Diagnostic Value of Histological Analysis of Punch Biopsies in Suspected Cutaneous Buruli Ulcer: A Study on 32 Cases of Confirmed Buruli Ulcer in Cameroon.

BACKGROUND: Buruli ulcer (BU) is a cutaneous infectious disease caused by Mycobacterium ulcerans. In this prospective study, we aim to clarify the main histopathological features of cutaneous BU based on 4-mm skin punch biopsies and to evaluate the diagnostic value of this method. METHODS: Between 2011 and 2013, a prospective study was conducted in Cameroon. Dry swabs from ulcerative lesions and fine-needle aspirates of nonulcerative lesions were examined for Ziehl-Neelsen (ZN) staining, followed by PCR targeting IS2404 and culture. Two 4-mm punch biopsies were performed in the center and in the periphery of each lesion. RESULTS: The 364 patients included in the study had 422 lesions (381 were ulcerative and 357 lesions were biopsied). Among the 99 ulcerated lesions with a final diagnosis of BU, histological features for BU were fulfilled in 32 lesions. 32/32 showed subcutaneous necrosis with a neutrophilic inflammatory infiltrate. 26/32 presented alcohol-resistant bacilli confirmed by ZN stain on histology. CONCLUSION: Punch biopsies help in establishing the correct diagnosis of BU and also in the differential diagnosis of chronic ulcers. The main histological feature for BU is diffuse coagulative necrosis of subcutaneous tissue, with acid-fast bacilli detected by ZN stain.

Ingenol Mebutate for Recalcitrant Chronic Actinic Cheilitis.

We present the case of a healthy 76-year-old man with a whitish, hyperkeratotic lesion of the lower lip diagnosed as actinic cheilitis (AC) previously treated with classic red light photodynamic therapy 5 years ago. Initial treatment with 5% imiquimod cream - also with intensified application - failed. After 2 cycles thrice daily, consecutive applications of 150 µg/g ingenol mebutate gel at 3 weeks' interval, the lesions cleared completely. Surprisingly, no pustular or crusting reaction or other side effect occurred contrary to expectation. Remission was stable for 10 months, when recurrence occurred. Ingenol mebutate proved to be a feasible and safe treatment in this otherwise refractory case of AC.

Clinical features and management of a severe paradoxical reaction associated with combined treatment of Buruli ulcer and HIV co-infection.

BACKGROUND: In West and Central Africa Buruli ulcer (BU) and HIV co-infection is increasingly recognised and management of these two diseases combined is an emerging challenge for which there is little published information. In this case we present a severe paradoxical reaction occurring after commencing antibiotic treatment for BU combined with antiretroviral therapy for HIV, and describe its clinical features and management. This includes to our knowledge the first reported use of prednisolone in Africa to manage a severe paradoxical reaction related to BU treatment. CASE PRESENTATION: A 30 year old immunosuppressed HIV positive man from Cameroon developed a severe paradoxical reaction 24 days after commencing antibiotic treatment for BU and 14 days after commencing antiretroviral therapy for HIV. Oral prednisolone was successfully used to settle the reaction and prevent further tissue loss. The antiretroviral regimen was continued unchanged and the BU antibiotic treatment not prolonged beyond the recommended duration of 8 weeks. A second small local paradoxical lesion developed 8 months after starting antibiotics and settled with conservative treatment only. Complete healing of lesions occurred and there was no disease recurrence 12 months after commencement of treatment. CONCLUSIONS: Clinicians should be aware that severe paradoxical reactions can occur during the treatment of BU/HIV co-infected patients. Prednisolone was effectively and safely used to settle the reaction and minimize the secondary tissue damage.

[Heparins and skin reactions: from diagnostic to management]. / Héparines et réactions cutanées: du diagnostic a la prise en charge.

The use of heparins in the prophylaxis and treatment of thromboembolic diseases shows non-negligible risks of cutaneous lesions, which are very frequently underestimated by physicians. As this risk often originates from either delayed-type hypersensitivity reactions or life-threatening heparin-induced thrombocytopenia, a quick diagnosis and an adapted management are thus crucial. Furthermore, as most cases are emergencies, allergologic testing cannot be considered in the first place. In this context, an overview of the therapeutic as well as management recommendations is presented, taking into account the most recent treatments.

Dermoscopy of desmoplastic trichoepithelioma reveals other criteria to distinguish it from basal cell carcinoma.

BACKGROUND: Desmoplastic trichoepithelioma (DT) is a rare benign adnexal neoplasm considered to have follicular differentiation. It usually presents as an asymptomatic, firm, annular plaque with a raised border. The diagnosis of DT is based on clinical and histological features which can be similar to those of morpheiform basal cell carcinoma. Taking this into consideration, the use of another diagnostic technique would be very useful. Dermoscopy is a noninvasive diagnostic technique allowing a more accurate diagnosis. OBJECTIVES: To differentiate DT and morpheiform basal cell carcinoma by means of dermoscopy. METHODS: Here, we describe the dermoscopic features of a case of DT, and make a direct clinicopathological correlation by using the horizontally cut slides provided by Mohs micrographic surgery. RESULTS: On dermoscopy, DT shows well-defined borders and an ivory-white color, as well as prominent arborizing telangiectasias in the central area and on the right side. There are no leaf-like structures and no ovoid nests. This observation confirms and completes the first dermoscopic analysis of DT reported in the literature so far.

Linear sclerodermic lupus erythematosus, a distinct variant of linear morphea and chronic cutaneous lupus erythematous.

