- Email: [email protected]
Alopecia-associated pseudocyst of the scalp
J AM ACAD DERMATOL
e114 Letters
SEPTEMBER 2012
point of view, it must be differentiated from certain papillomatous lesions secondary to human papilloma virus infection, traumatic fibromas, Abrikossoff’s tumor, exophytic epidermoid carcinomas, pyogenic granuloma, and sialadenoma papilliferum.5 Leticia Sempau, MD,a Luis Miguel Valladares, MD,a Jes us Lomas-Garcıa, MD,b Nieves Alonso-Orcajo, MD,b Jose Marıa Garcıa Ruiz de Morales, MD, Rodrıguez-Prieto, MDa PhD,c and Manuel Angel Departments of Dermatology,a Pathology,b and Immunology,c Complejo Asistencial Universitario de Leon, Altos de Nava SN, 24071 Leon, Spain Funding sources: None. Conflicts of interest: None declared. Fig 1. EBV-induced lingual papule. A pedicled lesion with two small whitish ulcerations at its base on posterolateral surface of the tongue.
Fig 2. Immunohistochemical study of EBV-induced lingual papule for CD20 (original magnification 340); CD30 (3200) and Epstein-Barr virus (immunohistochemistry for LMP-1) (3400) showed intense positivity in activated lymphocytes.
Correspondence to: Leticia Sempau, MD, Department of Dermatology, Complejo Asistencial de Leon, Altos de Nava SN, 24071 Leon, Spain E-mail: [email protected] REFERENCES 1. Mendoza N, Diamantis M, Arora A, Bartlett B, Gewirtzman A, Tremaine AM, et al. Mucocutaneous manifestations of Epstein-Barr virus infection. Am J Clin Dermatol 2008;9: 295-305. 2. Ebell MH. Epstein-Barr virus infectious mononucleosis. Am Fam Physician 2004;70:1279-87. 3. Macsween KF, Crawford DH. Epstein-Barr virus-recent advances. Lancet Infect Dis 2003;3:131-40. 4. Kutok JL, Wang F. Spectrum of Epstein-Barr ViruseAssociated Diseases. Annu Rev Pathol Mech Dis 2006;1:375-404. 5. Wood NK, Goaz PW, editors. Maxillo-differential diagnosis of oral and maxillofacial lesions. Philadelphia: Mosby-Year Book, Inc; 1997. http://dx.doi.org/10.1016/j.jaad.2011.09.020
Alopecia-associated pseudocyst of the scalp EBV is transmitted via the saliva. The target within the mouth is the oral lymphoid tissue B cells. Once the B cells are infected, proliferation takes place in the germinal centers3 with later dissemination. We are unaware of any previous reports of a pedicled tumor at the tongue as the manifestation of infectious mononucleosis, though the pathological changes of EBV in our patient were identical to those described in involvement of the palatine tonsils by this virus.4 This is to be expected, given that the lingual tonsil forms part of Waldeyer’s tonsillar ring, where the EBV enters the organism and starts replicating. Pathologically, the differential diagnosis includes lymphoproliferative processes (high-grade B lymphoma or Hodgkin’s lymphoma). From a clinical
To the Editor: Pseudocyst of the scalp is a rarely described condition that has only been reported in the Japanese and European literature. The author has documented 11 cases observed over the last 10 years (Table I), two of which are detailed below. A 19-year-old man presented for the first time with a 2-week history of a tender and tense slightly erythematous cystic swelling at the vertex (Fig 1). He noted hair loss at the onset of the swelling. A bacterial culture of the slightly viscous yellow fluid aspirated was sterile. The cyst was treated with intralesional triamcinolone acetonide (2.5 mg/mL) and the swelling resolved after 2 days. Early hair growth was noted 5 weeks later without any evidence of scarring.
