• Users Online: 401
  • Print this page
  • Email this page

CORRESPONDENCE Table of Contents  
Ahead of print publication
Laugier–Hunziker syndrome with coexisting palmoplantar psoriasis


1 Department of Dermatology, Faculty of Medicine, Trakya University, Edirne, Turkey
2 Department of Pathology, Faculty of Medicine, Trakya University, Edirne, Turkey

Click here for correspondence address and email

Date of Submission25-Aug-2020
Date of Decision12-Dec-2020
Date of Acceptance17-Jan-2021
Date of Web Publication24-Apr-2021
 


How to cite this URL:
Urun M, Urun YG, Can N. Laugier–Hunziker syndrome with coexisting palmoplantar psoriasis. Dermatol Sin [Epub ahead of print] [cited 2021 May 7]. Available from: https://www.dermsinica.org/preprintarticle.asp?id=314514




Dear Editor,

Laugier–Hunziker syndrome (LHS) is a rare disorder characterized by acquired macular hyperpigmentation of the oral mucosa and the nails.[1] Different phenomena accompanying this syndrome are described in the literature: esophageal melanocytosis, actinic lichen planus, hypocellular marrow, thrombocytopenia, invasive melanoma, and lupus erythematosus.[2]

With this case, we present LHS that developed in a patient who had been diagnosed with palmoplantar psoriasis for 10 Xyears.

A 55-year-old male patient presented with numerous brown, hyperpigmented, macular lesions with sharply demarcated borders on both palms and soles, the fingers, and the oral mucosa. The patient also had noticeable hyperkeratosis of both palms and soles, subungual hyperkeratosis of the fingers and toenails, and longitudinal melanonychia on the third nail of the right foot [Figure 1]a,[Figure 1]b[Figure 1]c[Figure 1]d[Figure 1]e[Figure 1]f. His medical history revealed that he had had palmoplantar psoriasis for 10 years and hyperpigmentation on the hands and feet for 2 years and on the lips for a few weeks. He had received various topical treatments periodically for palmoplantar psoriasis but had not received psoralen plus ultraviolet A treatment. Examinations of the genital and oral mucosa of the patient, who does not have a known chronic disease, a drug that he constantly uses, and does not smoke, were normal. In the accompanying gastrointestinal tract examination, no signs of bleeding, diarrhea, and malabsorption were detected. No other family member had similar lesions.
Figure 1: Clinical manifestations of the lesions; (a) brown, hyperpigmented macules on the lower lip (b) over the buccal mucosa, and (c) on the fingers, (d) subungual hyperkeratosis of the toenails and longitudinal melanonychia on the third nail of the right foot, (e and f) palmar and plantar hyperkeratosis

Click here to view


The results of biochemical tests (liver and renal function tests and electrolytes), hemogram, hormonal tests, tumor markers, antinuclear antibody level, and HIV and syphilis serology tests were found to be normal. In the ultrasound of the entire abdomen with colonoscopy examination for malignancy screening, no pathological signs were found. The hyperkeratotic plaque, including the hyperpigmented macule on the patient's sole, and the hyperpigmented macule on the lips were examined histopathologically. Upon detection of suprapapillary thinning accompanied by hyperkeratosis, parakeratosis, and psoriasiform hyperplasia in the biopsy performed from the sole, the patient was histopathologically diagnosed with psoriasis [Figure 2]a. In addition, increased pigmentation in basal keratinocytes was consistent with hyperpigmented macules associated with LHS [Figure 2]b. The biopsy sample taken from the lip showed increased pigmentation in basal keratinocytes and an increase in melanophages in the papillary dermis [Figure 2]c and [Figure 2]d. Based on all the findings, the patient was diagnosed with LHS with palmoplantar psoriasis.
Figure 2: Histopathological examination of the biopsy specimens; (a) from the hyperkeratotic plaque, including the hyperpigmented macule on the sole showed suprapapillary thinning accompanied by hyperkeratosis, parakeratosis, psoriasiform hyperplasia, (b) and increased pigmentation in basal keratinocytes, (c and d) from the lip showed increased pigmentation in basal keratinocytes and an increase in melanophages in the papillary dermis (H and E, original magnification: a ×100, b ×1000, c ×200, d ×400) (The image in [Figure 2]b is a higher magnification of the area demarcated by the rectangle in [Figure 2]a)

Click here to view


LHS, whose etiology is not fully elucidated, is more common in white people and the average age of onset is 52 years.[2],[3] It has been shown that pigmentation in LHS is most often seen on the lower lip and the buccal mucosa. Other mucosal surfaces where similar lesions can be seen include conjunctiva, oropharynx, esophagus, vulva, anus, and perineum.[4] The extraoral involvement is most often observed in the form of longitudinal melanonychia on the nails.[1],[4] In addition, hyperpigmentation can be observed on the neck, chest, abdomen, and acral surfaces, especially on the fingertips.[3]

A diagnosis of LHS is made based on the age of the onset, the distribution of pigmentation, and the absence of pathological manifestations in colonoscopy, patient's personal medical history of cancer, and gastrointestinal tract symptoms.[3] In differential diagnosis, the causes of hyperpigmentation in appearance and etiologically should be reviewed individually. Drug-induced pigmentation, smoking, idiopathic melanoplakia, Addison's disease, and other syndromes such as Peutz–Jeghers syndrome, Bandler syndrome, and McCune–Albright syndrome must be investigated.[2],[3]

In the literature, when cases of LHS with psoriasis were reviewed, there was only one case with subacute cutaneous lupus erythematosus and psoriasis published by Kemmet et al. in 1990.[5] The case we present here, to our knowledge, is the first LHS case associated with palmoplantar psoriasis. LHS's relationship with other diseases is not entirely clear, and these conditions are considered coincidences.[2] However, it is also believed that this syndrome is more common than reported.[1]

In conclusion, it is important to know that LHS is a chronic but benign syndrome, asymptomatic pigmentation may increase gradually, and there is no effective treatment. Furthermore, distinguishing it from other malignant conditions will save the patient from invasive procedures. We believe that as more awareness develops about this syndrome, reporting of concomitant diseases will increase.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Nayak RS, Kotrashetti VS, Hosmani JV. Laugier-Hunziker syndrome. J Oral Maxillofac Pathol 2012;16:245-50.  Back to cited text no. 1
  [Full text]  
2.
Lalosevic J, Zivanovic D, Skiljevic D, Medenica L. Laugier-Hunziker syndrome-Case report. An Bras Dermatol 2015;90:223-5.  Back to cited text no. 2
    
3.
Paul J, Harvey VM, Sbicca JA, O'Neal B. Laugier-Hunziker syndrome. Cutis 2017;100:E17-9.  Back to cited text no. 3
    
4.
Nikitakis NG, Koumaki D. Laugier-Hunziker syndrome: Case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol 2013;116:e52-8.  Back to cited text no. 4
    
5.
Kemmett D, Ellis J, Spencer MJ, Hunter JA. The Laugier-Hunziker syndrome – A clinical review of six cases. Clin Exp Dermatol 1990;15:111-4.  Back to cited text no. 5
    

Top
Correspondence Address:
Mustafa Urun,
Department of Dermatology, Faculty of Medicine, Trakya University, Edirne
Turkey
Login to access the Email id

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ds.ds_2_21



    Figures

  [Figure 1], [Figure 2]



 

Top
 
  Search
 
   Ahead Of Print
  
 Article in PDF
     Search Pubmed for
 
    -  Urun M
    -  Urun YG
    -  Can N


References
Article Figures

 Article Access Statistics
    Viewed99    
    PDF Downloaded7    

Recommend this journal