Danis et al. Journal of Medical Case Reports 2010, 4:46 http://www.jmedicalcasereports.com/content/4/1/46 C A S E R E P O R T JOURNAL OF MEDICAL CASE REPORTS Open Access Unusual presentation of eosinophilic fasciitis: two case reports and a review of the literature Ramazan Danis1, Sami Akbulut2*, Abdullah Altintas3, Sehmus Ozmen1, Cihan Akgul Ozmen4 Abstract Introduction: Eosinophilic fasciitis is an uncommon disorder with unknown etiology and a poorly understood pathogenesis. We present the cases of two patients with eosinophilic fasciitis with unusual presentation, and describe the clinical characteristics and laboratory findings related to them. Case presentation: The first case involves a 29-year-old Turkish man admitted with pain, edema and induration of his right-upper and left-lower limbs. Unilateral edema and stiffness with prominent pretibial edema was noted upon physical examination. A high eosinophil count was found on the peripheral smear. The second case involves a 63-year-old Turkish man who had pain, edema, erythema, and itching on his upper and lower extremities, which developed after strenuous physical activity. He had cervical lymphadenopathy and polyarthritis upon physical examination, and rheumatoid factor and antinuclear antibody upon laboratory examination. Conclusion: Eosinophilic fasciitis can present with various symptoms. When patients exhibit eosinophilia, arthralgia and myalgia, eosinophilic fasciitis should be considered as a possible diagnosis. Introduction Eosinophilic fasciitis (EF) is an uncommon disorder with unknown etiology and a poorly understood pathogen- esis. It has symmetrical involvement and in its early phase is characterized by limb or trunk erythema and edema, and later by collagenous thickening of the der- mis and subcutaneous fascia. EF is a scleroderma-like syndrome that was first described in 1974 by Shulman in patients with diffuse fasciitis and eosinophilia [1-3]. This syndrome was later named EF by Rodnan et al. [2]. Its onset is typically acute and findings include erythema, swelling and induration of the extremities, usually accompanied by eosinophilia. Here, we present two cases of EF with unusual presen- tation, and describe their corresponding clinical charac- teristics and laboratory findings. The first patient displayed unusual features that included high eosinophi- lia count and asymmetry. The second patient had cervi- cal lymphadenopathy and polyarthritis with rheumatoid factor (RF) and antinuclear antibody (ANA). * Correspondence: akbulutsami@gmail.com 2Department of Surgery, Diyarbakir Education and Research Hospital, 21400, Diyarbakir, Turkey Case presentation Case report 1 A 29-year-old Turkish man was admitted to our clinic with disability because of significant pain, edema and stiffness of his right-upper and left-lower limbs. He reported that the same clinical picture first appeared 3 years prior to this presentation and had since been repeated many times. His condition sometimes improved spontaneously and other times with the use of non-steroidal anti-inflammatory drugs (NSAIDs). Unilat- eral edema and stiffness in his right-upper limb (left arm circumference was 28.5 cm and right arm circumference was 30.5 cm) and left-lower limb (left thigh circumfer- ence was 53 cm and right thigh circumference was 46.4 cm), with prominent non-pitting pretibial edema were detected upon physical examination. His white blood cell count (WBC) was 22.8 × 109/L with 26.4% neutro- phils, 11.2% lymphocytes, and 60% eosinophils. His hemoglobin was 14.6 gdL, and his erythrocyte sedimen- tation rate (ESR) was 3 mm/h. Our patient’s stool specimens were examined for ova and parasites. Meanwhile, his renal, thyroid and liver function tests yielded negative results. His electrolytes were also within normal limits. Results were also nega- tive for RF, C-reactive protein and ANA. Results of his © 2010 Danis et al; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.