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Bacillary angiomatosis of acquired immunodeficiency syndrome: Case report and literature review
Brief communications Bacillaryangiomatosis of acquired immunodeficiency syndrome: Case report and literature review F. Jimenez-Acosta, MD,a Rube J. Pardo, MD, PhD,a Robert J. Cohen, MD,a Edwin W. Gould, MD,band Neal S. Penneys, MD, PhDa Miami. Florida Bacillary angiomatosis (BA), also called epithelioid angiomatosis, is a newly described disorder in patients with acquired immunodeficiency syndrome (AIDS). In 1983Stoleret al.' described a patientwithAIDS whohad an atypical subcutaneous infection that was recognized retrospectively to be the first recordedcase of BA. Since then severalreports have described AIDS patientswith angiomatous nodules that clinically resemble Kaposi's sarcoma or pyogenic granuloma but histologically havea distinct epithelioid hemangioma-like appearance.r" For these reasons, BA wasinitiallyconsidered a vascularneoplasm and was called epithelioid angiomatosis.? In 1988 LeBoit et al.3 characterized BA as an infectious process on the basis of the histochemical and ultrastructural identification of bacilliwithin the lesions. The bacilli appear to be identical to the organism that causes catscratch disease.b 9 We report a caseof BA sothat dermatologists mightbecomemoreawareofthis easily treatable disease. A brief review of the literature is included. Case report. A 50-year-old white homosexual man with AIDS had skinlesions of 2 months' duration. He denied fever, chills, or bonepainbut had a historyofweightloss. He had been feeding cats in his backyard for approximately 1 year and had received bites and scratchesfrom the cats. Physical examination revealed an afebrile, thin man with generalized lymphadenopathy and oral thrush. He had 15 red papules and friabledome-shaped nodules on the nose (Fig. 1), trunk, and lowerextremities (Fig. 2, A). The nodules varied from 0.5 to 4 em in diameterand somewerecrusted. A nodule on the right pretibial area was surrounded by an indurated erythematous border with a superficial scale (Fig. 3). Laboratory studies showed normal electrolytes and liver function. A CBCshowed the WBe countto be 2.0 X 103 cells/ mm'' with 33% neutrophils, 17% band forms, 2% eosinophils, 16%lymphocytes; hematocrit was 31% and hemoglobin, lOA gm/dl, The rapid plasma reagin (RPR) was nonreactive and urinalysis was normal. X-ray films of the involved extremities showed no evidence of bonechanges. A wedgebiopsy specimen From the Department of Dermatology and Cutaneous Surgery," and theDepartment ofPathology," University ofMiami School ofMedicine. Reprint requests: Neal S.Penneys, MD, University ofMiami School of Medicine, Department ofDermatologyandCutaneous Surgery, P.O. Box 016250 (R-250), Miami, FL 33101. 16/4/17604
of one of the leg nodules wasprocessed for light and electron microscopy. Hematoxylin-and-eosin-stained sections showed a vascular lesion involving the dermis aswell asthe subcutaneous tissue. The lesion wascomposed of abundant, thin-walled, vascularchannels ofvarying sizes that wereseparated byan edematousneutrophilic connective tissue(Fig. 4).Someofthevessels werelinedbyplumpendothelial cells that protruded intothe lumen.Fociof basophilic material couldbeseen adjacent tosome of thevessels (Fig.5).Special stains,including Giemsa's, ZiehlNeelsen, Steiner,Brown and Brenn, and periodic acid-Schiff, did notdetectmicroorganisms. The Warthin-Starry stain(performed by Colonel D. Wear at the ArmedForces Institute of Pathology) showed numerous clumps of darkly staining filamentous formsin thestromaand aroundblood vessels (Fig. 6). Ultrastructurally, bacilli could be seeninterspersed among collagen bundles. Morphologically the bacilli had a core of electron-dense material and a trilaminarcell wall (Fig. 7). The patient wastreatedwithoral erythromycin, 500mg every 6 hours,for 1 monthwithcomplete resolution of thelesions (Fig. 2, B).
