Recurrent Bullous Scabies - Journal of Cutaneous Medicine and
Transcription
Recurrent Bullous Scabies - Journal of Cutaneous Medicine and
CASE REPORT Recurrent Bullous Scabies David Serra, José Pedro Reis, Angelina Mariano, Álvaro Machado, and Américo Figueiredo Background: Scabies is a common infestation that usually presents as a pruriginous eruption consisting of erythematous papules, nodules, and excoriations. Diagnosis relies mostly on clinical features, including the identification of scabietic burrows. Several atypical clinical manifestations have been reported; bullous presentations are particularly rare and intriguing. Objective: A case of recurrent bullous scabies is reported. Methods and Results: An 87-year-old institutionalized woman developed a pruriginous eruption that mimicked bullous pemphigoid. Histopathologic examination revealed blistering at the subepidermal level and a mixed inflammatory infiltrate with numerous eosinophils. Scabies mites were identified on dermoscopy and on skin scrapings, allowing a diagnosis of bullous scabies. Despite effective treatment with topical lindane, failure to control scabies in her institution led to recurrences of her peculiar blistering infestation. Immunofluorescence studies were always negative. Conclusions: These observations underscore the mite’s role in the pathogenesis of blister formation and illustrate current challenges in scabies’ diagnosis and management. Antécédents: La gale sarcoptique humaine est une infestation qui se présente habituellement sous forme d’éruption prurigineuse, caractérisée par des papules érythémateuses, des nodules, et des excoriacions. Le diagnostic se fonde surtout sur les signes cliniques, notamment l’identification de sillons. Plusieurs manifestations cliniques atypiques ont été rapportées; les manifestations bulleuses sont particulièrement rares et étranges. Objectif: Rapporter un cas de gale bulleuse. Méthodes et résultats: Une femme de 87 ans, résidant dans un établissement de soins, a développé une éruption prurigineuse évocatrice d’une pemphigoı̈de. L’examen histopathologique a révélé des vésications de l’épiderme et un infiltrat inflammatoire avec de nombreux éosinophiles. Une dermoscopie et un grattage cutané ont permis d’identifier des mites ce qui a mené au diagnostic de gale bulleuse. Malgré le traitement efficace au lindane topique, le manque de contrôle de la gale dans l’établissement a produit une récurrence de l’infestation. Les tests d’immunofluorescence ont tous été négatifs. Conclusions: Ces observations soulignent le rôle des mites dans la pathogenèse des formations bulleuses et illustrent les défis actuels auxquels font face les médecins dans le diagnostic et la gestion de la gale. E REPORT A CASE of recurrent bullous scabies in an 87-year-old institutionalized woman. She presented for the first time at our department with a 2-month history of a pruritic eruption. Erythematous papules, along with excoriations and some blisters, were seen on examination. Skin scraping examination confirmed scabietic infestation, and topical lindane was prescribed. She W From the Dermatology Department, University Hospital of Coimbra, Coimbra, Portugal. Presented as an oral free communication at the 17th Congress of the European Academy of Dermatology and Venereology, Paris, France, September 17–20, 2008. Address reprint requests to: David Serra, MD, Dermatology Department, University Hospital of Coimbra, Praceta Prof Mota Pinto, 3000-075 Coimbra, Portugal; e-mail: [email protected]. DOI 10.2310/7750.2010.09028 # 2010 Canadian Dermatology Association missed her follow-up appointment and was reobserved only 3 months later. At that time, she presented a blistering, pruriginous eruption mimicking bullous pemphigoid, with tense bullae, erosions, and crusts (Figure 1). No scabietic burrows were found. Skin biopsy revealed blistering at the subepidermal level, with an inflammatory infiltrate in the upper dermis composed of neutrophils and eosinophils (Figure 2). Direct immunofluorescence (DIF) studies were negative. Diagnostic workup also included blood studies that revealed eosinophilia (1,000/mL; normal range 0–500 mL) and a high IgE titer (812 IU/mL; normal , 100 IU/mL). She was treated with topical clobetasol propionate, and her condition improved during the first 2 weeks of treatment. However, 1 month later, she complained of intense pruritus and presented numerous typical scabietic burrows. Application of topical lindane, followed by the regular use of emollients, allowed marked improvement. Journal of