A case with two unusual findings: cutaneous leishmaniasis presenting as panniculitis and pericarditis after antimony therapy

Background Cutaneous leishmaniasis is a parasitic disease caused by a Protozoan. Clinically and histopathologically, it can be confused with various dermatologic diseases. Methods We report a case of cutaneous leishmaniasis (CL) with two unusual findings. A 49‐year‐old male patient presented to our clinic with a 3‐month history of multiple nodules exhibiting arciform arrangement on the lateral side of the left leg. Results Histopathologic examination revealed it as nodular vasculitis. Leishmania smear showed suspicious parasites. Although leishmania culture was negative, PCR was positive for Leishmania. The patient was considered to have CL and was treated with systemic meglumine antimoniate for 14 d. Three days after the end of the treatment, the patient presented to emergency room with a sharp, pleuritic chest pain. He was diagnosed with pericarditis based on clinical and electrocardiogram findings. As other causes of pericarditis were absent, it was thought to be related to antimony therapy. Conclusion The histopathologic presentation of CL as panniculitis is a very rare and this is the first case of pericarditis after the antimony treatment.     

Necrotizing Black Tattoo Reaction

We report the rare case of an 18-year-old man who developed a necrotizing cutaneous reaction 5 days after having a permanent black tattoo on his left forearm spelling his name. Three cases of reactions to permanent black tattoos have been reported within the literature. These cases described the development of cellulitis of the skin adjacent to the tattoo but none reported florid necrotizing cutaneous reactions. The initial management with oral antibacterials failed to resolve the symptoms and use of intravenous antibacterials and topical corticosteroids was needed. Six weeks after presentation the tattoo lettering showed the presence of hyperpigmented skin. Subsequent patch testing confirmed that the patient had no allergy to black tattoo pigments suggesting that the necrotizing cutaneous reaction was secondary to infection. We show that successful treatment of this rare infective complication of permanent black tattoos involves the early institution of intravenous antibacterial agents and topical corticosteroids.     

原发性皮肤毛霉病

报告1例原发性皮肤毛霉病.患者男,57岁.右前臂埋置静脉套管针17 d后局部出现红肿,迅速坏死并扩大.患者患2型糖尿病及慢性肾功能不全.皮肤科检查示右前臂大片坏死区,上覆黑色焦痂及渗出.取黑痂及渗液行真菌镜检,镜下可见大量粗大、无分隔、垂直分枝的菌丝,培养第2天即有棉花糖样菌落生长,初步诊断为皮肤毛霉病.皮损行组织病理检查,PAS染色示真皮及皮下组织可见较多粗大、无分隔菌丝,确诊为皮肤毛霉病.给予两性霉素B脂质体泵入、坏死组织清创术、术后外敷两性霉素B脂质体溶液及外用人成纤维细胞生长因子.患者伤口愈合良好,随访4个月无复发.     

First case of typical Old World cutaneous leishmaniasis treated with miltefosine

A 26-year-old man presented to our clinic for advice on treatment options regarding cutaneous leishmaniasis (CL) with multiple lesions. The biopsy revealed Leishmania amastigotes; Leishmania major was identified by polymerase chain reaction (PCR).(11 )Leishmania serology (indirect immunofluorescence test) was positive at 1 : 160 (normal, < or = 1 : 40). Ten weeks earlier, the patient had spent a 1-week biking holiday in central Tunisia. Two weeks after returning to Germany, he noticed the flaring up of multiple insect bites (> 40) contracted in Tunisia on both arms. The lesions increased in size and axillary lymph node enlargement occurred. Some of the nodular lesions disappeared spontaneously and some increased in size and showed central ulceration. When we first saw the patient, he had six lesions on his right arm and one on his left arm. The largest lesion was on the dorsum of the right hand (Fig. 1) and showed an indurated edge and central ulceration. Before starting systemic treatment, the patient decided to wait a few weeks in case spontaneous improvement occurred. However, 8 weeks later, the ulceration had further increased in size (Fig. 2) from a diameter of 2 cm initially to a diameter of 5 cm. Regional lymphatic spread with palpable nodules along the adjacent lymphatic vessel had occurred. With the patient's informed consent, treatment with oral miltefosine, according to his body weight of 96 kg, was started at 50 mg three times daily for 28 days. The treatment was well tolerated without any subjective side-effects reported. Liver enzymes, serum creatinine, and urea were monitored during treatment. There was a mild increase in liver enzymes during the third week of treatment: aspartate aminotransferase (AST), 49 U/L (normal, < 35 U/L); alanine aminotransferase (ALT), 107 U/L (normal, < 45 U/L); this resolved spontaneously. Ten days after completion of treatment, the rolled edge had disappeared and the central ulcer had almost healed (Fig. 3). Figure 4 shows the lesion 5 months after completion of treatment.     