Discussion. Our patientdeveloped progressive onset of red,friable, dome-shaped papules andnodules. Thisisthe mostcommon presentation in the reported patients,2-5 as summarized in Table I. Skinlesions havebeendescribed as red to purple, exophytic or pedunculated nodules, which can be easily confused withKaposi's sarcoma-3 or pyogenic granuloma." Lesions vary from a few to hundreds and can be found on the face, trunk, and extremities. Another type of presentation less often noted is an erythematous, induratedplaque" or a deepsubcutaneous masswithoverlying changes that resemble cellulitis/' Osseous lesions can occurundertheseinduratedmasses and can be demonstrated by roentgenographic studies or technetium-labeled bone scanning.v 6 The diagnosis ofBAissupported by histologic criteria. In BA,therearesmallvessel proliferations linedbyplump or epithelioid-like endothelial cells that may show cytologic atypia and mitotic activity.' In the stromathere is a polymorphousinfiltrate, neutrophilic debris, andedema. Hematoxylin-and-eosin-stained tissue sections mayshow a granularbasophilic material(asin ourcase)that isfrequently adjacentto the vascular channels. Thisgranular material is composed of numerous bacilli that are only demonstrable by a fastidious modified Warthin-Starry technique, as is shown in the present case. Other silver stains as well as other conventional histochemical techniques for visualizing bacteria fail to stain this bacillus. Ultrastucturalexamination ofthis material, however, reveals clusters of bacilli that usually lie between collagen bundles and contain an electron-dense centersurrounded bytrilaminarcellwalls. I, 6 Similarfindings wereobserved in our case. Lesions histologically similar to those found in the
525
526
Briefcommunications
Fig. 1. Fig. 2. 15 days Fig. 3.
Journal of the American Academy of Dermatology
Red, dome-shaped nodule on the nose. A, Friable and crusted exophytic nodule on dorsum of the foot. B, Resolution after of treatment with oral erythromycin. Red nodule on right pretibial area shows erythematous border and superficial scale.
skin have been described in oral, nasal, conjunctival, and anal mucosa2, 5 as well as in various internal organs. 2, 4 A relation between BA and cat-scratch disease has been reported.': 7,8 Some patients, like ours, have had a history ofbeing scratched by a cat. 3,5,6 Histologically, the bacilli of both cat-scratch disease and BA take up the Warthin-Starry stain, have similar ultrastructural features, and react with antiserum to cat-scratch disease bacillus on immunoperoxidase staining.'
The cutaneous and bone lesions of BA resolve rapidly with oral erythromycin at a dose of 500 mg every 4 to 6 hours. Doxycycline, isoniazid, and rifampin have also been used successfully.f In our patient, the lesions resolved in 15 days with 2 gm daily of oral erythromycin, although treatment was continued for 30 days. No recurrence has been observed after 4 months of follow-up. Some patients who have not received any treatment have been reported to progress to disseminated disease that has proved occasionally to be fatal.'
Volume 22 Number 3 March 1990
Brief communications 527
Fig. 4. Telangiectatic vessels are lined by plump endothelial cells. Intervening edematous stroma contains numerous neutrophils. (Hematoxylin-eosin stain; X600.)
Fig. 6. Clumps of darkly staining bacilli are seen in stroma, adjacent to blood vessels. (Warthin-Starry stain; X1000.)
Fig. 5. Clumps of amorphous basophilic material adjacent to a vessel (arrow). (Hematoxylin-eosin stain; XlOOO.)
Fig. 7. Multiple bacilli composed of electron-dense material and a trilaminar cell wall interspersed between collagen bundles. (X12,OOO.)
Table I. Cases of bacillary angiomatosis described in the English language literature Authons)
and reported cases (Age/Race/Sex)
Stoler et al:' 1 (32/B/M)* Cockerell et aU 1 (37/B/M)
Clinical presentation (Description; size; distribution)
Treatment
Follow-up
Multiple subcutaneous nodules; 2-6 em; head, extremities
Erythromycin
Resolved
Purple dome-shaped papules; 0.4-2.0 em; generalized
None
Died; autopsy showed widespread angiomatosis
B. Black; W. white; M, male; F. female. *The case reported by Stoler et al. (1983) is the same as case 1 reported by Lelsoitet al. (1988). tPatients 4, 5, 6, and 7 correspond to same patients numbered 4, J, 2, and 3, respectively, in Koehler et al, tHistory of cat-scratch exposure.
