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January 1989, Vol 125, No. 1 >
Article | January 1989

Penicillamine Dermatopathy With Lymphangiectases: Title and subTitle BreakA Clinical, Immunohistologic, and Ultrastructural Study

Jeffrey B. Goldstein, MD; N. Scott McNutt, MD; George W. Hambrick, Jr, MD; Hsu Amy
[+] Author Affiliations

Accepted for publication Aug 4, 1988.

Presented in part at the annual meeting of the American Academy of Dermatology, San Antonio, Tex, Dec 7, 1987.

Reprint requests to Dermatopathology (F-309), New York Hospital, 525 E 68th Street, New York, NY 10021 (Dr McNutt).


From the Departments of Medicine (Dermatology) (Drs Goldstein, McNutt, and Hambrick), Pathology (Dr McNutt and Ms Hsu), New York Hospital-Cornell University Medical College, New York.


Arch Dermatol. 1989;125(1):92-97. doi:10.1001/archderm.1989.01670130094014
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References
Comments

• The term penicillamine dermatopathy refers to the characteristic hemorrhagic skin lesions found in persons receiving long-term penicillamine therapy for either Wilson's disease or cystinuria. These lesions are thought to develop as a result of faulty collagen and elastin synthesis. We describe a patient with Wilson's disease who developed extensive penicillamine dermatopathy. In addition, histologic, immunochemical, and ultrastructural studies revealed multiple lymphangiectases with blood vessel to lymphatic anastomosis within these lesions, a finding not previously reported. The possible relationship to defective collagen and elastin formation are considered.

(Arch Dermatol 1989;125:92-97)