Kasabach-Merritt Syndrome Arising from Tufted Angioma Successfully Treated with Systemic Corticosteroid

We report a case of Kasabach-Merritt syndrome arising from a tufted angioma successfully treated with systemic corticosteroid. A 2-month-old male infant presented with a palm-sized, erythematous induration on his left pubis. The lesion was diagnosed as tufted angioma histopathologically. After 1 month, the lesion suddenly expanded to the abdomen and scrotum. Initial laboratory tests were consistent with consumptive coagulopathy. He was diagnosed with Kasabach-Merritt syndrome and treated with intravenous dexamethasone at 0.32 mg/kg/day (equivalent to prednisolone 2.0 mg/kg/day). Two days after initiating the treatment, his platelet counts recovered and the lesion ceased to expand. Steroid therapy was converted to oral prednisolone and the dosage was subsequently tapered, and the lesion gradually involuted with no signs of recurrence for a year.     

Nocardia asteroides septicemia in a pemphigus patient. Successful treatment with trimethoprim-sulfamethoxazole and amikacin association

We report the case of a patient with pemphigus who presented Nocardia asteroides septicemia. The infection was controlled with an original association of trimethoprim-sulfamethoxazole and amikacin.     

Tinea Capitis in a Newborn Caused by Two Organisms

Abstract: Tinea capitis is a common infection of childhood. There have been several reports of tinea capitis in newborns. Our patient presented at 19 days of age to the emergency room with a scalp lesion of 5 days duration. The fungal culture grew both Trichophyton rubrum and Trichophyton mentagrophyles. The patient was successfully treated with oral griseofulvin.     

WIDESPREAD PRIMARY CUTANEOUS INFECTION WITH MYCOBACTERIUM FORTUITUM

Background. Cutaneous infection with Mycobacterium fortuitum, a ubiquitous rapid growing atypical mycobacterium, most often occurs as a postsurgical wound complication or at the site of a penetrating injury to the skin. Rarely, disseminated infection with cutaneous involvement can occur in immunocompromised patients. Case Report. A 47-year-old black woman presented with a 10-year history of numerous draining abscesses and tender nodules on the back and buttocks unresponsive to oral and intravenous antibiotics. Biopsy showed a granulomatous and suppurative dermatitis and panniculitis and special stains did not reveal organisms. M. fortuitum was cultured from involved skin on two separate occasions. The patient improved with a 2-week course of intravenous amikacin and cefoxitin combined with oral probenecid followed by a course of doxycycline and ciprofloxacin. Conclusions. Widespread primary cutaneous infection with M. fortuitum may occur in an immunocompetent patient. Chronic draining skin abscesses unresponsive to routine antibiotics may represent infection with an atypical mycobacterium; tissue cultures of affected skin should be performed to rule out this possibility. Therapy should be directed by culture sensitivity results.     

Immunoablative high-dose cyclophosphamide without stem cell rescue in a patient with pemphigus vulgaris

The use of ablative intravenous cyclophosphamide (50 mg/kg per day for 4 days) without stem cell rescue has been described in patients with refractory autoimmune diseases such as paraneoplastic pemphigus, systemic lupus erythematosus, and aplastic anemia. We describe a 33-year-old patient with pemphigus vulgaris recalcitrant to multiple therapies. The patient presented with numerous oral ulcerations, erosions, and hyperpigmented crusted plaques on his face, trunk, and arms. Findings of a skin biopsy and direct immunofluorescence were consistent with pemphigus vulgaris. The circulating pemphigus vulgaris autoantibodies were present at a titer of 1:640. The patient received immunoablative therapy (50 mg/kg of cyclophosphamide for a total of 4 days) and tolerated the regimen well. Complications such as thrombocytopenia and Pseudomonas septicemia were quickly treated. Four months after the 4-day therapy, his oral and skin lesions completely healed, and his pemphigus titers have decreased to zero. He is no longer receiving prednisone and no new lesions have developed. This provides further evidence that this regimen is relatively safe and provides a potential "cure" for refractory autoimmune diseases such as pemphigus vulgaris.     

