Journal of Dermatological Case Reports

Publications in this journal

• Article: Gardner-Diamond syndrome associated with complex regional pain syndrome.

  Lara K Edinger, Robert J Schwartzman
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  ABSTRACT: Gardner-Diamond syndrome (GDS) is also known as psychogenic purpura, autoerythrocyte sensitization syndrome and painful bruising syndrome. This is a case report of 27-year-old woman who presented with unexplained bruising and intramuscular hematomas after a seven year history of complex regional pain syndrome. Her evaluation consisted of hematological studies, skin and muscle biopsy; it failed to reveal an underlying coagulopathy, vasculitis or other demonstrable cause. In the absence of any other etiology, she was diagnosed as Gardner-Diamond syndrome. This patient is unique because of intramuscular hematomas and the association of Complex regional pain syndrome with Gardner-Diamond syndrome.
  Journal of Dermatological Case Reports 03/2013; 7(1):10-4.
• Article: Photoletter to the editor: Dermoscopy for discriminating between pityriasis rubra pilaris and psoriasis.

  Aimilios Lallas, Zoe Apalla, Anatoli Karteridou, Ioanna Lefaki
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  ABSTRACT: Pityriasis rubra pilaris (PRP) is a relatively uncommon entity that often has to be clinically differentiated from other erythematosquamous skin diseases, such as psoriasis. Dermoscopy has already been shown to enhance clinical evaluation of inflammatory skin conditions and dermoscopic patterns of various diseases, including psoriasis, have been documented. In the current manuscript we present the dermoscopic findings observed in two patients suffering from PRP and psoriasis, respectively. The dermoscopic pattern of PRP consisted of round/oval yellowish areas surrounded by vessels of mixed morphology. The latter findings are clearly distinct from the dermoscopic features of psoriasis, which have been extensively investigated previously and are presented also in the psoriatic patient herein. This observation represents an initial indication that dermoscopy could be of value in differentiating between the two entities.
  Journal of Dermatological Case Reports 03/2013; 7(1):20-2.
• Article: Photoletter to the editor: A papular eruption on the face. A distinct subtype of lichen planopilaris?

  Israel D Andrews, Catherine Breen, Linda H Zhou
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  ABSTRACT: Lichen planopilaris is a scarring alopecia resulting from a lymphocytic inflammatory process of unknown etiology. We report a case of a 46-year-old man, who presented with an asymptomatic papular eruption over the face. Histologic examination was consistent with lichen planopilaris. This case is unusual because the disease affects the facial vellus hair only, without scalp involvement or other features of lichen planopilaris and its variants.
  Journal of Dermatological Case Reports 03/2013; 7(1):23-4.
• Article: Photoletter to the editor: Disseminated histoplasmosis with initial oral manifestations.

  Surabhi Sinha, Kabir Sardana, Vijay K Garg
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  ABSTRACT: Histoplasmosis is a systemic fungal disease that may present in a variety of clinical manifestations. Involvment of the oral mucosa is very rare and may occur as part of disseminated histoplasmosis or as isolated involvement. We present a patient with disseminated histoplasmosis, in whom oral lesions were the initial manifestation of the disease.
  Journal of Dermatological Case Reports 03/2013; 7(1):25-6.
• Article: Clinical, dermoscopic and histological features of a Merkel cell carcinoma of the hand.

  Massimiliano Scalvenzi, Franco Palmisano, Gennaro Ilardi, Silvia Varricchio, Claudia Costa
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  ABSTRACT: Merkel cell carcinoma (MCC) is a rare and typically aggressive form of skin cancer. The benign appearance of the tumor usually on exposed skin parts, contrasting with its extensive microscopic invasion, can delay timely diagnosis. We report a case of a 71-year-old man with a slowly progressive nodule on the left hand. At the dermoscopic examination, the presence of a polymorphous vascular pattern, including milky-red areas may constitute an additional clinical clue to accurately diagnose this rare tumor. Clinical, dermoscopic and histological features are discussed.
  Journal of Dermatological Case Reports 03/2013; 7(1):15-7.
• Article: Photoletter to the editor: Congenital atrichia associated with an uncommon mutation of HR gene.

