[Actinomycosic mycetoma of the foot in Morocco due to Actinomycetes viscosus]. / Mycétome actinomycosique du pied à Actinomycetes viscosus au Maroc.

We present the case of an actinomycotic mycetoma of the foot due to Actinomycetes viscosus. It evolved for nine years on the foot of a 26-year-old patient from a rural environment: Douar Inezgane (city in southern Morocco). Bacteriological study of the skin and grains confirmed the diagnosis. It showed positive bacilli on direct examination and on Gram staining and in positive culture. Histological study showed a polymorphous granulomatous inflammation without signs of malignancy with actinomycotic grains. Then we retained the diagnosis of primary cutaneous actinomycosis without visceral locations. The treatment was based on antibiotics: penicillin G by intravenous infusion for five weeks, relayed orally by amoxicillin associated with trimethoprim-sulfamethoxazole for long periods. After six months of treatment, we observed a favorable outcome with reduction of the swelling, nodules, lymphadenopathy, fistula's number and extension of time of issue of grains. The current follow up is 15 months. The primary cutaneous actinomycosis is still relevant in Morocco.

[Deep-seated granulomatous pyoderma gangrenosum: A novel variant?]. / Pyoderma gangrenosum granulomateux profond : une forme originale de pyoderma gangrenosum ?

BACKGROUND: We report an extremely rare case of pyoderma gangrenosum with the clinical features of pyoderma gangrenosum but with unusual histopathological findings and deep massive granulomatous infiltration through the dermis. PATIENTS AND METHODS: A 15-year-old girl presented with a two-month history of deep ulcers on the lower legs and forehead. She also presented abdominal pain with diarrhea and vomiting that disappeared after symptomatic treatment. Histology showed extensive granulomatous infiltration in the dermis and hypodermis and a micro-abscess in the dermis. An abdominopelvic CT scan revealed peritoneal fluid and tissue formation in a lateral carotid cave (6.56 cm/4.56 cm). Colonoscopy was normal. The patient was treated with oral prednisone, resulting in resolution of the problems of cutaneous lesions, peritoneal fluid and tissue formation. DISCUSSION: Our case involves a rare presentation of pyoderma gangrenosum (PG) inconsistent with any of the four clinical variants of PG and characterized histologically by the presence of deep granulomas in the dermis and hypodermis. Despite the atypical clinical presentation and unusual histopathological findings, PG seemed the most likely diagnosis after infectious diseases had been ruled out; the good outcome achieved with oral corticosteroids supported our diagnosis.

[Xanthoma disseminatum with asymptomatic multisystem involvement]. / Xanthoma disseminatum avec atteinte viscérale multifocale asymptomatique.

BACKGROUND: Xanthogranulomas belong to non-Langerhans histiocytosis of the second group in the Histiocyte Society classification. They comprise a heterogeneous group of rare entities frequently involving cutaneous tropism. Xanthoma disseminatum belongs to this group of non-Langerhans histiocytosis. We report a case of xanthoma disseminatum (XD) in which localized skin and mucous impairment revealed multisystem involvement. PATIENTS AND METHODS: A 28-year-old man presented with a two-year history of progressive yellow-orange and infiltrated xanthomatous papulonodular lesions of the face. Lesions of the oral mucosa and genital region were seen, with no functional repercussions. No ophthalmic or other complications were found. Histopathology showed a dense histiocytic infiltrate within the dermis with Touton giant cells, foamy multinucleated giant cells and inflammatory cells, without necrobiosis. Histiocytes were positive for CD68 but negative for CD1a. Gastric and lung involvement was seen and was confirmed at histology. Bone scintigraphy showed suspicious left ulnar hyperfixation suggesting bone involvement. No monoclonal gammopathy or diabetes insipidus was seen. Our patient was treated with corticosteroids 1mg/kg/day and thalidomide 100 mg/day. The outcome was marked by regression and exfiltration of the cutaneous lesions from the second week of treatment, with subsidence continuing at 3 months. DISCUSSION: This case involves a very rare form of xanthoma disseminatum. The localized facial skin lesions revealed multifocal non-Langerhans histiocytosis that was in fact asymptomatic. The diagnosis of XD was based on clinical, histological and immunohistochemical criteria. Xanthoma disseminatum is a non-Langerhans histiocytic proliferation first described by Montgomery in 1938. This rare entity is characterized by skin and mucous membrane xanthomatosis in which the facial involvement is common, together with diabetes insipidus and normal lipid metabolism. The prognosis is determined by the presence of mucosal xanthomas and visceral involvement. Thus, all xanthogranulomas involving multiple lesions warrant screening for visceral involvement. Diagnosis of this entity can be difficult and is usually based on clinical and histopathological findings. In addition, treatment is complex and non-consensual.

