Case report
Leukocytoclastic vasculitis associated with sulfuric acid inhalation Zaira Pellicer-Oliver1, MD, Jose Marı´a Martin1, MD, Rebeca Bella-Navarro1, MD, Carlos Monteagudo2, MD, and Esperanza Jorda´1, MD
Departments of 1Dermatology and 2 Pathology, University Hospital Clinic, University of Valencia, Valencia, Spain Correspondence Zaira Pellicer-Oliver, MD Department of Dermatology Hospital Clı´nico Universitario Universidad de Valencia Avd. Blasco Iba´n˜ez 17. 46010 Valencia Spain E-mail: [email protected] Conflicts of interest: None.
Cutaneous leukocytoclastic vasculitis (CLV) is confined to capillaries and post-capillary venules of the superficial plexus.1 The principal clinical lesions in CLV are purpuric papules, although other clinical findings secondary to ischemia, including ulceration, may occur. Causes of CLV include drugs, infection, connective tissue disease, and malignancy.2 Cutaneous leukocytoclastic vasculitis has very rarely been reported in association with the inhalation of drugs or chemical products.3 We present an atypical case of leukocytoclastic vasculitis secondary to sulfuric acid inhalation in a 53-year-old man. Complementary examinations ruled out systemic involvement and other causes of CLV. To our knowledge, this is the first case of CLV following inhalation of sulfuric acid to be reported. Case report
228
A 53-year-old man presented with cutaneous lesions located on the limbs that had progressively developed over the previous three days. Physical examination revealed an extensive eruption of palpable purpuric papular lesions on the limbs (Fig. 1). The lesions were slightly pruriginous, and Koebner’s phenomenon could be seen in some areas. Moreover, several International Journal of Dermatology 2014, 53, 228–230
Figure 1 Extensive eruption of palpable purpuric papular lesions on the limbs
linear lesions suggestive of burn scabs were observed on the right leg and abdomen. There were no associated systemic symptoms and, despite the striking cutaneous manifestations, the patient’s general health was good. Anamnesis revealed that five days previously the patient had used the chemical product MELT (95% sulfuric acid) to unblock his drains. The burn scabs had formed ª 2013 The International Society of Dermatology
Pellicer-Oliver et al.
Leukocytoclastic vasculitis associated with sulfuric acid inhalation
Case report
(a) Figure 2 Burn scabs formed by the direct bonding of the product on the leg
as a result of the direct bonding of the product with the skin on the right leg and on the abdomen (Fig. 2). The subject also reported nausea and dizziness but denied any signs of fever, dyspnea, ocular alterations, and other symptoms suggestive of respiratory or systemic involvement after inhalation. The patient reported that slight cutaneous lesions had appeared on the day he used the product, but 48 hours later, the flare became severe and rapidly involved the entire limbs. A skin biopsy of a cutaneous purpuric lesion revealed a superficial venular neutrophilic inflammatory infiltration with extensive leukocytoclasis and endothelial damage, together with red blood cell extravasation (Fig. 3), all of which are characteristic of leukocytoclastic vasculitis. Laboratory studies demonstrated a normal blood cell count, serum chemistry, coagulation profile, antineutrophil cytoplasmic antibodies (ANCA), antinuclear antibodies (ANA), anti-DNA antibodies, antibodies to extractable nuclear antigens (ENA), anti-Ro antibodies, anticardiolipin antibodies, circulating immune complexes, rheumatoid factor, and cryoglobulins. Moreover, serological examinations for human immunodeficiency virus (HIV), hepatitis B virus (HBV), and hepatitis C virus (HCV) were also negative. The patient initially received symptomatic treatment with oral antihistamines and was advised to rest. During the course of the next week, the vasculitic lesions faded progressively.