REFERENCES

1 +
Sternlieb I, Fisher M, Scheinberg IH:  Penicillamine-induced skin lesions . J Rheumatol 1981;;8( (suppl 7) ):149-154.
2 +
Sternlieb I, Scheinberg IH:  Penicillamine therapy for hepatolenticular degeneration . JAMA 1964;;189:748-754.
3 +
Katz R:  Penicillamine-induced skin lesions: A possible example of human lathyrism . Arch Dermatol 1977;;95:196-198.
4 +
Kueppers F, Daniels F Jr:  Penicillamine-induced skin lesions in patients with Wilson's disease . Cutis 1969;;5:35-39.
5 +
Greer KS, Asker FC, Richardson DR:  Skin lesions induced by penicillamine: Occurrence in a patient with hepatolenticular degeneration (Wilson's disease) . Arch Dermatol 1976;;112:1267-1269.
6 +
Walshe JM:  Toxic reactions to penicillamine in patients with Wilson's disease . Postgrad Med J 1968;;44( (suppl) ):6-8.
7 +
Levy RS, Fisher M, Alter JN:  Penicillamine: Review and cutaneous manifestation . J Am Acad Dermatol 1983;;8:548-558.
8 +
Beer WE, Cooke KB:  Epidermolysis bullosa induced by penicillamine . Br J Dermatol 1967;;79:123-125.
9 +
Hashimoto K, McEvoy B, Belcher R:  Ultrastructural penicillamine-induced skin lesions . J Am Acad Dermatol 1981;;4:300-315.
10 +
Lyle WH:  Penicillamine in metal poisoning . J Rheumatol 1981;;8( (suppl 7) ):96-99.
11 +
MacDonald WB, Fellers FX:  Penicillamine in the treatment of patients with cystinuria . JAMA 1966;;197:306-402.
12 +
Epstein O, de Villiers D, Jain S, et al:  Reduction of immune complexes and immunoglobulins induced by D-penicillamine in primary biliary cirrhosis . N Engl J Med 1979;;300:274-278.
13 +
Camp AV:  Penicillamine in the treatment of rheumatoid arthritis . J Rheumatol 1981;;8( (suppl 7) ):103-106.
14 +
Baum J:  The use of penicillamine in the treatment of rheumatoid arthritis and scleroderma . Scand J Rheumatol 1979;;28( (suppl) ):65-70.
15 +
Fulgham DD, Katz R:  Penicillamine for scleroderma . Arch Dermatol 1968;;98:51-52.
16 +
Perrett D:  The metabolism and pharmacology of D-penicillamine in man . J Rheumatol 1981;;8( (suppl 7) ):41-50.
17 +
Miller EJ, Mantin GR, Mecca CE, et al:  The biosynthesis of elastin crosslinks: The effects of copper deficiency and a lathyrogen . J Biol Chem 1965;;240:3623-3627.
18 +
Fisher I, Orkin M:  Acquired lymphangioma (lymphangiectasis) . Arch Dermatol 1970;;101:230-234.
19 +
Prioleau PG, Santa Cruz DJ:  Lymphangioma circumscriptum following radical mastectomy and radiation therapy . Cancer 1978;;42:1989-1991.
20 +
Capo V, Ozzello L, Fenoglio CM, et al:  Angiosarcomas arising in edematous extremities: Immunostaining for factor VIII-related antigen and ultrastructural features . Hum Pathol 1985;;16:144-150.
21 +
Jaffe EA, Hoyer LW, Nachman RL:  Synthesis of antihemophilic factor antigen by cultured human endothelial cells . J Clin Invest 1973;;52:2757-2764.
22 +
Miettinen M, Holthofer H, Lehto V, et al:  Ulex europaeus I lectin as a marker for tumors derived from endothelial cells . Am J Clin Pathol 1983;;79:32-36.
23 +
Miettinen M, Lehto V, Virtanen I:  Postmastectomy angiosarcoma (Stewart-Treves syndrome): Light-microscopic, immunohistological, and ultrastructural characteristics of two cases . Am J Surg Pathol 1983;;7:329-339.
24 +
Weiss SW, Enzinger FM:  Spindle cell hemangioendothelioma: A low-grade angiosarcoma resembling a cavernous hemangioma and Kaposi's sarcoma . Am J Surg Pathol 1986;;10:521-530.
25 +
Suzuki Y, Hashimoto K, Crissman J, et al:  The value of blood group-specific lectin and endothelial associated antibodies in the diagnosis of vascular proliferations . J Cutan Pathol 1986;;13:408-419.
26 +
Tufanelli DL:  Lymphangiectasis due to scleroderma . Arch Dermatol 1975;;3:1216.
27 +
DiLeonardo M, Jacoby R:  Acquired cutaneous lymphangiectasias secondary to scarring from scrofuloderma . J Am Acad Dermatol 1986;;14:688-690.
28 +
Bardach H, Gebhart W:  Elastic fiber changes induced by penicillamine . J Am Acad Dermatol 1982;;6:398-399.
29 +
Bardach H, Gebhart W, Niuebauer G:  `Lumpy-bumpy' elastic fibers in the skin and lungs of a patient with a penicillamineinduced elastosis perforans serpiginosa . J Cutan Pathol 1979;;6:243-252.
30 +
Weiss L:  Lymphatic vessels and lymph nodes , in Weiss L, Greep RO (eds): Histology , ed 4. New York, McGraw-Hill International Book Co, 1977;, pp 523-544.
31 +
Reed BR, Clark RAF:  Cutaneous tissue repair: Practical implications of current knowledge . J Am Acad Dermatol 1985;;13:919-941.

