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University of León, 24071 León, Spain Bone changes in an adult from a medieval mountain population in north-western Spain (12th-13th century): a possible case of leprosy 1Area of Physical Anthropology, Department of Biodiversity and Environmental Management University of León, 24071 León, Spain 1,2 2,3 2* 2CIAS - Research Centre for Anthropology and Health, Department of Life Sciences Laura González-Garrido , Célia Lopes , Sofia N. Wasterlain University of Coimbra, 3000-456 Coimbra, Portugal 3Department of Biology, School of Science and Technology, University of Évora, 7002-554 Évora, Portugal *E-mail: [email protected] (S.N. Wasterlain) INTRODUCTION DISCUSSION The lesions identified in a skeleton of an adult male (COR XVIII), Differential diagnosis recovered from the medieval necropolis of Barrejo, León (Spain), are presented and discussed. Differential diagnosis of the rhinomaxillary lesions must take into consideration several pathological conditions, namely fungal and oral The possible diagnosis of leprosy opens a debate on its diagnosis in infections, maxillary sinusitis, leishmaniosis, neoplastic disease, medieval populations in the Iberian Peninsula (Spain and Portugal). treponematoses and leprosy (Ortner, 2003). In fungal infections the formation of new bone is uncommon, causing only unilateral perforations of the bone (Robbins et al., MATERIAL AND METHODS 2009), which is not the case for this individual. Oral infections and rhinomaxillary sinusitis can cause Necropolis: A total of 26 individuals have been recovered: 17 males a b inflammation and destruction of the alveolar region of the maxilla and 7 females (20-50 years-old) and 2 non-adults (5-8 years-old). Figure 3. Resorption of the anterior nasal spine and osteolysis of the (Robbins et al., 2009), which was not observed in the present case. Chronology: 12th-13th centuries. piriform aperture are noticeable (a). Lytic lesions are also identified in Mucocutaneous leishmaniosis can also cause destructive Localization: Barrejo medieval necropolis is located in the valley of the nostrils, which is indicative of an inflammatory condition, alterations on the bones of the rhinomaxillary region, but the atrophy Valdeón, National Park of Picos de Europa, province of León (north- culminating in the total resorption of the vomer and nasal conchae, of the nasal spine and piriform aperture is not present (Ortner, 2003; western Spain) (Figure 1). It is delimited by the Cantabrian with the ethmoid also showing signs of intranasal destructive Robbins et al., 2009). Mountains. The weather is rainy Atlantic, with a harsh winter and alterations (b). These lesions are accompanied by pitting and In neoplastic diseases, osteolytic lesions may manifest in the skull short mild summers. proliferation of woven bone in the lateral margins of the nasal cavity. or the facial bones (Ortner, 2003). However, their morphological appearance is distinct from those presented here. Although in treponematoses, such as acquired syphilis, nasal lesions can be present, the rhinomaxillary alterations here observed together with the absence of pathognomonic lesions of this condition, such as caries sicca, turns this diagnosis unlikely (Hacket, 1976; Ortner, 2003). The association of rhinomaxillary remodelling, appendicular involvement, abnormal bone formation and destruction presented by the male COR-XVIII, point to lepromatous leprosy but in an early a b stage. Leprosy is a chronic and slowly progressive disease that, in later stages, leads inevitably to neurotrophic alterations of the hands Figure 1. Localization and landscape of the medieval necropolis of Figure 4. Hard palate presents evidence of pitting and abnormal and feet (Milner and Boldsen, 2017). It should also be highlighted that Barrejo. Province of León in red. The star shows the localization of proliferative bone (a). Perforation in palatine transverse suture and some hand and feet bones are missing, precluding their analysis. the necropolis. the floor of the nasal cavity are noted probably related to inflammatory processes (b). Cribra orbitalia and enamel hypoplasia observed emphasize the fact that this individual underwent a period of physiological stress caused Sex estimation: Visual method using the human hip bone (Bruzek, by disease and/or malnutrition. 2002). Age-at-death estimation: Morphological changes in the auricular surface of the ilium (Lovejoy et al., 1985). Archaeological background th Stature estimation: Regression equations based on femur length In the 13 century, leprosy was widespread in the north of Spain, (Mendonça, 2000). there being 24 leprosy hospitals established on the main pilgrim routes of the Asturias province (Tolivar, 1966). At that time, valley of Paleopathological analysis: The skeleton was macroscopically Valdeón was also provided by a small commercial exchange that examined by the three authors at the same time and the bones could bring infirmed people to the village (Rodríguez, 1999). radiographed. Like other individuals buried in this necropolis (Figure 8), individual CORXVIII was