- Email: [email protected]
Neutrophilic eccrine hidradenitis masquerading as facial cellulitis
Case reports 693
J AM ACAD DERMATOL VOLUME 56, NUMBER 4
tissues.5,7 Sweet’s-like syndrome in APML patients receiving ATRA therapy may represent a marker of the effectiveness of ATRA to induce promyelocyte differentiation.
4.
5. REFERENCES 1. Cohen PR. Subcutaneous Sweet’s syndrome: a variant of acute febrile neutrophilic dermatosis that is included in the histopathologic differential diagnosis of neutrophilic panniculitis. J Am Acad Dermatol 2005;52:927-8. 2. Levi I, Raanani P, Shalmon B, Schiby-Brilliant R, Ben-Bassat I. Acute neutrophilic dermatosis induced by all-trans-retinoic acid treatment for acute promyelocytic leukemia. Leuk Lymphoma 1999;34:401-4. 3. Requena L, Kutzner H, Palmedo G, Pascual M, Ferna´ndezHerrera J, Fraga J, et al. Histiocytoid Sweet syndrome: a dermal
6.
7.
infiltration of immature neutrophilic granulocytes. Arch Dermatol 2005;141:834-42. Ueno R, Takeuchi J, Shimizu T, Kumagai T, Sawada U, Horie T. Development of Sweet’s syndrome during all-trans retinoic acid therapy for acute promyelocytic leukemia. Rinsho Ketsueki 2000;41:718-22. Astudillo L, Loche F, Reynish W, Rigal-Huguet F, Lamant L, Pris J. Sweet’s syndrome associated with retinoic acid syndrome in a patient with promyelocytic leukemia. Ann Hematol 2002;81: 111-4. Paydas S, Yavuz S, Disel U, Sahin B, Canbolat T, Tuncer I. Vasculitis associated with all trans retinoic acid (ATRA) in a case with acute promyelocytic leukemia. Leuk Lymphoma 2003; 44:547-8. Piette WW, Trapp JF, O’Donnell MJ, Argenyi Z, Talbot EA, Burns CP. Acute neutrophilic dermatosis with myeloblastic infiltrate in a leukemia patient receiving all-trans-retinoic acid therapy. J Am Acad Dermatol 1994;30:293-7.
Neutrophilic eccrine hidradenitis masquerading as facial cellulitis Monika Srivastava, MD,a Susan Scharf, MD,b Shane A. Meehan, MD,a and David Polsky, MD, PhDa New York, New York Neutrophilic eccrine hidradenitis typically manifests as erythematous plaques on the face, trunk, or extremities. This eruption has been associated with numerous factors, but most commonly is seen with chemotherapy, particularly cytarabine. We report a 73-year-old woman with acute myelogenous leukemia who developed rapidly expansive neutrophilic eccrine hidradenitis mimicking facial cellulitis only after a course of cytarabine was followed by granulocyte-colony stimulating factor. Prompt diagnosis is imperative to prevent prolonged antimicrobial therapy. ( J Am Acad Dermatol 2007;56:693-6.)
