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Localized unilateral hyperhidrosis: Eccrine nevus
Volume 27 Number I July 1992
Briefcommunications 115
logic findings in our patient excluded white fibrous papulosis of the neck, a recently reported entity.6 In agreement with Brenner et aI. 4 we propose to add "focal" to the term noninflammatory dermal elastolysis to emphasize the localized variety ofthis rare condition. REFERENCES 1. Rae V, Falanga V. Wrinkling due to midderrnal elastolysis. Arch Dermatol 1989;125:950-1.
2. Shelley WB, Wood MG. Wrinkles due to idiopathic loss of mid-dermal elastic tissue. Br J Dermatol1977;97:441-5. 3. RUdolph RI. Mid dermal elastolysis. J AM ACAD DERMATOL 1990;22:203-6. 4. Brenner W, Gschnait F, Konrad K, et a1. Noninflammatory mid dermal elastolysis. Br J Dermato1 1978;99:335-8. 5. Heudes AM, Bouillie Me, Thomine E, et al. Elasto1yse acquise en nappe du derme moyen. Ann Dermatol Venereal 1988;115:1041-5. 6. Shimizu H, Kimura S, Harada T, et al. White fibrous papulosis of the neck: A new clinicopathologic entity? J AM ACAD DERMATOL [989;20:1073-7.
Localized unilateral hyperhidrosis: Eccrine nevus F. Ruiz de Erenchun,a F. J. Vizquez-Doval,a F. Contreras Mejuto,b and E. Quintanillaa Pamplona, Spain
Eccrine hamartoma is an unusual entity with varied clinical manifestations.1-4 We report the case of an 81-year-old man who had a localized area of hyperhidrosis on the right side of the thorax and abdomen. CASE REPORT An 81-year-old man had a 2-year history of chronic B-celilymphocytic leukemia (stage II) for which he was taking prednisone, 8 mg/day, and chlorambucil, 2 mgtwo times weekly. He was also taking digoxin, 0.25 mg/day, furosemide, 40 mg twice weekly, and captopril, 25 mg/ day, for systemic arterial hypertension and atrial fibrillation. He also had a localized area of hyperhidrosis, lOX 10 cm, on the right lateral aspect of the trunk that started 1 year before consultation. The profuse sweating drenched his clothes and obliged him to change shirts three to four times each day. Heat did not increase sweating but it was more profuse at night. Examination did not show any cutaneous lesions but increased sweating was noted in the localized area. Results of neurologic examination were normal. A biopsy specimen revealed marked hyperplasia of the ducts and secretory coils of the eccrine sweat glands (Fig. 1). The interior of the secretory ducts showed a material that reacted with the periodic acid-Schiff (PAS) stain, and the internal cellular wall layer was covered by a fine cuticle of PAS-positive malerial (Fig. 2). A biopsy specimen of the contralateral aspect of the trunk showed a normal number and size of eccrine sweat glands.
From the Departments of Dermatology· and Patho1ogy,b University Clinic, School of Medicine, University of Navarra. No reprint~ available. 16/54/36088
Fig. 1. Increase in both size and number of eccrine coils.