Discussion Chemical agents are widely distributed in a diverse range of products. Sulfuric acid has a wide range of applications and is used in many industries.4 Our patient used a proª 2013 The International Society of Dermatology
(b) Figure 3 Histopathology shows (a) superficial dermal neutro-
philic infiltrates centered in small vessels and (b) endothelial damage, leukocytoclasis and red blood cell extravasation. (Hematoxylin and eosin stain; original magnification [a] ·40, [b] ·400)
duct called MELT to unblock his water drains. This product consists of 95% sulfuric acid. Sulfuric acid is a desiccant and causes dehydration damage or produces excessive heat in tissues.5 Common adverse effects associated with sulfuric acid include ocular, respiratory, cutaneous, and systemic alterations. Inhalation of sulfuric acid vapor mainly affects the laryngeal, pharyngeal, and tracheobronchial mucosa.6,7 Sulfuric acid induces cutaneous inflammation and blistering.8 Cutaneous leukocytoclastic vasculitis involves the deposition of immune complexes in vessel walls, ultimately leading to cellular infiltrates, cytokine release, and vessel damage. Common causes of CLV include infections (HIV, HBV, HCV), malignancy, connective tissue disease (systemic lupus erythematosus, rheumatoid arthritis), autoimmune disease, and drugs (antibiotics, diuretics, sulfonamides, allopurinol, phenytoin).9 The disease has very International Journal of Dermatology 2014, 53, 228–230
229
230
Case report
Leukocytoclastic vasculitis associated with sulfuric acid inhalation
rarely been reported in association with inhalation of drugs or chemical products.3 In this case, the workup allowed us to exclude systemic involvement. Despite the expansion of the cutaneous lesions, the patient’s general good health and the absence of respiratory and systemic involvement allowed us to adopt a conservative treatment regime. Other possible causes of vasculitis were excluded by diagnostic tests. The temporal relationship between the inhalation of sulfuric acid, together with the absence of relapse after discontinuation of the exposition, support the hypothesis that the inhalation of sulfuric acid represented the cause of vasculitis in the present patient.10 We were unable to find any other cases of leukocytoclastic vasculitis resulting from the inhalation of sulfuric acid in the literature. Although rare, the inhalation of sulfuric acid vapor should be considered as another cause of leukocytoclastic vasculitis.
4
5
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7
8
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References 1 Crowson AN, Mihm MC Jr, Magro CM. Cutaneous vasculitis: a review. J Cutan Pathol 2003; 30: 161–173. 2 Khasnis A, Langford CA. Update on vasculitis. J Allergy Clin Immunol 2009; 123: 1226–1236. 3 Nepal M, Padma H. Fluticasone-associated cutaneous allergic granulomatous vasculitis: an under-recognized
International Journal of Dermatology 2014, 53, 228–230
10
Pellicer-Oliver et al.
but important cause of drug-induced cutaneous Churg–Strauss syndrome. South Med J 2008; 101: 761–763. Kilgour JD, Foster J, Soames A. Responses in the respiratory tract of rats following exposure to sulfuric acid aerosols for 5 or 28 days. J Appl Toxicol 2002; 22: 387–395. Ricketts S, Kimble FW. Chemical injuries: the Tasmanian Burns Unit experience. ANZ J Surg 2003; 73: 45–48. Kehe K, Thiermann H, Balszuweit F, et al. Acute effects of sulfur mustard injury – Munich experiences. Toxicology 2009; 263: 3–8. Husain MT, Hasanain J, Kumar P. Sulphuric acid burns: report of a mass domestic accident. Burns 1989; 15: 389– 391. Kehe K, Balszuweit F, Steinritz D, et al. Molecular toxicology of sulfur mustard-induced cutaneous inflammation and blistering. Toxicology 2009; 263: 12– 19. Blanco R, Martínez-Taboada VM, Rodríguez-Valverde V, García-Fuentes M. Cutaneous vasculitis in children and adults: associated diseases and etiologic factors in 303 patients. Medicine (Baltimore) 1998; 77: 403–418. Calabrese LH, Michel BA, Bloch DA, et al. The American College of Rheumatology 1990 criteria for the classification of hypersensitivity vasculitis. Arthritis Rheum 1990; 33: 1108–1113.