A
73-year-old white woman with acute myelogenous leukemia received induction chemotherapy with idarubicin and cytarabine. On day 6 of therapy, the patient developed a neutropenic fever (1038F; absolute neutrophil count, 100/mm3). Antimicrobial therapy including vancomycin, cefipime, acyclovir, and caspofungin was initiated. On day 10 of induction therapy, the patient
From the Ronald O. Perelman Department of Dermatologya and the Department of Internal Medicine,b New York University School of Medicine. Funding sources: None. Conflicts of interest: None identified. Reprint requests: David Polsky, MD, PhD, Ronald O. Perelman Department of Dermatology, New York University School of Medicine, 560 1st Avenue, H-100, New York, NY 10016. E-mail: [email protected]. Published online November 17, 2006. 0190-9622/$32.00 ª 2007 by the American Academy of Dermatology, Inc. doi:10.1016/j.jaad.2006.07.032
developed an erythematous indurated tender plaque over her right temple (Fig 1, A). On day 12, granulocyte-colony stimulating factor (G-CSF) was initiated to treat the patient’s ongoing neutropenia and subsequently (days 14-17) the erythematous plaque on the right temple spread rapidly to include the right and left periorbital areas, the nose, and the left temple (Fig 1, B). The ophthalmology and infectious diseases services were consulted on day 15 and diagnosed this rapidly expansive facial eruption as a cellulitis. Antimicrobial therapy was broadened to include linezolid and imipenem; however, the patient remained febrile and the facial eruption continued to rapidly expand for the next 2 days. Computed tomography and/or magnetic resonance imaging was recommended to rule out an orbital abscess. Dermatology was also consulted and skin punch biopsies were completed for histopathology and microbiology. Cultures failed to reveal bacterial or fungal pathogens. Histopathology demonstrated a perieccrine neutrophilic infiltrate with invasion
694 Case reports
J AM ACAD DERMATOL APRIL 2007
Fig 2. Photomicrographs of the biopsy specimen from the right temple. A, Neutrophilic infiltrate around eccrine glands. B, High-power view demonstrating neutrophils invading the walls of eccrine glands. (Hematoxylineeosin stain; original magnifications: A, 3100; B, 3400.) Fig 1. A, Tender erythematous plaque on right temple at day 10 of induction therapy with idarubicin and cytarabine. B, Rapid expansion of the erythematous tender plaque, here shown on day 17—five days after receiving granulocyte stimulating factor.
of eccrine gland walls with neutrophils. There was also necrosis of the eccrine coils. A significant dermal or interstitial neutrophilic infiltrate—suggesting Sweet’s syndrome, cellulitis, or other neutrophilic dermatoses—was not seen. These changes were consistent with a diagnosis of neutrophilic eccrine hidradenitis (NEH; Fig 2). Between days 17 and 20, the patient’s neutropenia reversed (absolute neutrophil count, 2800/mm3) and her fever defervesced. By day 20, when her bone marrow had fully reconstituted, the NEH had nearly completely resolved, leaving only a faintly erythematous patch.
Table I. Etiologic factors of neutrophilic eccrine hidradenitis Chemotherapeutic agents Cytarabine1 Anthracyclines20 Mitoxantrone21 Cyclophosphamide6 Bleomycin22 Imatinib mesylate3 Methotrexate9 Topotecan8 Non-chemotherapeutic agents Granulocyte colony stimulating factor18 Acetaminophen19 Anti-retroviral agents14
Malignancies Acute myelogenous leukemia11,16 Chronic myelogenous leukemia7 Infectious agents HIV12,14-15 Staphylococcus17 Enterobacter23 Serratia13 Nocardia4 Streptococcus2 Other Behc¸et’s disease5,10
DISCUSSION NEH is a benign, self-limiting dermatosis, characterized by the sudden onset of usually asymptomatic, asymmetric erythematous papules and plaques over the trunk, limbs, or face. It was first described by Harrist et al1 in 1982, and since then there have been more than 50 cases reported in the literature. It is typically seen in patients with acute myelogenous
leukemia following chemotherapy with cytarabine, but has also been associated with other chemotherapeutic and non-chemotherapeutic agents. There have also been reports of NEH related to infectious organisms (Table I).1-23 Occasionally, NEH is a marker for the onset of hematologic malignancies.7,11,16
J AM ACAD DERMATOL
Case reports 695
VOLUME 56, NUMBER 4
