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Kaposi Varicelliform Eruption in a Patient with Pemphigus Vulgaris: a Case Report and Review of the Literature

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Kaposi Varicelliform Eruption in a Patient with Pemphigus Vulgaris: a Case Report and Review of the Literature Hindawi Case Reports in Dermatological Medicine Volume 2020, Article ID 6695342, 8 pages https://doi.org/10.1155/2020/6695342 Case Report Kaposi Varicelliform Eruption in a Patient with Pemphigus Vulgaris: A Case Report and Review of the Literature Bibisha Baaniya and Sudha Agrawal Department of Dermatology and Venerology, B.P. Koirala Institute of Health Sciences, Dharan, Nepal Correspondence should be addressed to Bibisha Baaniya; [email protected] Received 30 October 2020; Revised 19 December 2020; Accepted 21 December 2020; Published 31 December 2020 Academic Editor: Jacek Cezary Szepietowski Copyright © 2020 Bibisha Baaniya and Sudha Agrawal. *is is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Haemorrhagic crusted lesions over pre-existing pemphigus vulgaris erosions should arouse suspicion of Kaposi varicelliform eruption (KVE). Immediate treatment with antivirals helps to prevent mortality and morbidities. Here, we report a case of a 67-year-old male who developed haemorrhagic crusted lesions on pre-existing pemphigus lesions during his hospital stay and obtained almost 90% resolution of cutaneous lesions of Pemphigus vulgaris as well as Kaposi varicelliform eruption within 2 weeks of acyclovir treatment along with the continuation of systemic steroids. We also highlight the review of the literature of other reported cases with its management. 1. Introduction high as 50%; now with specific acyclovir treatment, it is around 10% [6]. Here, a case of pemphigus vulgaris with Pemphigus vulgaris (PV) is an autoimmune intraepidermal KVE is being presented along with the review of the liter- blistering disorder of the skin and mucous membrane ature of other reported cases with its management. caused due to damage to intercellular adhesion structures, i.e., desmogleins resulting in acantholysis [1]. Kaposi vari- 2. Patient Information celliform eruption (KVE) is a rare and potentially fatal viral infection typically due to herpes simplex virus (HSV) 1 or 2, A 67-year-old male had presented to Dermatology OPD of rarely by coxsackie A16 virus and vaccinia virus over a pre- our hospital 8 months back with a history of oral erosions for existing dermatosis [2]. *is phenomenon has been well 2 months and cutaneous erosions for 1 month and was studied in atopic dermatitis; hence, alternatively, KVE is also diagnosed as pemphigus vulgaris (Figure 1). *e patient was called eczema herpeticum. However, it is only rarely known advised to take an oral steroid 60 mg daily along with other to occur in patients with pemphigus vulgaris [3]. supportive treatments. In spite of proper counselling of the *e mechanisms underlying the pathogenesis of KVE prognosis and long-term follow-up of pemphigus, he was remain incompletely understood. It is assumed that the taking prednisolone on and off and was unable to come for defective skin barrier, acting in conjunction with immune the follow-up because of the COVID-19 pandemic. deficiencies (both cell-mediated and humoral immunity), is implicated [4]. Furthermore, treatment of these patients with systemic immunosuppressants also compromises the 2.1. Investigations and Treatment. *e patient presented integrity of the immune system [3]. again 5 weeks before the admission with multiple erosions Antiviral therapy with acyclovir is the treatment of that extended to the anterior chest and was advised to choice, which is usually combined with a systemic antibiotic continue prednisolone 50 mg once daily, azathioprine to control heavy bacterial colonization [5]. Before the advent (100 mg once daily), doxycycline (100 mg once daily), and of acyclovir therapy, the mortality in cases of KVE was as nicotinamide (500 mg daily) for 5 weeks. 2 Case Reports in Dermatological Medicine Figure 1: 8 months prior to admission: hyperkeratotic plaque and erosions. However, he failed to show satisfactory improvement to follow up via teledermatology consultation during the (Figure 2), and his swab for bacterial culture and sensitivity COVID-19 pandemic as he came from remote hilly area. revealed Pseudomonas aeruginosa sensitive to piperacillin; hence, he was admitted to the dermatology ward and started on 3. Discussion the same antibiotic and intravenous, dexamethasone 8 mg once daily along with skin care. After receiving these medications for Austrian dermatologist Moriz Kaposi first described KVE or 2 weeks, once the pseudomonas infection resolved, dose of eczema herpeticum. Some authors, however, define these dexamethasone was increased to 16 mg over the next 2 weeks, two terms differently: eczema