  Ana Pedrosa, Ana Nogueira, Paulo Morais, Ana Filipa Duarte, Joana Pardal, Alberto Mota, Filomena Azevedo
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  ABSTRACT: Congenital atrichia (CA) is a rare form of irreversible alopecia with an autosomal recessive mode of inheritance, usually associated with a mutation in the human hairless (HR) gene located at chromosome 8. Papular lesions may develop as an additional phenotypic feature. Herein we describe a case of CA supported by trichoscopy, histology and genetic analysis. The patient's single brother had also universal alopecia. To our knowledge this is the second report of a specific pathogenic mutation (c.2818C>T) of the HR, which until now had only been identified in a family with CA and papular lesions, emphasizing the difficulty to establish a strict correlation between HR genotyping and the phenotype.
  Journal of Dermatological Case Reports 03/2013; 7(1):18-9.
• Article: Photoletter to the editor: Botryomycosis in an immunocompetent woman.

  Hugo Marques Barreiros, Henriqueta Cunha, Elvira Bártolo
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  ABSTRACT: Botryomycosis is a chronic, granulomatous infection of the skin, soft tissue and viscera. It is uncommon and mainly occurs in immunocompromised patients. Splendore-Hoeppli phenomenon represents an immunological host reaction to antigens of infectious and non-infectious agents. We report a case of a 64-year-old immunocompetent woman presented at our department with a 2-month history of abdominal papules and discharging nodules. She was treated with several antibiotics with no success. Skin biopsy showed granular bodies in the dermis with a Splendore-Hoeppli phenomenon. Microbiologic study isolated a Staphylococcus aureus. Patient was successfully treated 4 weeks with oral clindamycin 300 mg bid. Lack of drug penetration into the "encapsulated-like" microcolonies could explain the therapeutic challenge of this case.
  Journal of Dermatological Case Reports 03/2013; 7(1):29-30.
• Article: The risk of herpes zoster in the anti-TNF-α era: a case report and review of the literature.

  Luisa Di Costanzo, Fabio Ayala, Matteo Megna, Francesca Gaudiello, Angela Patrì, Nicola Balato
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  ABSTRACT: Tumor necrosis factor-α (TNF-α) inhibitors represent efficacious therapeutic agents in many chronic inflammatory diseases such as psoriasis and rheumatoid arthritis. However they have been connected with increased risk of infection and reactivation of a variety of infectious agents, such as viruses. The reactivation of varicella zoster virus infection causes herpes zoster (HZ), a self-limiting, dermatomally localized, vesicular rash that can be accompanied by postherpetic neuralgia and severe neurological complications. Limited information has been published regarding HZ during therapy with TNF-α inhibitors especially for the occurrence of HZ during adalimumab treatment. We report the case of a 58-year-old immunocompetent man with a 18-year history of plaque psoriasis who develops ophthalmic HZ during treatment with adalimumab. We report this case to enrich the literature and to highlight the increased risk of HZ infections in patient on anti-TNF-α therapy (incidence of HZ is about 3-fold increased respect to general population). Clinically, these infections often have atypical presentations that may hamper prompt diagnosis. Therefore, it is very important to identify early signs and symptoms of herpes zoster in patients on biologic therapy in order to start prompt efficient antiviral treatment to prevent the development of severe complications.
  Journal of Dermatological Case Reports 03/2013; 7(1):1-4.
• Article: Long-term ustekinumab treatment for refractory type I pityriasis rubra pilaris.

  Alessandro Di Stefani, Marco Galluzzo, Marina Talamonti, Andrea Chiricozzi, Antonio Costanzo, Sergio Chimenti
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  ABSTRACT: Pityriasis rubra pilaris is a rare, chronic erythematous squamous disorder of unknown etiology. The disease is characterized initially by small follicular papules that coalesce into yellowish pink scaly plaques, palmoplantar keratoderma, diffuse furfuraceous scale of the scalp, and frequent progression to exfoliative erythroderma. Generally it is difficult to discern pityriasis rubra pilaris from other skin conditions but key-clinical features help in the diagnosis such as "islands" of spared skin within generalized erythroderma, follicular keratotic plugs, and an orange hue of the involved skin. Treatment options include topical vitamin D analogues, keratolytics, systemic acitretin, methotrexate, cyclosporine, azathioprine, fumaric acid esters, phototherapy, and anti-TNFα agents. Cases, of pityriasis rubra pilaris, successfully treated with a short-course ustekinumab therapy, have been reported. We report a 31-year-old man with pityriasis rubra pilaris, refractory to conventional treatments, successfully treated with ustekinumab monotherapy for over 64 weeks. After failing conventional systemic agents (cyclosporine, aciretin and methotrexate), ustekinumab 45 mg has been prescribed, with the same dosing regimen as in psoriasis. The patient improved dramatically within 4 weeks of the first injection, with markedly less erythema and pruritus. Long-term control of the disease of the disease was achieved (64 weeks of treatment). We report this case in order to show the striking and rapid efficacy of ustekinumab in reducing the signs and symptoms of the disease. Complete remission was achieved after the third injection, but also a long-term control of the disease. The therapy was well-tolerated in our patient and no adverse events occurred.
  Journal of Dermatological Case Reports 03/2013; 7(1):5-9.
• Article: Photoletter to the editor: Atrophic dermatofibrosarcoma protuberans with minimal clinical manifestation.