NEH occurs rarely. One study reported a 2.6% incidence of all neutrophilic dermatoses in granulocytopenic patients.24 NEH is usually seen 8 to 15 days after the start of chemotherapy (mean, 9.7 days) and lasts approximately 6 to 33 days before resolution without sequellae.25-27 Most patients who develop NEH in the setting of chemotherapy are febrile and neutropenic at the time clinical lesions are observed, hence the concern for cellulitis. Neutropenia, however, does not appear to be a necessary or sufficient etiologic factor for the development of NEH, because patients other than those neutropenic from chemoinduction therapy have been afflicted with this neutrophilic dermatosis.1-23 Patients who develop NEH during chemoinduction therapy do not always experience recurrence with consolidation therapy. One report cites a 60% recurrence rate.28 Dapsone 100 mg daily for 2 weeks has been shown to prevent recurrence in a case of NEH secondary to lomustine therapy for Hodgkin’s disease.28 Entities other then recurrent NEH which need to be considered in these patients, include Sweet’s syndrome, leukemia cutis, and cellulitis. Therefore, biopsies for histopathology and microbiology should be completed in a timely manner. The pathogenesis of NEH is unclear. Histopathologically, NEH is characterized by a neutrophilic infiltrate around eccrine glands and coils with necrosis. In patients who have received chemotherapy, it is thought that the cytotoxicity of the drugs secreted in sweat glands leads to necrosis, which secondarily attracts neutrophils.29 However, other pathogenetic mechanisms are likely, because NEH is associated with etiologic factors other than chemotherapeutic agents. Our patient’s clinical picture is quite unusual. NEH mimicking facial and orbital cellulitis has been described in 2 other patients who received chemoinduction with cytarabine for acute myelogenous leukemia30,31; however, to our knowledge there have been no cases in which a small stable lesion of NEH that arose during cytarabine chemoinduction therapy dramatically expanded, resembling infectious cellulitis, only after the initiation of G-CSF therapy 4 days later. Interestingly, there has been a case report implicating G-CSF in the development of NEH.18 G-CSF is often administered to neutropenic patients in attempts to hasten bone marrow reconstitution and reverse neutropenia. It enhances chemotaxis and stimulates the proliferation of neutrophils. In the case reported by Bachmeyer et al18 the patient developed NEH after chemoinduction with busulphan, when he was receiving G-CSF. In our patient, the initial plaque
of NEH developed during chemoinduction with cytarabine and expanded only after starting G-CSF. We therefore propose the following mechanism of disease progression in our patient. It is possible that in our patient, cytarabine chemotherapy initiated the cytotoxic events that led to the development of NEH on the right temple on day 10. Then, G-CSF, introduced on day 12, led to neutrophil proliferation. This increase in neutrophil activity and chemotaxis led to rapid expansion on day 14 of the previously stable NEH plaque. As the patient’s bone marrow became reconstituted over the next several days, the NEH resolved. This is not surprising, because resolution of NEH is usually seen with reversal of neutropenia in cases related to chemotherapy. In conclusion, NEH should be considered in the differential diagnosis of an erythematous tender or nontender plaque which begins to rapidly expand even a few days after initial development, especially in patients receiving chemoinduction therapy with cytarabine. It is possible that G-CSF therapy may exacerbate otherwise stable disease secondary to neutrophil proliferation and chemotaxis. Although infection should be ruled out with appropriate tissue cultures, a prompt diagnosis of NEH should be made with histopathology to prevent unnecessary broad spectrum antimicrobial therapy and imaging studies.
REFERENCES 1. Harrist TJ, Fine JD, Berman RS, Murphy GF, Mihm MC Jr. Neutrophilic eccrine hidradenitis. A distinctive type of neutrophilic dermatosis associated with myelogenous leukemia and chemotherapy. Arch Dermatol 1982;118:263-6. 2. Takai T, Matsunaga A. A case of neutrophilic eccrine hidradenitis associated with streptococcal infectious endocarditis. Dermatology 2006;212:203-5. 3. Dib EG, Ifthikharuddin JJ, Scott GA, Partilo SR. Neutrophilic eccrine hidradenitis induced by imatinib mesylate (Gleevec) therapy. Leuk Res 2005;29:233-4. 4. Antonovich DD, Berke A, Grant-Kels JM, Fung M. Infectious eccrine hidradenitis caused by Nocardia. J Am Acad Dermatol 2004;50:315-8. 5. Mercader-Garcia P, Vilata-Corell JJ, Pardo-Sanchez J, ForteaBaixauli JM. Neutrophilic eccrine hidradenitis in a patient with Behc¸et’s disease. Acta Derm Venereol 2003;83:395-6. 6. Lienesch DW, Mutasim DF, Singh RR. Neutrophilic eccrine hidradenitis mimicking cutaneous vasculitis in a lupus patient: a complication of cyclophosphamide. Lupus 2003; 12:707-9. 7. Gomez Vazquez M, Peteiro C, Toribio J. Neutrophilic eccrine hidradenitis heralding the onset of chronic myelogenous leukaemia. J Eur Acad Dermatol Venereol 2003;17:328-30. 8. Marini M, Wright D, Ropolo M, Abbruzzese M, Casas G. Neutrophilic eccrine hidradenitis secondary to topotecan. J Dermatolog Treat 2002;13:35-7. 9. Tojo M, Iwatsuki K, Furukawa H, Takahashi M, Kaneko F. Neutrophilic eccrine hidradenitis in actinic reticuloid syndrome. Eur J Dermatol 2002;12:198-200.