The drugs for his chronic lymphocytic leukemia, arterial hypertension, and atrial fibrillation were continued, and treatment with oral mianserine, 30 mg/day, for depressive episodes was started. He refused topical treatment. However, his hyperhidrosis was markedly de-
Journal of the American Academy of Dermatology
116 Brief communications
Fig. 2. Eccrine coils lined with cuboidal cells. Lumina are dilated and some walls are thickened. creased after 2 months of treatment with mianserine. One year later, the patient died of heart failure. DISCUSSION
Localized unilateral hyperhidrosis has diverse causes. 4,5 Alterations of the sympathetic nervous system is the most common cause. It can also be caused by lesions in the central nervous system. Spinal cord injuries may be associated with increased sweating in some cases. Localized hyperhidrosis can also appear in patients with peripheral neuropathies after sympathectomy and in association with intrathoracic neoplasms. Cutaneous diseases associated with localized hyperhidrosis include blue rubber bleb nevus, glomus tumors, POEMS syndrome, causalgia, pachydermoperiostosis, and eccrine nevus. In some cases the cause cannot be determined and these are considered idiopathic. 2 Purely eccrine nevi are not common. I Three types of clinical presentation have been described a solitary pore-draining mucoid material, 3 localized area ofhyperhidrosis, 4, 5 and a centrally depressed nodule surrounded by a slightly scaly border. I In these three presentations, the histologic features are similar to hyperplasia of the eccrine sweat glands. In our patient, the symptoms began at an ad-
vanced age. Previous cases reported were diagnosed at an earlier age; our patient is the oldest reported case. Surgical excision is the usual treatment of eccrine nevus, but it was not possible in our patient because of the extensive area of involvement. Mianserine is an antidepressive agent with weak anticholinergic effects. Its mechanism of action seems to be due to an increase in noradrenaline concentrations in the synaptic space by blocking ex- 2 presynaptic receptors, although it also blocks 5-hydroxytryptamine and serotonin receptors. The clinical improvement observed in our patient was probably coincidental but may indicate that this treatment should be considered in extensive forms of localized hyperhidrosis. REFERENCES I. Pippione M, Depaoli MA, Sartoris S. Naevus eccrine. Dermatologica 1976;152:40-6. 2. Sata K, Kang WH, Saga K, et al. Biology of sweat glands and their disorders. 1. Normal sweat gland function. J AM ACAD DERMATOL 1989;20:537-63. 3. Herzberg J1. Ekkrines syringcystadenam. Arch Klin Exp Dermato1 1962;214:600-21. 4. Martius 1. Lokalisierte ekkrine Schweissdriisenhyperplasie. Dermatol Monatsschr 1979;165:327-30. 5. Goistein eN. Ephidrosis (local hyperhidrosis): nevus sudoriferus. Arch Dermatol 1967;96:67-8.
Briefcommunications 115
logic findings in our patient excluded white fibrous papulosis of the neck, a recently reported entity.6 In agreement with Brenner et aI. 4 we propose to add "focal" to the term noninflammatory dermal elastolysis to emphasize the localized variety ofthis rare condition. REFERENCES 1. Rae V, Falanga V. Wrinkling due to midderrnal elastolysis. Arch Dermatol 1989;125:950-1.
2. Shelley WB, Wood MG. Wrinkles due to idiopathic loss of mid-dermal elastic tissue. Br J Dermatol1977;97:441-5. 3. RUdolph RI. Mid dermal elastolysis. J AM ACAD DERMATOL 1990;22:203-6. 4. Brenner W, Gschnait F, Konrad K, et a1. Noninflammatory mid dermal elastolysis. Br J Dermato1 1978;99:335-8. 5. Heudes AM, Bouillie Me, Thomine E, et al. Elasto1yse acquise en nappe du derme moyen. Ann Dermatol Venereal 1988;115:1041-5. 6. Shimizu H, Kimura S, Harada T, et al. White fibrous papulosis of the neck: A new clinicopathologic entity? J AM ACAD DERMATOL [989;20:1073-7.
