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Facial and axillary apocrine chromhidrosis

  • Author(s): Tato, Berta Pérez
  • Martínez, Elena Zamora
  • Albisua, Begoña Sánchez
  • González, Yosmar C Pérez
  • Olabarrieta, Isabel Polimón
  • Escobedo, Silvia Marinero
  • López, Paloma Fernández
  • et al.
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Facial and axillary apocrine chromhidrosis
Berta Pérez Tato MD¹, Elena Zamora Martínez MD¹, Begoña Sánchez Albisua MD¹, Yosmar C Pérez González MD², Isabel Polimón Olabarrieta MD¹, Silvia Marinero Escobedo MD¹, Paloma Fernández López MD¹
Dermatology Online Journal 18 (3): 13

Departments of Dermatology¹ and Pathology²
Hospital Universitario de Móstoles, Madrid, Spain


Abstract

Apocrine chromhidrosis is a rare, chronic, idiopathic disorder, characterized by the excretion of pigmented sweat. A 26-year-old woman presented with a 3-year history of dark blue secretions on bilateral malar cheeks. On examination, upon pressure on the cheeks, a dark blue fluid was expressed, which appeared to arise primarily from the follicle. The patient had not appreciated it, but on examination of her axillae, a very subtle blue coloring was observed. Histopathologic examination revealed apocrine glands in the deep reticular dermis. Bluish cytoplasmic granules were observed in the apocrine epithelium lining, which correspond to lipofuscin granules. The diagnosis of apocrine cromhidrosis was made. We began treatment with 20 percent aluminum chloride hexahydrate solution and capsaicin cream with poor tolerance. Finally, we treated with botulinum toxin type A with a successful response. We report a case of facial and axillary apocrine cromhidrosis with good response to botulinum toxin type A.



Introduction

Apocrine chromhidrosis is a rare, chronic, idiopathic disorder, characterized by the excretion of pigmented sweat. Cases of yellow, blue, green, or black sweat have been described [1].


Case report

A 26-year-old woman presented with a 3-year history of dark blue secretions on bilateral malar cheeks. Her medical history was unremarkable and she was not on any topical or oral medications.


Figure 1aFigure 1b
Figure 1a. Right cheek showing dark blue discharge.

Figure 1b. Closer view of the dark blue fluid expressed.

On examination, upon pressure on the cheeks, a dark blue fluid was expressed, which appeared to arise primarily from the follicle.

Although the patient had not appreciated it, on examination of her axillae, a very subtle blue coloring was observed. The patient's urine, stool, tears, and oral and nasal secretions were all of normal color.


Figure 2aFigure 2b
Figure 2a. Bluish cytoplasmic granules in the apocrine epithelium lining (H&E, panoramic view)

Figures 2b and 2c. Granules in the apocrine epithelium (black arrow). (H&E, high power)

Figure 2cFigure 2d
Figure 2d. Positive granules to PAS stain.

A 4 mm punch biopsy specimen was obtained from the axillary region. Histopathologic examination revealed apocrine glands in the deep reticular dermis. Bluish cytoplasmic granules were observed in the apocrine epithelium lining. These granules, which correspond to lipofuscin granules, were positive to PAS and Oil Red O stains. Electronic microscopy showed vacuoles with myelinoid bodies compatible with degraded phospholipids.


Figure 2eFigure 2f
Figure 2e. Oil Red O positive granules.

Figure 2f. Electronic microscopy showed vacuoles with myelinoid bodies (black arrow) compatible with degraded phospholipids.

A 4 mm punch biopsy specimen was obtained from axillary region. Histopathologic examination revealed apocrine glands in the deep reticular dermis. Bluish cytoplasmic granules were observed in the apocrine epithelium lining. These granules, which correspond to lipofuscin granules, were positive to PAS and Oil Red O stains. Electronic microscopy showed vacuoles with myelinoid bodies compatible with degraded phospholipids.

A complete blood count, urinalysis and homogentisic levels were within normal limits. Skin bacterial and fungal cultures were negative.