Cutaneous Medicine and Surgery, Vol 14, No 4 (July/August), 2010: pp 175–177 175 176 Serra et al Figure 1. Tense bullae in the left arm (left) and the left foot (right). Six months later, she had a scabietic reinfestation, with intense pruritus, typical scabietic lesions, and some tense blisters. Scabies mites and eggs were once again identified in skin scrapings; sample collection was guided by dermoscopy (Figure 3). Immunofluorescence studies (including indirect immunofluorescence) remained negative. She was once more treated with lindane. Vigorous measures were implemented at her institution to avoid further outbreaks or reinfestations. Since then, she has been asymptomatic for more than a year of follow-up. Figure 2. Blistering at the subepidermal level and a dermal infiltrate with eosinophils (hematoxylin-eosin stain, 3 100 original magnification). Discussion Bullous lesions are very rare in scabies.1 A few cases have been reported in which scabies mimicked bullous pemphigoid.1,2 Recurrent bullous scabies, as seen in the present case, is particularly interesting. The recurrence of a blistering eruption raised suspicion of an underlying autoimmune blistering disorder, such as bullous pemphigoid. However, immunofluorescence studies were always negative and against this possibility. Furthermore, we did not need any immunosuppressive therapy to control her blistering disease. Clobetasol treatment was temporarily used and indeed stopped the blistering process while uncovering the true nature of the disease. In fact, antiscabietic treatment cured both the infestation and the blistering eruption, providing a strong argument in favor of the mite’s role in the pathogenesis of bullae formation. The mechanism of blister formation in scabies remains unexplained. Scabies-induced hypersensitivity mediated by histamine and eosinophils has been proposed as a putative mechanism.3 Another theory states that the parasite might induce an autoimmune blistering reaction similar to that seen in bullous pemphigoid. Enzymes released by the mites could damage the basement membrane zone and expose antigens that on physiologic conditions are not detected by the immune system, thereby allowing the development of autoimmunity.4 Indeed, immune deposits have been occasionally found on DIF studies of scabietic lesions from otherwise healthy patients and from patients with bullous scabies.1,5 However, the pathogenic role of such Recurrent Bullous Scabies 177 Figure 3. The ‘‘jet with contrail’’ or the ‘‘circumflex’’ sign (arrows), seen on dermoscopy (left), marks the spot where mites and eggs can be found, allowing successful collection of skin scrapings and a higher rate of positive microscopic examinations: Sarcoptes scabiei and an egg (right). deposits is still unclear, and they were not detected in the majority of cases. Diagnosing scabies is not always a straightforward task. Dermoscopy is a helpful technique.6 It can be easily performed in everyday practice to obtain quick diagnostic confirmation. We also use it to locate mites, thus allowing a higher rate of positive skin scraping examinations. Microscopic examination of skin scrapings is timeconsuming but remains useful to establish a definite diagnosis, especially in difficult cases. Finally, our patient’s successive reinfestations illustrate how challenging it can be to control scabies in institutional settings. Old or overcrowded facilities are particularly prone to epidemics. Inadequate implementation of disease control measures and the need to treat a high number of contactants explain why failure is so frequent in such settings. The morbidity and very high costs resulting from outbreaks underscore the value of an effective first-time approach in such situations.7 Acknowledgment Financial disclosure of authors and reviewers: None reported. References 1. Shahab RKA, Loo DS. Bullous scabies. J Am Acad Dermatol 2003;49: 346–50. 2. Viraben R, Dupre A. Scabies mimicking bullous pemphigoid. J Am Acad Dermatol 1989;20:134–6. 3. Haustein UF. Bullous scabies. Dermatology 1995;190:83–4. 4. Veraldi S, Scarabelli G, Zerboni R, et al. Bullous scabies. Acta Dermatol Venereol 1996;34(5 Pt 2):878–9. 5. Frentz G, Veien NK, Eriksen K. Immunofluorescence studies in scabies. J Cutan Pathol 1977;4:191–3. 6. Dupuy A, Dehen L, Bourrat E, et al. Accuracy of standard dermoscopy for diagnosing scabies. J Am Acad Dermatol 2007;56: 53–62. 7. Scheinfeld N. Controlling scabies in institutional settings. A review of medications, treatment models and implementation. Am J Clin Dermatol 2004;5:31–7.