Eruptive disseminated lobular capillary hemangioma (pyogenic granuloma)

Eruptive disseminated lobular capillary hemangioma (pyogenic granuloma)

Volume 21 Number 2, Part 2 August 1989 12. 13. 14. 15. 16. 17. refractory genital warts. J Invest Dermatol 1986; 86:485A. Edwards L. Interferon: pr...

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Volume 21 Number 2, Part 2 August 1989

12. 13. 14. 15.

16. 17.

refractory genital warts. J Invest Dermatol 1986; 86:485A. Edwards L. Interferon: promises, disappointments, and tempered optimism. Arch Dermatol 1987;123:743-4. Tyring SK. Antitumor actions of interferons: direct, indirect, and synergy with other treatment modalities. Int J Dermatol 1987;26:549-56. Morhenn V. The etiology of lichen planus: a hypothesis. Am J Dermatopathol 1986;18:154-6. Jacob CO, van der Meide PH, McDevitt HO. In vivo treatment of (NZB • NZW) Ft lupus-like nephritis with monoclonal antibody to 1' interferon. J Exp Med 1987; 166:798-802. Trichieri G, M-Kobayashi M, Clark SV, et al. Response of resting human peripheral blood natural killer cells to interleukin 2. J Exp Med 1984;160:1147-69. Pace J, Russel SW, LeBlanc PA. Comparative effects of

Interferon-3, as adjuvant therapy for resistant warts various classes of mouse interferons on maerophage activation for tumor cell kilting. J Inamunol 1985;134:97781. 18. Tsujilnoto M, Yip YK, Vilcek J. /nterferon-,r enhances expression of cellular receptors for tumor necrosis factor. J Immunol 1986;136:2441-4. 19. Shiohara T, Nickoloff BJ, Moriya N, et al. In vivo effects of interferon-3, and anti-interferon-3, antibody on the experimentally induced lichenoid tissue reaction. Br J Dermatol 1988;I 19:199-206. 20. Nickoloff BJ, Lewinsohn DM, Butcher EC, et al. Recombinant gamma interferon increases the binding of peripheral blood mononuclear leukocytes and a Leu-3 + T lymphocyte cell clone to cultured keratinocytes and a malignant cutaneous squamous carcinoma cell line which is blocked by antibody against the LFA-1 molecule. J Invest Dermatol 1988;90:17-22.

Eruptive disseminated lobular capillary hemangioma (pyogenic granuloma) Barbara Braunstein Wilson, MD, ~ Kenneth E. Greer, MD," and Philip H. Cooper, MD a.b

Charlottesville, Virginia A 43-year-old man reported a 2-month history of numerous widespread vascular lesions that had developed suddenly on his scalp, trunk, and extremities. The lesions were 2 to 6 m m in diameter, firm, dark red papules that bled easily with minor trauma. Histopathologic examination of four lesions revealed lobular capillary hemangioma, also known as pyogenie granuloma. Treated lesions did not recur after surgical excision or electrocauterization. Five months after the onset of the eruption, no new lesions were developing, and some were resolving spontaneously after bleeding. Eruptive, disseminated lobular capillary hemangioma is a rare, self-limited cutaneous disorder that occurs in otherwise healthy persons. (J AM ACAO DERMATOL 1989;21:391-4.)

Lobular capillary hemangioma, 1 also known as pyogenic granuloma, is a common, benign vascular tumor of the skin and mucous membranes that can develop spontaneously or in response to trauma. Subcutaneous 2 and intravenous lobular capillary hemangiomas 3 also have been described. These vascular tumors are usually solitary, although multiple satellite lesions occasionally develop near a primary lobular capillary hemangioma, usually after destruction of that lesion. 4,s Rarely, lobular

Fromthe Departmentsof Dermatology'and Pathology,bUniversityof Virginia School of Medicine. Reprint requests: Barbara Braunstein Wilson, MD, Box 134, Department of Dermatology,Universityof Virginia School of Medicine, Charlottesville, VA 22908.

capillary hemangioma presents in an eruptive, disseminated form. 6-9 We report such a case and review the pertinent literature. CASE R E P O R T A 43-year-old man was in good health until 2 months before evaluation, when he noted the sudden appearance of approximately 10 red papules on his chest and back. The lesions bled easily and profusely, often during the night while he was sleeping. Histologie examination of two biopsy specimens revealed lobular capillary hemangioma. During the next several weeks, new lesions continued to develop on his trunk, scalp, and extremities. Findings from the following laboratory tests were normal: WBC and differential cell counts, platelet counts, prothrombin time, partial thromboplastin time, fibrinogen level, serum protein electrophoresis, bleeding time,

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Fig. 1. Isolated 2 mm lobular capillary hemangioma on finger.