696 Case reports
J AM ACAD DERMATOL APRIL 2007
10. Bilic M, Mutasim DF. Neutrophilic eccrine hidradenitis in a patient with Behc¸et’s disease. Cutis 2001;68:107-11. 11. Roustan G, Salas C, Cabrera R, Simon A. Neutrophilic eccrine hidradenitis unassociated with chemotherapy in a patient with acute myelogenous leukemia. Int J Dermatol 2001;40:144-7. 12. Bachmeyer C, Reygagne P, Aractingi S. Recurrent neutrophilic eccrine hidradenitis in an HIV-1einfected patient. Dermatology 2000;200:328-30. 13. Combemale P, Faisant M, Azoulay-Petit C, Dupin M, Kanitakis J. Neutrophilic eccrine hidradenitis secondary to infection with Serratia marcescens. Br J Dermatol 2000;142:784-8. 14. Krischer J, Rutschmann O, Roten SV, Harms M, Saurat JH, Pechere M. Neutrophil eccrine hidradenitis in a patient with AIDS. J Dermatol 1998;25:199-200. 15. Sevila A, Morell A, Banuls J, Silvestre JF, Betlloch I. Neutrophilic eccrine hidradenitis in an HIV-infected patient. Int J Dermatol 1996;35:651-2. 16. Pierson JC, Helm TN, Taylor JS, Elston DM, Tuthill RJ. Neutrophilic eccrine hidradenitis heralding the onset of acute myelogenous leukemia. Arch Dermatol 1993;129:791-2. 17. Shih IH, Huang YH, Yang CH, Yang LC, Hong HS. Childhood neutrophilic eccrine hidradenitis: a clinicopathologic and immunohistochemical study of 10 patients. J Am Acad Dermatol 2005;52:963-6. 18. Bachmeyer C, Chaibi P, Aractingi S. Neutrophilic eccrine hidradenitis induced by granulocyte colony-stimulating factor. Br J Dermatol 1998;139:354-5. 19. Kuttner BJ, Kurban RS. Neutrophilic eccrine hidradenitis in the absence of an underlying malignancy. Cutis 1988;41:403-5. 20. Keane FM, Munn SE, Buckley DA, Hopster D, Mufti GJ, du Vivier AW. Neutrophilic eccrine hidradenitis in two neutropaenic patients. Clin Exp Dermatol 2001;26:162-5.
21. Burg G, Bieber T, Langecker P. Localized neutrophilic eccrine hydradenitis in mitoxantrone therapy: a typical side-effect of cytostatic drugs [in German]. Hautarzt 1988;39:233-6. 22. Scallan PG, Kettler AH, Levy ML, Tschen JA. Neutrophilic eccrine hidradenitis. Evidence implicating bleomycin as a causative agent. Cancer 1988;62:2532-6. 23. Allegue F, Rocamora A, Martin-Gonzalez M, Alonso ML, Ledo A. Infectious eccrine hidradenitis. J Am Acad Dermatol 1990;22(6 pt 1):1119-20. 24. Aractingi S, Mallet V, Pinquier L, Chosidow O, Vignon-Pennamen MD, Degos L, et al. Neutrophilic dermatoses during granulocytopenia. Arch Dermatol 1995;131:1141-5. 25. Wong GC, Lee LH, Chong YY. A case report of neutrophilic eccrine hidradenitis in a patient receiving chemotherapy for acute myeloid leukaemia. Ann Acad Med Singapore 1998;27: 860-3. 26. Cho KH, Han KH, Kim SW, Youn SW, Youn JI, Kim BK. Neutrophilic dermatoses associated with myeloid malignancy. Clin Exp Dermatol 1997;22:269-73. 27. Chng WJ, Thamboo TP. Unusual presentation of neutrophilic eccrine hidradenitis. Br J Dermatol 2004;151:507-8. 28. Shear NH, Knowles SR, Shapiro L, Poldre P. Dapsone in prevention of recurrent neutrophilic eccrine hidradenitis. J Am Acad Dermatol 1996;35(5 Pt 2):819-22. 29. Brehler R, Reimann S, Bonsmann G, Metze D. Neutrophilic hidradenitis induced by chemotherapy involves eccrine and apocrine glands. Am J Dermatopathol 1997;19:73-8. 30. Bardenstein DS, Haluschak J, Gerson S, Zaim MT. Neutrophilic eccrine hidradenitis simulating orbital cellulitis. Arch Ophthalmol 1994;112:1460-3. 31. Crawford GH, Chu AY, Halpern M, James WD. Erythematous facial plaques in a patient with leukemia. Neutrophilic eccrine hidradenitis. Arch Dermatol 2003;139:531-6.