inhumed in supine position with upper limbs by the RESULTS sides of the torso on a west-east axis, in stone lined graves and lacking grave goods, suggesting that people with leprosy were not Conservation: The skeleton COR XVIII is relatively well-preserved necessarily stigmatised in death. In fact, this was not uncommon in although its structure was affected by chemical diagenesis due to the isolated populations where coexistence with patients was normal and necropolis proximity to the River Cares. Only the left side of the routine (Tolivar, 1966; Etxeberria et al., 1997; Rubini et al., 2017). mandible is present. The proximal epiphyses of both fibulae were not Figure 5. Remodelling and thinning of the anterior surface of the right preserved. maxilla, which led to post-mortem fracture. Missing bones: vertebrae (2), tarsals (2), carpals (3), metacarpals (2), and hand (2) and foot (21) phalanges. Sex and age-at-death: Adult male >30 years-old. Stature: 154.5 ± 6.90 cm. Pathology: Several lesions were identified in the skull (Figures 2-5), tibiae and fibulae (Figure 6,7). Figure 8. Overview of the inhumations in the medieval necropolis of Barrejo. Cribra orbitalia was observed in both orbits. Porotic hyperostosis was present in the supraorbital area. c Dental enamel hypoplasias were recorded in the upper anterior teeth. Upper central incisors were lost post-mortem. CONCLUSION No lesions were found in either hand or foot bones. According to historical documentary sources, leprosy was a relatively common disease in the medieval Iberian Peninsula documented by the presence of leprosy hospitals (De las Aguas, 2005). However, as a a b b d with many seemingly frequent diseases, the number of cases diagnosed in paleopathology is sorely lacking (Rubini et al., 2014). Figure 6. Cortical periosteal reaction, bilateral and symmetrical, The present case is the first to be described for Middle Ages in north recorded at the diaphyses of both tibiae (a, c) and fibulae (b). A post- western Spain, enriching the osteoarcheological record of leprosy mortem fracture of the right fibulae allows for the observation of and contributing to reducing the discrepancies between the historical subperiosteal bone reaction (d). and paleopathological evidence of this disease. REFERENCES Bruzek J. 2002. A method for visual determination of sex, using the human hip bone. American Journal of Physical Anthropology, 117: 157-168. De las Aguas JT. 2005. Historia de la lepra en España. Piel, 20: 485-49. Etxeberria F, Herrasti L, Beguiristain MA. 1997. Signos de lepra en un individuo Altomedieval de Navarra. IV Congreso Nacional de Paleopatología (San Fernando, Spain): La enfermedad en los restos humanos arqueológicos: actualización conceptual y metodológica, pp. 319-323. Hackett CJ. 1976. Diagnostic criteria of syphilis, yaws and treponarid (treponematoses) and some other diseases in dry bones. Heidelberg; Springer-Verlag. Lovejoy C, Meindl R., Pryzbeck, T, Mensforth R. 1985. Chronological metamorphosis of the auricular surface of the ilium: a new method for the determination of adult skeletal age at death. American Journal of Physical Anthropology, 68, 15-28. Mendonça M. (2000), Estimation of height from the length of long bones in a Portuguese adult population. American Journal of Physical Anthropology, 112: 39-48. Milner GR, Boldsen JL. 2017. Life not death: Epidemiology from skeletons. International Journal of Paleopathology, 17:26-39. Ortner DJ. 2003. Identification of Pathological Conditions in Human Skeletal Remains. Academic Press, New York. Robbins G, Tripathy VM, Misra VN, Mohanty RK, Shinde VS. 2009. Ancient Skeletal Evidence for Leprosy in India (2000 B.C.). PLoS ONE 4(5): e5669. Rodríguez Díaz E. 2000. Valdeón: Historia y Colección Diplomática. El occidente de Picos de Europa en la Edad Media. Real Instituto de Estudios Asturianos. Oviedo, Spain. Rubini M, Erdal YS, Spigelman M, Zaio P, Donoghue HD. 2014. Paleopathological and Molecular Study on Two Cases of Figure 7. The radiographic analysis of the tibiae and fibulae shows Ancient Childhood Leprosy from the Roman and Byzantine Empires. International Journal of Osteoarchaeology, 24, 570- 582. the reduction of the medullary cavities, particularly in tibiae, and Rubini M, Zaio P, Spigelman M, Donoghue HD. 2017. Leprosy in a Lombard-Avar cemetery in central Italy (Campochiaro, maintenance of cortical thickness. No tumours were found. No Molise, 6th-8th century AD): ancient DNA evidence and demography. Annals of Human Biology, 44 (6): 510-521. Tolivar Faes J. 1966. Hospitales de leprosos en Asturias durante las Edades Media y Moderna. Real Instituto de Estudios Figure 2. Skull of COR XVIII. radiolucent lines, suggestive of fractures, were identified. Asturianos. Oviedo, Spain. Acknowledgements. Dr. José Manuel Gonzalo Orden. Head of Radiology. Veterinary Clinic Hospital of University of León, Spain. Mr. Donal Savage for the English language revision This study was financed by national funds by FCT – Fundação para a Ciência e Tecnologia, under the project Reference UID/ANT/00283/2013.
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