J AM ACAD DERMATOL
Letters e115
VOLUME 67, NUMBER 3
Table I. Demographics and clinical features of patients Case No. y,z
1 2 3 4y 5x 6x 7x 8 9 10z 11
Age, y
Race/ethnicity
Sex
Site
No/size (cm) of cysts
Onset before visit, wk
No. of occurrences*
24 22 24 18 32 16 22 48 19 40 28
Hispanic White Black White Asian Arabic Arabic White White Black White
M M M M M M F F M M F
Vertex Vertex Vertex Occiput Vertex Vertex Vertex Vertex Vertex Vertex Vertex
1/4 1/3 1/2.5 1/2 1/2.5 2/3 1/2.5 1/2.5 1/2 1/2 1/2
4 1 4 1 4 4 6 8 8 4 12
3 1 2 2 1 4 1 1 1 4 2
F, Female; M, male. *All patients (except patient 11, who received no treatment) were treated only with intralesional triamcinolone acetonide 2.5 mg/mL, except patient 1 who also received courses of tetracycline and cephalexin. Recurrences occurred in same area of scalp. Not all occurrences were treated; some resolved spontaneously. Complete hair regrowth was documented in 8 of 11 patients; 3 patients were unavailable for follow-up. y History of alopecia areata. z Resolved by self-induced rupture on occasion. x Associated with acne.
Fig 1. Alopecia-associated pseudocyst of the scalp.
A 16-year-old boy presented with a 1-month history of hair loss in a localized area of swelling at the vertex. He declined treatment and showed spontaneous improvement with some hair growth noted when he was seen again 1 month later. He returned 3 years later with two adjacent and nearly confluent areas of cystic swelling with overlying hair loss at the vertex. He noted that the hair came out easily after the appearance of the cysts. The cysts cleared after two consecutive monthly treatments with intralesional triamcinolone acetonide (2.5 mg/mL) and there was complete hair regrowth. He had two similar episodes during the next 2 years and each resolved spontaneously within 1 month. He has been free from recurrences for the last 2 years. The original report in the Japanese literature described 19 cases of pseudocysts and associated alopecia.1 Pathology was obtained in 9 cases by
complete excision, and the histology showed cystic cavities without a wall structure located in the middle and papillary dermis surrounded by inflammatory granulation tissue. Subsequently, 29 additional cases were described in the Japanese, French, and European English-language literature.2-5 The 11 cases reported here (Table I) presented with alopecic cysts in mostly young healthy men. The cysts showed surface erythema and a few residual vellus-like hairs, and were either firm or soft and fluctuant. The hair loss was noted as the cyst began to form, and tenderness occurred with increasing cyst size. Patients responded quickly in almost all cases to intralesional triamcinolone acetonide (2.5 mg/mL), patients healed without any evidence of scarring, and hair growth was noted within 2 months of treatment. Most cases presented as isolated events but with a tendency to relapse. Cultures were sterile, confirming previous observations.1-5 It is important to note that once the swelling resolved, the appearance closely resembled alopecia areata. The rapid response to intralesional corticosteroid, and spontaneous resolution after self-induced rupture in some cases, suggest a mild inflammatory process. Future investigations with biopsies performed at the onset of inflammation, and a chemical analysis of the cyst contents, might provide additional clues to the pathogenesis. Because this condition is not well recognized, it may be misdiagnosed as alopecia areata, pilar or acne cysts, or dissecting cellulitis. It is suspected that it is far more common than the literature suggests.
