Caprine nodular thelitis due to Mycobacterium uberis: A series of 26 cases in 11 dairy goat farms in Western France.

Bovine Nodular Thelitis (BNT) is a granulomatous dermatitis of teat skin associated with acid-fast bacilli. A similar condition has been recorded in a dairy goat flock in France recently. The causative agent was shown to be related to the leprosy-causing bacilli Mycobacterium leprae and M. lepromatosis, then sequenced and named M. uberis. Following the initial report in goats, the aim of this study was to investigate new cases of Caprine Nodular Thelitis (CNT) in the same area to confirm the presence of M. uberis by molecular techniques and to get a better description of the clinical signs and of the affected flocks. Twenty-six animals (25 females and 1 male) from 11 flocks were included in the study. Lesions were located on the udder/teat skin (24/25), on the body skin (6/25) or on the scrotum skin (1/1). Udder skin lesions were circular, nodular and/or ulcerate covered with a crust and associated with supramammary lymph node enlargement. Body skin lesions were located at different parts of the body, showed large necrotizing ulcers with undetermined edges and were associated with regional lymph node enlargement. Histopathological results indicated granulomatous dermatitis and lymphadenitis of varying intensity with no acid-fast bacilli seen after Fite-Faraco staining. M. uberis DNA was amplified from 26 samples out of 47 (udder: 11/22; lymph node: 11/20; body: 4/5). The female goats were mostly older than 4 year of age and originated from breeding units characterized by large flock size and high proportion of goat in continuous lactation.

Lymphangiomas: Rare presentations in oral cavity and scrotum in pediatric age group.

The incidence of lymphangioma is 1.2 to 2.8/1000 newborns. They present at birth/before 2 years, with predilection for the head and neck (50%-70%). The buccal mucosa is the second most common site reported (14 cases reported) after the anterior two-thirds of tongue. The scrotum is a rare site with less than 50 cases reported (till 2002). Involvement of vital structures, aesthetic, and functional requirements may necessitate treatments such as surgical excision, radiation, cryotherapy, electrocautery, sclerotherapy, embolization, ligation, and laser. Two rare cases - the first being primary, late-onset buccal lymphangioma, with vesicular presentation, and the second being genital lymphangioma involving the right side of scrotum, thigh, and groin with extension to the left groin - are highlighted.

First-time versus recurrent penoscrotal extramammary Paget's disease: Clinicopathological characteristics and risk factors in 164 Chinese male patients.

BACKGROUND: Penoscrotal extramammary Paget's disease is a rare, slow-growing neoplasm with high frequency of local recurrence. AIMS: The aim of this study was to investigate the difference in clinicopathological characteristics between first-time and recurrent penoscrotal Paget's disease, and to discover the potential risk factors of recurrence. METHODS: Between January 2007 and February 2014, a total of 164 Chinese patients with biopsy-proven tramammary Paget's diseaseex in penis and scrotum underwent wide local resection in our institution. Among them, 142 patients with first-time disease and other 22 patients with recurrent disease were enrolled in this retrospective analysis. RESULTS: The median duration of symptoms was much shorter in recurrent disease than in first-timers (3 vs. 24 months, P < 0.001). Patients with recurrent disease tended to have lower lesion exudation rates (27.3% vs. 51.8%, P= 0.032). In addition, patients with distant stage were more likely to obtain recurrent disease compared with first-time disease (P = 0.005). Through immunohistochemical detection of extramammary Paget's specimen, we found that HER2/neu protein expression in the recurrent group was significantly higher than first-timers (P = 0.036). LIMITATIONS: In this study, the information on familial history of most patients was insufficient. Moreover, due to the lack of follow-up data of our included cases, we were unable to evaluate the prognosis after diagnosis of extramammary Paget's disease. CONCLUSION: Patients with penoscrotal Paget's disease, especially those with shorter duration of symptoms, exudation of lesions, distant-stage, Paget cells infiltrating into adnexa, and HER2/neu expression, should be followed up more carefully after surgery, as they were more likely to suffer recurrence.

Successful treatment of metastatic extramammary Paget's disease with pemetrexed monotherapy systemically and 5-fluorouracil topically.