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1 +
Sternlieb I, Fisher M, Scheinberg IH:  Penicillamine-induced skin lesions . J Rheumatol 1981;;8( (suppl 7) ):149-154.
2 +
Sternlieb I, Scheinberg IH:  Penicillamine therapy for hepatolenticular degeneration . JAMA 1964;;189:748-754.
3 +
Katz R:  Penicillamine-induced skin lesions: A possible example of human lathyrism . Arch Dermatol 1977;;95:196-198.
4 +
Kueppers F, Daniels F Jr:  Penicillamine-induced skin lesions in patients with Wilson's disease . Cutis 1969;;5:35-39.
5 +
Greer KS, Asker FC, Richardson DR:  Skin lesions induced by penicillamine: Occurrence in a patient with hepatolenticular degeneration (Wilson's disease) . Arch Dermatol 1976;;112:1267-1269.
6 +
Walshe JM:  Toxic reactions to penicillamine in patients with Wilson's disease . Postgrad Med J 1968;;44( (suppl) ):6-8.
7 +
Levy RS, Fisher M, Alter JN:  Penicillamine: Review and cutaneous manifestation . J Am Acad Dermatol 1983;;8:548-558.
8 +
Beer WE, Cooke KB:  Epidermolysis bullosa induced by penicillamine . Br J Dermatol 1967;;79:123-125.
9 +
Hashimoto K, McEvoy B, Belcher R:  Ultrastructural penicillamine-induced skin lesions . J Am Acad Dermatol 1981;;4:300-315.
10 +
Lyle WH:  Penicillamine in metal poisoning . J Rheumatol 1981;;8( (suppl 7) ):96-99.
11 +
MacDonald WB, Fellers FX:  Penicillamine in the treatment of patients with cystinuria . JAMA 1966;;197:306-402.
12 +
Epstein O, de Villiers D, Jain S, et al:  Reduction of immune complexes and immunoglobulins induced by D-penicillamine in primary biliary cirrhosis . N Engl J Med 1979;;300:274-278.
13 +
Camp AV:  Penicillamine in the treatment of rheumatoid arthritis . J Rheumatol 1981;;8( (suppl 7) ):103-106.
14 +
Baum J:  The use of penicillamine in the treatment of rheumatoid arthritis and scleroderma . Scand J Rheumatol 1979;;28( (suppl) ):65-70.
15 +
Fulgham DD, Katz R:  Penicillamine for scleroderma . Arch Dermatol 1968;;98:51-52.
16 +
Perrett D:  The metabolism and pharmacology of D-penicillamine in man . J Rheumatol 1981;;8( (suppl 7) ):41-50.
17 +
Miller EJ, Mantin GR, Mecca CE, et al:  The biosynthesis of elastin crosslinks: The effects of copper deficiency and a lathyrogen . J Biol Chem 1965;;240:3623-3627.
18 +
Fisher I, Orkin M:  Acquired lymphangioma (lymphangiectasis) . Arch Dermatol 1970;;101:230-234.
19 +
Prioleau PG, Santa Cruz DJ:  Lymphangioma circumscriptum following radical mastectomy and radiation therapy . Cancer 1978;;42:1989-1991.
20 +
Capo V, Ozzello L, Fenoglio CM, et al:  Angiosarcomas arising in edematous extremities: Immunostaining for factor VIII-related antigen and ultrastructural features . Hum Pathol 1985;;16:144-150.
21 +
Jaffe EA, Hoyer LW, Nachman RL:  Synthesis of antihemophilic factor antigen by cultured human endothelial cells . J Clin Invest 1973;;52:2757-2764.
22 +
Miettinen M, Holthofer H, Lehto V, et al:  Ulex europaeus I lectin as a marker for tumors derived from endothelial cells . Am J Clin Pathol 1983;;79:32-36.
23 +
Miettinen M, Lehto V, Virtanen I:  Postmastectomy angiosarcoma (Stewart-Treves syndrome): Light-microscopic, immunohistological, and ultrastructural characteristics of two cases . Am J Surg Pathol 1983;;7:329-339.
24 +
Weiss SW, Enzinger FM:  Spindle cell hemangioendothelioma: A low-grade angiosarcoma resembling a cavernous hemangioma and Kaposi's sarcoma . Am J Surg Pathol 1986;;10:521-530.
25 +
Suzuki Y, Hashimoto K, Crissman J, et al:  The value of blood group-specific lectin and endothelial associated antibodies in the diagnosis of vascular proliferations . J Cutan Pathol 1986;;13:408-419.
26 +
Tufanelli DL:  Lymphangiectasis due to scleroderma . Arch Dermatol 1975;;3:1216.
27 +
DiLeonardo M, Jacoby R:  Acquired cutaneous lymphangiectasias secondary to scarring from scrofuloderma . J Am Acad Dermatol 1986;;14:688-690.
28 +
Bardach H, Gebhart W:  Elastic fiber changes induced by penicillamine . J Am Acad Dermatol 1982;;6:398-399.
29 +
Bardach H, Gebhart W, Niuebauer G:  `Lumpy-bumpy' elastic fibers in the skin and lungs of a patient with a penicillamineinduced elastosis perforans serpiginosa . J Cutan Pathol 1979;;6:243-252.
30 +
Weiss L:  Lymphatic vessels and lymph nodes , in Weiss L, Greep RO (eds): Histology , ed 4. New York, McGraw-Hill International Book Co, 1977;, pp 523-544.
31 +
Reed BR, Clark RAF:  Cutaneous tissue repair: Practical implications of current knowledge . J Am Acad Dermatol 1985;;13:919-941.

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