Hyperimmunoglobulin E syndrome: two cases and a review of the literature

Hyperimmunoglobulin E syndrome (HIES) is a rare immunodeficiency associated with elevated serum IgE levels, eczematous skin, recurrent cutaneous infections, and distinctive musculoskeletal features. We report two cases seen at our institution and review the current literature. Patient 1 was an 18-month-old African American boy with recurrent staphylococcal cold abscesses, pneumonia, and bacteremia. He had severely eczematous skin, ultimately complicated by eczema herpeticum. After treatment of systemic infections with culture-directed antibiotics, a brief course of cyclosporine, 5 mg/kg, improved the dermatitis and allowed transition to long-term therapy with oral trimethoprim-sulfamethoxazole. Patient 2 was a 15-year-old Caucasian boy with long-standing HIES. He has been maintained on a regimen of interferon gamma injections given 3 times weekly and monthly intravenous immunoglobulin since the age of 3 years, prophylactic antibiotics, and low-dose fluconazole. He has occasional episodes of cold abscesses and sinusitis, but has had excellent control since institution of this regimen and has not experienced any adverse effects.     

The allergic emergency – management of severe allergic reactions

Anaphylaxis is characterized by the sudden onset of acute allergic symptoms involving two or more organ systems. An acute allergic emergency is a challenge for physicians due to its life‐threatening potential. The incidence of anaphylactic reactions has increased in recent years. Most frequent elicitors of mast cell and primarily histamine dependent anaphylactic reactions are food, insect venom or drugs. Allergic ­reactions are graded into four groups according to the classification by Ring and Messmer; grade I is defined by the onset of cutaneous symptoms only whereas grade IV is characterized by cardiovascular shock as well as cardiac and/or respiratory arrest. The treatment of allergic reactions should be guided by the severity of the reaction. Initially an intramuscular epinephrine injection into the lateral thigh should be given if cutaneous, mucosal and cardiovascular/respiratory symptoms occur. Additionally, the patient should receive intravenous antihistamines and corticosteroids. For self‐treatment in the case of an allergic emergency, oral antihistamines and corticosteroids should be prescribed to the patient.     

Cutaneous Paecilomyces lilacunis infection in a hospitalized patient taking corticosteroids

A 72-year-old Caucasian man presented with tender, hyperkeratotic, violaceous, ill-defined, scattered plaques associated with minimal pitting edema of the right upper extremity. He had been discharged 3 days earlier from an outlying community hospital after a 33-day admission for exacerbation of chronic obstructive lung disease requiring large doses of systemic corticosteroids. A right subclavian central venous line for intravenous access was placed after he developed a gastrointestinal bleed and myocardial infarction. The clinical differential diagnosis of the eruption on the right upper extremity at the time he presented to our service included cellulitis, deep venous thrombosis, and brachial plexus neuropathy. Brachial plexus neuropathy was ruled out with normal electromyography, nerve conduction studies, and cervical spine radiographs. Deep venous thrombosis was never completely eliminated, because the patient refused a venogram and the venous Doppler exam was nondiagnostic. Treatment for a presumed bacterial cellulitis was initiated with Nafcillin intravenously. As there was clinical disease progression without appreciable improvement by day nine ( Fig. 1 and 2), a skin biopsy was performed and topical Nystatin powder was added for possible yeast etiology. Bacterial cultures were negative; however, fungal cultures on Sabouraud’s agar were positive for Paecilomyces lilacunis after 15 days. Histologic sections demonstrated skin with an essentially normal epidermis. Within the dermis, there was an extensive, mixed, granulomatous inflammatory infiltrate with abundant multinucleated giant cells. Necrosis was not abundant. Occasional 8–12 μm yeast-like organisms were apparent within the cytoplasm of multinucleated cells and in an extracellular location, together with occasional hyphal forms with irregular branching. Periodic acid–Schiff (PAS) with diastase and Gomori methenamine silver accentuated the staining and confirmed the fungal nature of the organisms ( Fig. 3 ). The histologic differential included various other fungi. Cryptococcus is usually smaller, endospores characteristic of coccidiomycosis were not present, and a thick capsule usually seen in blastomycosis was not observed in these organisms. The diagnosis of Paecilomyces lilacunis was established, and itraconazole 200 mg orally twice a day was instituted. Clinical improvement as measured by decreased edema, increased range of motion, and resolution of cutaneous lesions was dramatic during the first week. Itraconazole was decreased to 200 mg a day and the patient continued to improve. He was discharged to home. He has followed up in our clinic every month with complete resolution to date 7 months after discharge.