Continued
Journal of the American Academy of Dermatology
528 Brief communications Table I. Cont'd Author(s) andreported cases (Age/Race/Sex)
Cockerell et al. 2 2 (26/B/M) 3 (32/B/M)
4 (33/W/M) 5 (52/B/M)
Leboitet al,3 1 (32/M) 2 (39/M) 3 (31/M) 4,5,6,7t Angritt et aU 1 (26)
Knobler et al. 4 1 (46/M) 2 (24/M)
3 (311M)
4 (37/F)
Koehler et al.6 1 (43/M)*
Clinical presentation (Description; size; distribution)
Red-purple, dome-shaped papules; 0.2-1.0 em; face, trunk, extremities Red papules and nodules; 0.1-1.0 em; head, trunk, extremities; oral, conjunctival, and anal mucosa Solitary, flesh-colored nodule; forearm Reddish papules (15); scalp
Treatment
Follow-up
None
Spontaneous resolution
Excision, dessicated and curetted
Died; autopsyshowed widespread angiomatosis
Excision
No recurrence
None
Spontaneous resolution
Multiple colorless nodules; head, extremities Singleviolaceous subcutaneous nodule; forehead Multiple violaceous subcutaneous nodules; 2-3 em
Erythromycin; ketoconazole Excision
Resolved
Erythromycin; isoniazid; rifampin
Resolved
Erythematous, pedunculated, scaly, or ulcerated papules and nodules; 0.3-1.0 em; generalized
Not discussed
Not discussed
Red-purple papules; 3-4 mm; arms, buttocks, thighs, legs Red-purple accuminated and filiform papules (100); 2-4 mm; face, back, arms, legs Plaque; 1 em; forearm Erythematous indurated mass; 2 cm; on tibia with underlying bone lesion Red papules; 0.5-1.0 em; generalized. Liver biopsy finding: angiomatosis
None
Spontaneous resolution
Isoniazid; rifampin; ethambutol
Resolved
Not discussed
Not discussed
None
Spontaneous resolution
Penicillin Erythromycin
Not resolved Resolved
Nodules (5); face, thigh, back;mass; 3 x 5 ern; right wristwith underlying bone lesion
No recurrence
For legends see page527.
continued
Volume 22 Number 3 March 1990
Brief communications 529
Table I. Cont'd Author(s) and reportedcases (Age/Race/Sex)
Koehler et al.6
Clinical presentation (Description; size; distribution)
Treatment
Follow-up
2 (52jWjM)
Erythematous, exophytic angiomatous lesions (60); 0.5-2.0 cm; face, back, chest, arms, legs
Radiation Vinblastine Isoniazid; rifampin
Not resolved Not resolved Resolved
3 (51jWjM):j:
Red lesion; 1 cm; cellulitic appearance on finger Red papule; 0.8 em; inguinal region Pedunculated nodules; face, trunk, extremities (60) Subcutaneous mass; pretibial area with underlying bonelesion
Dicloxacillin; cephradine Doxycycline
Not resolved
Penicillin Nafcillin
Not resolved Not resolved
Dicloxacillin Isoniazid; rifampin; ethambutol; clofazamine
Not resolved Resolved
Electron beam Erythromycin
Not resolved Resolved
4 (39jBjM)
Axiotis et al. 5 1 (32jBjM):j:
Red papules and nodules (20); 0.5-4.0 cm; face and back Oral, nasal, and conjunctival mucosa
Resolved
For legend see page 527. REFERENCES 1. Stoler MH, Bonfiglio TA, Steigbigel RT, et al. An atypical subcutaneous infection associated with acquired immune deficiency syndrome. Am J Clin PathoI1983;80:714-8. 2. Cockerell Cl, Whitlow MA, WebsterGF, et al. Epithelioid angiomatosis: a distinct vasculardisorderin patients withthe acquiredimmunodeficiency syndrome orAIDS-related complex. Lancet 1987;2:654-6. 3. LeBoit PE, Berger TG, Egbert BM, et al. Epithelioid haemangioma-Iike vascular proliferation in AIDS:manifestation of cat scratch disease bacillus infection? Lancet 1988;1:960-3. 4. Knobler EH, Silvers ON, Fine KC, et al. Uniquevascular skin lesions associated with human immunodeficiency virus. JAMA 1988;260:524-7. 5. AxiotisCA, SchwartzR, Jennings TA, et al. AIDS-related angiomatosis. Am J OermatopathoI1989;1l:177-81. 6. KoehlerJE, LeBoit PE, Egbert BM,et al. Cutaneous vascular lesions and disseminated cat-scratch disease in patients with the acquired immunodeficiency syndrome (AIDS)and AIDS-related complex. Ann Intern Med 1988;109:449-55. 7. Angritt P, Tuur SM, Macher AM, et al. Epithelioid angiomatosisin HIV infection: neoplasm or cat-scratch disease? Lancet 1988;1 :96. 8. Cockerell CJ, Friedman-Kien AE. Epithelioid angiomatosis and cat-scratch disease bacillus. Lancet 1988;1:1334-5. 9. English CK, Wear OJ, Margileth AM, et al. Cat-scratch disease: isolation and culture of the bacterial agent.JAMA 1988;259:1347-52.
