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Palmar Petechiae in Dermatitis Herpetiformis: a Case Report and Clinical Review

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Palmar Petechiae in Dermatitis Herpetiformis: a Case Report and Clinical Review CONTINUING MEDICAL EDUCATION Palmar Petechiae in Dermatitis Herpetiformis: A Case Report and Clinical Review Capt Patrick E. McCleskey, USAF, MC; Capt Quenby L. Erickson, USAF, MC; COL Kathleen M. David-Bajar, MC, USA; COL Dirk M. Elston, MC, USA GOAL To have a comprehensive understanding of dermatitis herpetiformis (DH) OBJECTIVES Upon completion of this activity, dermatologists and general practitioners should be able to: 1. Explain the clinical presentation of DH. 2. Discuss the differential diagnoses for DH. 3. Identify the treatment options for DH. CME Test on page 224. This article has been peer reviewed and Medicine is accredited by the ACCME to provide approved by Michael Fisher, MD, Professor of continuing medical education for physicians. Medicine, Albert Einstein College of Medicine. Albert Einstein College of Medicine designates Review date: September 2002. this educational activity for a maximum of 1.0 hour This activity has been planned and implemented in category 1 credit toward the AMA Physician’s in accordance with the Essential Areas and Policies Recognition Award. Each physician should claim of the Accreditation Council for Continuing Medical only those hours of credit that he/she actually spent Education through the joint sponsorship of Albert in the educational activity. Einstein College of Medicine and Quadrant This activity has been planned and produced in HealthCom, Inc. The Albert Einstein College of accordance with ACCME Essentials. Drs. McCleskey, Erickson, David-Bajar, and Elston report no conflict of interest. The authors report no discussion of off-label use. Dr. Fisher reports no conflict of interest. Dr. McCleskey is performing a transitional internship at David Palmar petechiae or purpura is an unusual finding Grant Medical Center, Fairfield, California. Drs. Erickson, in dermatitis herpetiformis (DH) that occurs in chil- David-Bajar, and Elston are from the Department of Dermatology, dren but is only rarely reported in adults. We Brooke Army Medical Center, San Antonio, Texas. Dr. Erickson is a describe a 46-year-old man with DH who presented dermatology resident. Dr. David-Bajar is staff dermatologist at the San Antonio Uniformed Services Health Education Consortium and with the classic pruritic papulovesicular eruption Dermatology Consultant to the Army Surgeon General. Dr. Elston is and associated volar finger and palmar petechiae. Assistant Clinical Professor in the Department of Medicine at the We discuss recent advances in the pathogenesis University of Texas Health Sciences Center, San Antonio. The opinions expressed are those of the authors and should not and treatment of DH. be construed as official or as representing those of the Army Medical Department, the United States Air Force, or the Department of Defense. The authors were full-time federal employees at the Case Report time this work was completed. It is in the public domain. Reprints: Dirk M. Elston, MD, Department of Dermatology, A 46-year-old man presented to our department Geisinger Medical Center, 100 N Academy Ave, Danville, PA complaining of severe pruritus associated with a 17822-1406 (e-mail: [email protected]). rash on his elbows and knees. He also reported VOLUME 70, OCTOBER 2002 217 Dermatitis Herpetiformis Figure 1. Erythematous papules on the extensor surfaces. Figure 2. Volar finger petechiae following the skin lines. pinpoint burning on the palmar surface of his Comment hands after which a black spot would appear in the Diagnosis and Treatment—DH is a chronic disease respective location. He denied any associated diar- associated with gluten sensitivity that usually pre- rhea, cramping, bloating, or other gastrointestinal sents as an extremely pruritic papulovesicular erup- symptoms. Physical examination revealed many tion on the extensor surfaces. Its onset is typically 1- to 5-mm round and oval erythematous papules during early adulthood to 50 years of age,1 and its with overlying excoriations distributed symmetri- incidence is higher among Europeans than people of cally over the extensor surfaces of his elbows and Asian or African descent. Male predominance is knees (Figure 1). Petechiae less than 1 mm were seen in adults but not in children. noted on the palms and volar aspect of the fingers The characteristic distribution is symmetrical bilaterally and seemed to follow the dermato- and involves the elbows, knees, buttocks, sacrum, glyphics (Figure 2); they were more prominent over back, shoulders, posterior neck, and posterior hair- the first and second digits of each hand. There was line.1-3 Oral mucosa and larynx involvement rarely no involvement of the soles. The patient’s elbow had