J AM ACAD DERMATOL
e116 Letters
SEPTEMBER 2012
Eric L. Eisenberg, MD, FRCPC Division of Dermatology, Toronto Western Hospital, University Health Network, University of Toronto, Ontario, Canada Funding sources: None. Conflicts of interest: None declared. Correspondence to: Eric L. Eisenberg, MD, FRCPC, 3420 Hurontario St, Suite 206, Mississauga, Ontario, Canada L5B 4A9 E-mail: [email protected] REFERENCES 1. Iwata T, Hashimoto T, Niimura M. A pseudocyst with inflammatory granulation tissue on the scalpepseudocyst of the scalp. Rinsho Derma (Tokyo) 1992;46:9-16. 2. Abdennader S, Vignon-Pennamen MD, Hatchuel J, Reygagne P. Alopecic and aseptic nodules of the scalp ( pseudocyst of the scalp): a prospective clinicopathological study of 15 cases. Dermatology 2011;222:31-5. 3. Tsuruta D, Hayashi A, Kobayashi H, Nakagawa K, Furukawa M, Ishii M. Pseudocyst of the scalp. Dermatology 2005;210:333-5. 4. Abdennader S, Reygagne P. Alopecic and aseptic nodules of the scalp. Dermatology 2009;218:86. 5. Tsuruta D. Reply [letter]. Dermatology 2009;218:87. http://dx.doi.org/10.1016/j.jaad.2011.09.028
Chemotherapy-induced acral erythema sparing the palms To the Editor: Jean Bolognia et al1 have suggested that the variously named cutaneous eruptions resulting from toxic insult caused by high-dose chemotherapy share a new clinical term: ‘‘toxic erythema of chemotherapy.’’ Although proposing a further clinical term rarely provides resolution for the problem of conditions having too many names, ‘‘toxic erythema of chemotherapy’’ is, indeed, useful. In support of this proposal, we present a case where the cause is clearly chemotherapy toxicity but the clinical presentation differs from the classic descriptions of chemotherapy-induced acral erythema. A 63-year-old Russian woman with stage IV adenocarcinoma of the breast, who completed 6 treatments of paclitaxel (140 mg) administered weekly, presented with a pruritic and painful eruption on the back of her hands, beginning after her second paclitaxel treatment 2 months earlier and progressing with subsequent treatments. On physical examination, the skin overlying her metacarpal-phalangeal joints and to a lesser extent her interphalangeal joints had moderately illdefined smooth slightly violaceous and erythematous edematous thin plaques (Fig 1). Her proximal nailfolds were edematous and had telangiectases and ruffled cuticles. This eruption spared her palms and
Fig 1. Toxic erythema of chemotherapy. Right hand after treatment with potent topical steroids for 2 weeks, left hand untreated.
palpebrae. The proximal and distal muscle strength was normal. The distribution and slightly violaceous color of the eruption along with the cuticle involvement supported a diagnosis of dermatomyositis-like drug eruption. Complete sparing of the palms made chemotherapy-induced acral erythema less likely. Punch biopsy specimens revealed moderate compact orthokeratosis, moderate to marked mostly regular epidermal hyperplasia, pallor of all layers of the epidermis, several dyskeratotic keratinocytes, several atypical mitotic figures within the basal layer, diffuse mild basal vacuolization, and a moderate superficial perivascular lymphocytic infiltrate focally obscuring the dermoepidermal junction. These findings were characteristic of chemotherapy-induced acral erythema. Of her recent medications, only paclitaxel frequently associates with chemotherapyinduced acral erythema. Discontinuation of the inciting agent is the preferable treatment for toxic erythema of chemotherapy; palliative treatments include topical corticosteroids and emollients.1,2 Oral pyridoxine,3 cyclo-oxygenase-2 inhibitors,4 and local vasoconstrictive therapy via skin cooling5 have also showed efficacy. In the aforementioned patient, in addition to paclitaxel discontinuation, high-potency topical corticosteroids significantly decreased the duration of this eruption. She applied clobetasol propionate ointment to her right hand for 2 weeks, with improvement compared with her untreated left hand (Fig 1). The eruption markedly improved in 1 month with clobetasol propionate ointment use twice daily on both hands, at which point the patient was lost to follow-up; however, her oncologist has not noted recrudescence. High-potency topical steroids likely offer both symptomatic improvement and decreased time to resolution. Although the clinical presentation initially favored dermatomyositis-like drug eruption, the histologic
e114 Letters
SEPTEMBER 2012
point of view, it must be differentiated from certain papillomatous lesions secondary to human papilloma virus infection, traumatic fibromas, Abrikossoff’s tumor, exophytic epidermoid carcinomas, pyogenic granuloma, and sialadenoma papilliferum.5 Leticia Sempau, MD,a Luis Miguel Valladares, MD,a Jes us Lomas-Garcıa, MD,b Nieves Alonso-Orcajo, MD,b Jose Marıa Garcıa Ruiz de Morales, MD, Rodrıguez-Prieto, MDa PhD,c and Manuel Angel Departments of Dermatology,a Pathology,b and Immunology,c Complejo Asistencial Universitario de Leon, Altos de Nava SN, 24071 Leon, Spain Funding sources: None. Conflicts of interest: None declared. Fig 1. EBV-induced lingual papule. A pedicled lesion with two small whitish ulcerations at its base on posterolateral surface of the tongue.