Localized unilateral hyperhidrosis: Eccrine nevus F. Ruiz de Erenchun,a F. J. Vizquez-Doval,a F. Contreras Mejuto,b and E. Quintanillaa Pamplona, Spain
Eccrine hamartoma is an unusual entity with varied clinical manifestations.1-4 We report the case of an 81-year-old man who had a localized area of hyperhidrosis on the right side of the thorax and abdomen. CASE REPORT An 81-year-old man had a 2-year history of chronic B-celilymphocytic leukemia (stage II) for which he was taking prednisone, 8 mg/day, and chlorambucil, 2 mgtwo times weekly. He was also taking digoxin, 0.25 mg/day, furosemide, 40 mg twice weekly, and captopril, 25 mg/ day, for systemic arterial hypertension and atrial fibrillation. He also had a localized area of hyperhidrosis, lOX 10 cm, on the right lateral aspect of the trunk that started 1 year before consultation. The profuse sweating drenched his clothes and obliged him to change shirts three to four times each day. Heat did not increase sweating but it was more profuse at night. Examination did not show any cutaneous lesions but increased sweating was noted in the localized area. Results of neurologic examination were normal. A biopsy specimen revealed marked hyperplasia of the ducts and secretory coils of the eccrine sweat glands (Fig. 1). The interior of the secretory ducts showed a material that reacted with the periodic acid-Schiff (PAS) stain, and the internal cellular wall layer was covered by a fine cuticle of PAS-positive malerial (Fig. 2). A biopsy specimen of the contralateral aspect of the trunk showed a normal number and size of eccrine sweat glands.
From the Departments of Dermatology· and Patho1ogy,b University Clinic, School of Medicine, University of Navarra. No reprint~ available. 16/54/36088
Fig. 1. Increase in both size and number of eccrine coils.
The drugs for his chronic lymphocytic leukemia, arterial hypertension, and atrial fibrillation were continued, and treatment with oral mianserine, 30 mg/day, for depressive episodes was started. He refused topical treatment. However, his hyperhidrosis was markedly de-
Journal of the American Academy of Dermatology
116 Brief communications
Fig. 2. Eccrine coils lined with cuboidal cells. Lumina are dilated and some walls are thickened. creased after 2 months of treatment with mianserine. One year later, the patient died of heart failure. DISCUSSION
Localized unilateral hyperhidrosis has diverse causes. 4,5 Alterations of the sympathetic nervous system is the most common cause. It can also be caused by lesions in the central nervous system. Spinal cord injuries may be associated with increased sweating in some cases. Localized hyperhidrosis can also appear in patients with peripheral neuropathies after sympathectomy and in association with intrathoracic neoplasms. Cutaneous diseases associated with localized hyperhidrosis include blue rubber bleb nevus, glomus tumors, POEMS syndrome, causalgia, pachydermoperiostosis, and eccrine nevus. In some cases the cause cannot be determined and these are considered idiopathic. 2 Purely eccrine nevi are not common. I Three types of clinical presentation have been described a solitary pore-draining mucoid material, 3 localized area ofhyperhidrosis, 4, 5 and a centrally depressed nodule surrounded by a slightly scaly border. I In these three presentations, the histologic features are similar to hyperplasia of the eccrine sweat glands. In our patient, the symptoms began at an ad-
vanced age. Previous cases reported were diagnosed at an earlier age; our patient is the oldest reported case. Surgical excision is the usual treatment of eccrine nevus, but it was not possible in our patient because of the extensive area of involvement. Mianserine is an antidepressive agent with weak anticholinergic effects. Its mechanism of action seems to be due to an increase in noradrenaline concentrations in the synaptic space by blocking ex- 2 presynaptic receptors, although it also blocks 5-hydroxytryptamine and serotonin receptors. The clinical improvement observed in our patient was probably coincidental but may indicate that this treatment should be considered in extensive forms of localized hyperhidrosis. REFERENCES I. Pippione M, Depaoli MA, Sartoris S. Naevus eccrine. Dermatologica 1976;152:40-6. 2. Sata K, Kang WH, Saga K, et al. Biology of sweat glands and their disorders. 1. Normal sweat gland function. J AM ACAD DERMATOL 1989;20:537-63. 3. Herzberg J1. Ekkrines syringcystadenam. Arch Klin Exp Dermato1 1962;214:600-21. 4. Martius 1. Lokalisierte ekkrine Schweissdriisenhyperplasie. Dermatol Monatsschr 1979;165:327-30. 5. Goistein eN. Ephidrosis (local hyperhidrosis): nevus sudoriferus. Arch Dermatol 1967;96:67-8.