- Email: [email protected]
Unilateral plantar erythema nodosum
654
Journal of the American Academy of Dermatology April 1994
Brief communications
Unilateral plantar erythema nodosum Naoto Ohtake, MD, Tatsuyoshi Kawamura, MD, Chie Akiyama, MD, Masutaka Fume, MD, and Kunihiko Tamaki, MD Yamanashi, Japan We report a case of erythema nodosum on the right sale. To our knowledge this is the second reported case of unilateral plantar erythema nodosum. CASE REPORT A 27-year-old Japanese woman had recurrent painful nodules on her right sole for 6 months. Individual nodules spontaneously resolved within I to 4 weeks. She had no systemic symptoms and no history of trauma. Examination revealed five erythematous, firm, tender, subcutaneous, 2 em nodules on the plantar aspect of the right foot (Fig. I). The results of the following studies were within normal limits or negative: erythrocyte sedimentation rate, blood cell counts, C-reactive protein, antistreptolysin 0 antibodies, antistreptokinase antibodies, hepatitis B surface antigen, antibodies for syphilis, antinuclear antibodies, rheumatoid factor, C3, C4, urinalysis, and bacterial culture from the tonsils. Roentgenograms of the chest and right foot were also normal. A biopsy specimen showed infiltration of lymphohistiocytes in the septae of fat lobules and inflammation of septal blood vessels (Fig. 2). The lesions were diagnosed as erythema nodosum and treated with oralloxoprofen sodium, 120 mg three times per day. The lesions completely regressed 20 days later. The lesions recurred after stopping loxoprofen sodium, but complete control was again achieved with readministration of the drug. No recurrence has been noted after 6 months.
Fig. 1. Tender nodules on plantar aspect of right foot.
DISCUSSION
Lesions of erythema nodosum usually appear symmetrically on the extensor aspects of the legs, with occasional involvement of the thighs or forearms. Unilateral l or plantar- 3 involvement uncommonly occurs. Hem and Shwayder" first reported unilateral plantar erythema nodosum. To our knowl-
From the Department of Dermatology, Yamanashi Medical University. Reprint requests: Naoto Ohtake, MD, Department of Dermatology, Yarnanashi Medical University, 1110 Shimokatoh, Tarnaho, Nakakoma, Yamanashi 409-38, Japan. JAM ACAD DERMATOL
1994;30:654-5.
Copyright ® [994 by the American Academy of Dermatology, Inc. 0[90-9622/94 $3.00 + 0
16/54/50277
Fig. 2. Infiltration of lymphohistiocytes in septae of fat lobules with inflammation of septal blood vessels. (Hematoxylin-eosin stain; X9.)
Journal of the American Academy of Dermatology Volume 30, Number 4
edge, ours is the second reported case of this disorder. Erythema nodosum is frequently associated with streptococcal infection, mycoses, tuberculosis, sarcoidosis, or drug allergy. None of these was present in our patient. Although mycoplasma infection does not usually induce erythema nodosum, antimycoplasma IgM was positive in Hem and Shwayder's case." Because we did not check antimycoplasma titer during the active stage of disease in our patient, we cannot comment on whether mycoplasma infection is
Brief communications 655 associated with unilateral plantar erythema nodosum. REFERENCES I. Hicks JH. Erythema nodosum in patients with tinea pedis and onychomycosis. South Med J 1977;70:27-8. 2. Berger TG, Tappero J. Traumatic plantar urticaria or plantar erythema nodosum [Letter]. J AM ACAD DERMATOL 1989;20:701-2. 3. Hosokawa H. Erythema nodosum on sales in a patient with pulmonary tuberculosis [in Japanese]. Skin Res 1990;
32:34-9. 4. Hem AE, Shwayder TA. Unilateral plantar erythema nodosum. J AM ACAD DERMATOL 1992;26:259-60.
Becker's nevus associated with chromosomal mosaicism and congenital adrenal hyperplasia Julien R. Lambert, MD,a Patrick Willems, PhD,b Roger Abs, MD,c and Bernadette Van Roy, BScb Antwerp, Belgium Becker's nevus is a relatively common unilateral hyperpigmented hairy cutaneous hamartoma. It is usually acquired, but a few cases have been considered congenital.' It can be associated with structural abnormalities, such as hypoplasia of the ipsilateral breast, areola and nipple, with limb asymmetry, spina bifida, scoliosis, and pectus carinatum. Although Becker's nevus occurs much more frequently in males (male/female ratio: 4.5: 1), these associated anomalies have been reported more often in females. The pathogenesis of Becker's nevus has not been elucidated, but several characteristics suggest that androgen stimulation may contribute. We describe a patient with Becker's nevus associated with hypotrophy of the shoulder girdle, congenital adrenal hyperplasia with severe hirsutism, and chromosomal mosaicism. CASE REPORT
A 20-year-oldwoman had hirsutism. At birth she had a blue discoloration on the left shoulder. As a childdiffuse From the Departments of Dermatology," Medical Genetics," and Endocrinology," University of Antwerp. Reprint requests: Julien R. Lambert, Department of Dermatology, University Hospital of Antwerp (U.I.A.), Wilrijkstraat 10, 2650 Edegem, Belgium.
