Comparison of outcomes of azathioprine, leflunomide and allergen avoidance in patients with patch test-positive pigmented contact dermatitis: a randomized comparative study.

BACKGROUND: Pigmented contact dermatitis (PCD) is a noneczematous form of allergic contact dermatitis characterized by dermal hyperpigmentation. Allergen avoidance is the cornerstone of therapy, but it is difficult to achieve. The use of immunosuppressives seems rational, but data are lacking. OBJECTIVES: To compare outcomes with azathioprine (AZA), leflunomide and allergen avoidance (AA) in patients with PCD. METHODS: A comparative study was conducted on 28 patients with patch test-positive PCD who were randomly allocated to one of three treatment groups: AZA 2 mg kg-1 daily for 24 weeks + AA (n = 10); leflunomide (LEF) 20 mg daily for 24 weeks + -AA (n = 8); AA alone (n = 10). Patients were followed up for an additional 24 weeks. The Dermal Pigmentation Area and Severity Index (DPASI) score and Hindi Melasma Quality of Life scale (MELASQOL) were used to assess hyperpigmentation and quality of life (QoL). respectively. RESULTS: Hair colorants (n = 12) and paraphenylenediamine (n = 8) were the most common allergens. Mean (SD) DPASI score decreased from 30.97 (3.69), 32.35 (3.90) and 31.86 (3.47) to 13.78 (4.25), 21.67 (2.99) and 20.64 (3.82) at 48 weeks in the three groups, respectively (P < 0.001); the maximum percentage decline was seen with AZA (56%). Mean (SD) MELASQOL score was reduced in the three treatment groups from 48.0 (6.46), 46.75 (3.69) and 46.6 (4.65) to 19.6 (6.98), 24.5 (5.80) and 24.0 (5.49), respectively, at 48 weeks (P < 0.001). Reductions in DPASI and Hindi MELASQOL scores were significantly correlated. The most frequent adverse event was transaminitis in both the AZA and LEF groups. CONCLUSIONS: Patients on AZA achieved a statistically significantly greater reduction in DPASI and MELASQOL score; therefore, AZA may fulfil an unmet need in PCD treatment. An objective reduction in hyperpigmentation was paralleled by an improvement in QoL score, reiterating the need for active management of this disease.

A case of orofacial granulomatosis evolving into Melkersson Rosenthal syndrome in a child treated with a steroid free regimen of Clofazimine and Minocycline.

Orofacial granulomatosis (OFG) is a rare disorder with varied etiological, immunological and infectious mechanisms implicated and is believed to be a umbrella term which includes Melkersson Rosethal syndrome (MRS). We describe a 17 year old female who was diagnosed with OFG and was successfully treated with a combination of minocycline and clofazimine without oral steroids with significant improvement within 1 month of therapy.

Lichenoid drug reaction due to anti-tubercular therapy presenting as erythroderma.

First-line anti-tubercular therapy (ATT) is very effective in management of tuberculosis and is usually well tolerated. Varied spectrum of cutaneous adverse drug reactions is associated with ATT, of which lichenoid drug eruption (LDR) constitutes approximately 10% of the cases. However, LDR presenting as erythroderma is very rare. Here, we report a case of exfoliative dermatitis secondary to LDR which developed after 5 months of ATT.

Microcystic lymphatic malformation in a child and his mother.

A 3-year-old boy presented to us with swelling of the right upper lip and the surrounding perioral area with the multiple clear fluid-filled grouped vesicles on the mucosal surface. The patient's mother had a similar swelling located at the same anatomic location extending to the cheek and the ala of nose on the same side. Magnetic resonance imaging and histopathological examination were suggestive of microcystic lymphatic malformation (LM) in both mother and child. Although an autosomal recessive inheritance pattern has been reported for isolated cystic hygromas, no familial case of microcystic LM has been reported previously.

Exploring the combination of SSKI and topical heparin in a case of erythema nodosum migrans.

Erythema nodosum migrans (subacute nodular migratory panniculitis) is an uncommon type of panniculitis characterized by migrating subcutaneous nodules or plaque on the lower extremity. There are a very few cases of Erythema nodosum migrans reported and thus its appropriate treatment modality is not defined. We describe a case of a 30-year-old male with idiopathic erythema nodosum migrans which was manifest centrifugally spreading, slightly morpheaform erythematous plaque on the lower left leg. The patient was diagnosed initially and treated as a case of furunculosis with poor clinical response. The skin biopsy showed features consistent with subacute nodular panniculitis. Saturated Solution of Potassium Iodide along with topical Heparin successfully treated the patient, when the conventional treatment modalities failed. In a morpheaform centrifugally expanded plaque, erythema nodosum migrans should be kept in mind in the differential diagnosis, especially in the lower extremities in cases of unknown etiology.

Lymphangioma circumscriptum treated with combination of Bleomycin sclerotherapy and Radiofrequency ablation.

