Letters to the Editor

899

endothelial cells characterized by an epithelioid or ‘hobnail’ appearance line ectatic vascular lumina. In the deeper dermis, collagen dissecting, rather narrow neoplastic vessels are present.4,5 The histogenetic origin is still debated. There is evidence for a vascular origin, but also for a lymphatic involvement. Clinical differential diagnosis includes melanocytic naevus, dermatofibroma, Kaposi’s sarcoma, haemangioma and insect bite reaction, depending on the time of evaluation. Dermatoscopic examination can be of help to differentiate between these diagnoses. Especially if symptomatic, lesions can be treated by surgical excision. This case is unusual in its presentation because of the young age at presentation and the cyclical changes at this age. Furthermore, the skin lesion is large and there were two lesions, whereas it generally presents as a solitary lesion. Figure 1 An indurated plaque on the skin overlying the second metacarpal bone of the right hand, on the thumb, a comparable smaller lesion.

Funding sources None. L. Timmer-de Mik,1,* F.C.P. Moll2 1

2

Isala Dermatologic Center, Department of Pathology, Isala Klinieken, Zwolle, The Netherlands *Correspondence: L. Timmer-de Mik. E-mail: [email protected]

References

Figure 2 Prominent, dilated, thin-walled vessels in the papillary dermis with prominent endothelial cells, with a ‘hobnail’ appearance. (Haematoxylin–eosin stain; original magnification: 920.)

1 Santa Cruz DJ, Aronberg J. Targetoid hemosiderotic hemangioma. J Am Acad Dermatol 1988; 19: 550–558. 2 Christenson IJ, Stone MS. Trauma-induced simulator of targetoid hemosiderotic hemangioma. Am J Dermatopathol 2001; 23: 221–223. 3 Carlson JA, Daulat S, Goodheart HP. Targetoid hemosiderotic hemangioma – a dynamic vascular tumor: report of 3 cases with episodic and cyclic changes and comparison with solitary angiokeratomas. J Am Acad Dermatol 1999; 41: 215–224. 4 Mentzel T, Partanen TA, Kutzner H. Hobnail hemangioma (targetoid hemosiderotic hemangioma) clinicopathologic and immunohistochemical analysis of 62 cases. J Cut Pathol 1999; 26: 279–286. 5 Guillou L, Calonje E, Speight P, Rosai J, Fletcher CD. Hobnail hemangioma: a pseudomalignant vascular lesion with a reappraisal of targetoid hemosiderotic hemangioma. Am J Surg Pathol 1999; 23: 97–105. DOI: 10.1111/jdv.13062

Prevalence of a targetoid haemosiderotic haemangioma is low. Male-to-female ratio is 1.4:1. Patient age at presentation varies from 5–72 years, occurring mostly in young and middle-aged persons. Several theories state that trauma plays a role in the development.2 Clinically, targetoid haemosiderotic haemangioma appears as a small, solitary, red, purple and/or brown papule or macule that is typically encircled by a thin pale area and peripheral ecchymotic ring, giving an appearance of a targetoid lesion. Overall dimensions are generally less than one cm in diameter. The lesions affect mainly the trunk or the extremities and are generally asymptomatic. There may be pain, change in colour, increase in size or cyclical change,3 as was the case in our patient. Histological examination is characterized by a dermal vascular proliferation. In the papillary dermis, a single layer of

JEADV 2016, 30, 852–909

Long-term remission of primary cutaneous neutrophil-rich CD30+ anaplastic large cell lymphoma treated with topical imiquimod. A case report Editor A 25-year-old male presented with a 2-week history of a welldefined, nodule on the right aspect of his nose (Fig. 1a). Physical