The melanocyte: An essential link in hydroquinoneinduced ochronosis PeterRobinHull,MB,BCh, MMED, FFDERM (SA), PhD (Med), and Peter Robert Procter, MB, BCh Pretoria, South Africa In southern Africatheprolonged and uncontrolled use ofhydroquinone-containing cosmetic bleaching products has resulted in an epidemic of exogenous ochronosis. I, 2 Despite the use of hydroquinone preparations in many countries, theprevalence ofhydroquinone-induced ochronosis is unrivaled outside the African subcontinent.l" Hydroquinone has been considered the sole factor in the pathogenesis of ochronosis. An essential and unsuspected roleplayed bythe melanocyte in the development of ochronosis is illustrated by the following case. Case report. A 60-year-old black woman, in whom vitiligo developed a yearbefore, began using ahydroquinone-containing bleaching creamto lighten her normal skin. A statutory maxFrom the Department ofDermatology, University ofPretoria. Reprint requests: Dr. P. R. Hull, P.O. Box 28815, Sunnyside, 0132 Pretoria, South Africa. 16/4/14141
of one of the leg nodules wasprocessed for light and electron microscopy. Hematoxylin-and-eosin-stained sections showed a vascular lesion involving the dermis aswell asthe subcutaneous tissue. The lesion wascomposed of abundant, thin-walled, vascularchannels ofvarying sizes that wereseparated byan edematousneutrophilic connective tissue(Fig. 4).Someofthevessels werelinedbyplumpendothelial cells that protruded intothe lumen.Fociof basophilic material couldbeseen adjacent tosome of thevessels (Fig.5).Special stains,including Giemsa's, ZiehlNeelsen, Steiner,Brown and Brenn, and periodic acid-Schiff, did notdetectmicroorganisms. The Warthin-Starry stain(performed by Colonel D. Wear at the ArmedForces Institute of Pathology) showed numerous clumps of darkly staining filamentous formsin thestromaand aroundblood vessels (Fig. 6). Ultrastructurally, bacilli could be seeninterspersed among collagen bundles. Morphologically the bacilli had a core of electron-dense material and a trilaminarcell wall (Fig. 7). The patient wastreatedwithoral erythromycin, 500mg every 6 hours,for 1 monthwithcomplete resolution of thelesions (Fig. 2, B).
Discussion. Our patientdeveloped progressive onset of red,friable, dome-shaped papules andnodules. Thisisthe mostcommon presentation in the reported patients,2-5 as summarized in Table I. Skinlesions havebeendescribed as red to purple, exophytic or pedunculated nodules, which can be easily confused withKaposi's sarcoma-3 or pyogenic granuloma." Lesions vary from a few to hundreds and can be found on the face, trunk, and extremities. Another type of presentation less often noted is an erythematous, induratedplaque" or a deepsubcutaneous masswithoverlying changes that resemble cellulitis/' Osseous lesions can occurundertheseinduratedmasses and can be demonstrated by roentgenographic studies or technetium-labeled bone scanning.v 6 The diagnosis ofBAissupported by histologic criteria. In BA,therearesmallvessel proliferations linedbyplump or epithelioid-like endothelial cells that may show cytologic atypia and mitotic activity.' In the stromathere is a polymorphousinfiltrate, neutrophilic debris, andedema. Hematoxylin-and-eosin-stained tissue sections mayshow a granularbasophilic material(asin ourcase)that isfrequently adjacentto the vascular channels. Thisgranular material is composed of numerous bacilli that are only demonstrable by a fastidious modified Warthin-Starry technique, as is shown in the present case. Other silver stains as well as other conventional histochemical techniques for visualizing bacteria fail to stain this bacillus. Ultrastucturalexamination ofthis material, however, reveals clusters of bacilli that usually lie between collagen bundles and contain an electron-dense centersurrounded bytrilaminarcellwalls. I, 6 Similarfindings wereobserved in our case. Lesions histologically similar to those found in the
525
526
Briefcommunications
Fig. 1. Fig. 2. 15 days Fig. 3.