Lymphangioma circumscriptum (LC) is a lymphatic malformation presenting with vesiculo-papules or warty lesions, resembling a "frog-spawn". Many treatment modalities have been described but random excision is still often needed. Bleomycin is a safe sclerosant with specific action on the vascular endothelial cells. It has been used extensively in cystic hygromas but its use in LC of the skin has not been documented previously. We present this case with extensive LC of the flank treated with a combination of intralesional bleomycin and radiofrequency ablation in the same sitting. As the lesion was extensive, treatment was done in three sittings, one to each third. Near complete resolution, with no significant recurrence was achieved with a single sitting to each third.

Pheniramine maleate: an apparently safe drug causing bullous fixed drug eruption.

Fixed drug eruption is a delayed type hypersensitivity reaction to a drug seen most frequently with antibiotics such as tetracyclines, sulfonamides, and NSAIDs such as naproxen and ibuprofen. Although H1-antihistamines rarely elicit cutaneous adverse effects, there have been a few reports in the literature implicating them in causing fixed drug eruption, particularly the piperazine derivatives (hydroxyzine, cetirizine, levocetirizine), and loratadine. However, cutaneous drug reactions with the alkylamine derivatives like pheniramine maleate are extremely uncommon and fixed drug eruptions have not been reported with any of the alkylamine antihistamines to date. We herein report a case of multifocal bullous fixed drug eruption following ingestion of pheniramine maleate.

Case Report: Successful Outcome of Primary Infertility with Multidrug Therapy in a Long-Standing Case of Hypogonadism Resulting from Lepromatous Leprosy: Shattering the Dogma of Irreversible Infertility in Leprosy.

We present a case of primary infertility with features of hypogonadism in a male patient with lepromatous leprosy who had remained undiagnosed for 3 years. On investigation, azoospermia and deranged gonadotropin levels with normal serum testosterone were noted and the patient was initiated on multibacillary-multidrug therapy with the primary aim of treating the disease. Although the cutaneous lesions improved within 6 weeks, remarkably infertility was reversed in 2 months-with concomitant normalization of luteinizing hormone, follicle stimulating hormone, and sperm count-an outcome that was unexpected. While reiterating leprosy as a cause of infertility, we discuss the probable mechanism for the efficacy of multidrug therapy in what seemed to be an irreversible outcome of advanced lepromatous leprosy.

Optical damage limit of efficient spintronic THz emitters.

THz pulses are generated from femtosecond pulse-excited ferromagnetic/nonmagnetic spintronic heterostructures via inverse spin Hall effect. The highest possible THz signal strength from spintronic THz emitters is limited by the optical damage threshold of the corresponding heterostructures at the excitation wavelength. For the thickness-optimized spintronic heterostructure, the THz generation efficiency does not saturate with the excitation fluence even up till the damage threshold. Bilayer (Fe, CoFeB)/(Pt, Ta)-based ferromagnetic/nonmagnetic (FM/NM) spintronic heterostructures have been studied for an optimized performance for THz generation when pumped by sub-50 fs amplified laser pulses at 800 nm. Among them, CoFeB/Pt is the best combination for an efficient THz source. The optimized FM/NM spintronic heterostructure having α-phase Ta as the nonmagnetic layer shows the highest damage threshold as compared to those with Pt, irrespective of their generation efficiency. The damage threshold of the Fe/Ta heterostructure on a quartz substrate is ∼85 GW/cm2.

Griscelli Syndrome Type 3 with Coexistent Universal Dyschromia-An Uncommon Association of a Rare Entity.

Griscelli syndrome type 3 is an autosomal recessive disorder caused by mutations in the melanophilin gene and does not have any mucocutaneous or systemic abnormalities other than a pigmentary dilution of skin and hair. We report a case of an 8-year-old girl who presented with silvery grey hair of scalp, eyebrows, eyelashes, and entire body surface with associated universal dyschromia of the skin. After establishing a definite diagnosis of Griscelli syndrome 3, the prognosis was explained and counseling was given. A review of the literature revealed only 27 cases of Griscelli syndrome type 3 in the English language of which only one case by Batrani et al. has reported an associated dyschromia. We report this case to add to the existing literature on this rare condition and to highlight the coexistence of universal dyschromia with Griscelli syndrome type 3.

Congenital Triangular Alopecia Associated with Phakomatosis Pigmentovascularis Type II along with Klippel Trenaunay Syndrome.

Phakomatosis pigmentovascularis (PPV) is characterized by the association of a vascular nevus with a pigmentary nevus and is divided into five subtypes. PPV type II or Happle's phakomatosis cesioflammea is the most common subtype comprising of nevus flammeus along with pigmentary nevus in the form of aberrant Mongolian spots, nevus of Ota or less frequently nevus of Ito. It is estimated that around 50% of patients with PPV have systemic involvement, most frequently involving the central nervous system and eye. Other associated features include vascular abnormalities such as Sturge-Weber syndrome, and klippel trenaunay syndrome (KTS), and cutaneous lesions such as nevus anemicus (most common), cafe'-au-lait macules, generalized vitiligo and congenital triangular alopecia (CTA). There are only four reports of PPV associated with CTA in literature, and only a single previous report with associated KTS and this association has not been reported previously from India. We describe a case of a 30-year-old male having phakomatosis pigmentovascularis type II along with klippel trenaunay syndrome and associated with congenital triangular alopecia.

