Review Is imported onchocerciasis a truly rare entity? Case report and review of the literature Spinello Antinori a, b, * , Carlo Parravicini c , Laura Galimberti b , Antonella Tosoni c , Paolo Giunta d , Massimo Galli a, b , Mario Corbellino b , Anna Lisa Ridolfo b a Luigi Sacco Department of Biomedical and Clinical Sciences, University of Milano, Italy b III Division of Infectious Diseases, Luigi Sacco Hospital, Fatebenefratelli Sacco ASST, Milano, Italy c Pathology Unit, Luigi Sacco Hospital, Fatebenefratelli Sacco ASST, Milano, Italy d Pathology Unit, Melegnano and Martesana ASST, Vizzolo Predabissi, Italy article info Article history: Received 5 October 2016 Received in revised form 13 February 2017 Accepted 16 February 2017 Available online xxx Keywords: Onchocerciasis River blindness Pruritus Imported infections Immigrants Travellers abstract Background: Onchocerciasis is endemic in a number of tropical countries in Africa and South America, and it is occasionally diagnosed as an imported disease in non-endemic areas. Methods: We describe the case of an African migrant with long-lasting pruritus and a cutaneous nodule who was diagnosed with onchocerciasis after nodulectomy, and review the medical literature regarding imported cases of onchocerciasis in the period 1994e2014. Results: Twenty-nine cases of onchocerciasis diagnosed in migrants from endemic countries, and in expatriates and travellers from non-endemic areas were retrieved. They were predominantly males (73.3%), had a median age of 37 years (two were aged <15 years), and acquired the diseases in sub- Saharan Africa, most frequently in Cameroon (43.3%). Diagnosis of onchocercosis was proven in 73.3% of patients. The most frequent clinical manifestations in these and our own patient were pruritus (23/30, 76.7%), unilateral leg or forearm swelling (13/30, 43.3%) and rash (12/30, 40.0%), whereas only two (6.9%) complained of eye symptoms. Eosinophilia was observed in almost all of the patients (92.0%), with median counts of 2915/mL among migrants and 1960/mL among travellers/expatriates. Eighteen patients underwent a skin snip biopsy, which was positive in 10 cases (55.5%); in the other 13 patients the parasite was directly demonstrated by means of a skin or nodule biopsy (n ¼ 5), nodulectomy (n ¼ 5) or slit lamp examination (n ¼ 3). Eighteen received ivermectin, alone, and seven ivermectin combined with diethylcarbamazine or doxycycline. Outcome details were available for only 14 patients, all of whom were asymptomatic after a median follow-up of 10 months (range 1e48). Conclusions: Onchocerciasis is a neglected tropical disease whose subtle and non-specific features may lead to under-diagnosis or underreporting in non-endemic areas. Physicians should consider this tropical disease when caring for migrants and travellers/expatriates with pruritus, skin lesions and eosinophilia. © 2017 Published by Elsevier Ltd. 1. Introduction Onchocerciasis or “river blindness” is a multisystem infestation caused by Onchocerca volvulus, a nematode worm that is trans- mitted to humans by the bites of infected blackflies (Simulium spp.) [1]. Microfilariae are found in peripheral blood, eye, skin and urine, and develop into adult worms in sub-dermal connective tissue. Onchocerciasis is considered a neglected tropical disease and the world's second most frequent infectious cause of blindness [2]. Typically pruritic cutaneous manifestations are very frequent dur- ing the course of the disease, and may appear several years before the ocular complications. At present, human onchocerciasis is endemic in 31 countries of sub-Saharan Africa (where 99% of infected people live), two coun- tries of Latin America (Brazil, Venezuela) and in Yemen [3]. By 2016 the World Health Organization (WHO) verified the interruption of transmission of onchocerciasis in four countries in Latin America: Colombia (2013), Mexico (2015), Ecuador (2015), Guatemala (2016) [4,5]. Despite the great efforts of African Programme for Oncho- cerciasis Control (APOC) based on community-wide mass drug * Corresponding author. Luigi Sacco Department of Biomedical and Clinical Sci- ences, Universit a degli Studi di Milano, Via GB Grassi 74, 20157, Milano, Italy. E-mail address: spinello.antinori@unimi.it (S. Antinori). Contents lists available at ScienceDirect Travel Medicine and Infectious Disease journal homepage: www.elsevierhealth.com/journals/tmid http://dx.doi.org/10.1016/j.tmaid.2017.02.006 1477-8939/© 2017 Published by Elsevier Ltd. Travel Medicine and Infectious Disease xxx (2017) 1e7 Please cite this article in press as: Antinori S, et al., Is imported onchocerciasis a truly rare entity? Case report and review of the literature, Travel Medicine and Infectious Disease (2017), http://dx.doi.org/10.1016/j.tmaid.2017.02.006