Journal of the American Academy of Dermatology
Red, dome-shaped nodule on the nose. A, Friable and crusted exophytic nodule on dorsum of the foot. B, Resolution after of treatment with oral erythromycin. Red nodule on right pretibial area shows erythematous border and superficial scale.
skin have been described in oral, nasal, conjunctival, and anal mucosa2, 5 as well as in various internal organs. 2, 4 A relation between BA and cat-scratch disease has been reported.': 7,8 Some patients, like ours, have had a history ofbeing scratched by a cat. 3,5,6 Histologically, the bacilli of both cat-scratch disease and BA take up the Warthin-Starry stain, have similar ultrastructural features, and react with antiserum to cat-scratch disease bacillus on immunoperoxidase staining.'
The cutaneous and bone lesions of BA resolve rapidly with oral erythromycin at a dose of 500 mg every 4 to 6 hours. Doxycycline, isoniazid, and rifampin have also been used successfully.f In our patient, the lesions resolved in 15 days with 2 gm daily of oral erythromycin, although treatment was continued for 30 days. No recurrence has been observed after 4 months of follow-up. Some patients who have not received any treatment have been reported to progress to disseminated disease that has proved occasionally to be fatal.'
Volume 22 Number 3 March 1990
Brief communications 527
Fig. 4. Telangiectatic vessels are lined by plump endothelial cells. Intervening edematous stroma contains numerous neutrophils. (Hematoxylin-eosin stain; X600.)
Fig. 6. Clumps of darkly staining bacilli are seen in stroma, adjacent to blood vessels. (Warthin-Starry stain; X1000.)
Fig. 5. Clumps of amorphous basophilic material adjacent to a vessel (arrow). (Hematoxylin-eosin stain; XlOOO.)
Fig. 7. Multiple bacilli composed of electron-dense material and a trilaminar cell wall interspersed between collagen bundles. (X12,OOO.)
Table I. Cases of bacillary angiomatosis described in the English language literature Authons)
and reported cases (Age/Race/Sex)
Stoler et al:' 1 (32/B/M)* Cockerell et aU 1 (37/B/M)
Clinical presentation (Description; size; distribution)
Treatment
Follow-up
Multiple subcutaneous nodules; 2-6 em; head, extremities
Erythromycin
Resolved
Purple dome-shaped papules; 0.4-2.0 em; generalized
None
Died; autopsy showed widespread angiomatosis
B. Black; W. white; M, male; F. female. *The case reported by Stoler et al. (1983) is the same as case 1 reported by Lelsoitet al. (1988). tPatients 4, 5, 6, and 7 correspond to same patients numbered 4, J, 2, and 3, respectively, in Koehler et al, tHistory of cat-scratch exposure.
Continued
Journal of the American Academy of Dermatology
528 Brief communications Table I. Cont'd Author(s) andreported cases (Age/Race/Sex)
Cockerell et al. 2 2 (26/B/M) 3 (32/B/M)
4 (33/W/M) 5 (52/B/M)
Leboitet al,3 1 (32/M) 2 (39/M) 3 (31/M) 4,5,6,7t Angritt et aU 1 (26)
Knobler et al. 4 1 (46/M) 2 (24/M)
3 (311M)
4 (37/F)
Koehler et al.6 1 (43/M)*
Clinical presentation (Description; size; distribution)
Red-purple, dome-shaped papules; 0.2-1.0 em; face, trunk, extremities Red papules and nodules; 0.1-1.0 em; head, trunk, extremities; oral, conjunctival, and anal mucosa Solitary, flesh-colored nodule; forearm Reddish papules (15); scalp
Treatment
Follow-up
None
Spontaneous resolution
Excision, dessicated and curetted
Died; autopsyshowed widespread angiomatosis
Excision
No recurrence
None
Spontaneous resolution
Multiple colorless nodules; head, extremities Singleviolaceous subcutaneous nodule; forehead Multiple violaceous subcutaneous nodules; 2-3 em
Erythromycin; ketoconazole Excision
Resolved
Erythromycin; isoniazid; rifampin
Resolved
Erythematous, pedunculated, scaly, or ulcerated papules and nodules; 0.3-1.0 em; generalized
Not discussed
Not discussed
Red-purple papules; 3-4 mm; arms, buttocks, thighs, legs Red-purple accuminated and filiform papules (100); 2-4 mm; face, back, arms, legs Plaque; 1 em; forearm Erythematous indurated mass; 2 cm; on tibia with underlying bone lesion Red papules; 0.5-1.0 em; generalized. Liver biopsy finding: angiomatosis
None
Spontaneous resolution
Isoniazid; rifampin; ethambutol
Resolved
Not discussed
Not discussed
None
Spontaneous resolution
Penicillin Erythromycin
Not resolved Resolved
Nodules (5); face, thigh, back;mass; 3 x 5 ern; right wristwith underlying bone lesion
No recurrence
For legends see page527.