Cavitary pyoderma gangrenosum treated with local infusion of corticosteroid Ben Tallon, MBCHB,a Marius Rademaker, DM,a Grant Parkinson, FRACS,b Brian Whitley, FRACDS,c and Michael J. Swarbrick, FRCRd Hamilton, New Zealand Background: Pyoderma gangrenosum (PG) is a rare inflammatory skin condition, characterized by progressive and recurrent skin ulceration, often representing a pathergy response. Observations: We report a case of spontaneous retrosternal PG that developed on the background of previous PG of the skin of the back. The patient had underlying ulcerative colitis. After failure of prednisone, minocycline, and dapsone, a novel method was instituted. Local flushing with dexamethasone, by a radiologically inserted retrosternal catheter with precordial suction, induced a rapid and sustained
From the Departments of Dermatology,a Cardiac Surgery,b Oral and Maxillo-Facial Surgery,c and Radiology,d Waikato Hospital. Funding sources: None. Conflicts of interest: None identified. Reprint requests: Ben Tallon, MBCHB, Department of Dermatology, Waikato Hospital, Hamilton, New Zealand. E-mail: bentallon@ gmail.com.
Published online December 12, 2006. 0190-9622/$32.00 ª 2007 by the American Academy of Dermatology, Inc. doi:10.1016/j.jaad.2006.10.043
J AM ACAD DERMATOL VOLUME 56, NUMBER 4
tissues.5,7 Sweet’s-like syndrome in APML patients receiving ATRA therapy may represent a marker of the effectiveness of ATRA to induce promyelocyte differentiation.
4.
5. REFERENCES 1. Cohen PR. Subcutaneous Sweet’s syndrome: a variant of acute febrile neutrophilic dermatosis that is included in the histopathologic differential diagnosis of neutrophilic panniculitis. J Am Acad Dermatol 2005;52:927-8. 2. Levi I, Raanani P, Shalmon B, Schiby-Brilliant R, Ben-Bassat I. Acute neutrophilic dermatosis induced by all-trans-retinoic acid treatment for acute promyelocytic leukemia. Leuk Lymphoma 1999;34:401-4. 3. Requena L, Kutzner H, Palmedo G, Pascual M, Ferna´ndezHerrera J, Fraga J, et al. Histiocytoid Sweet syndrome: a dermal
6.
7.
infiltration of immature neutrophilic granulocytes. Arch Dermatol 2005;141:834-42. Ueno R, Takeuchi J, Shimizu T, Kumagai T, Sawada U, Horie T. Development of Sweet’s syndrome during all-trans retinoic acid therapy for acute promyelocytic leukemia. Rinsho Ketsueki 2000;41:718-22. Astudillo L, Loche F, Reynish W, Rigal-Huguet F, Lamant L, Pris J. Sweet’s syndrome associated with retinoic acid syndrome in a patient with promyelocytic leukemia. Ann Hematol 2002;81: 111-4. Paydas S, Yavuz S, Disel U, Sahin B, Canbolat T, Tuncer I. Vasculitis associated with all trans retinoic acid (ATRA) in a case with acute promyelocytic leukemia. Leuk Lymphoma 2003; 44:547-8. Piette WW, Trapp JF, O’Donnell MJ, Argenyi Z, Talbot EA, Burns CP. Acute neutrophilic dermatosis with myeloblastic infiltrate in a leukemia patient receiving all-trans-retinoic acid therapy. J Am Acad Dermatol 1994;30:293-7.