have been reported.4 The various morphologic pre- a single 2-mm vesicle (Figure 3). A shave biopsy sentations include pruritic erythematous papules specimen from perilesional skin showed strongly pos- and papulovesicles, erosions with or without crusts, itive granular IgA staining at the dermal-epidermal urticarial papules, vesicles, and bullae. Patients junction (DEJ) and dermal papillae (Figure 4), with sometimes note burning or itching that occurs 12 to weaker C3 and fibrin staining in the same distribu- 24 hours after the lesions present. tion; the findings are diagnostic of dermatitis her- DH in children has a distribution similar to that petiformis (DH). Antigliadin IgG antibodies were of adults; however, children also may present with positive, but antigliadin IgA antibodies and anti- palmar and plantar lesions. In addition, children endomysial antibodies were negative. Results were may exhibit red-brown macules and papules, hemor- normal for a complete blood count (CBC) and for rhagic vesicles, or petechiae and purpura over the liver, renal, and thyroid function tests, as well as for palms and volar fingers but excluding the dorsal 1,5 glucose-6-phosphate dehydrogenase, vitamin B12, aspect of the hands. Often these hand lesions are folate, and iron studies. The patient responded to asymptomatic and favor the dominant hand, sug- 25 mg/d of dapsone and a gluten-free diet (GFD); all gesting a possible traumatic etiology. Soles are more symptoms have since resolved. rarely involved. Although this presentation of hand 218 CUTIS® Dermatitis Herpetiformis Figure 3. Single 2-mm vesicle on the elbow. Figure 4. Granular IgA deposits at dermal papillae on direct immunofluores- cence (original magnification ϫ200). lesions is more common in children, it has been deserves mention because some older classifications reported in 10 adults.6-9 Despite early description as grouped it with DH because of the presence of IgA. pseudopurpura,8 the purpura seen in DH had Childhood LAD (also known as chronic bullous der- been shown histologically to have both extravasated matosis of childhood) is characterized by large bullae erythrocytes6,9,10 and papillary neutrophilic micro- in rosettes that may cluster in the genital and per- abscesses,6,8-11 the latter being typical histological ineal distribution and is not associated with gluten- findings in DH. sensitive enteropathy (GSE). Adult LAD is similar The differential diagnosis of DH lesions includes but typically occurs in the seventh decade and may linear IgA disease (LAD), erythema multiforme, involve mucous membranes, as well as skin. pemphigoid gestationis, bullous pemphigoid, pem- Histopathology of early DH lesions reveals a phigus vulgaris, pityriasis lichenoides et varioliformis neutrophilic infiltrate in the dermal papillary tips acuta, transient acantholytic dermatosis, papular (microabscesses) with variable eosinophilia and urticaria, scabies, insect bites, neurotic excoriations, subepidermal vesicles with neutrophil cellular bullous impetigo, and atopic dermatitis.1,2 LAD debris and fibrin.2,3,12 The vesicle expands to involve VOLUME 70, OCTOBER 2002 219 Dermatitis Herpetiformis several adjacent papillae. The histopathologic dif- phoma in both diseases2,17-19 and oral, pharyngeal, ferential diagnosis of early DH lesions includes and esophageal carcinoma in CD.17 The most com- LAD, bullous lupus erythematosus, epidermolysis mon associated malignancy is non-Hodgkin’s lym- bullosa acquisita, and bullous pemphigoid. LAD, phoma of the gastrointestinal tract,19,20 which has bullous lupus erythematosus, and epidermolysis bul- been referred to as enteropathy-associated T-cell losa acquisita tend toward a more continuous array lymphoma. Lymphomas have not been reported in of neutrophils at the DEJ. Bullous pemphigoid tends association with DH in childhood.1 toward a more eosinophil-rich infiltrate. Later CD is a gluten-sensitive enteropathy character- lesions may resemble any of the inflammatory ized by malabsorption and atrophy of the small subepidermal blistering diseases, including bullous intestine associated with dietary gluten. Patients pemphigoid, bullous drug reactions, erythema multi- often complain of diarrhea, cramps, and bloating. forme, and pemphigoid gestationis. The pathologic hallmarks revealed by results of Direct immunofluorescence (DIF) of peri- jejunal biopsies are jejunal villous blunting, elon- lesional skin should be performed by taking a 3- or gation of intestinal crypts, flattening of surface 4-mm punch biopsy within 1 cm of a lesion and epithelial cells, reduction of microvillous forma- either placing it in the immunofluorescence trans- tions, and lymphohistiocytic infiltrate in the lam- port medium or immediately freezing it. Lesional ina propria.15 CD is diagnosed by the finding of skin should not be used because DIF results are villous atrophy and
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