© 2015 European Academy of Dermatology and Venereology

Letters to the Editor

900

examination showed a 15-mm ulcerated nodule, with a large plug of horny material in the centre. Initial diagnosis was keratoacanthoma (KA) and a biopsy punch for histopathologic confirmation was performed on the border of the lesion. Since surgery would have been disfiguring, off-label treatment with imiquimod 5% cream was started with the patient’s written consent. The patient was asked to apply imiquimod at bedtime once a day for ten consecutive days, then every other day for 2 months. After 7 days, erythema, erosions, a burning sensation and bleeding appeared, and by the 15th the cutaneous lesion started to regress disappearing completely within the first month of therapy and only leaving a mild atrophic scar (Fig. 1b). Histopathologic examination showed a polymorphous cellular infiltrate occupying the entire dermis and part of the subcutis. There were sheets of atypical lymphoid cells with enlarged, horseshoe-shaped nuclei and prominent nucleoli. Numerous mitoses were observed, some of which atypical. No epidermotropism was detectable. Immunohistochemical analysis showed that the atypical lymphocytes were intensely positive for CD45, CD3, CD4 and CD30. Partial, dim expression of the cytotoxic molecule granzyme-B was also observed in tumour cells. They tested negative for CD8, CD56, CD20, ALK-protein, S-100 protein, epithelial membrane antigen (EMA), cytokeratin, CD34 and myeloperoxidase. Ki67 (Mib-1) staining demonstrated a high proliferation rate. In situ hybridization for Epstein–Barr virus (EBV)–encoded small nuclear RNA (EBER) was negative. On the basis of histopathological and immunohistochemical findings a diagnosis of primary cutaneous CD30+ neutrophilrich ALCL was made. The patient is now under observation and is free from recurrence after a 60-month follow-up period. Anaplastic large cell lymphoma is a rare, highly aggressive T-cell lymphoma characterized by constant expression of the CD30 antigen, a transmembrane glycoprotein belonging to the tumour necrosis factor receptor family.1 Two histopathological indistinguishable subtypes, both characterized by the presence of hallmark cells with horseshoe-shaped nuclei and abundant cytoplasm, have been categorized as positive or negative for the anaplastic lymphoma tyrosine kinase (ALK) receptor, an enzyme encoded by the ALK (a)

Table 1 Salient data of six patients with primary cutaneous CD30+ ALCL treated with imiquimod Cases

Sex

Age

Site

Previous treatments

Coors et al.8

F

63

NR

Surgery, PUVA, IFN

Ehst et al.9

M

65

Arm

Multi agent chemotherapy, MTX, topical and intralesional corticosteroids and minocycline

Ehst et al.9

M

78

Temple

Radiotherapy, multi agent chemotherapy, PUVA, MTX

4

Didona et al.7

M

23

Knee

None

8

Didona et al.7

M

55

Leg

None

8

Current case

M

25

Nose

None

60

(b)

Figure 1 Response of primary cutaneous neutrophil-rich CD30+ anaplastic large cell lymphoma to imiquimod. (a) Nodule of the nose. (b) The same patient 10 weeks later.

JEADV 2016, 30, 852–909

gene. ALK-positive ALCL predominantly affects young adults and children, mostly male, and has an overall 5-year survival rate of 70–80%. On the contrary, ALK-negative ALCL is an aggressive disease with a dismal prognosis.1,2 According to the European Organization for Research and Treatment of Cancer criteria, the diagnosis of primary cutaneous ALCL should be restricted to skin-limited lesions for at least 6 months after initial identification.1 Clinical diagnosis is very difficult both because of the rarity of ALCL and the absence of specific symptoms. Surgery or radiation therapy is the treatments of choice. Spontaneous remission may occur in a few cases.1–4 Imiquimod is an immunomodulating drug which is able to induce the synthesis of a number of cytokines by binding on a synthetic ligand for toll-like receptor 7.5 Although imiquimod has only been approved for use by the FDA for the treatment of external anogenital warts and superficial basal cell carcinomas in adults, its immunomodulatory properties have led to extensive off-label therapeutic use for a number of disease such as: cutaneous melanoma, cutaneous T-cell lymphoma, actinic keratosis, KA, keloids and molluscum contagiosum.6 International literature only reports three articles in which imiquimod was adopted for treating primary cutaneous ALCL, all of which gave excellent cosmetic results, although the follow-up period after treatment was very short (Table 1).7–9 In our patient, the absence of recurrence after 5 years may be considered a sufficient evidence of complete healing. Even if further investigations are necessary to establish the real efficacy of imiquimod, this topical agent might represent an additional and effective option for the conservative management of primary cutaneous ALCL.

Follow-up (months) 5 12

NR, not reported; MTX, methotrexate; IFN, interferon; PUVA, Psoralens + UVA.