Figure 1 Open in figure viewer PowerPoint Right upper extremity demonstrating violaceous hue, edema, and scale taken after 9 days of Nafcillin. Note progression of disease to involve the left upper extremity     

Exaggerated insect bite reaction exacerbated by a pyogenic infection in a patient with chronic lymphocytic leukaemia

A 69-year-old woman with chronic lymphocytic leukaemia presented with an 18-month history of pruritic, tender, erythematous plaques. The recurrent lesions simulated insect bites and cellulitis, but failed to respond to appropriate treatments. A recent severe flare was associated with markedly elevated inflammatory markers and swelling of the left leg. The swelling settled rapidly with ibuprofen, leaving cellulitis around a small ulcer infected with Staphylococcus aureus and Streptococcus pyogenes. The cellulitis responded to oral flucloxacillin. Subsequent multiple small exaggerated insect bite reaction lesions settled with oral prednisolone. Eosinophilic cellulitis (Wells' syndrome) was considered unlikely based on the lack of a morphoea-like resolution phase, histological lack of flame figures and presence of vasculitis; however, the distinction is probably artificial.     

Can a simple outpatient‐based treatment be used to treat cutaneous leishmaniasis in young children? A Critically Appraised Topic

A 5‐year‐old boy from rural Afghanistan presented with a 1‐year history of a skin lesion on his left knee, confirmed by polymerase chain reaction to be cutaneous leishmaniasis (Leishmania tropica). Conventional treatment of cutaneous leishmaniasis involves intravenous or intralesional pentavalent antimonials. The aim of this Critically Appraised Topic (CAT) is therefore to determine what alternative effective but less painful treatments (such as oral or topical therapies) can be used to treat cutaneous leishmaniasis in children. Embase and PubMed were searched for ‘cutaneous leishmania*’ AND ‘treatment’ AND ‘children’ in August 2014. All abstracts from April 2008 to August 2014 were reviewed. This search period was chosen to follow on from the Cochrane reviews on Old World and American leishmaniasis. Five randomized controlled trials met our inclusion criteria and have been included in this CAT. The study design and reporting quality in most of the trials included in both Cochrane reviews was found to be poor, and neither Cochrane review investigated the effect of patient age on response to treatment. This CAT identified two nonpainful treatments, topical paromomycin and oral miltefosine, whose effective use in children is supported in the literature. However, both of these treatments are currently unlicensed in the U.K. Our patient was successfully treated with miltefosine 20 mg twice daily for 4 weeks, leading to good resolution of the leishmaniasis plaque but with residual scarring.     

Pyoderma gangrenosum-like lesions in leukocyte adhesion deficiency I treated with intravenous immunoglobulin

A 31-year-old Caucasian male with leukocyte adhesion deficiency I and a 20-year history of recurrent, painful cutaneous ulcerations on the extremities presented with fatigue and worsening pain in both legs. He had experienced minimal improvement in his leg ulcers from treatment with systemic steroids, numerous courses of systemic antibiotics, and brief trials of infliximab and mycophenolate mofetil. He was treated with monthly intravenous immunoglobulin infusions. Upon completion of six courses of intravenous immunoglobulin his ulcerations had nearly healed for the first time in a decade.     

Stevens-Johnson Syndrome Due to Concomitant Use of Lamotrigine and Valproic Acid

Stevens-Johnson syndrome (SJS) is a rare but life-threatening acute mucocutaneous hypersensitivity reaction, usually related to drugs. Severe cutaneous adverse effects such as SJS and toxic epidermal necrolysis can arise during treatment with antiepileptic drugs (AEDs). A 23-year-old female patient with idiopathic epilepsy was referred to the emergency service with fever, oral and genital mucosal lesions, generalized rash, and weakness. She had been taking carbamazepine, valproic acid, and lamotrigine until 1 week prior to referral. Following consultations with a range of clinicians, the patient was diagnosed with SJS related to lamotrigine on the basis of her history and physical findings, and on consideration of current consensus definitions of this condition. The patient was followed up and treated in the emergency intensive care unit with intravenous fluids, antibacterial therapy, and methylprednisolone. After 18 days, the patient was considered to have made a sufficient recovery and was discharged. Patients who undergo therapy with AEDs, particularly new users of these agents, should be informed of and monitored for possible systemic and cutaneous adverse effects of AEDs. Polytherapy should be avoided for as long as possible as this increases the frequency of adverse effects, and treatment should be discontinued if any rash appears.     

