[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.

[Clinical characteristics of nail lichen planus and follow-up: a descriptive study of 20 patients]. / Caractéristiques cliniques et évolutives du lichen plan unguéal : étude descriptive de 20 patients.

BACKGROUND: Nail lichen planus (NLP) occurs in 10% of patients with disseminated disease but it can also occur in isolation. The outcome of treatment is usually disappointing and data about its long-term prognosis are lacking. STUDY AIM: To study clinical features, response to treatment and follow-up of a series of 20 patients with NLP. PATIENTS AND METHODS: A descriptive study was conducted of 20 patients with histologically confirmed NLP seen at consultations for nail disorders between September 2009 and April 2013. RESULTS: The mean age was 35 years (9-56 years) with no gender preponderance. Forty percent of patients were children. Stress was an evident triggering factor in 20% of patients. The mean duration was around 48 months (2 months to 10 years). Only 25% of patients had extra-ungual lesions. All 20 nails were affected in 55% of patients. Nail matrix involvement was observed in 85% of cases and 55% had nail-bed involvement. Twenty-five percent of patients were presenting severe involvement such as pterygium or anonychia. Intramuscular corticosteroids were given to 10 patients. In 80% of patients, the NLP was limited or regressed rapidly from the third injection. The average follow-up was 24 months. DISCUSSION: Our study highlights the frequency of paediatric forms and of often aesthetically unacceptable nail scarring. Early diagnosis of NLP, notably in children, would allow initiation of adequate treatment that could perhaps limit the risk of such sequelae.

[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.

[Acrodermatitis enteropathica-like disease in a full-term exclusively breast-fed infant]. / Pseudo-acrodermatite entéropathique chez un nourrisson né à terme, exclusivement nourri au sein.

UNLABELLED: Symptomatic zinc deficiency can occur in exclusively breast-fed infants. We report a case in a 5-month-old infant. CASE REPORT: A 5-month-old exclusively breast-fed boy presented with persistent erythema of breech since the age of 2 months as well as perioral and acral erythematous scaly lesions. Laboratory investigations revealed low zinc levels in the infant's serum, the mother's plasma zinc level was normal, and breast milk zinc was low. Rapid healing occurred after oral zinc supplementation. DISCUSSION: Zinc deficiency in breast-fed infants is a rare disease caused by a low level of zinc in the mother's milk. The clinical features resemble those of acrodermatitis enteropathica. Oral zinc supplementation is required until weaning.

[Renal ultrasound in fat necrosis]. / Intérêt de l'échographie rénale dans la cytostéatonécrose sous-cutanée.

Subcutaneous fat necrosis is an uncommon disease that may be complicated with potentially fatal hypercalcemia or with nephrocalcinosis. We report on the case of a patient with a history of significant perinatal asphyxia, hospitalized for a urinary tract infection. Lesions of subcutaneous fat necrosis were noted, with asymptomatic hypercalcemia at 3.9mmol/L. A renal ultrasound was performed and showed echogenic medullary pyramids bilaterally, consistent with nephrocalcinosis and left nephrolithiasis. The treatment of hypercalcemia included hyperhydration, a diuretic and corticosteroids. Progression was characterized by the total regression of skin lesions and normalization of serum calcium. Hypercalcemia is a rare complication of subcutaneous fat necrosis. It develops within days to weeks after the appearance of skin lesions. Nephrocalcinosis appears after several weeks or months. Hypercalcemia must be treated in due time to avoid the impact on the kidney.

[Purpura fulminans, venous thrombosis and constitutional thrombophilia in an infant]. / Purpura fulminans et thrombose veineuse compatible avec une thrombophilie constitutionnelle chez un nourrisson.

The association of idiopathic purpura fulminans (PF) and venous thrombosis (VT) seldom reveals constitutional thrombophilia in an infant. We report a case of PF in an 18-month-old infant. Laboratory tests showed disseminated intravascular coagulation (DIVC) with normal rates of C and S proteins and antithrombin. The echo-Doppler examination conveyed venous thrombosis of the lower limbs, while the genetic study showed heterozygous mutation of Factor II (G 20210A). Precocious and multidisciplinary management included frozen fresh plasma supplementation and necrosectomy with skin grafts. The diagnosis and therapeutic problems posed by PF combined with deep venous thrombosis are discussed.

[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.