal CDla+ Langerhans cell density in both lesional and non-lesional skin of patients with PR in its active phase compared to the Sweet’s syndrome and genital infection: Two case won To the Editor: Two patients with Sweet’s syndrome and genital infection by Neisseria gonorrhoeae were observed in our department. This association has not been previously reported in the literature. The patients, a 50-year-old Caucasian and a 35 year-old Caucasian gypsy, both women, presented with fever, arthralgia and tender erythematous plaques on the face, neck and arms, with a mamillated surface (Fig. 1). Both patients presented leucocytosis with neutrophilia and the light microscopic
to the editor
clinically normal skin of the same patients after complete cure. The decrease of the number of LC both in lesional and non-lesional skin might suggest the involvement of LC in the presentation of an undefined immunogen to CD4+ cells [3,5,6]. G. Calabro
*,
F. De Natale, A. Schettino
Clinica Demzatologica, Universitir Ii”, 80131 Naples, Italy
degli Shcdi di Napoli
“Federico
111Garsia de Silva L, Gardner PS. Pityriasis rosea: A virological study. Br J Dermato11968,80:514-515.
[21Sechar, L, Weismann K, Kobayasi T. Pitytiasis rosea eruption in secondary syphilis: An isomorphic phenomenon. Cutis 1985;35:403-404. [31 Aiba S, Tagami H. Immunohistologic studies in pityriasis rosea: Evidence for cellular immune reaction in the lesional epidennis.‘Arch Dermatol 1985;121:761-765. [41 Aiba S, Tagami H. Hla-Dr antigen expression on cheratinocytes surface in dermatoses characterized by lymphocytic exocytosis (e.g., pityriasis rosea). Br J Dermatol 19&15111:285-294. 151 Takaki Y, Miyaxaki M. Cytolytic degeneration of keratinocytes adjacent to Langerhans’ cell in pityriasis rosea (Sibert). Acta Derm Venereol (Stockh) 1976;56:99-103. 161Sugiura H, Miysuchi H and Uehara M. Evolutionary changes of immunohistological characteristics of secondary lesions in pityriasis rosea. Arch Dermatol Res 1988;280:405-410. Corresponding author. Tel.: ( + 39)81-7462440; Fax: ( + 39)81-7462442. l
SSDIO926-9959(95)00021-6
examination of the biopsy specimens showed a dense perivascular dermal infiltrate consisting predominantly of neutrophils (Fig. 2). The first patient had a urinary infection and Enterococci was demonstrated in the urine culture; the second one referred a history of tonsilitis two weeks before the onset of the skin lesions. In both cases, Neisseria gonorrhoeae was cultured from an endouterine smear. A good response to antimicrobial and corticosteroid therapy was observed. The second patient relapsed a month after cure (while corticosteroid dosage was being reduced) and Neisseria gonorrhoeae was again isolated from the cervical dis-
/
Letter to the editor
Letter
charge. Therapy with spectinomicin and oral steroids achieved remission. No further relapses were noted in either patient. Sweet’s syndrome was first described by Sweet in 1964 [l]. In spite of the numerous patient series reported in the literature [2-51, the etiopathogenesis of this syndrome remains obscure. Sweet’s syndrome has been reported in association with multiple diseases, including myeloproliferative and malignant disorders [6], connective tissue diseases [7], vaccinations [8] and, most of all, infectious conditions [2-51. Four of the eight patients originally described by Sweet [l] had either a previous or coincidental infectious disorder. Furthermore, there was evidence of an infectious process, usually an upper respiratory tract infection, in about one-third of the patients reported in the larger series [2-51. Sweet’s syndrome has also been occasionally reported in association with urinary infections [9]. However, genital infections were rarely reported. Su and Liu [2] described a patient with Sweet’s syndrome and vaginitis. A review of the literature indicates that Sweet’s syndrome has been preceded by genital tuberculosis [4], bartholinitis [9] and urethroprostatitis caused by Ureoplasma urealitycum [lo]. To the best of our knowledge, no case of associated gonococcal infection has been reported previously. Both our patients had gonococcal cervicitis in association with the typical clinical and histologic features of Sweet’s syndrome. In addition, our first patient had a urinary infection; in the other patient, the clinical picture of acute neutrophilic dermatosis was preceded by an upper respiratory infection. It is interesting to note that this last patient experienced a recurrence of the skin lesions along with a reinfection by Nezkseria gonorrhoeae, suggesting that the association between the two conditions was more than casual.
Fig. 1. Symmetric Fig. 2. H&E neutrophils.
X 10: Epidermal
multiple
and subepidermal
erythematous
to the editor
We believe that the association of genital infection with Sweet’s syndrome may occur more frequently than previously reported. Therefore, appropriate investigations for gonococcal cervicitis should always be performed in sexually active women with Sweet’s syndrome. Cecilia Mouca *, Cristina Gabciela Marques Pinto
Tapadinhas,
Department of Dermatology, Curry Beneficik?ncia, 1000 Lisbon, Portugal
Cabral
Isabel Conchon, Hospital,
Run
da
ill
Sweet R. An acute febrile neutrophilic dermatosis. Br J Dermatol 1964,76:349-356. 121Su W. Liu H. Diagnostic criteria for Sweet’s syndrome. Cutis 1986;37:167-170. [31 Dias M, Conchon I, Afonso A et al. Sindrome de Sweet-RevisHo dos cases diagnosticados no Hospital Curry Cabral, de 1980 a 1991. Arqui Med 1992;6:253-257. 141 Silva R, Garcia e Silva L, Morais Cardoso J. Sidrome de Sweet - Revisio de 35 cases. Acta Mtdica Portuguesa 1985;6;221-227. neutrophilic dermatosis - A 151 Jordaan H. Acute febrile histopathological study of 37 patients and a review of the literature. Am J Dermatophatol 1989;11:99-111. [61 Cooper P. Imtes D, Greer K Acute febrile neutrophilic dermatosis (Sweet’s syndrome) and myeloproliferative disorders. Cancer 1983;51:1518-1526. E, Gaveau D, Piette F. et al. Acute febrile neu[71 Delaporte trophilic dermatosis (Sweet’s syndrome). Arch Dermatol 1989;125:1101-1104. Bl Maddox R, Motley R. Sweet’s syndrome: A severe complication of pneumococcal vaccination following emergency splenectomy. Br J Surg 1990;70:809-810. [91 Sitjas D, Puig L, Cuatrecasas M et al. Acute febrile neutrophilic dermatosis (Sweet’s syndrome). Int J Dermatol 1993;32:261-268. DOI Stieler W, Schulte C. Localized Sweet syndrome in urethroprostatitis caused by ureoplasma Hautarxt 1988;39:658-661.
Corresponding 7969515. l
author.
Tel.:
351-797494-1;
fax:
351-l-
SSDI 0926-9959(95)00022-4
nodules and plaques on the neck and arms (First patient).
edema, exocytosis
of neutrophils,
and intense dermal
infihrate
consisting
predominantly
of