BACKGROUND: Overlap syndromes represent disorders that combine diagnostic criteria of two or more different connective tissue diseases. METHODS: We herein describe the case of a 34-year-old patient. RESULTS: Our patient developed a lesion on the scalp and forehead following Blaschko's line typical for linear morphea "en coup de sabre", while histopathological features were consistent with both chronic cutaneous lupus erythematosus and linear morphea, a cutaneous overlap syndrome previously described as linear sclerodermiform lupus erythematosus. The patient was given oral antimalarials in association with topical steroids and calcineurin inhibitors with good response. CONCLUSIONS: Knowledge of this peculiar cutaneous overlap syndrome is warranted, since its management and prognosis is probably different from classical linear morphea.

Primary atrophic solitary morphea profunda.

Solitary morphea profunda (SMP) is a distinct form of localized scleroderma, a cutaneous disorder of unknown origin. Here, we describe a patient presenting with noninflammatory cupuliform depressed plaques, without any significant skin induration, pigmentation or texture change, that appeared on the left shoulder at a site of previous intramuscular vaccination. Light microscopy studies revealed excessive dermal collagen deposition with thickened hyalinized collagen bundles. Magnetic resonance imaging studies demonstrated tissue fibrosis with thinning of the subcutaneous fat tissue and involvement of the adjacent deltoid muscle, which was confirmed by electromyographic tests. Borrelia serology was negative. Our observation of SMP differed from previously described cases, since it mimicked localized lipoatrophy. Our observation highlights the wide spectrum of clinical presentations of localized scleroderma. The latter should be considered in the presence of lipoatrophy-like lesions for proper workup and therapy.

An illustrative case of Muir-Torre syndrome: contribution of immunohistochemical analysis in identifying indicator sebaceous lesions.

BACKGROUND: Muir-Torre syndrome (MTS) is an autosomal dominant genodermatosis characterized by the association of at least 1 cutaneous sebaceous tumor and 1 internal malignancy, often arising in the gastrointestinal tract. It is secondary to germline mutations in DNA mismatch repair genes, mainly MLH-1 and MSH-2. OBSERVATIONS: We report the case of a 54-year-old man with a 2-year history of skin-colored papules clinically reminiscent of large sebaceous hyperplasias on the nose and back, but histologically diagnosed as sebaceous adenomas and epitheliomas. His family history was positive for colon cancer in the mother and 2 brothers. A colonoscopy done during the hospitalization revealed 2 sessile polyps in the left colon, both showing a low-grade dysplasia on the biopsy specimen. Immunohistochemical staining performed on the cutaneous and colic biopsy specimens revealed a lack of expression of MSH-2 and MSH-6. Genetic testing revealed microsatellite instability in the colon and cutaneous tumors. CONCLUSION: The immunohistochemical testing for MSH-2, MSH-6, and MLH-1 is useful for rapid identification of an underlying mismatch repair defect and early diagnosis of MTS.

Histopathologic correlation in dermoscopy: a micropunch technique.

BACKGROUND: Dermoscopy is a simple-to-use, in vivo method for the diagnosis of malignant melanoma and the differential diagnosis of pigmented skin lesions. It uses an immersion technique and optical magnification to visualize structures not visible to the naked eye. The anatomoclinical correlation of dermoscopic with histopathologic findings is important, and while many articles have described different techniques to achieve this goal, no direct correlation with a visual control has been described. We recently developed a micropunch technique that allows for the first time this direct correlation. OBSERVATIONS: After applying local anesthesia, the physician makes a superficial round incision using a 1-mm micropunch in the area of interest and leaves the punch in place. The lesion is documented using digital dermoscopy before and after surgery. Using these images, the laboratory technicians can easily visualize the precise site of the punch and its correlation with the initial dermoscopic image, and the sections are chosen in a way that they pass through the punch incision. The punch incision can be easily identified in the histopathologic slides because of its clear-cut borders. Since the punch always stays in place, this technique does not interfere with the interpretation of the slides (eg, measurement of the Breslow thickness). CONCLUSIONS: The advantages of our technique are that it is easy to perform by any clinician in any setting after a short setup and training period for the clinician and the laboratory technicians. Unlike with other techniques, the physician need not be present at the laboratory at the moment of the step sectioning. It can be performed in private practice and for many other indications besides pigmented skin lesions. Finally, since this technique allows for the first time a direct correlation between dermoscopic and histopathologic findings, the clinician will be able to "guide" the pathologist and indicate the precise areas of interest or suspicion.

Hyaluronate accumulation and decreased CD44 expression in perifollicular solitary cutaneous myxoma.

BACKGROUND: Myxomas are rare cutaneous tumors which may be solitary or associated with Carney's complex, NAME or LAMB syndromes. The mucinous material which constitutes the stroma of cutaneous myxomas is predominantly composed of hyaluronate (HA), the major component of the extracellular matrix. CD44 is a polymorphic integral membrane glycoprotein which serves as the principal cell surface receptor for HA. OBJECTIVE AND METHODS: Here we present 2 cases of solitary cutaneous myxomas displaying microscopically a perifollicular localization, in which we explored the nature of the accumulated mucinous material by colloidal iron and HA-binding protein stainings, as well as the epidermal expression of CD44 protein by immunohistochemistry. RESULTS: We show that HA is accumulated in the stroma of the cutaneous myxoma lesions and that the protein expression of CD44 in the keratinocytes of the trichofolliculoma-like epithelial buds projecting from the hair follicle centering these lesions is significantly decreased. CONCLUSIONS: Our results suggest that a decrease in CD44 expression in follicular epithelial proliferations may be correlated with an abnormal HA accumulation in cutaneous myxoma.