Two cases of pachydermodactyly

Byung Duk Kang, Seung Hyun Hong, Hwan Kim, Woo Kyung Kim, Chil Hwan Oh

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Abstract

Case 1 A 27-year-old man had a 15-year history of progressive, asymptomatic, fusiform swelling on the medial and lateral aspects of several fingers. There was no history of significant trauma and pain or arthritic symptoms. The family history was noncontributory. His mother said that he had a habit of rubbing his fingers. Physical examination revealed fusiform swellings limited to the medial and lateral sides of the proximal interphalangeal joints of the index, middle, and ring fingers of both hands (Fig. 1). These lesions were firm tumors, and the overlying skin was lichenified. He had a full range of motion of all of his joints with no tenderness. The thumbs, outer aspects of the index and little fingers, and toes were spared. The roentgenogram of the hands showed soft tissue swellings without bony or articular abnormalities. Routine laboratory findings, including complete blood count, erythrocyte sedimentation rate, urine analysis, liver function test, concentration of nitrogen in the form of urea in the blood (BUN), creatinine, blood sugar, hepatitis B antigen, venereal disease research laboratory (VDRL) test, rheumatoid factor, anti-nuclear antibody, and chest X-ray, were either normal or negative. An excisional biopsy was performed (Fig. 2), which revealed hyperkeratotis, acanthosis, and reticular dermis thickened by deposits of collagen. In the reticular dermis, the collagen was arranged in a haphazard array. A proliferation of thin collagen fibers encircled the adnexal structures and blood vessels. Fibroblasts which were cytologically benign were slightly increased in the lesion. Silver nitrate impregnation staining revealed an increased number of reticulum fibers in the lesion. An ultrastructural examination of the reticular dermis from the lesional and adjacent normal skin was performed (Fig. 3, Table 1). We tried to measure the diameter and size of the collagen fibers using an image analysis system (AIC Inc., Roswall, GA). Collagen fibers on the lesion were smaller in diameter and area. In the lesion, the mean collagen fiber diameter was 80.43 nm and the mean area was 6.75 x 103 nm2. In the control, the mean collagen fiber diameter was 160.04 nm and the mean area was 27.83 x 103 nm2. The coefficient of variation in the diameter and area of the collagen fibers revealed a higher value in the lesional site than in the normal control, indicating that the collagen fibers on the lesion were less uniform in diameter and area than those of the normal control. The patient had subcutaneous and dermal resection over the PIP joint of the right ring finger by way of trial, and has been followed up until now. Case 2 A 33- year-old woman presented to our department with a 19-year history of swelling of the middle and ring fingers of the left hand. There was no history of significant trauma and pain or arthritic symptoms. Other past and family history was noncontributory, but she had a habit of rubbing her fingers. Physical examination revealed fusiform swelling limited to the medial and lateral sides of the proximal interphalangeal joints of the middle and ring fingers of the left hand (Fig. 4). The roentgenogram of her hands showed soft tissue swelling without bony or articular abnormalities. Routine laboratory findings were either normal or negative. The skin biopsy specimen revealed hyperkeratosis, acanthosis, and reticular dermis thickened by deposits of collagen. In the silver nitrate impregnation study, there was an increased amount of reticulum fiber. In image analysis, the diameters of the lesional collagen fibers were decreased and less uniform than the normal collagen fibers. With these clinical and histologic findings, a diagnosis of pachydermodactyly was made. After five intralesional injections of triamcinolone (4 mg/ml), the size of the skin lesion was decreased considerably.

Original languageEnglish
Pages (from-to)768-772
Number of pages5
JournalInternational Journal of Dermatology
Volume36
Issue number10
Publication statusPublished - 1997 Oct 1

ASJC Scopus subject areas

  • Dermatology

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  • Cite this

    Kang, B. D., Hong, S. H., Kim, H., Kim, W. K., & Oh, C. H. (1997). Two cases of pachydermodactyly. International Journal of Dermatology, 36(10), 768-772.