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CORRESPONDENCE
Year : 2020  |  Volume : 65  |  Issue : 1  |  Page : 69-70
Black maculae between the toes: A rare form of Laugier–Hunziker syndrome?


Department of Dermatology, First Affiliated Hospital of Nanjing Medical University, Nanjing, Jiangsu, China

Date of Web Publication13-Jan-2020

Correspondence Address:
Zhi Qiang Yin
Department of Dermatology, First Affiliated Hospital of Nanjing Medical University, Nanjing, Jiangsu
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.IJD_18_19

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How to cite this article:
Guo J, Hu Y, Tu J, Yin ZQ. Black maculae between the toes: A rare form of Laugier–Hunziker syndrome?. Indian J Dermatol 2020;65:69-70

How to cite this URL:
Guo J, Hu Y, Tu J, Yin ZQ. Black maculae between the toes: A rare form of Laugier–Hunziker syndrome?. Indian J Dermatol [serial online] 2020 [cited 2020 Jul 11];65:69-70. Available from: http://www.e-ijd.org/text.asp?2020/65/1/69/275756




Sir,

A 65-year-old man presented with irregular black maculae between all 10 toes with no progress for 30 months [Figure 1]. He had white maceration between some toes due to hyperhidrosis of feet over the past decades. He had been given intermittent itraconazole in a local hospital at a dosage of 400 mg daily for 1 week every month for 3 months to treat toenail onychomycosis, which was subsequently cured; however, the black maculae between the toes had no change. We gave this patient mycological microscopy and culture of samples taken from black maculae between the toes several times but the results were negative. Dermoscopy on the dark spot between the third and fourth toes of the left foot exhibited unsymmetrical pigmentation without blue-white veil, which did not show a typical character of melanoma [Figure 2]a. After a detailed examination of the skin, we found black spots on the lip and oral mucosa [Figure 2]b with unknowing disease course, but no abnormity on fingernail, toenails, or genitalia was seen. Biopsy was conducted on the dark spot between the third and fourth toes of the left foot and histopathology showed epidermal acanthosis and trochanterellus extension [Figure 2]c, and periodic acid-Schiff (PAS) staining was negative. Immunohistochemistry staining of Sox10 [Figure 2]d and HMB45 [Figure 2]e antibodies both presented some positive melanocytes in trochanterellus. The patient underwent enteroscopy examination which exhibited that colorectum mucosa had no obvious abnormity [Figure 2]f. Based on the clinical manifestation and the results of the accessory examinations, we diagnosed this case as Laugier–Hunziker syndrome (LHS), which to our knowledge was the first case report of LHS presenting melanotic macules between all the toes. There was no family history of similar complaints. This elderly patient did not care deeply about these “unbeautiful” melanotic macules and refused to accept our treatment suggestions including any topical and systemic management.
Figure 1: Irregular black maculae between all 10 toes, and white maceration was seen between some toes

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Figure 2: (a) Unsymmetrical pigmentation without blue-white veil under dermoscopy, (b) labial and oral melanotic macules, (c) histopathology showed epidermal acanthosis and trochanterellus extension, (d and e) Sox10 and HMB45 staining both exhibited positive melanocytes in trochanterellus (×200), (f) colorectum mucosa without obvious abnormity

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Black maculae between the toes are not a very common symptom, and the most probable diagnosis might be black ringworm or melanoma. This patient had a long history of feet hyperhidrosis, which would increase the water content of epidermal horny layer and induce the occurrence of maceration between the toes where dermatomycosis is commonly secondary to maceration. However, consideration of black ringworm was excluded due to a negative response to broad-spectrum systemic antifungal therapy, and negative mycological microscopy and culture results and PAS staining. Melanoma merely involving the skin between all the 10 toes seems to be unlikely. A small number of epidermal melanocytes with positive staining of HMB-45 antibody are not ample evidence for diagnosing melanoma; sometimes, normal melanocytes can also present positive results with HMB-45 staining. The diagnosis of melanoma was not supported by the evidence of dermoscopy manifestation, histopathology, and immunohistochemical staining. Concerning the possibility of progression to melanoma, we think the prognosis was good because of black maculae on nonrubbing parts and long-term “inactive” clinical manifestation.

Labial and oral melanotic macules are commonly encountered in many diseases, such as LHS and Peutz–Jeghers syndrome (PJS). LHS is an acquired benign condition commonly associated with longitudinal melanonychia.[1] Gerbig and Hunziker[2] first suggested “idiopathic lenticular mucocutaneous pigmentation” in 1996 to deal with LHS with atypical features; however, more literature regarded LHS and “idiopathic lenticular mucocutaneous pigmentation” as the same notion.[3],[4] PJS is characterized by mucocutaneous pigmentation and multiple gastrointestinal hamartomatous polyps that belong to autosomal dominant inherited disease due to LKB1/STK11 gene mutation or some other genetic disorders.[5] However in our case, the examination result of enteroscopy did not support PJS. Taking all factors into consideration, we think this patient presented a rare form of LHS: melanotic macules on lip, oral mucosa, and the skin between the toes without manifestation of longitudinal melanonychia. While encountering the black maculae between the toes, the clinicians should examine the labial and oral mucosa of the patient for the possibility of LHS.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Acknowledgments

We thank Prof. Yan Lu and Prof. Mei Hua Zhang for kind assistance for the diagnosis.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Duan N, Zhang YH, Wang WM, Wang X. Mystery behind labial and oral melanotic macules: Clinical, dermoscopic and pathological aspects of Laugier-Hunziker syndrome. World J Clin Cases 2018;6:322-34.  Back to cited text no. 1
    
2.
Gerbig AW, Hunziker T. Idiopathic lenticular mucocutaneous pigmentation or Laugier-Hunziker syndrome with atypical features. Arch Dermatol 1996;132:844-5.  Back to cited text no. 2
    
3.
Mignogna MD, Lo Muzio L, Ruoppo E, Errico M, Amato M, Satriano RA. Oral manifestations of idiopathic lenticular mucocutaneous pigmentation (Laugier-Hunziker syndrome): A clinical, histopathological and ultrastructural review of 12 cases. Oral Dis 1999;5:80-6.  Back to cited text no. 3
    
4.
Zaki H, Sabharwal A, Kramer J, Aguirre A. Laugier-Hunziker syndrome presenting with metachronous melanoacanthomas. Head Neck Pathol 2018 Feb 15. doi: 10.1007/s12105-018-0897-3. [Epub ahead of print]  Back to cited text no. 4
    
5.
Duan FX, Gu GL, Yang HR, Yu P-F, Zhang Z. Must Peutz-Jeghers syndrome patients have the LKB1/STK11 gene mutation? A case report and review of the literature. World J Clin Cases 2018;6:224-32.  Back to cited text no. 5
    


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