Sequential Multiple Analyzer 12/60, erythrocyte sedmentation rate, and urinalysis. Results of an electrocardiogram and chest roentgenogram also were normal. Because of frequent episodes of bleeding, 25 to 30 lesions were treated by eleetrocauterizafion. The patient was then referred to the University of Virginia Department of Dermatology for further evaluation. Physical examination revealed eight firm, shiny, dark red papules, 2 to 6 ram, scattered on the patient's scalp, trunk, finger, and thigh (Fig. 1). Some of the papules had a slightly scaly surface as well as a collarette of scale around their bases. Two papules were excised, and microscopic examination of this material revealed lobules of capillaries in the papillary dermis characteristic of lobular capillary hemangioma (Fig. 2). During the next 2 months, the patient continued to develop new lesions (approximately 15) and many of the original lobular capillary hemangiomas enlarged to about 5 or 6 ram. Lesions that bled spontaneously resolved and did not recur. Treated lesions also did not recur. Five months after the lesions first appeared, no new lobular capillary hemangiomas were developing. DISCUSSION Clinically, lobular capillary hemangioma (pyogenie granuloma) typically presents as a dark red papule, 2 to 10 mm, that usually develops during a period of a few weeks and bleeds with minimal trauma. Solitary lesions are seen most frequently on the face, scalp, acral locations, and oronasal mucous membranes in otherwise healthy persons. Microscopically, lobular capillary hemangioma demonstrates diagnostic changes composed of a characteristic lobular arrangement of capillaries at the base of the lesion. ~ T h e more superficial portions may exhibit edema, capillary dilation,

Journal of the American Academy of Dermatology inflammation, and changes consistent with granulation tissue. There have been several reports of multiple satellite lesions developing near a primary lobular capillary hemangioma, usually within 1 to 4 months after surgical excision or fulguration of the primary lesion. 4,5 Less commonly, satellite lobular capillary hemangiomas develop spontaneously before the primary lesion has been treated? ~ Most lobular capillary hemangiomas with satellites occur in adolescents and young adults and are located on the upper portion of the trunk, especially in the interscapular region. It is not uncommon for treated satellite lesions to recur. Spontaneous resolution of satellite lesions also has been reported. 4 Multiple grouped lobular capillary hemangiomas also have been described in nevus flammeus 4, ~1 and after trauma? 2 Rarely, lobular capillary hemangioma presents in an eruptive, disseminated form. In a review of the literature published in English we found five well-documented examples of this phenomenon (Table I). Eruptive, disseminated lobular capillary hemangioma is characterized by the sudden development of numerous widespread lobular capillary hemangiomas that range in size from 1 to 10 mm. The benign vascular tumors usually continue to erupt for days to months. The number of lobular capillary hemangiomas has been as few as five and as many as 700, and a major complaint in all but one case 8 has been the tendency for the lesions to bleed with minimal trauma. The age at onset is highly variable, ranging from 15 months to 63 years. In most instances, the lobular capillary hemangiomas were distributed in a generalized fashion. In one case, however, the lesions were confined to the neck and upper portion of the trunk. 6 The tumors developed spontaneously in four patients. In one patient they occurred in skin that had sustained second-degree burns 1 week before the development of the vascular tumors] and in another patient multiple lobular capillary hemangiomas developed after removal of a solitary lesion near the eye? In all cases the lobular capillary hemangiomas showed evidence of regression or complete spontaneous resolution within several months to approximately 1 year. Lesions that were treated did not recur. Three of the six patients with eruptive, disseminated lobular capillary hemangioma had other medical problems, including hypogammaglobulin-

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Eruptive disseminated lobular capillary hemangioma 393

Fig. 2. Characteristic microscopic appearance of lesion. Overlying epidermis is intact; therefore stroma lacks inflammation or granulation tissue response. (Hematoxylin-eosin stain; x65.) Table I. Clinical findings of patients with eruptive disseminated lobular capillary hemangioma Size (mm)