Papillary tumor in pineal region: Immunohistochemistry meeting the diagnostic challenge.

Papillary tumor of pineal region (PTPR) is extremely rare and poses diagnostic challenge with other central nervous system tumors having papillary architecture. Immunohistochemistry is crucial for a definitive diagnosis of PTPR.

Cutaneous Histoplasmosis: An Unusual Presentation with Nasal Obstruction.

Histoplasmosis is a systemic fungal disease that may be presented with a variety of clinical manifestations, usually as an opportunistic infection in immunocompromised individuals. We present an HIV seropositive patient with a large fleshy growth causing left-sided nasal obstruction, as an unusual presentation. The lesions shrunk dramatically and almost completely on intravenous amphotericin-B lipid complex (ABLC) given for 2 weeks followed by long-term oral itraconazole.

Reverse Koebner's phenomenon in a case of Lymphohemangioma circumscriptum: A diagnostic clue representative of the Whimster hypothesis.

BACKGROUND: Lymphangioma circumscriptum are benign hamartomatous lymphatic growths, diagnosed clinically by the morphology of groups of clear fluid filled to serosanguinous fluid vesicles. MRI is an essential investigation to assess the depth of the lesions. AIM: To report an interesting hitherto unreported observation of Reverse Koebner's phenomenon, which can be used as a new diagnostic technique in Lymphohemagioma. METHOD: Biopsy was taken for the confirmation of the lesion which is an inflicted iatrogenic trauma which showed Reverse koebner's phenomenon in the lesion. RESULTS: Irregular shaped reddish blue painless plaque with rough lobulated surface with discrete firm reddish-blue painless papules, of varying sizes were biopsied. Post biopsy, significant reduction in size and redness of the lesions noticed. CONCLUSION: Disappearance of the lesions on biopsy indicates reminiscensce of the reverse koebner's phenonmenon.

Non-bullous neutrophilic lupus erythematosus-Muted bullous disease?

Non-bullous neutrophilic lupus erythematosus is a rare form of cutaneous lupus erythematosus (LE). We hereby present a case of 24-year-old female, known case of discoid LE (DLE) with negative ANA stabilized on hydroxychloroquine for 2 years. She reported new occurrence of erythematous, mildly pruritic, papular lesions and painful mucosal ulceration. The ANA became strongly positive by ELISA and urine showed proteinuria. A provisional diagnosis of Rowell syndrome was made, skin biopsy was taken, and patient started on steroids. Histopathology showed interface vacuolar change and many neutrophils in the dermis with leukocytoclasia without any bulla formation. The skin lesions responded promptly to addition of dapsone following biopsy report. We conclude that the presence of neutrophils associated with interface pathology on biopsy represents a muted form of bullous LE, especially in patients on immunosuppression. This case highlights the importance of histopathologic examination in the evaluation of any new skin lesions in a patient of lupus on therapy.

Complete Remission in a Patient with Treatment Refractory Bullous Pemphigoid after a Single Dose of Omalizumab.

Bullous pemphigoid (BP) is an autoimmune disorder known to be mediated by immunoglobulin G (IgG) autoantibodies. The role of immunoglobulin E (IgE) antibodies is being investigated as their presence has been described in severe cases. Herein, we report a patient of BP who was refractory to most conventional agents and developed hypotension after rituximab but achieved lasting remission after a single dose of the anti-IgE monoclonal antibody omalizumab.

Poorly differentiated thyroid carcinoma mimicking adenoid cystic carcinoma on aspiration cytology: a case report.

BACKGROUND: Spherical hyaline globules present a characteristic cytologic picture classically seen in salivary gland tumors. This pattern in fine needle aspiration cytology (FNAC) of thyroid malignancies is rare. Sporadic reports of papillary and follicular carcinoma with adenoid cystic pattern are described in the literature. CASE: We report, to our knowledge, the first documented case of poorly differentiated thyroid carcinoma with adenoid cystic pattern on aspiration cytology. An 80-year-old man presented with a thyroid nodule of 3 years' duration. FNAC smears consisted predominantly of cells arranged around spherical hyaline globules resembling adenoid cystic carcinoma. Histopathologic examination showed a poorly differentiated thyroid carcinoma. Immunohistochemically, the tumor cells showed positive reaction for thyroglobulin and cytokeratin. CONCLUSION: Poorly differentiated carcinoma of the thyroid presents a variable cytologic picture. Awareness of the cytomorphologic spectrum reduces the possibility of diagnostic error. Reliance on hyaline globules as the sole criterion results in erroneous diagnosis.