.J
AM ACAD DERMATOL
1994;30:655-7.
Copyright @ 1994 by the American Academy of Dermatology, Inc. 0190-9622/94 $3.00
+0
16/54/49937
excessive hair growth gradually developed. She had a regular menstrual cycle from the age of 12 years. Severe hirsutism with a score of 34/44 according to the scale of Ferriman and Gallwey was observed.? The left shoulder and upper portion of the arm were more hairy than other parts of the body and showed a dark brown pigmentation that suggested a diagnosis of Becker's nevus. The left shoulder girdle was hypotrophic compared with the right but without functional deficit. Thin frontal hair was present, but no other signs of masculinization were seen. Blood samples were taken during the follicular phase of the menstrual cycle. Serum luteinizing hormone and follic1e-stimula ting hormone levels were normal. Serum testosterone levelwas7.5 nmoljL (normal <4,0 nmoljL), and serum sex-hormone binding globulin level was 30 nmol/L (normal 20 to 100 nmol/L), resulting in an increased free androgen index of 25 (normal <10). Serum dehydroepiandrosterone 3-sulfate (DHAS) levelwas elevated at 13.3 nmolfL (normal 2.2 to 9.2 nmol/L), Serum l7-a-hydroxyprogesterone level was 2.0 nmol/L (normal 0.5 to 3.0 nmoljL) and serum 8-4-androstenedione level was 6.0 nmoljL (normal 3.0 to 10.5 nmol/L). Oral administration of 2 mg of dexamethasone for 5 days resulted in significant suppression of serum DHAS concentration. A diagnosis of late-onset congenital adrenal hyperplasia caused by 3-iJ-hydroxysteroiddehydrogenase deficiency was made. The diagnosis of Becker's nevus was confirmed by a biopsy specimen that showed a normal epidermis with basal hyperpigmentation caused by an increase in mel-
Journal of the American Academy of Dermatology April 1994
Brief communications
Unilateral plantar erythema nodosum Naoto Ohtake, MD, Tatsuyoshi Kawamura, MD, Chie Akiyama, MD, Masutaka Fume, MD, and Kunihiko Tamaki, MD Yamanashi, Japan We report a case of erythema nodosum on the right sale. To our knowledge this is the second reported case of unilateral plantar erythema nodosum. CASE REPORT A 27-year-old Japanese woman had recurrent painful nodules on her right sole for 6 months. Individual nodules spontaneously resolved within I to 4 weeks. She had no systemic symptoms and no history of trauma. Examination revealed five erythematous, firm, tender, subcutaneous, 2 em nodules on the plantar aspect of the right foot (Fig. I). The results of the following studies were within normal limits or negative: erythrocyte sedimentation rate, blood cell counts, C-reactive protein, antistreptolysin 0 antibodies, antistreptokinase antibodies, hepatitis B surface antigen, antibodies for syphilis, antinuclear antibodies, rheumatoid factor, C3, C4, urinalysis, and bacterial culture from the tonsils. Roentgenograms of the chest and right foot were also normal. A biopsy specimen showed infiltration of lymphohistiocytes in the septae of fat lobules and inflammation of septal blood vessels (Fig. 2). The lesions were diagnosed as erythema nodosum and treated with oralloxoprofen sodium, 120 mg three times per day. The lesions completely regressed 20 days later. The lesions recurred after stopping loxoprofen sodium, but complete control was again achieved with readministration of the drug. No recurrence has been noted after 6 months.
Fig. 1. Tender nodules on plantar aspect of right foot.
DISCUSSION
Lesions of erythema nodosum usually appear symmetrically on the extensor aspects of the legs, with occasional involvement of the thighs or forearms. Unilateral l or plantar- 3 involvement uncommonly occurs. Hem and Shwayder" first reported unilateral plantar erythema nodosum. To our knowl-
From the Department of Dermatology, Yamanashi Medical University. Reprint requests: Naoto Ohtake, MD, Department of Dermatology, Yarnanashi Medical University, 1110 Shimokatoh, Tarnaho, Nakakoma, Yamanashi 409-38, Japan. JAM ACAD DERMATOL
1994;30:654-5.