Leucemia cutánea en un paciente con leucemia mieloide aguda M2

—The leukemia cutis is the cutaneous infiltration by leukemics cells. Patients with acute myelogenous leukemia (AML) present specific cutaneous involvement in approximately 10% of the cases.We report the case of a leukemia cutis in a 73-year-old male with AML-M2. The patient presented with a one-week history of general malaise, asthenia, cough, dysnea and fever. Physical examination revealed and indurated red-brown plaque on his back of two moths duration, which was pruritic. Concomitance blood and bone marrow findings were diagnostic of AML-M2. A cutaneous biopsy was consistent with leukemia cutis. A CT and a bronchial biopsy showed pulmonary involvement. Since starting chemotherapy the patient had complete remission and the cutaneous lesion cleared, but 15 days later the skin lesion reappeared. A new bone marrow examination revealed recurrent leukemia. He died one month later.     

Sarcoidosis with subcutaneous lesions

A 30-year-old man presented for evaluation of multiple, hypopigmented macules and erythematous papules on his upper and lower lips and upper extremities. He subsequently developed firm, non-tender, subcutaneous, mobile nodules on the left forearm, left calf, and right thigh. Clinical and histopathologic features were consistent with cutaneous and subcutaneous sarcoidosis. Systemic evaluation showed no evidence of extracutaneous involvement. Subcutaneous sarcoidosis is a rare form of nodular cutaneous sarcoidosis. Typical skin findings are painless, firm, mobile nodules without overlying epidermal involvement and with a predilection for the trunk and extremities. Histopathologic alterations include epilthelioid cell tubercles in the subcutaneous fat. As this is a diagnosis of exclusion, other etiologies of granulomatous inflammation must be ruled out with tests and special stains. The mainstay of treatment is with oral glucocorticoids.     

Lichenoid nail changes as sole external manifestation of graft vs. host disease

A 56-year-old-man who had refractory anemia with an excess of blasts underwent an allogeneic peripheral blood stem cell transplantation (PBSCT) from his brother after preparation with melphalan and fludarabin. He received GvHD (graft-vs.-host disease) prophylaxis with cyclosporine from day -1 at a daily dose of 5 mg/kg of body weight. The daily dosage was tapered gradually from day +20. On post-PBSCT day 68 he developed acute cutaneous GvHD grade 3 and acute gastrointestinal GvHD grade 2-3, which was resolved with a daily dose of 1 mg/kg of body weight of prednisone. The patient was discharged in good clinical condition and without signs of GvHD, and he started tapering his immunosuppressive treatment. By day 160 he developed oral lichen planus-like changes, with several reticulate white lesions on the oral mucosa. A biopsy specimen was microscopically consistent with lichenoid GvHD (Fig. 1). By day 150 after PBSCT, when he was being treated with CsA 100 mg once daily and prednisone 10 mg once daily, his fingernails started to grow abnormally and gradually became dystrophic and painful. Two months later his toenails became similarly affected. Although affecting all finger and toe nails, the lesions were especially important in both thumbs. Physical examination revealed multiple findings on his nails (Fig. 2): thickening, fragility, onycholysis, longitudinal striations, and even pterygium. The micological cultures were negative. A biopsy specimen showed an sparse papillary dermis lymphoid infiltrate with focal exocytosis and presence of isolated multiple necrotic keratinocytes (Fig. 3). These findings were interpreted as a lichenoid GvHD with oral and nail involvement. The patient did not have other associated cutaneous lesions. He did not develop signs or symptoms consistent with hepatic GvHD. In May 2000 thalidomide was added to the immunosuppressive therapy, at a daily dose from 100 to 300 mg according to tolerance (constipation, sedation, ...). The lesions on the oral mucous showed a substantial improvement, but the nail changes remained more or less stable. Thalidomide was discontinued after 7 months because the patient displayed numbness and tingling in the hands and feet consistent with a peripheral neuropathy. Twenty days later he stopped taking thalidomide and the oral lichenoid lesions worsened, resulting in difficulty in eating. He also developed periungueal erythema, swelling and intense pain after minimal trauma. The daily dose of prednisone increased to 20-30 mg with moderate improvement. However, the dose could not be increased because of the secondary immunosuppressive effects. Twenty-three months post-PBSCT the patient remains with intense oral and nail lichenoid lesions.