continued
Volume 22 Number 3 March 1990
Brief communications 529
Table I. Cont'd Author(s) and reportedcases (Age/Race/Sex)
Koehler et al.6
Clinical presentation (Description; size; distribution)
Treatment
Follow-up
2 (52jWjM)
Erythematous, exophytic angiomatous lesions (60); 0.5-2.0 cm; face, back, chest, arms, legs
Radiation Vinblastine Isoniazid; rifampin
Not resolved Not resolved Resolved
3 (51jWjM):j:
Red lesion; 1 cm; cellulitic appearance on finger Red papule; 0.8 em; inguinal region Pedunculated nodules; face, trunk, extremities (60) Subcutaneous mass; pretibial area with underlying bonelesion
Dicloxacillin; cephradine Doxycycline
Not resolved
Penicillin Nafcillin
Not resolved Not resolved
Dicloxacillin Isoniazid; rifampin; ethambutol; clofazamine
Not resolved Resolved
Electron beam Erythromycin
Not resolved Resolved
4 (39jBjM)
Axiotis et al. 5 1 (32jBjM):j:
Red papules and nodules (20); 0.5-4.0 cm; face and back Oral, nasal, and conjunctival mucosa
Resolved
For legend see page 527. REFERENCES 1. Stoler MH, Bonfiglio TA, Steigbigel RT, et al. An atypical subcutaneous infection associated with acquired immune deficiency syndrome. Am J Clin PathoI1983;80:714-8. 2. Cockerell Cl, Whitlow MA, WebsterGF, et al. Epithelioid angiomatosis: a distinct vasculardisorderin patients withthe acquiredimmunodeficiency syndrome orAIDS-related complex. Lancet 1987;2:654-6. 3. LeBoit PE, Berger TG, Egbert BM, et al. Epithelioid haemangioma-Iike vascular proliferation in AIDS:manifestation of cat scratch disease bacillus infection? Lancet 1988;1:960-3. 4. Knobler EH, Silvers ON, Fine KC, et al. Uniquevascular skin lesions associated with human immunodeficiency virus. JAMA 1988;260:524-7. 5. AxiotisCA, SchwartzR, Jennings TA, et al. AIDS-related angiomatosis. Am J OermatopathoI1989;1l:177-81. 6. KoehlerJE, LeBoit PE, Egbert BM,et al. Cutaneous vascular lesions and disseminated cat-scratch disease in patients with the acquired immunodeficiency syndrome (AIDS)and AIDS-related complex. Ann Intern Med 1988;109:449-55. 7. Angritt P, Tuur SM, Macher AM, et al. Epithelioid angiomatosisin HIV infection: neoplasm or cat-scratch disease? Lancet 1988;1 :96. 8. Cockerell CJ, Friedman-Kien AE. Epithelioid angiomatosis and cat-scratch disease bacillus. Lancet 1988;1:1334-5. 9. English CK, Wear OJ, Margileth AM, et al. Cat-scratch disease: isolation and culture of the bacterial agent.JAMA 1988;259:1347-52.
The melanocyte: An essential link in hydroquinoneinduced ochronosis PeterRobinHull,MB,BCh, MMED, FFDERM (SA), PhD (Med), and Peter Robert Procter, MB, BCh Pretoria, South Africa In southern Africatheprolonged and uncontrolled use ofhydroquinone-containing cosmetic bleaching products has resulted in an epidemic of exogenous ochronosis. I, 2 Despite the use of hydroquinone preparations in many countries, theprevalence ofhydroquinone-induced ochronosis is unrivaled outside the African subcontinent.l" Hydroquinone has been considered the sole factor in the pathogenesis of ochronosis. An essential and unsuspected roleplayed bythe melanocyte in the development of ochronosis is illustrated by the following case. Case report. A 60-year-old black woman, in whom vitiligo developed a yearbefore, began using ahydroquinone-containing bleaching creamto lighten her normal skin. A statutory maxFrom the Department ofDermatology, University ofPretoria. Reprint requests: Dr. P. R. Hull, P.O. Box 28815, Sunnyside, 0132 Pretoria, South Africa. 16/4/14141