  Hugo Marques Barreiros, Pedro Nuno Serrano, José Carlos Parreira, Elvira Bártolo
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  ABSTRACT: Dermatofibrosarcoma protuberans is an uncommon soft tissue neoplasm. In the vast majority of cases it presents as a nodule or a firm tumor that can reach massive dimensions producing the protuberant nodules for which it is named. We report a case of a 34-year-old woman presented at our department with an 8-year history of a small and discretely erythematous supraclavicular atrophic plaque. Skin biopsy lead to the diagnosis of dermatofibrosarcoma protuberans and a wide local excision of the tumor was performed in collaboration with the Plastic Surgery department. In this clinical case we describe an uncommon variant of the disease with minimal clinical manifestation that can cause serious diagnostic difficulties. The small and discrete atrophic plaque of our patient could have been easily ignored with serious clinical and prognostic implications for the patient.
  Journal of Dermatological Case Reports 03/2013; 7(1):27-8.
• Article: Peripheral stellate telangiectasias: a clinical-dermoscopic clue for diganosing cutaneous melanoma metastases.

  Yamina Julian, Giuseppe Argenziano, Elvira Moscarella, Raquel de Paula Ramos Castro, Caterina Longo, Alejandra Abeldaño, Iris Zalaudek
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  ABSTRACT: BACKGROUND: The clinical and dermoscopic diagnosis of cutaneous melanoma metastases may be challenging especially in patients with unknown primary melanoma. MAIN OBSERVATIONS: We observed repetitive dermoscopic patterns of peripheral stellate telangiectasias in cutaneous melanoma metastases from 3 patients, of whom 2 had an unknown primary melanoma. CONCLUSIONS: Stellate telangiectasias surrounding bluish to purple or red nodules with recent onset may represent a clue for cutaneous melanoma metastases.
  Journal of Dermatological Case Reports 12/2012; 6(4):102-104.
• Article: Spontaneous abortion during ustekinumab therapy.

  Christina Fotiadou, Elizabeth Lazaridou, Eleni Sotiriou, Demetrios Ioannides
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  ABSTRACT: BACKGROUND: Psoriasis affects a considerable proportion of women in their reproductive years. Limited published data exist about the possible negative impact of the disease itself in the prognosis of pregnancy. On this background, the emergence of newer biologic agents for psoriasis treatment - such as ustekinumab - raises safety issues concerning the exposure to the drug during pregnancy. To our knowledge this is the first report in the literature describing a pregnancy outcome under ustekinumab treatment. OBSERVATION: We report a 35-year-old female psoriasis patient who was under treatment with ustekinumab for a year when she inadvertedly became pregnant. The drug was discontinued immediately and the patient did not opt for termination. During the 12th week of gestation she experienced a spontaneous abortion. CONCLUSION: Although the patient's profile fulfilled 2 general risk factors for spontaneous abortion - she was a smoker and this was her third pregnancy - one could not underestimate the possible role of the drug and of psoriasis per se in this adverse pregnancy outcome. Pregnancy registries and large prospective studies are needed in order to determine whether poorer pregnancies outcomes in psoriatic women are due to the disease itself, associated comorbidities or side-effects of new therapies such as ustekinumab.
  Journal of Dermatological Case Reports 12/2012; 6(4):105-107.
• Article: Drug-induced linear IgA bullous dermatosis after discontinuation of cefuroxime axetil treatment.