[Efficacy of pulsed-dye laser on residual red lesions of cutaneous leishmaniasis]. / Efficacité du laser à colorant pulsé sur les lésions résiduelles rouges de la leishmaniose cutanée.

BACKGROUND: Cutaneous leishmaniasis caused by Leishmania tropica can leave troublesome and unsightly lesions. Treatment of these scars remains difficult. Pulsed-dye laser (PDL) is one therapeutic approach that may improve the clinical appearance of erythematosus lesions. The purpose of this retrospective study was to evaluate the effectiveness of PDL on the residual red lesions of erythematous facial leishmaniasis in three patients. PATIENTS AND METHODS: Case no. 1: a 14-year-old girl presented an ulcerative and erythematous nodular lesion on her left cheek. One month after treatment, an erythematous lesion measuring 3 cm persisted on the patient's cheek, without atrophy or hyperpigmentation. PDL 595nm was used at the following settings: duration: 3ms; spot size: 7mm; energy: 8 j/cm(2). Case no. 2: a 43-year-old woman presented an erythematous papular lesion on her right cheek. Following treatment, a 4-cm hypertrophic, red telangiectasic lesion remained. PDL 595nm was used with the following settings: pulse duration: 3 ms; spot-size: 10mm; energy: 8 j/cm(2). Case no. 3: a 60-year-old woman presented an erythematous papular lesion on her cheek. After treatment, an infiltrated erythematous macule with surface telangiectasia measuring 3.5cm remained. PDL 595nm was also given using the following settings: pulse duration: 3 ms; spot size: 10mm; energy: 8 j/cm(2). All three patients underwent three sessions of PDL. The erythematous and telangiectasic lesions showed improvement after the initial session and had completely disappeared after the third session. Post-laser purpura subsided within around 10 days. Therapeutic response was assessed clinically by comparing photographs taken before and after treatment and follow-up lasted 12 months. DISCUSSION: Cutaneous leishmaniasis caused by L. tropica is endemo-epidemic in Morocco. A number of treatments are available for red residual lesions but thanks to its effect on erythematous and vascular lesions, PDL has been shown to provide the most reproducibly good results and is the laser method of choice for this type of scar. A recent study of the dermoscopic features of DL identified the presence of vascular patterns in 100% of cases in this infection, which may account for the efficacy of PDL. PDL results in selective thermolysis that destroys small vessels. Our study showed improvement with PDL regarding scar size, pliability, erythema and texture. Further larger-scale studies could better determine the place of PDL in treating the sequelae of cutaneous leishmaniasis.

[Palmoplantar keratoderma: a rare manifestation of myxoedema]. / Kératodermie palmo-plantaire : une manifestation rare du myxœdème.

BACKGROUND: Herein we report a rare case of acquired palmoplantar keratoderma in association with myxoedema and hypothyroidism. PATIENTS AND METHODS: A 53-year-old woman presented with palmoplantar keratoderma, dry skin, muscular weakness and cramps for 9 months. The laboratory work-up revealed autoimmune thyroiditis with hypothyroidism. Skin biopsy showed chronic eczema. Other causes of acquired palmoplantar keratoderma were ruled out. Rapid improvement was achieved within 3 weeks of institution of hormone replacement therapy. DISCUSSION: Although this association is very rare, hypothyroidism must be suspected in patients with acquired palmoplantar keratoderma, particularly when it occurs in association with systemic symptoms.

[Generalized chromomycosis caused by Phialophora verrucosa]. / Chromomycose cutanée diffuse àPhialophora verrucosa.