ª 2013 The International Society of Dermatology
Leukocytoclastic vasculitis associated with sulfuric acid inhalation Zaira Pellicer-Oliver1, MD, Jose Marı´a Martin1, MD, Rebeca Bella-Navarro1, MD, Carlos Monteagudo2, MD, and Esperanza Jorda´1, MD
Departments of 1Dermatology and 2 Pathology, University Hospital Clinic, University of Valencia, Valencia, Spain Correspondence Zaira Pellicer-Oliver, MD Department of Dermatology Hospital Clı´nico Universitario Universidad de Valencia Avd. Blasco Iba´n˜ez 17. 46010 Valencia Spain E-mail: [email protected] Conflicts of interest: None.
Cutaneous leukocytoclastic vasculitis (CLV) is confined to capillaries and post-capillary venules of the superficial plexus.1 The principal clinical lesions in CLV are purpuric papules, although other clinical findings secondary to ischemia, including ulceration, may occur. Causes of CLV include drugs, infection, connective tissue disease, and malignancy.2 Cutaneous leukocytoclastic vasculitis has very rarely been reported in association with the inhalation of drugs or chemical products.3 We present an atypical case of leukocytoclastic vasculitis secondary to sulfuric acid inhalation in a 53-year-old man. Complementary examinations ruled out systemic involvement and other causes of CLV. To our knowledge, this is the first case of CLV following inhalation of sulfuric acid to be reported. Case report
228
A 53-year-old man presented with cutaneous lesions located on the limbs that had progressively developed over the previous three days. Physical examination revealed an extensive eruption of palpable purpuric papular lesions on the limbs (Fig. 1). The lesions were slightly pruriginous, and Koebner’s phenomenon could be seen in some areas. Moreover, several International Journal of Dermatology 2014, 53, 228–230
Figure 1 Extensive eruption of palpable purpuric papular lesions on the limbs
linear lesions suggestive of burn scabs were observed on the right leg and abdomen. There were no associated systemic symptoms and, despite the striking cutaneous manifestations, the patient’s general health was good. Anamnesis revealed that five days previously the patient had used the chemical product MELT (95% sulfuric acid) to unblock his drains. The burn scabs had formed ª 2013 The International Society of Dermatology
Pellicer-Oliver et al.
Leukocytoclastic vasculitis associated with sulfuric acid inhalation
Case report
(a) Figure 2 Burn scabs formed by the direct bonding of the product on the leg
as a result of the direct bonding of the product with the skin on the right leg and on the abdomen (Fig. 2). The subject also reported nausea and dizziness but denied any signs of fever, dyspnea, ocular alterations, and other symptoms suggestive of respiratory or systemic involvement after inhalation. The patient reported that slight cutaneous lesions had appeared on the day he used the product, but 48 hours later, the flare became severe and rapidly involved the entire limbs. A skin biopsy of a cutaneous purpuric lesion revealed a superficial venular neutrophilic inflammatory infiltration with extensive leukocytoclasis and endothelial damage, together with red blood cell extravasation (Fig. 3), all of which are characteristic of leukocytoclastic vasculitis. Laboratory studies demonstrated a normal blood cell count, serum chemistry, coagulation profile, antineutrophil cytoplasmic antibodies (ANCA), antinuclear antibodies (ANA), anti-DNA antibodies, antibodies to extractable nuclear antigens (ENA), anti-Ro antibodies, anticardiolipin antibodies, circulating immune complexes, rheumatoid factor, and cryoglobulins. Moreover, serological examinations for human immunodeficiency virus (HIV), hepatitis B virus (HBV), and hepatitis C virus (HCV) were also negative. The patient initially received symptomatic treatment with oral antihistamines and was advised to rest. During the course of the next week, the vasculitic lesions faded progressively.