  Maciej Pastuszczak, Sylwia Lipko-Godlewska, Andrzej K Jaworek, Anna Wojas-Pelc
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  ABSTRACT: BACKGROUND: Linear immunoglobulin A (IgA) bullous dermatosis (LABD) is a rare autoimmune blistering disorder. The disease may be either idiopathic or druginduced. Over the past 30 years, approximately one hundred LABD cases have been described as induced by a wide range of drugs, chiefly antibiotics. MAIN OBSERVATIONS: We report the case of 37-year-old woman who developed pruritic bullous lesions spread all over the body three weeks after her last dose of cefuroxime axetil. Antibiotic therapy was started due to rhino-sinusitis. CONCLUSIONS: In most reported cases of drug-induced LABD, skin lesions occur within the time of drug administration. However, the onset of disease may be even after discontinuation of treatment. It seems that in such cases, other clinical conditions (like infection) act, as cofactors of immunologic response.
  Journal of Dermatological Case Reports 12/2012; 6(4):117-119.
• Article: Photoletter to the editor: Proliferating pilomatricoma with no recurrence during a 3-year follow-up.

  Masataka Satoh, Masayuki Ookouchi, Toshiyuki Yamamoto
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  ABSTRACT: Proliferating pilomatricoma is a proliferative variant of pilomatricoma. Although it is considered as a benign tumor, local recurrence may occur. We report a case of a 49-year-old Japanese man with 3-year history of an asymptomatic subcutaneous tumor in the nuchal area. Histological evaluation demonstrated a cystic lesion lined by a basaloid epithelium at the periphery and filled with eosinophilic cornified material containing shadow cells in upper part of the tumor, and multilobular proliferation of basaloid cells in association with small foci of shadow cells in the remaining part. Based on these findings, the diagnosis of proliferating pilomatricoma was made. No recurrence has been observed during a 3-year follow-up.
  Journal of Dermatological Case Reports 12/2012; 6(4):127-129.
• Article: Photoletter to the editor: Fibrous histiocytoma developing at the site of tuberculin skin test.

  Erika Nomura, Toshiyuki Yamamoto
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  ABSTRACT: Benign fibrous histiocytomas of the skin sometimes extend into the deeper dermis with higher cellularity and show more aggressive clinical courses in comparison with typical dermatofibromas. They may occur either as a true neoplasm or in a reactive process. We describe a case of fibrous histiocytoma which was triggered by tuberculin skin test.
  Journal of Dermatological Case Reports 12/2012; 6(4):130-131.
• Article: Photoletter to the editor: Linear atrophoderma of Moulin progressing slowly over 46 years.

  Maryam Afshar, Jeff Melancon, Tissa Hata
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  ABSTRACT: Linear atrophoderma of Moulin is a rare acquired disorder arising most commonly during childhood or adolescence, occurring equally in both sexes and characterized by hyperpigmented atrophoderma in a unilateral bandlike distribution along the lines of Blaschko. Since Moulin et al described the condition in 1992, only a few dozen cases have been reported. It has been postulated that linear atrophoderma of Moulin may be due to mosaicism.A 66-year-old man pre-sen-ted with a 46-year his-to-ry of evol-ving tan soft atro-phic con-fluent plaques in a striking-ly Blasch-koid dis-tri-bu-tion, in-vol-ving the left up-per back, shoulder, up-per arm, chest and flank. Ini-tial on-set, at age 20, con-sis-ted of a single mildly pru-ritic pink patch on the left back that was un-res-pon-sive to topical anti-fun-gals. Each new le-sion arose simi-larly as a pink pru-ritic patch, sub-se-quent-ly be-co-ming de-pressed, hy-per-pig-men-ted, and asym-pto-ma-tic over se-ve-ral years. Le-sions were never scaly, firm, or indu-rated. Punch biopsy speci-mens were obtained. The clinical and histo-patho-lo-gi-cal features con-firmed the diagnosis of linear atrophoderma of moulin.Our present case has the characteristic clinical and histopathological features of linear atrophoderma of Moulin, but is the first reported case with mild pruritus at the onset of each new lesion and progressing slowly over 46 years. The lack of any systemic symptoms or other complications in our patient reaffirms the benign nature of this skin disease.
  Journal of Dermatological Case Reports 12/2012; 6(4):125-126.
• Article: Letter to the editor: Application of dapsone 5% gel in a patient with dermatitis herpetiformis.