Neutrophilic eccrine hidradenitis masquerading as facial cellulitis Monika Srivastava, MD,a Susan Scharf, MD,b Shane A. Meehan, MD,a and David Polsky, MD, PhDa New York, New York Neutrophilic eccrine hidradenitis typically manifests as erythematous plaques on the face, trunk, or extremities. This eruption has been associated with numerous factors, but most commonly is seen with chemotherapy, particularly cytarabine. We report a 73-year-old woman with acute myelogenous leukemia who developed rapidly expansive neutrophilic eccrine hidradenitis mimicking facial cellulitis only after a course of cytarabine was followed by granulocyte-colony stimulating factor. Prompt diagnosis is imperative to prevent prolonged antimicrobial therapy. ( J Am Acad Dermatol 2007;56:693-6.)
A
73-year-old white woman with acute myelogenous leukemia received induction chemotherapy with idarubicin and cytarabine. On day 6 of therapy, the patient developed a neutropenic fever (1038F; absolute neutrophil count, 100/mm3). Antimicrobial therapy including vancomycin, cefipime, acyclovir, and caspofungin was initiated. On day 10 of induction therapy, the patient
From the Ronald O. Perelman Department of Dermatologya and the Department of Internal Medicine,b New York University School of Medicine. Funding sources: None. Conflicts of interest: None identified. Reprint requests: David Polsky, MD, PhD, Ronald O. Perelman Department of Dermatology, New York University School of Medicine, 560 1st Avenue, H-100, New York, NY 10016. E-mail: [email protected]. Published online November 17, 2006. 0190-9622/$32.00 ª 2007 by the American Academy of Dermatology, Inc. doi:10.1016/j.jaad.2006.07.032
developed an erythematous indurated tender plaque over her right temple (Fig 1, A). On day 12, granulocyte-colony stimulating factor (G-CSF) was initiated to treat the patient’s ongoing neutropenia and subsequently (days 14-17) the erythematous plaque on the right temple spread rapidly to include the right and left periorbital areas, the nose, and the left temple (Fig 1, B). The ophthalmology and infectious diseases services were consulted on day 15 and diagnosed this rapidly expansive facial eruption as a cellulitis. Antimicrobial therapy was broadened to include linezolid and imipenem; however, the patient remained febrile and the facial eruption continued to rapidly expand for the next 2 days. Computed tomography and/or magnetic resonance imaging was recommended to rule out an orbital abscess. Dermatology was also consulted and skin punch biopsies were completed for histopathology and microbiology. Cultures failed to reveal bacterial or fungal pathogens. Histopathology demonstrated a perieccrine neutrophilic infiltrate with invasion
694 Case reports
J AM ACAD DERMATOL APRIL 2007
Fig 2. Photomicrographs of the biopsy specimen from the right temple. A, Neutrophilic infiltrate around eccrine glands. B, High-power view demonstrating neutrophils invading the walls of eccrine glands. (Hematoxylineeosin stain; original magnifications: A, 3100; B, 3400.) Fig 1. A, Tender erythematous plaque on right temple at day 10 of induction therapy with idarubicin and cytarabine. B, Rapid expansion of the erythematous tender plaque, here shown on day 17—five days after receiving granulocyte stimulating factor.
of eccrine gland walls with neutrophils. There was also necrosis of the eccrine coils. A significant dermal or interstitial neutrophilic infiltrate—suggesting Sweet’s syndrome, cellulitis, or other neutrophilic dermatoses—was not seen. These changes were consistent with a diagnosis of neutrophilic eccrine hidradenitis (NEH; Fig 2). Between days 17 and 20, the patient’s neutropenia reversed (absolute neutrophil count, 2800/mm3) and her fever defervesced. By day 20, when her bone marrow had fully reconstituted, the NEH had nearly completely resolved, leaving only a faintly erythematous patch.