Discussion Chemical agents are widely distributed in a diverse range of products. Sulfuric acid has a wide range of applications and is used in many industries.4 Our patient used a proª 2013 The International Society of Dermatology
(b) Figure 3 Histopathology shows (a) superficial dermal neutro-
philic infiltrates centered in small vessels and (b) endothelial damage, leukocytoclasis and red blood cell extravasation. (Hematoxylin and eosin stain; original magnification [a] ·40, [b] ·400)
duct called MELT to unblock his water drains. This product consists of 95% sulfuric acid. Sulfuric acid is a desiccant and causes dehydration damage or produces excessive heat in tissues.5 Common adverse effects associated with sulfuric acid include ocular, respiratory, cutaneous, and systemic alterations. Inhalation of sulfuric acid vapor mainly affects the laryngeal, pharyngeal, and tracheobronchial mucosa.6,7 Sulfuric acid induces cutaneous inflammation and blistering.8 Cutaneous leukocytoclastic vasculitis involves the deposition of immune complexes in vessel walls, ultimately leading to cellular infiltrates, cytokine release, and vessel damage. Common causes of CLV include infections (HIV, HBV, HCV), malignancy, connective tissue disease (systemic lupus erythematosus, rheumatoid arthritis), autoimmune disease, and drugs (antibiotics, diuretics, sulfonamides, allopurinol, phenytoin).9 The disease has very International Journal of Dermatology 2014, 53, 228–230
229
230
Case report
Leukocytoclastic vasculitis associated with sulfuric acid inhalation
rarely been reported in association with inhalation of drugs or chemical products.3 In this case, the workup allowed us to exclude systemic involvement. Despite the expansion of the cutaneous lesions, the patient’s general good health and the absence of respiratory and systemic involvement allowed us to adopt a conservative treatment regime. Other possible causes of vasculitis were excluded by diagnostic tests. The temporal relationship between the inhalation of sulfuric acid, together with the absence of relapse after discontinuation of the exposition, support the hypothesis that the inhalation of sulfuric acid represented the cause of vasculitis in the present patient.10 We were unable to find any other cases of leukocytoclastic vasculitis resulting from the inhalation of sulfuric acid in the literature. Although rare, the inhalation of sulfuric acid vapor should be considered as another cause of leukocytoclastic vasculitis.
4
5
6
7
8
9
References 1 Crowson AN, Mihm MC Jr, Magro CM. Cutaneous vasculitis: a review. J Cutan Pathol 2003; 30: 161–173. 2 Khasnis A, Langford CA. Update on vasculitis. J Allergy Clin Immunol 2009; 123: 1226–1236. 3 Nepal M, Padma H. Fluticasone-associated cutaneous allergic granulomatous vasculitis: an under-recognized
International Journal of Dermatology 2014, 53, 228–230
10
Pellicer-Oliver et al.
but important cause of drug-induced cutaneous Churg–Strauss syndrome. South Med J 2008; 101: 761–763. Kilgour JD, Foster J, Soames A. Responses in the respiratory tract of rats following exposure to sulfuric acid aerosols for 5 or 28 days. J Appl Toxicol 2002; 22: 387–395. Ricketts S, Kimble FW. Chemical injuries: the Tasmanian Burns Unit experience. ANZ J Surg 2003; 73: 45–48. Kehe K, Thiermann H, Balszuweit F, et al. Acute effects of sulfur mustard injury – Munich experiences. Toxicology 2009; 263: 3–8. Husain MT, Hasanain J, Kumar P. Sulphuric acid burns: report of a mass domestic accident. Burns 1989; 15: 389– 391. Kehe K, Balszuweit F, Steinritz D, et al. Molecular toxicology of sulfur mustard-induced cutaneous inflammation and blistering. Toxicology 2009; 263: 12– 19. Blanco R, Martínez-Taboada VM, Rodríguez-Valverde V, García-Fuentes M. Cutaneous vasculitis in children and adults: associated diseases and etiologic factors in 303 patients. Medicine (Baltimore) 1998; 77: 403–418. Calabrese LH, Michel BA, Bloch DA, et al. The American College of Rheumatology 1990 criteria for the classification of hypersensitivity vasculitis. Arthritis Rheum 1990; 33: 1108–1113.
ª 2013 The International Society of Dermatology