  Marc Z Handler, Anna H Chacon, Michael I Shiman, Lawrence A Schachner
  Journal of Dermatological Case Reports 12/2012; 6(4):132-133.
• Article: Granulomatous slack skin. Histopathology diagnosis preceding clinical manifestations by 12 years.

  Karen O Goldsztajn, Beatriz Moritz Trope, Maria Elisa Ribeiro Lenzi, Tullia Cuzzi, Marcia Ramos-E-Silva
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  ABSTRACT: BACKGROUND: Granulomatous slack skin is a very rare subtype of T-cell cutaneous lymphoma, characterized by the slow development of cutaneous sagging, especially on flexural areas. Its behavior is indolent and the treatment, in the majority of cases, disappointing. MAIN OBSERVATION: We report a 54-year-old black patient with granulomatous slack skin, who at the beginning of the investigation showed intense xeroderma and generalized lymph node enlargement. The diagnosis was established based on histopathologic findings long before the disease's characteristic clinical presentation appeared. CONCLUSIONS: During the twelve years of follow-up, the clinical manifestation evolved to marked skin looseness, most predominant in flexural regions, illustrating the clinical hallmark of granulomatous slack skin, long after first histological abnormalities were observed.
  Journal of Dermatological Case Reports 12/2012; 6(4):108-112.
• Article: Geographic stomatitis: an oral manifestation of psoriasis?

  Bruna Picciani, Geraldo Silva-Junior, Sueli Carneiro, Ana Luisa Sampaio, Daniel Cohen Goldemberg, Juliana Oliveira, Luis Cristóvão Porto, Eliane Pedra Dias
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  ABSTRACT: BACKGROUND: Geographic stomatitis is an uncommon oral lesion that presents similar clinical, histopathological and genetic features as those of psoriasis. These findings suggest that this lesion may actually represent an oral manifestation of psoriasis. We report one case of geographic stomatitis and discuss a possible connection between this condition and psoriasis. MAIN OBSERVATIONS: A 37-year-old woman presented with red patches, surrounded by a white border on the labial mucosa and a positive family history of psoriasis. Histopathological examination, immunohistochemical analysis with antibodies against CD4, CD8, CD20, CD68, CD31, and Ki-67 and HLA-A*, -B*, -C*, -DRB1*, -DQA1* and -DQB1* genotyping were performed. Histopathological examination revealed parakeratosis, marked elongation of rete ridges with acanthosis and clubbing, exocytosis, Munro microabscesses, pustule of Kogoj, dilated tortuous vessels at the tip of dermal papillae, and predominant superficial and perivascular lymphocytic chronic inflammatory cell infiltrate. Immunohistochemistry analysis revealed a predominant T-cell subepithelial infiltrate. Based on the referred clinicopathological findings and in the absence of cutaneous lesions, the diagnosis of geographic stomatitiswas confirmed. CONCLUSIONS: This case and theoretical data indicate that geographic stomatitis may be an oral manifestation of psoriasis. Moreover, to improve our understanding, psoriatic patients should routinely undergo a detailed oral examination and patients with geographic stomatitis should routinely be submitted to a cutaneous routine examination.
  Journal of Dermatological Case Reports 12/2012; 6(4):113-116.
• Article: Alternaria scalp infection in a patient with alopecia areata. Coexistence or causative relationship?

  Lidia Rudnicka, Malgorzata Lukomska
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  ABSTRACT: BACKGROUND: Alopecia areata is an autoimmune disease that is affecting anagen hair follicles. The triggers of autoimmunity in patients with alopecia areata remain unknown. MAIN OBSERVATION: A 13-year-old boy developed multiple hairless patches of focal hair loss with typical clinical and trichoscopy features of alopecia areata. Mycology examination of the scalp hair and epidermal scrapings reveled massive growth of Alternaria chlamydospora. CONCLUSION: We hypothesize that fungal antigens (e.g. antigens involved in fungal melanin synthesis) may be possible triggers, contributing to autoimmune reactions in patients with alopecia areata. We discuss research data, which may indirectly support this hypothesis, however the concept has yet to be verified.
  Journal of Dermatological Case Reports 12/2012; 6(4):120-124.