A 50-year-old man, a known case of human immunodeficiency virus infection for the past 1 year, was on antiretroviral therapy in the form of stavudine, lamivudine, and nevirapine. Three days after replacing stavudine with tenofovir, he developed redness on the face and neck and within 48 h the rash became generalized. Dermatological examination revealed involvement of photoexposed areas of the face in the form of erythema and ill-defined hyperpigmented plaques, with mild periorbital edema. There was specific involvement of V and nape of the neck. Extensive erythema and scaling were also present on buttocks, thighs, and upper third of legs. A diagnosis of photoallergic dermatitis to tenofovir was considered and confirmed by histopathology and photopatch test. He responded well to the stoppage of the drug and oral corticosteroids. This is the first report of a photoallergic reaction to tenofovir in the literature.
Drug reaction with eosinophilia and systemic symptoms (DRESS) is a severe adverse cutaneous drug reaction with mortality up to 10%. It is a rare condition with risk varying between 1 in 1000 and 1 in 10 000 drug exposures. The aim of the study was to describe clinical features, management and drugs responsible for causing DRESS. The study was retrospective, observational study. The data of patients admitted to hospital with diagnosis of DRESS during study period (March 2018 to February 2020), were retrieved and analyzed. The descriptive data of patients were summarized. The continuous variables were summarized as mean ± SD and/or median, depending on the skewness of the data. The categorical variables were expressed as absolute numbers, frequency, and proportions (%). The data was tabulated and analyzed in Microsoft Excel 2019 version. A total of 20 patients who met inclusion criteria (probable or definite DRESS as per RegiSCAR criteria) were included in the study. The mean age of the patients was 41.2 ± 15.7 years. The average latency period was 26.45 ± 5.65 days (range: 7‐60). The commonest culprit drugs were dapsone and phenytoin, each in five (25%) patients. Commonest morphology of rash was morbilliform in 13 (65%) patients. One patient with targetoid rash had multi‐organ involvement. Facial edema, periorbital edema, and conjunctival injection were seen in 17 (85%), seven (35%), and six (30%) cases, respectively. Eosinophilia was present in 18 (90%) patients with mean (±SD) value of 1976 ± 840 cells/μl. Liver was the commonest internal organ involved in 14 (70%) patients and kidney in three (15%) patients. The initial dose of prednisolone for treatment varied from 0.75 to 2 mg/kg/day. The mean duration of steroid treatment was 64 ± 21 days. Two patients were treated with intravenous methylprednisolone and one with intravenous immunoglobulin. Two patients (10%) had recurrence of adverse drug reaction >6 months after completion of initial treatment and two (10%) developed autoimmune thyroiditis during follow‐up. Small sample size and retrospective nature of the study were main limitations. Selection bias is a possibility as study was carried out in tertiary care center. Tests for incriminating culprit drugs such as patch test, intradermal test, and lymphocyte transformation test were not performed. DRESS is a rare disease that can be diagnosed early with high index of suspicion and treated successfully with steroids. The internal organ involvement is common in DRESS and requires a thorough evaluation.
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.
investigation, the median age of HGPS diagnosis was 3 month, much earlier than 2.9 years reported in the United States and 2.6 years in Europe. 2 We found the facial dysmorphism, sclerotic skin, and a 'protuberant bikini sign' are the most valuable clue for early clinical diagnosis, and could help to facilitate first sight recognition. The significant 'protuberant bikini sign' was first reported in our article and might also help to make differential diagnosis with other reasons of sclerotic skin such as sclerema neonatorum.Some of the manifestations were not commonly observed as literature described, such as loss of scalp hair, dystrophic nails, cardiovascular and endocrine complications. 4,5 These could be explained as the clinical manifestation gradually progresses depending on ages, and the evaluation ages of our patients were too young to develop these complications.Skin histopathology is not helpful in diagnosis. LMNA sequencing is essential when HGPS is suspected. The c.1824C>T mutation is most frequent that refers to classic type HGPS, 4 as in 66.7% of our cases. The atypical c.1968+1G>T mutation in case 3 results in the most severe manifestation and the only death among these patients. It is also the second report about this mutation that causes severe progeroid syndromes. 6 In case 6, the atypical heterozygous IVS8, À4 C>A was a novel mutation in the Human Gene Mutation Database (HGMD).In conclusion, HGPS have characteristic clinical features that facilitate first sight recognition, and LMNA gene mutation is necessary for early diagnosis. A detailed multi-system examination with close monitoring is important to extend the lifespan.
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