The diagnosis of apocrine chromhidrosis was made. We began treatment with 20 percent aluminum chloride hexahydrate solution. Later, capsaicin cream was used. Irritation and burning sensation, respectively, resulted in poor tolerance by the patient. Consequently, we had to discontinue treatment.


Figure 3
Figure 3. Right cheek after botulinum toxin type A injection.

Because of the psychological concerns of the patient, we decided to begin treatment with botulinum toxin (botox) type A. A 100-unit vial of lyophilized botox was reconstituted with 5.0 cc of unpreserved normal saline. Ten units were injected, intradermally, into each side of the face, 0.05 cc per injection site (concentration 1 unit/0.05 cc).

One week after injection the patient reported a reduction in sweating and we observed a decrease in discoloration, although without complete remission. On examination 4 months after treatment the improvement was still present.


Discussion

Cromhidrosis is an unusual disorder that usually occurs in puberty, when apocrine secretory function is activated. As apocrine glands regress with age, a parallel regression of the disease is often seen. The majority of cases report in the literature, are confined to the face or axillae [2, 3, 4], but areolar chromhidrosis has been reported [5, 6]. Yonge first described facial chromhidrosis in 1709. Shelley and Hurley in 1954 associated the color to a higher concentration of lipofuscine granules in apocrine glands [7].

Clinical diagnosis is not difficult. If in doubt, the presence of lipofuscin granules in apocrine cells can be used to confirm the diagnosis. The apocrine glands appear normal in size and morphology, but the number of glands varies. The increased number of yellow-brown lipofuscin granules is observed in the cytoplasm of secretory cells on routine hematoxylin-eosin staining. The granules are positive on periodic acid-Schiff and Oil Red O stains. The lipofuscin granules are responsible for the pigmented sweat.

Lipofuscin is a yellow pigment that is not specific to apocrine glands. In apocrine chromhidrosis the lipofuscin is in higher concentration or in a higher state of oxidation than in normal secretions, adopting a darker coloration like blue, green, or black. The reason for the development in only a few patients is unknown [1].

It is necessary to consider differentiation from true eccrine chromhidrosis and pseudochromhidrosis. True eccrine chromhidrosis is associated with water-soluble pigments excreted by the eccrine glands, as occurs when some drugs are taken as quinines. Pseudochromhidrosis is the result of colorless perspiration mixed with an external chromogen such as dyed clothing, colored chemicals, or microorganisms such as Piedraia or Cornynebacterium [8].

Satisfactory therapy for this condition remains a challenge. Success has been reported with capsaicin cream and 20 per cent aluminum chloride hexahydrate solution although relapse tends to occur when therapy is discontinued [9].

Botulinum toxin type A (BTX-A) has been shown to be effective for facial chromhidrosis [10, 11]. The mechanism by which BTX-A suppresses apocrine chromhidrosis is unclear. It is thought that the mechanism by which BTX-A suppresses apocrine secretion is related to different mechanisms. Although apocrine glands are thought to be unresponsive to cholinergic stimulation, some authors have demonstrated a response to local administration of cholinergic compounds [12]. Cholinergic nerve fibers have been demonstrated around the secretory coils of apocrine sweat glands, although with a lower density of innervation than the adjacent eccrine glands [13, 14]. Therefore, it is possible that BTX-A suppresses apocrine secretion by blockade of cholinergic stimulation of apocrine glands. In addition, the toxin has also been shown to inhibit substance P release [15]. Successful treatment with capsaicin cream supports the role of substance P in chromhidrosis [9]. As in our own experience, on previous reports, results persists for 4 to 5 months after treatment.

We report an unusual case of facial and axillary chromhidrosis with improvement with botulinum toxin type A treatment. We consider it is an adequate second line treatment when capsaicin or 20 percent aluminum chloride hexahydrate are ineffective or poorly tolerated by the patient.

Acknowledgements: The authors thank M A Martínez González MD, Department of Pathology, Hospital 12 de Octubre, for the electronic microscopy technique.

References

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