Table I. Etiologic factors of neutrophilic eccrine hidradenitis Chemotherapeutic agents Cytarabine1 Anthracyclines20 Mitoxantrone21 Cyclophosphamide6 Bleomycin22 Imatinib mesylate3 Methotrexate9 Topotecan8 Non-chemotherapeutic agents Granulocyte colony stimulating factor18 Acetaminophen19 Anti-retroviral agents14
Malignancies Acute myelogenous leukemia11,16 Chronic myelogenous leukemia7 Infectious agents HIV12,14-15 Staphylococcus17 Enterobacter23 Serratia13 Nocardia4 Streptococcus2 Other Behc¸et’s disease5,10
DISCUSSION NEH is a benign, self-limiting dermatosis, characterized by the sudden onset of usually asymptomatic, asymmetric erythematous papules and plaques over the trunk, limbs, or face. It was first described by Harrist et al1 in 1982, and since then there have been more than 50 cases reported in the literature. It is typically seen in patients with acute myelogenous
leukemia following chemotherapy with cytarabine, but has also been associated with other chemotherapeutic and non-chemotherapeutic agents. There have also been reports of NEH related to infectious organisms (Table I).1-23 Occasionally, NEH is a marker for the onset of hematologic malignancies.7,11,16
J AM ACAD DERMATOL
Case reports 695
VOLUME 56, NUMBER 4
NEH occurs rarely. One study reported a 2.6% incidence of all neutrophilic dermatoses in granulocytopenic patients.24 NEH is usually seen 8 to 15 days after the start of chemotherapy (mean, 9.7 days) and lasts approximately 6 to 33 days before resolution without sequellae.25-27 Most patients who develop NEH in the setting of chemotherapy are febrile and neutropenic at the time clinical lesions are observed, hence the concern for cellulitis. Neutropenia, however, does not appear to be a necessary or sufficient etiologic factor for the development of NEH, because patients other than those neutropenic from chemoinduction therapy have been afflicted with this neutrophilic dermatosis.1-23 Patients who develop NEH during chemoinduction therapy do not always experience recurrence with consolidation therapy. One report cites a 60% recurrence rate.28 Dapsone 100 mg daily for 2 weeks has been shown to prevent recurrence in a case of NEH secondary to lomustine therapy for Hodgkin’s disease.28 Entities other then recurrent NEH which need to be considered in these patients, include Sweet’s syndrome, leukemia cutis, and cellulitis. Therefore, biopsies for histopathology and microbiology should be completed in a timely manner. The pathogenesis of NEH is unclear. Histopathologically, NEH is characterized by a neutrophilic infiltrate around eccrine glands and coils with necrosis. In patients who have received chemotherapy, it is thought that the cytotoxicity of the drugs secreted in sweat glands leads to necrosis, which secondarily attracts neutrophils.29 However, other pathogenetic mechanisms are likely, because NEH is associated with etiologic factors other than chemotherapeutic agents. Our patient’s clinical picture is quite unusual. NEH mimicking facial and orbital cellulitis has been described in 2 other patients who received chemoinduction with cytarabine for acute myelogenous leukemia30,31; however, to our knowledge there have been no cases in which a small stable lesion of NEH that arose during cytarabine chemoinduction therapy dramatically expanded, resembling infectious cellulitis, only after the initiation of G-CSF therapy 4 days later. Interestingly, there has been a case report implicating G-CSF in the development of NEH.18 G-CSF is often administered to neutropenic patients in attempts to hasten bone marrow reconstitution and reverse neutropenia. It enhances chemotaxis and stimulates the proliferation of neutrophils. In the case reported by Bachmeyer et al18 the patient developed NEH after chemoinduction with busulphan, when he was receiving G-CSF. In our patient, the initial plaque
of NEH developed during chemoinduction with cytarabine and expanded only after starting G-CSF. We therefore propose the following mechanism of disease progression in our patient. It is possible that in our patient, cytarabine chemotherapy initiated the cytotoxic events that led to the development of NEH on the right temple on day 10. Then, G-CSF, introduced on day 12, led to neutrophil proliferation. This increase in neutrophil activity and chemotaxis led to rapid expansion on day 14 of the previously stable NEH plaque. As the patient’s bone marrow became reconstituted over the next several days, the NEH resolved. This is not surprising, because resolution of NEH is usually seen with reversal of neutropenia in cases related to chemotherapy. In conclusion, NEH should be considered in the differential diagnosis of an erythematous tender or nontender plaque which begins to rapidly expand even a few days after initial development, especially in patients receiving chemoinduction therapy with cytarabine. It is possible that G-CSF therapy may exacerbate otherwise stable disease secondary to neutrophil proliferation and chemotaxis. Although infection should be ruled out with appropriate tissue cultures, a prompt diagnosis of NEH should be made with histopathology to prevent unnecessary broad spectrum antimicrobial therapy and imaging studies.