Underlying disease

12 18

-10

None Hypogammaglobulinemia

M/52 yr F/61 yr

130 5

1-3 4-10

Hodgkin's disease Alcoholic cirrhosis

Nappi and Wick 9

F/60 yr

700

2-10

None

Our case

M/38 yr

45

1-6

None

Reference

Sex/Age

Juhlin et al.6 DeKaminsky et al. 7

F/11 yr F/15 mo

Pembroke et al) Nappi and Wick9

No. of lesions

emia, 7 alcoholic cirrhosis, 9 and Hodgkin's disease. 8 One patient had a decreased helper/suppressor T cell ratio, but no associated immunodeficiency disease could be found? Pembroke et al. 8 described four patients with "eruptive angiomata" who had underlying malignancies. The features in one of their patients (case 1) dearly fit the clinical and histologic picture of multiple disseminated lobular capillary hemangioma. This patient is included in Table I. Two of their patients had lesions that resembled lobular capillary hemangioma clinically, but the microscopic findings were not clearly diagnostic of lobular capillary hemangioma. The

Course

Resolved in months No recurrence after therapy Resolved in months Resolved after approximately 1 yr Resolving spontaneously at 5 mo No new lesions developing after 5 mo; ruptured lesions resolving spontaneously

fourth patient developed widespread, deep-blue, vascular nodules that clinically and histologically resembled cavernous hemangiomas, Thus it does not appear that eruptive, disseminated lobular capillary hemangioma is clearly associated with a specific underlying disease. As more patients with this rare disorder are described, however, it is possible that an association may be found. The cause of lobular capillary hemangioma is unknown, but it has been suggested that these benign cutaneous growths may develop in areas where there is an underlying vascular abnormality that may be predisposed to the development of

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angiomatous proliferations:," A hypothesis for the development of the variant with multiple satellite lesions is that destruction of the primary lobular capillary hemangioma may cause hydrostatic pressure to be transferred to surrounding skin, thereby stimulating capillary proliferation? This, however, would not explain the spontaneous development of mukiple satellite lobular capillary hemangioma around an untreated primary lesion. It also has been suggested that an angiogenic factor released by a primary lobular capillary hemangiom a may stimulate the proliferation of satellite lesions. As with the solitary and multiple satellite lobular capillary hemangiomas, the pathogenesis for eruptive, disseminated lobular capillary hemangioma is unknown. Eruptive, disseminated lobular capillary hemangioma is a rare variant of a common lesion and is not well documented in the dermatologic literature. The eruption is often alarming for the patient because of the abrupt onset, the multiplicity of lesions, and the tendency of the lesions to bleed. It is important for dermatologists to be aware of the benign nature of this disorder so that patients can be reassured of the likelihood of spontaneous resolution. REFERENCES 1. Mills SE, Cooper PH, Feehner RE, Lobular capillary hemangioma: the underlying lesion of pyogenic granulo-

2. 3. 4. 5. 6. 7. 8. 9.

I0. 11. 12.

ma--a study of 73 cases from the oral and nasal mucous membranes. Am J Surg Pathol 1980;4:471-9. CooperPH, MilksSE. Subcutaneous granuloma pyogenicum. Lobular capillary hemangioma. Arch Dermatol 1982;118:30-3. Cooper PH, MeAllister HA, Helwig EB. Intravenous pyogenie granuloma: a study of 18 cases. Am J Surg Pathol 1979;3:221-8. Warner J, Wilson Jones E. Pyogenicgranuloma recurring with multiple satellites: a report of 11 cases. Br J Dermatol 1968;80:218-27. Zaynoun ST, Juljulian HH, Kurban AK. Pyogenicgranulorna with multiple satellites. Arch Dermatol 1974; 109:689-91. Juhlin L, Hjertquist S-O, Pont6n J, et al. Disseminated granuloma pyogenicum. Acta Derm Venereol (Stockh) 1970;50:134-6. DeKaminsky AR, Otero AC, Kaminsky CA, et al. Multiple disseminated pyogenicgranuloma. Br J Dermatol 1978;98:461-4. Pembroke AC, Griee K, Levantine AV, et al. Eruptive angiomata in malignant disease. Clin Exp Dermatol 1978;3:147-56. Nappi O, Wick MR. Disseminated lobular capillary hemangioma (pyogenic granuloma): a clinicopathologic study of two cases. Am J Dermatopathol 1986;8:37985. Frain-Bell W. Multiple pyogenic granulomata. Br J Dermatol 1958;70:428-9. Swerlick RA, Cooper PH. Pyogenie granuloma (lobular capillary hemangioma) within port-wine stains. J AM ACADDERt~ATOL1983;8:627-30. Foldvari F. Post traumatic angioma. Br J Dermatol 1935;47:463-7.