Dear Editor:
Chromhidrosis is a rare condition characterized by secretion of colored sweat from the eccrine or apocrine glands. Not only is this condition epidemiologically rare, but also its mimicry of eczematous clinical features may lead to misdiagnosis.
A 55-year-old man was examined for abnormal pigmentation on right palm and soles, which lasted a week. He was a local farmer in an apple orchard and he had diabetes, alcoholic liver cirrhosis, and hepatic regenerative nodules as underlying diseases. He was hospitalized due to longstanding diarrhea and fever. On clinical examination, yellow and green papules were observed on his right palm and soles (Fig. 1) and levels of total (19.3 mg/dl) and direct (13.8 mg/dl) bilirubin were elevated. Histological findings revealed hyperkeratosis, diffuse acanthosis, and subcorneal vesicles in the epidermis (Fig. 2A). Homogenous eosinophilic materials were observed surrounding the vesicles and both the number and size of eccrine glands were increased (Fig. 2A, inset). Hence, the patient was diagnosed with eccrine chromhidrosis and treated with emollient. After three weeks, abnormal pigmentation was almost resolved, levels of total (2.18 mg/dl, normal range: 0.3~1.9 mg/dl) and direct (0.71 mg/dl, normal range: 0~0.3 mg/dl) bilirubin were lower and body temperature had decreased (Fig. 2B).
Green pigmentation on the palms and soles in patients with hyperbilirubinemia is a rare condition1. While our case presented with eczematous lesions on the right palm and soles (where sweat glands are most abundant), the patient's clinical features resembled pompholyx, a primarily spongiotic dermatitis23. However, the yellow-green pigmentation in our case appeared to be bile-filled vesicles, which could not be explained by spongiotic changes alone. Two weeks of high fever led to increased sweating and high concentrations of bile components in the sweat may have acted as a sensitizer and induced eczematous lesions, exacerbating the inflammatory spongiosis. Eccrine chromhidrosis is a rare condition in which water-soluble pigments from certain dyes or drugs are excreted via the eccrine sweat glands-in our case, the bile components. It may be caused by chromogenic bacterial or fungal contamination or by extrinsic chemicals on the surface of the skin, which react with eccrine secretions and produce the color transformation. However, results of fungal and pseudomonal tests in this case were negative.
Kanzaki and Tsuda4 reported two cases of eccrine chromhidrosis with liver disease. In hepatocytes associated with liver disease, bile may become pigmented with brown color; however, no bile pigmentation was observed in this case. The bile pigment may have been washed out during histological fixation if it was located within the spaces of the eccrine ducts and vesicles, and not in the cellular spaces2. Possible pathomechanisms of three essential factors that could contribute towards the development of pigmentation: (1) increased plasma level of water-soluble direct bilirubin, (2) high fever with sweating, and (3) a thick horny layer. The green color is attributable to the switch from brown-colored bilirubin to green-colored biliverdin by oxidative processes5. We report a case of eccrine chromhidrosis resembling the clinical features of pompholyx with explanation of probable pathomechanism.
References
1. Triwongwaranat D, Kasemsarn P, Boonchai W. Green pigmentation on the palms and soles. Acral green pigmentation (eccrine chromhidrosis). JAMA Dermatol. 2013; 149:1339–1340.
2. Lee WJ, Lee DW, Kim CH, Won CH, Chang SE, Lee MW, et al. Pompholyx with bile-coloured vesicles in a patient with jaundice: are sweat ducts involved in the development of pompholyx? J Eur Acad Dermatol Venereol. 2010; 24:235–236.
3. Park SH, Kim SW, Noh TW, Hong KC, Kang YS, Lee UH, et al. A case of palmar lichen nitidus presenting as a clinical feature of pompholyx. Ann Dermatol. 2010; 22:235–237.