REFERENCES 1. Harrist TJ, Fine JD, Berman RS, Murphy GF, Mihm MC Jr. Neutrophilic eccrine hidradenitis. A distinctive type of neutrophilic dermatosis associated with myelogenous leukemia and chemotherapy. Arch Dermatol 1982;118:263-6. 2. Takai T, Matsunaga A. A case of neutrophilic eccrine hidradenitis associated with streptococcal infectious endocarditis. Dermatology 2006;212:203-5. 3. Dib EG, Ifthikharuddin JJ, Scott GA, Partilo SR. Neutrophilic eccrine hidradenitis induced by imatinib mesylate (Gleevec) therapy. Leuk Res 2005;29:233-4. 4. Antonovich DD, Berke A, Grant-Kels JM, Fung M. Infectious eccrine hidradenitis caused by Nocardia. J Am Acad Dermatol 2004;50:315-8. 5. Mercader-Garcia P, Vilata-Corell JJ, Pardo-Sanchez J, ForteaBaixauli JM. Neutrophilic eccrine hidradenitis in a patient with Behc¸et’s disease. Acta Derm Venereol 2003;83:395-6. 6. Lienesch DW, Mutasim DF, Singh RR. Neutrophilic eccrine hidradenitis mimicking cutaneous vasculitis in a lupus patient: a complication of cyclophosphamide. Lupus 2003; 12:707-9. 7. Gomez Vazquez M, Peteiro C, Toribio J. Neutrophilic eccrine hidradenitis heralding the onset of chronic myelogenous leukaemia. J Eur Acad Dermatol Venereol 2003;17:328-30. 8. Marini M, Wright D, Ropolo M, Abbruzzese M, Casas G. Neutrophilic eccrine hidradenitis secondary to topotecan. J Dermatolog Treat 2002;13:35-7. 9. Tojo M, Iwatsuki K, Furukawa H, Takahashi M, Kaneko F. Neutrophilic eccrine hidradenitis in actinic reticuloid syndrome. Eur J Dermatol 2002;12:198-200.
696 Case reports
J AM ACAD DERMATOL APRIL 2007
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Cavitary pyoderma gangrenosum treated with local infusion of corticosteroid Ben Tallon, MBCHB,a Marius Rademaker, DM,a Grant Parkinson, FRACS,b Brian Whitley, FRACDS,c and Michael J. Swarbrick, FRCRd Hamilton, New Zealand Background: Pyoderma gangrenosum (PG) is a rare inflammatory skin condition, characterized by progressive and recurrent skin ulceration, often representing a pathergy response. Observations: We report a case of spontaneous retrosternal PG that developed on the background of previous PG of the skin of the back. The patient had underlying ulcerative colitis. After failure of prednisone, minocycline, and dapsone, a novel method was instituted. Local flushing with dexamethasone, by a radiologically inserted retrosternal catheter with precordial suction, induced a rapid and sustained
From the Departments of Dermatology,a Cardiac Surgery,b Oral and Maxillo-Facial Surgery,c and Radiology,d Waikato Hospital. Funding sources: None. Conflicts of interest: None identified. Reprint requests: Ben Tallon, MBCHB, Department of Dermatology, Waikato Hospital, Hamilton, New Zealand. E-mail: bentallon@ gmail.com.
Published online December 12, 2006. 0190-9622/$32.00 ª 2007 by the American Academy of Dermatology, Inc. doi:10.1016/j.jaad.2006.10.043