Fig 2. Immunohistochemical study of EBV-induced lingual papule for CD20 (original magnification 340); CD30 (3200) and Epstein-Barr virus (immunohistochemistry for LMP-1) (3400) showed intense positivity in activated lymphocytes.
Correspondence to: Leticia Sempau, MD, Department of Dermatology, Complejo Asistencial de Leon, Altos de Nava SN, 24071 Leon, Spain E-mail: [email protected] REFERENCES 1. Mendoza N, Diamantis M, Arora A, Bartlett B, Gewirtzman A, Tremaine AM, et al. Mucocutaneous manifestations of Epstein-Barr virus infection. Am J Clin Dermatol 2008;9: 295-305. 2. Ebell MH. Epstein-Barr virus infectious mononucleosis. Am Fam Physician 2004;70:1279-87. 3. Macsween KF, Crawford DH. Epstein-Barr virus-recent advances. Lancet Infect Dis 2003;3:131-40. 4. Kutok JL, Wang F. Spectrum of Epstein-Barr ViruseAssociated Diseases. Annu Rev Pathol Mech Dis 2006;1:375-404. 5. Wood NK, Goaz PW, editors. Maxillo-differential diagnosis of oral and maxillofacial lesions. Philadelphia: Mosby-Year Book, Inc; 1997. http://dx.doi.org/10.1016/j.jaad.2011.09.020
Alopecia-associated pseudocyst of the scalp EBV is transmitted via the saliva. The target within the mouth is the oral lymphoid tissue B cells. Once the B cells are infected, proliferation takes place in the germinal centers3 with later dissemination. We are unaware of any previous reports of a pedicled tumor at the tongue as the manifestation of infectious mononucleosis, though the pathological changes of EBV in our patient were identical to those described in involvement of the palatine tonsils by this virus.4 This is to be expected, given that the lingual tonsil forms part of Waldeyer’s tonsillar ring, where the EBV enters the organism and starts replicating. Pathologically, the differential diagnosis includes lymphoproliferative processes (high-grade B lymphoma or Hodgkin’s lymphoma). From a clinical
To the Editor: Pseudocyst of the scalp is a rarely described condition that has only been reported in the Japanese and European literature. The author has documented 11 cases observed over the last 10 years (Table I), two of which are detailed below. A 19-year-old man presented for the first time with a 2-week history of a tender and tense slightly erythematous cystic swelling at the vertex (Fig 1). He noted hair loss at the onset of the swelling. A bacterial culture of the slightly viscous yellow fluid aspirated was sterile. The cyst was treated with intralesional triamcinolone acetonide (2.5 mg/mL) and the swelling resolved after 2 days. Early hair growth was noted 5 weeks later without any evidence of scarring.