Copyright ® [994 by the American Academy of Dermatology, Inc. 0[90-9622/94 $3.00 + 0
16/54/50277
Fig. 2. Infiltration of lymphohistiocytes in septae of fat lobules with inflammation of septal blood vessels. (Hematoxylin-eosin stain; X9.)
Journal of the American Academy of Dermatology Volume 30, Number 4
edge, ours is the second reported case of this disorder. Erythema nodosum is frequently associated with streptococcal infection, mycoses, tuberculosis, sarcoidosis, or drug allergy. None of these was present in our patient. Although mycoplasma infection does not usually induce erythema nodosum, antimycoplasma IgM was positive in Hem and Shwayder's case." Because we did not check antimycoplasma titer during the active stage of disease in our patient, we cannot comment on whether mycoplasma infection is
Brief communications 655 associated with unilateral plantar erythema nodosum. REFERENCES I. Hicks JH. Erythema nodosum in patients with tinea pedis and onychomycosis. South Med J 1977;70:27-8. 2. Berger TG, Tappero J. Traumatic plantar urticaria or plantar erythema nodosum [Letter]. J AM ACAD DERMATOL 1989;20:701-2. 3. Hosokawa H. Erythema nodosum on sales in a patient with pulmonary tuberculosis [in Japanese]. Skin Res 1990;
32:34-9. 4. Hem AE, Shwayder TA. Unilateral plantar erythema nodosum. J AM ACAD DERMATOL 1992;26:259-60.
Becker's nevus associated with chromosomal mosaicism and congenital adrenal hyperplasia Julien R. Lambert, MD,a Patrick Willems, PhD,b Roger Abs, MD,c and Bernadette Van Roy, BScb Antwerp, Belgium Becker's nevus is a relatively common unilateral hyperpigmented hairy cutaneous hamartoma. It is usually acquired, but a few cases have been considered congenital.' It can be associated with structural abnormalities, such as hypoplasia of the ipsilateral breast, areola and nipple, with limb asymmetry, spina bifida, scoliosis, and pectus carinatum. Although Becker's nevus occurs much more frequently in males (male/female ratio: 4.5: 1), these associated anomalies have been reported more often in females. The pathogenesis of Becker's nevus has not been elucidated, but several characteristics suggest that androgen stimulation may contribute. We describe a patient with Becker's nevus associated with hypotrophy of the shoulder girdle, congenital adrenal hyperplasia with severe hirsutism, and chromosomal mosaicism. CASE REPORT
A 20-year-oldwoman had hirsutism. At birth she had a blue discoloration on the left shoulder. As a childdiffuse From the Departments of Dermatology," Medical Genetics," and Endocrinology," University of Antwerp. Reprint requests: Julien R. Lambert, Department of Dermatology, University Hospital of Antwerp (U.I.A.), Wilrijkstraat 10, 2650 Edegem, Belgium.
.J
AM ACAD DERMATOL
1994;30:655-7.
Copyright @ 1994 by the American Academy of Dermatology, Inc. 0190-9622/94 $3.00
+0
16/54/49937
excessive hair growth gradually developed. She had a regular menstrual cycle from the age of 12 years. Severe hirsutism with a score of 34/44 according to the scale of Ferriman and Gallwey was observed.? The left shoulder and upper portion of the arm were more hairy than other parts of the body and showed a dark brown pigmentation that suggested a diagnosis of Becker's nevus. The left shoulder girdle was hypotrophic compared with the right but without functional deficit. Thin frontal hair was present, but no other signs of masculinization were seen. Blood samples were taken during the follicular phase of the menstrual cycle. Serum luteinizing hormone and follic1e-stimula ting hormone levels were normal. Serum testosterone levelwas7.5 nmoljL (normal <4,0 nmoljL), and serum sex-hormone binding globulin level was 30 nmol/L (normal 20 to 100 nmol/L), resulting in an increased free androgen index of 25 (normal <10). Serum dehydroepiandrosterone 3-sulfate (DHAS) levelwas elevated at 13.3 nmolfL (normal 2.2 to 9.2 nmol/L), Serum l7-a-hydroxyprogesterone level was 2.0 nmol/L (normal 0.5 to 3.0 nmoljL) and serum 8-4-androstenedione level was 6.0 nmoljL (normal 3.0 to 10.5 nmol/L). Oral administration of 2 mg of dexamethasone for 5 days resulted in significant suppression of serum DHAS concentration. A diagnosis of late-onset congenital adrenal hyperplasia caused by 3-iJ-hydroxysteroiddehydrogenase deficiency was made. The diagnosis of Becker's nevus was confirmed by a biopsy specimen that showed a normal epidermis with basal hyperpigmentation caused by an increase in mel-