herpeticum as disseminated and the patient got significant improvement. However, 1 week HSV infection over pre-existing eczematous skin disease and after initiating steroid dose escalation, the patient developed KVE as any disseminated cutaneous infection with HSV type few monomorphic haemorrhagic crusts on the cheeks over- 1 or 2 [5]. lying the pre-existing lesions (Figure 3), which progressed over After an incubation period of about 3–10 days, KVE the next week to involve bilateral cheeks, malar region, and presents with disseminated eruption of closely grouped, bridge of the nose with few lesions in the forehead, bilateral painful, monomorphic, umbilicated vesicles, accompanied ears, and chest. *ese lesions were associated with extensive by fever, malaise, and regional lymphadenopathy. *e cutaneous pain and myalgia (Figure 4). vesicles tend to evolve rapidly to pustules or dry out, forming Although he denied past history of herpes infection, his crusts over eroded areas during the course of the disease. *e HSV-1 IgM and IgG also came out to be positive and hence eruption is frequently located on the head, neck, and the was diagnosed as KVE. *en, he was immediately started on upper part of the trunk and usually heals within 2 to 6 weeks oral acyclovir 400 mg thrice daily, and dexamethasone was [2]. In our case, one week prior to developing haemorrhagic tapered rapidly to 9 mg from 16 mg once daily over a week crusts over pre-existing erosions, another patient residing (Figure 5). Further ophthalmological consultation was done, next to our patient had developed herpes labialis and was and herpes keratitis was ruled out. Within 1 week of the started on oral acyclovir 400 mg thrice daily. On inquiring, treatment, the crusts decreased significantly, and treatment our patient denied any previous herpes infection; hence, we was continued. *e erosions also healed rapidly thereafter. concluded it to be the primary infection he had acquired His comorbidities were type 2 diabetes mellitus, grade 2 from the nearby patient. We could not keep our patient in benign prostatic enlargement, right nephrolithiasis, and isolation due to logistic constraints secondary to ongoing osteopenia. COVID-19 pandemic. Recurrent episodes of KVE may occur [7]. Secondary bacterial infection, viremia, and multiorgan involvement are 2.2. Outcome and Follow-Up. Once there was 90% resolution the important causes for mortality; and hence, KVE is a in the haemorrhagic crust, the patient was discharged on dermatological emergency. Ocular complications such as 60 mg prednisolone and oral acyclovir 400 mg thrice daily keratitis, conjunctivitis, blepharitis, uveitis, and loss of vision (Figure 6). Oral acyclovir was continued with the same dose can also occur [2]. In our patient, herpes keratitis was ruled for a total of 4 weeks until almost 100% improvement was out after ophthalmological consultation. Here, we list the achieved (Figure 7). After that, monthly tapering of the dose case series and reports of KVE with pemphigus vulgaris of oral prednisolone was done, and the patient was instructed (Table 1). Case Reports in Dermatological Medicine 3 Figure 2: Day 1 of admission: extension of erosions despite high-dose oral steroids. Figure 3: Day 20 of admission: improvement of erosions but appearance of haemorrhagic crusts. Figure 4: Day 23 of admission: extensive haemorrhagic crusts in the face and few in the periphery of erosions in the chest. 4 Case Reports in Dermatological Medicine Figure 5: Day 27 of admission: further extension of haemorrhagic crusts. Figure 6: 90% resolution of lesions within 2 weeks of commencing oral acyclovir. Figure 7: Almost 100% improvement within 4 weeks of commencing antiviral treatment. Case Reports in Dermatological Medicine Table 1: Case series and reports of KVE with pemphigus vulgaris. Morphology of Age Treatment of pemphigus SN Author/year pemphigus Morphology of KVE Investigations Treatment of KVE Outcome of KVE (years)/sex vulgaris vulgaris Polymerase chain reaction Ganciclovir (PO) Mycophenolate mofetil, Resolved (time to Focal lesions with (PCR), skin swabs, viral 44.1/male Not mentioned (dose and duration prednisone (modification not resolution not oral involvement culture, direct not mentioned) mentioned) mentioned) immunofluorescence Widespread Acyclovir (IV) (dose Resolved (time to 51.6/ PCR, skin swabs, viral culture, Intramuscular corticosteroids lesions with oral Not mentioned and duration not resolution not female direct immunofluorescence (modification not mentioned) involvement mentioned) mentioned) Focal lesions with Valacyclovir Resolved (time to 70.2/ oral, conjunctival, PCR, skin swabs, viral culture, Azathioprine, prednisone Lehman and Not mentioned hydrochloride 1 gm resolution not female perianal direct immunofluorescence (modification not mentioned) 1 el-Azhary
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