J AM ACAD DERMATOL
Letters e115
VOLUME 67, NUMBER 3
Table I. Demographics and clinical features of patients Case No. y,z
1 2 3 4y 5x 6x 7x 8 9 10z 11
Age, y
Race/ethnicity
Sex
Site
No/size (cm) of cysts
Onset before visit, wk
No. of occurrences*
24 22 24 18 32 16 22 48 19 40 28
Hispanic White Black White Asian Arabic Arabic White White Black White
M M M M M M F F M M F
Vertex Vertex Vertex Occiput Vertex Vertex Vertex Vertex Vertex Vertex Vertex
1/4 1/3 1/2.5 1/2 1/2.5 2/3 1/2.5 1/2.5 1/2 1/2 1/2
4 1 4 1 4 4 6 8 8 4 12
3 1 2 2 1 4 1 1 1 4 2
F, Female; M, male. *All patients (except patient 11, who received no treatment) were treated only with intralesional triamcinolone acetonide 2.5 mg/mL, except patient 1 who also received courses of tetracycline and cephalexin. Recurrences occurred in same area of scalp. Not all occurrences were treated; some resolved spontaneously. Complete hair regrowth was documented in 8 of 11 patients; 3 patients were unavailable for follow-up. y History of alopecia areata. z Resolved by self-induced rupture on occasion. x Associated with acne.
Fig 1. Alopecia-associated pseudocyst of the scalp.
A 16-year-old boy presented with a 1-month history of hair loss in a localized area of swelling at the vertex. He declined treatment and showed spontaneous improvement with some hair growth noted when he was seen again 1 month later. He returned 3 years later with two adjacent and nearly confluent areas of cystic swelling with overlying hair loss at the vertex. He noted that the hair came out easily after the appearance of the cysts. The cysts cleared after two consecutive monthly treatments with intralesional triamcinolone acetonide (2.5 mg/mL) and there was complete hair regrowth. He had two similar episodes during the next 2 years and each resolved spontaneously within 1 month. He has been free from recurrences for the last 2 years. The original report in the Japanese literature described 19 cases of pseudocysts and associated alopecia.1 Pathology was obtained in 9 cases by
complete excision, and the histology showed cystic cavities without a wall structure located in the middle and papillary dermis surrounded by inflammatory granulation tissue. Subsequently, 29 additional cases were described in the Japanese, French, and European English-language literature.2-5 The 11 cases reported here (Table I) presented with alopecic cysts in mostly young healthy men. The cysts showed surface erythema and a few residual vellus-like hairs, and were either firm or soft and fluctuant. The hair loss was noted as the cyst began to form, and tenderness occurred with increasing cyst size. Patients responded quickly in almost all cases to intralesional triamcinolone acetonide (2.5 mg/mL), patients healed without any evidence of scarring, and hair growth was noted within 2 months of treatment. Most cases presented as isolated events but with a tendency to relapse. Cultures were sterile, confirming previous observations.1-5 It is important to note that once the swelling resolved, the appearance closely resembled alopecia areata. The rapid response to intralesional corticosteroid, and spontaneous resolution after self-induced rupture in some cases, suggest a mild inflammatory process. Future investigations with biopsies performed at the onset of inflammation, and a chemical analysis of the cyst contents, might provide additional clues to the pathogenesis. Because this condition is not well recognized, it may be misdiagnosed as alopecia areata, pilar or acne cysts, or dissecting cellulitis. It is suspected that it is far more common than the literature suggests.