BACKGROUND: Chromomycosis is a chronic fungal skin infection that generally presents in the form of verrucous or vegetative lesions on uncovered areas of skin. We report an unusual case of generalised chromomycosis due to Phialophora verrucosa. PATIENTS AND METHODS: A 42-year-old town-dwelling housewife was hospitalised for erythematous keratotic nodules on the arm showing sporotrichoid distribution, associated with a crusted ulcerative lesion on the homolateral index finger, as well as subcutaneous papulonodular lesions. In places, the lesions on the patient's back presented an umbilical and molluscoid appearance. The patient had suffered no previous injuries and had not visited any areas in which leishmaniasis is endemic. Her history included insulin-dependent diabetes. Screening for Leishman bodies was negative. Histopathological analysis of the skin biopsy revealed an epithelioid giant-cell granuloma with no caseous necrosis. The mycological study demonstrated the presence of fumagoid bodies and P. verrucosa was isolated. Treatment with terbinafine was initially given, followed by clarithromycin, but in the absence of any improvement, the patient was readmitted to hospital and is currently on itraconazole and amphotericin B. DISCUSSION: The novel features of our case comprise the clinical aspect of chromomycosis, the extent of the lesions, their unusual site on the back and upper limbs, and the isolation of a rare species, P. verrucosa (only the second observation in Morocco). It also highlights the therapeutic difficulties posed by this type of chromomycosis.

[Successive cutaneous adverse reactions to nilotinib and imatinib in a single patient]. / Toxidermie à l'imatinib suivie d'une toxidermie au nilotinib.

BACKGROUND: Since the advent of targeted molecules, the treatment and prognosis of many cancers, especially chronic myeloid leukemia (CML), have been substantially modified through the introduction of first- and second-generation tyrosine kinase inhibitors. Skin effects constitute the most common adverse effects of these new substances. Although such skin changes are not life-threatening, they can have extensive clinical impact, in some cases leading to discontinuation of treatment. PATIENTS AND METHODS: A 47-year-old woman with no past medical history was followed for chronic phase CML since 26/11/2010 with the presence of the t(9; 22) karyotype. Imatinib (IM) was started at a dose of 400mg/day and haematological response was good. After 4 months of treatment with IM the patient presented with erythematous plaques on both upper limbs and on the oral and vaginal mucosa. These lesions disappeared after discontinuation of IM. The patient was then put on nilotinib 400mg/d and skin lesions reappeared after 3 weeks in the more serious form of erythema multiform with acral distribution, but with no involvement of the mucosa, resulting in immediate cessation of nilotinib. Skin biopsy was consistent with a drug-induced eruption. The lesions disappeared after discontinuation of nilotinib. DISCUSSION: In case of intolerance to IM, a second-generation ITK (dasatinib or nilotinib) may be substituted, and while cross-sensitivities seem infrequent, therapy is problematic in these patients presenting potentially curable blood dyscrasias.

Syringocystadenoma papilliferum: Unusual location.

Syringocystadenocarcinoma papilliferum (SCAP) is rare. We describe a 55-year-old man with a partially eroded lesion on the trunk that developed over one year. Histopathological examination revealed syringocystadenoma papilliferum. A wide excision was performed to remove the tumor. The patient has been well without relapse or metastasis for 8 months.

[Fatal erythroderma in a young Moroccan]. / Erythrodermie fatale chez un jeune marocain.

The purpose of this report is to describe a case involving a young Moroccan who abruptly developed pruritic papulo-vesicular lesions with erythroderma. Secondary development of jaundice and tumoral syndrome lead to diagnosis of an acute form of adult T-cell leukemia/lymphoma associated with HTLV-1 infection. The patient died within three months. To o ur knowledge, this is the first such case reported in Morocco.

[Maxillary bone sarcoidosis]. / Sarcoïdose osseuse maxillaire.

INTRODUCTION: Sarcoidosis is an unexplained systemic granulomatosis. Bone localizations of the disease are rare. We report a maxillary localization. CASE: A 35-year-old patient consulted for facial dysmorphia first observed 2 years before. This swelling was associated to nodular panniculitis lesions and xerostomia. The facial asymmetry was due to maxillary gingival and alveolar swelling. It was associated with scarring in the legs and a purple nodular facial lesion. Lip and jaw biopsies revealed epithelioid and giant cell granulomas without caseous necrosis. The panoramic dental X-ray showed diffuse horizontal alveolar ridge lysis and CT scan revealed an osteolytic lesion of the right maxilla associated to a bone-condensing lesion of the left hemi-mandible. DISCUSSION: The diagnosis of sarcoidosis is made in case of epithelioid and giant cell granulomas without caseous necrosis and the ruling out of other possible diagnoses, including tuberculosis. Bone involvement is rare; face and maxillary localization are extremely rare. The recommended treatment is corticosteroids. Facial remodeling surgery is not recommended.