Advanced extramammary Paget's disease does not have a standardized treatment guideline as its incidence is low and has been rarely reported in literature. Here we describe a case of metastatic extramammary Paget's disease successfully treated with topical 5-fluorouracil (5-FU) and systemic pemetrexed. The therapy was safe without any appreciable adverse effects like diarrhea, rash, neutropenia or fatigue; maintaining remission for more than 6 months. Thus, we propose 5-FU and pemetrexed as the first-line therapy for advanced extramammary Paget's disease, especially for aged patients with unresectable skin lesions.

Acute scrotum: Hansen's disease versus filariasis.

Hansen's disease is caused by Mycobacterium leprae. The disease is known to involve the visceral organs including the testis apart from the skin and nerves in the lepromatous pole of leprosy due to widespread hematogenous dissemination of lepra bacilli. Furthermore, there can be testicular pain during the type 2 reaction in Hansen's disease. Filariasis is a disease caused by the parasitic nematode, Wuchereria bancrofti. This infection most commonly results in lymphedema and secondary vaginal hydrocele with an associated epididymo-orchitis. Acute epididymo-orchitis is either seen in the acute phase or as a part of secondary bacterial infections. The particular interest of this paper is to report the case of Hansen's disease who presented with testicular pain and posed a diagnostic dilemma when his pain did not respond to the standard mode of treatment and an alternate rare diagnosis was sought. This case report also emphasizes the need of reconsideration of diagnosis when the patient is not responding to standard therapy.

Extramammary Paget's disease: analysis of 17 Chinese cases.

BACKGROUND/OBJECTIVE: Extramammary Paget's Disease (EMPD) seems to be more common in Caucasians than Chinese. We report the clinical manifestations, management, and prognostic characteristics in 17 Chinese patients. METHODS: Medical records and biopsies of 17 patients who had been treated at a large university hospital in China between March 2005 and January 2012 were reviewed. RESULTS: Of the 17 patients, 14 were men. They had lesions on the scrotum and the penis. Of the three women, two had vulvar and one had inguinal lesions. All patients underwent Mohs micrographic surgery (MMS). Three men had metastasis to the inguinal lymph nodes and underwent an extensive local excision with inguinal lymphadenectomy. Eight patients who had positive excision margins received additional radiation therapy. The mean follow-up duration was 54 months (4-85 months). One patient had two recurrences. Three had metastasis to the inguinal lymph node. One had metastasis to the bone and concomitant prostate cancer. Two patients died of the disease. CONCLUSION: A striking difference in presentation of EMPD in Chinese compared with Caucasians is the male predominance and location on the penis and scrotum. Mohs micrographic surgery followed by radiotherapy is an effective treatment. Long-term follow-up suggests that the disease has a good prognosis when it does not metastasise.

Pruritic porokeratotic peno-scrotal plaques: porokeratosis or porokeratotic epidermal reaction pattern? A report of 10 cases.

BACKGROUND: Porokeratosis restricted to the genital region is rare with few cases described in the literature. Cases of porokeratosis restricted to the genital region are similar to plaque type of porokeratosis of Mibelli seen elsewhere on the body. We encountered 10 young males with pruritic plaques restricted to the peno-scrotal region, which clinically were not diagnosed as porokeratosis, but on biopsy revealed multiple cornoid lamellae, some of which were seen to arise from eccrine and follicular structures. AIMS: The aim of this study is to study lesions restricted to the peno-scrotal region in males, which on biopsy showed cornoid lamellae suggestive of porokeratosis. METHODS: Retrospective analysis of available data of patients who were rendered a histological diagnosis of genital porokeratosis. The database consisted of biopsies received in private consultation by the first author in the period January 2000 to March 2013. RESULTS: Ten young men, 8 in their third decade, presented with pruritic plaques restricted to the peno-scrotal region of variable duration. The lesions were well-demarcated on the penis, but ill-defined with a rough granular surface on the scrotum. None of patients were diagnosed clinically as porokeratosis. The lesions were poorly responsive to topical steroid/antifungal treatment, but two patients showed partial improvement with oral isotretinoin. Biopsy in nine patients revealed multiple cornoid lamellae involving epidermis (6) and adnexal structures (3). One patient had a single cornoid lamella. CONCLUSION: The clinical and histological presentation of these patients is different from typical genital porokeratosis described in the literature and we postulate that these patients have an unusual porokeratotic reaction pattern of the epidermis with multiple cornoid lamellae.

Phimosis and scrotal lesions in leprosy: two case reports.

We report two cases of borderline leprosy patients, one with scrotal lesions, the other presenting with leprosy on the prepuce with phimosis.