J AM ACAD DERMATOL
e116 Letters
SEPTEMBER 2012
Eric L. Eisenberg, MD, FRCPC Division of Dermatology, Toronto Western Hospital, University Health Network, University of Toronto, Ontario, Canada Funding sources: None. Conflicts of interest: None declared. Correspondence to: Eric L. Eisenberg, MD, FRCPC, 3420 Hurontario St, Suite 206, Mississauga, Ontario, Canada L5B 4A9 E-mail: [email protected] REFERENCES 1. Iwata T, Hashimoto T, Niimura M. A pseudocyst with inflammatory granulation tissue on the scalpepseudocyst of the scalp. Rinsho Derma (Tokyo) 1992;46:9-16. 2. Abdennader S, Vignon-Pennamen MD, Hatchuel J, Reygagne P. Alopecic and aseptic nodules of the scalp ( pseudocyst of the scalp): a prospective clinicopathological study of 15 cases. Dermatology 2011;222:31-5. 3. Tsuruta D, Hayashi A, Kobayashi H, Nakagawa K, Furukawa M, Ishii M. Pseudocyst of the scalp. Dermatology 2005;210:333-5. 4. Abdennader S, Reygagne P. Alopecic and aseptic nodules of the scalp. Dermatology 2009;218:86. 5. Tsuruta D. Reply [letter]. Dermatology 2009;218:87. http://dx.doi.org/10.1016/j.jaad.2011.09.028
Chemotherapy-induced acral erythema sparing the palms To the Editor: Jean Bolognia et al1 have suggested that the variously named cutaneous eruptions resulting from toxic insult caused by high-dose chemotherapy share a new clinical term: ‘‘toxic erythema of chemotherapy.’’ Although proposing a further clinical term rarely provides resolution for the problem of conditions having too many names, ‘‘toxic erythema of chemotherapy’’ is, indeed, useful. In support of this proposal, we present a case where the cause is clearly chemotherapy toxicity but the clinical presentation differs from the classic descriptions of chemotherapy-induced acral erythema. A 63-year-old Russian woman with stage IV adenocarcinoma of the breast, who completed 6 treatments of paclitaxel (140 mg) administered weekly, presented with a pruritic and painful eruption on the back of her hands, beginning after her second paclitaxel treatment 2 months earlier and progressing with subsequent treatments. On physical examination, the skin overlying her metacarpal-phalangeal joints and to a lesser extent her interphalangeal joints had moderately illdefined smooth slightly violaceous and erythematous edematous thin plaques (Fig 1). Her proximal nailfolds were edematous and had telangiectases and ruffled cuticles. This eruption spared her palms and
Fig 1. Toxic erythema of chemotherapy. Right hand after treatment with potent topical steroids for 2 weeks, left hand untreated.
palpebrae. The proximal and distal muscle strength was normal. The distribution and slightly violaceous color of the eruption along with the cuticle involvement supported a diagnosis of dermatomyositis-like drug eruption. Complete sparing of the palms made chemotherapy-induced acral erythema less likely. Punch biopsy specimens revealed moderate compact orthokeratosis, moderate to marked mostly regular epidermal hyperplasia, pallor of all layers of the epidermis, several dyskeratotic keratinocytes, several atypical mitotic figures within the basal layer, diffuse mild basal vacuolization, and a moderate superficial perivascular lymphocytic infiltrate focally obscuring the dermoepidermal junction. These findings were characteristic of chemotherapy-induced acral erythema. Of her recent medications, only paclitaxel frequently associates with chemotherapyinduced acral erythema. Discontinuation of the inciting agent is the preferable treatment for toxic erythema of chemotherapy; palliative treatments include topical corticosteroids and emollients.1,2 Oral pyridoxine,3 cyclo-oxygenase-2 inhibitors,4 and local vasoconstrictive therapy via skin cooling5 have also showed efficacy. In the aforementioned patient, in addition to paclitaxel discontinuation, high-potency topical corticosteroids significantly decreased the duration of this eruption. She applied clobetasol propionate ointment to her right hand for 2 weeks, with improvement compared with her untreated left hand (Fig 1). The eruption markedly improved in 1 month with clobetasol propionate ointment use twice daily on both hands, at which point the patient was lost to follow-up; however, her oncologist has not noted recrudescence. High-potency topical steroids likely offer both symptomatic improvement and decreased time to resolution. Although the clinical presentation initially favored dermatomyositis-like drug eruption, the histologic