© 2015 European Academy of Dermatology and Venereology

Letters to the Editor

Financial support None. D. Calista,1,* L. Riccioni,2 L. Bagli,3 F. Valenzano4 Department of Dermatology, “M. Bufalini” Hospital, 2Pathology Unit, “M. Bufalini” Hospital, 3Molecular Genetic Unit, Laboratorio Unico Area Vasta, Cesena, 4San Gallicano Dermatologic Institute, IRCCS, Rome, Italy *Correspondence: D. Calista, E-mail: [email protected] 1

References 1 Delsol G, Falini B, Muller-Hermelink HK et al. Anaplastic large cell lymphoma (ALCL) ALK-positive. In Swerdlow SH, Campo E, Harris NL, Jaffe E, Pileri SA, Stein H, Thiele J, Vardiman eds. WHO Classification of Tumours of Haematopoietic and Lymphoid Tissues. World health Organization IARC press, Lyon, 2008: 312–316. 2 Kempf W, Willemze R, Jaffe ES, Burg G, Kadin ME. CD30+ T-cell lymphoproliferative disorders. In LeBoit PE, Burg G, Weedon D, Sarasin A eds. Pathology & Genetics – Skin Tumours. World health Organization IARC press, Lyon, 2006: 179–181. 3 Mann KP, Hall B, Kamino H, Borowitz MJ, Ratech H. Neutrophil-rich, Ki-1-positive anaplastic large cell malignant lymphoma. Am J Surg Pathol 1995; 19: 407–416. 4 Burg G, Kempf W, Kazakov DV et al. Pyogenic lymphoma of the skin: a peculiar variant of primary cutaneous neutrophil-rich CD30+ anaplastic large-cell lymphoma. Clinicopathological study of four cases and review of the literature. Br J Dermatol 2003; 148: 580–586. 5 Garland SM. Imiquimod. Curr Opin Infect Dis 2003; 16: 85–89. 6 David CV, Nguyen H, Goldenberg G. Imiquimod: a review of off-label clinical applications. J Drugs Dermatol 2011; 10: 1300–1306. 7 Didona B, Benucci R, Amerio P, Canzona F, Rienzo O, Cavalieri R. Primary cutaneous CD30+ T-cell lymphoma responsive to topical imiquimod (Aldara). Br J Dermatol 2004; 150: 1198–1201. 8 Coors EA, Schuler G, Von Den Driesch P. Topical imiquimod as treatment for different kinds of cutaneous lymphoma. Eur J Dermatol 2006; 16: 391–393. 9 Ehst BD, Dreno B, Vonderheid EC. Primary cutaneous CD30+ anaplastic large cell lymphoma responds to imiquimod cream. Eur J Dermatol 2008; 18: 467–468.

901

Propranolol cream (at 1% concentration, prepared by the hospital pharmacy) was given to the patients to be applied on the PG overnight under occlusion (covered with a plastic wrap) for a maximum of 45 days. Nail clipping was performed when the PG was covered by the nail plate. Patients were followed up at days 15, 30 and 45. Ten patients (six female and four males) were included: their ages varied from 13 to 70 (mean age 40.8). Causes of PGs were: chemotherapy in three patients (1 trastuzumab, 1 capecitabine and lapatinib, 1 erlotinib), ingrown nail in 5, and friction in two cases. One patient had PGs of the fingernails, two were affected in both fingernails and toenails and seven had PGs limited to the toenails. Localizations of PGs in the nail apparatus were: proximal nail fold in three cases, lateral fold in 8 and nail bed in two cases. Propranolol cream induced cure of all (100%) PGs due to chemotherapy localized in the proximal and the lateral nail folds of the fingernails (Fig. 1a,b), despite the fact that the patients continued chemotherapy with an unmodified dose. Cure was still present at the follow-up visit. No side-effect was observed or reported.

(a)

DOI: 10.1111/jdv.13070

(b)

Topical propranolol 1% cream for pyogenic granulomas of the nail: open-label study in 10 patients Editor Pyogenic granuloma (PG) is a benign vascular proliferation that may affect the nails.1,2 Treatment is surgical, associated to or preceded by topical therapy.1–3 Based on previous studies showing that b-blockers increase the regression of PGs,4 we verified the effectiveness of topical 1% propranolol (1% cream in occlusion) cream on periungual and subungual PGs of hand and feet in 10 patients.

JEADV 2016, 30, 852–909

Figure 1 (a) Periungual PGs due to drug (trastuzumab) of the fingernails. (b) Complete disappearance of the PGs after 30 days of application of topical propanolol.

© 2015 European Academy of Dermatology and Venereology

Long-term remission of primary cutaneous neutrophil-rich CD30+ anaplastic large cell lymphoma treated with topical imiquimod. A case report.

Long-term remission of primary cutaneous neutrophil-rich CD30+ anaplastic large cell lymphoma treated with topical imiquimod. A case report. - PDF Download Free
390KB Sizes 0 Downloads 13 Views