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CORRESPONDENCE Table of Contents  
Ahead of print publication
Periungual xanthomas in a case of hepatocellular carcinoma and dyslipidemia


1 Department of Dermatology, Taipei Veterans General Hospital; Faculty of Medicine, School of Medicine, National Yang Ming Chiao Tung University, Taipei, Taiwan
2 Department of Dermatology, Taipei Veterans General Hospital; Faculty of Medicine, School of Medicine, National Yang Ming Chiao Tung University; Department of Dermatology, National Yang Ming Chiao Tung University, Taipei, Taiwan

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Date of Submission10-Jun-2022
Date of Decision03-Jul-2022
Date of Acceptance17-Jul-2022
Date of Web Publication20-Oct-2022
 


How to cite this URL:
Li CL, Lee DD. Periungual xanthomas in a case of hepatocellular carcinoma and dyslipidemia. Dermatol Sin [Epub ahead of print] [cited 2022 Nov 28]. Available from: https://www.dermsinica.org/preprintarticle.asp?id=359340




Dear Editor,

A 49-year-old man with metabolic syndrome and newly-diagnosed hepatocellular carcinoma (HCC) was brought to our outpatient clinic for multiple reddish papules and nodules on 10 fingers for 1 month. The patient claimed that these papules and nodules have been increasing in size with tenderness during this period. No significant drug or contact history was noted. He denied any other systemic discomfort and has been followed in the gastroenterology department due to an incidental finding of a liver tumor on computerized tomography scan 1 month ago. On examination, there were diffuse erythematous nodules and papules on bilateral hands, accentuated on the fingertips, periungual areas, and the distal interphalangeal joints [Figure 1]a,[Figure 1]b, [Figure 1]d, [Figure 1]e, [Figure 1]f. Dermoscopy revealed yellowish homogenous structure-less areas [Figure 1]g on the nodule of the right thumb. An incisional biopsy was performed.
Figure 1: (a-c) Diffuse erythematous nodules and papules on right hand, especially on the fingertips, periungual areas, and the distal interphalangeal joints. (d-f) Closer view of the xanthomatous lesions on the periungual area of right thumb and right index finger. (g) Dermoscopy revealed yellowish homogenous structure-less areas on the nodule of the right thumb.

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Histopathological examination revealed focal exocytosis of foamy histiocytes in the epidermis and a diffuse infiltrate of foamy histiocytes and lymphocytes in the dermis [Figure 2]a and [Figure 2]b. Multinucleated giant cells and Touton type giant cells were not found. For the foamy cells, immunohistochemical staining of cytokeratin was negative while CD68 was positive in the epidermis and dermis [Figure 2]c and [Figure 2]d. A diagnosis of xanthoma was established, with a unique anatomical distribution and a rare histological feature of foamy cells exocytosis. Blood tests showed elevated alanine transaminase (90 U/L; reference: <41 U/L), aspartate transaminase (357 U/L; reference: <40U/L), total cholesterol (460 mg/dL, reference: <200 mg/dL), low-density lipoprotein cholesterol (310 mg/dL; reference: <130 mg/dL), and triglyceride (319 mg/dL; reference: <150 mg/dL) levels, which was compatible with hepatitis and type IIb dyslipidemia according to Fredrickson's classification.[1] No family history of dyslipidemia was found in the patient. A treatment with oral lipid-lowering agent was started, and the patient reported gradual regression with improved pain in his fingers after 2 weeks. However, the patient died from intra-abdominal infection with septic shock during cancer treatment 2 weeks later.
Figure 2: (a) The histology finding revealed diffuse dermal infiltrate of foamy histiocytes on low-power field (H and E, ×40). (b) Aggregates of foamy cells were seen in the dermis with epidermal exocytosis (H and E, ×100). (c) Immunohistochemistry staining of cytokeratin was negative for the foamy cells, compared with the positive staining of keratinocytes (×200). (d) CD68 was positive for the foamy cells both in the epidermis and dermis (×100).

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Cutaneous xanthomas are plaques or nodules due to localized aggregates of histiocytes containing accumulated lipid. Conventionally, xanthomas are categorized into five main types according to their clinical features: eruptive, tendinous, tuberous, planar, and disseminated type.[2] While most xanthomas are associated with primary or secondary hyperlipidemia,[3] some subtypes occur in patients with a rather normolipidemic state, including verruciform xanthomas, xanthoma disseminatum, diffuse planar xanthoma, and xanthelasma. We herein present a unique clinical variant of xanthoma arising in the periungual area of a 49-year-old man, which does not fit well into any one of the five clinical types. The reports of periungual xanthoma are rare in previous literature, with only one case report of a 3-year-old girl, termed “papular xanthoma,” to date.[4] The case had a normal lipid profile and was initially diagnosed as multicentric reticulohistiocytosis clinically due to concurrent presentation of arthritis. It was later to be found that the case was more compatible with xanthoma pathologically; interestingly, the case also showed exocytosis of foamy cells as our patient, which is not commonly observed in other subtypes of xanthomatosis, suggesting that it may be an exclusive feature of periungual xanthoma.

The clinical course and laboratory data of our patient further supported the possible association between dyslipidemia, HCC, and xanthoma. Whether excess alcohol consumption and metabolic syndrome played a role in the synchronous development of HCC and xanthoma in this patient is yet to be clarified. Although no literature has reported the direct association between HCC and xanthoma, studies have shown that solid tumors may link to elevated low-density lipoprotein and triglyceride levels due to a shared pathway in cellular proliferation and cholesterol formation.[5] Future investigations to elucidate the causal effect between HCC and xanthoma should be conducted.

In summary, we propose a new subtype of xanthoma as “periungual xanthoma” with a special histopathological feature of foamy cell exocytosis. Larger clinical and histopathological studies are needed for the better understanding of this entity.

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 name and initials will not be published, and due efforts will be made to conceal his identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Hegele RA, Pollex RL. Hypertriglyceridemia: Phenomics and genomics. Mol Cell Biochem 2009;326:35-43.  Back to cited text no. 1
    
2.
Parker F. Xanthomas and hyperlipidemias. J Am Acad Dermatol 1985;13:1-30.  Back to cited text no. 2
    
3.
Cruz PD Jr., East C, Bergstresser PR. Dermal, subcutaneous, and tendon xanthomas: Diagnostic markers for specific lipoprotein disorders. J Am Acad Dermatol 1988;19:95-111.  Back to cited text no. 3
    
4.
Matiz C, Ferguson PJ, Zaenglein A, Groh B, Bingham CA. Papular xanthomas and erosive arthritis in a 3 year old girl, is this a new MRH variant? Pediatr Rheumatol Online J 2009;7:15.  Back to cited text no. 4
    
5.
Bielecka-Dąbrowa A, Hannam S, Rysz J, Banach M. Malignancy-associated dyslipidemia. Open Cardiovasc Med J 2011;5:35-40.  Back to cited text no. 5
    

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Correspondence Address:
Ding-Dar Lee,
Department of Dermatology, Taipei Veterans General Hospital, No. 201, Sec. 2, Shipai Road, Beitou District, Taipei City 11217
Taiwan
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/1027-8117.359340



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