Bone changes in an adult from a medieval mountain population in north-western Spain (12th-13th century): a possible case of leprosy

Laura González-Garrido1,2, Célia Lopes2,3, Sofia N. Wasterlain2*

1Area of Physical Anthropology, Department of Biodiversity and Environmental Management University of León, 24071 León, Spain

2CIAS - Research Centre for Anthropology and Health, Department of Life Sciences University of Coimbra, 3000-456 Coimbra, Portugal

3Department of Biology, School of Science and Technology, University of Évora, 7002-554 Évora, Portugal

*E-mail: sofiawas@antrop.uc.pt (S.N. Wasterlain)

DISCUSSION

Differential diagnosis

Differential diagnosis of the rhinomaxillary lesions must take into

consideration several pathological conditions, namely fungal and oral

infections, maxillary sinusitis, leishmaniosis, neoplastic disease,

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.,

2009), which is not the case for this individual.

Oral infections and rhinomaxillary sinusitis can cause

inflammation and destruction of the alveolar region of the maxilla

(Robbins et al., 2009), which was not observed in the present case.

Mucocutaneous leishmaniosis can also cause destructive

alterations on the bones of the rhinomaxillary region, but the atrophy

of the nasal spine and piriform aperture is not present (Ortner, 2003;

Robbins et al., 2009).

In neoplastic diseases, osteolytic lesions may manifest in the skull

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

stage. Leprosy is a chronic and slowly progressive disease that, in

later stages, leads inevitably to neurotrophic alterations of the hands

and feet (Milner and Boldsen, 2017). It should also be highlighted that

some hand and feet bones are missing, precluding their analysis.

Cribra orbitalia and enamel hypoplasia observed emphasize the fact

that this individual underwent a period of physiological stress caused

by disease and/or malnutrition.

Archaeological background

In the 13th century, leprosy was widespread in the north of Spain,

there being 24 leprosy hospitals established on the main pilgrim

routes of the Asturias province (Tolivar, 1966). At that time, valley of

Valdeón was also provided by a small commercial exchange that

could bring infirmed people to the village (Rodríguez, 1999).

Like other individuals buried in this necropolis (Figure 8), individual

CORXVIII was inhumed in supine position with upper limbs by the

sides of the torso on a west-east axis, in stone lined graves and

lacking grave goods, suggesting that people with leprosy were not

necessarily stigmatised in death. In fact, this was not uncommon in

isolated populations where coexistence with patients was normal and

routine (Tolivar, 1966; Etxeberria et al., 1997; Rubini et al., 2017).

Figure 8. Overview of the inhumations in the medieval necropolis of

Barrejo.

CONCLUSION

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

with many seemingly frequent diseases, the number of cases

diagnosed in paleopathology is sorely lacking (Rubini et al., 2014).

The present case is the first to be described for Middle Ages in north

western Spain, enriching the osteoarcheological record of leprosy

and contributing to reducing the discrepancies between the historical

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 PhysicalAnthropology, 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 CongresoNacional de Paleopatología (San Fernando, Spain): La enfermedad en los restos humanos arqueológicos: actualizaciónconceptual 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: anew 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 Journalof 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. RealInstituto de Estudios Asturianos. Oviedo, Spain.

Rubini M, Erdal YS, Spigelman M, Zaio P, Donoghue HD. 2014. Paleopathological and Molecular Study on Two Cases ofAncient Childhood Leprosy from the Roman and Byzantine Empires. International Journal of Osteoarchaeology, 24, 570-582.

Rubini M, Zaio P, Spigelman M, Donoghue HD. 2017. Leprosy in a Lombard-Avar cemetery in central Italy (Campochiaro,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 EstudiosAsturianos. Oviedo, Spain.

INTRODUCTION

The lesions identified in a skeleton of an adult male (COR XVIII),

recovered from the medieval necropolis of Barrejo, León (Spain), are

presented and discussed.

The possible diagnosis of leprosy opens a debate on its diagnosis in

medieval populations in the Iberian Peninsula (Spain and Portugal).

MATERIAL AND METHODS

Necropolis: A total of 26 individuals have been recovered: 17 males

and 7 females (20-50 years-old) and 2 non-adults (5-8 years-old).

Chronology: 12th-13th centuries.

Localization: Barrejo medieval necropolis is located in the valley of

Valdeón, National Park of Picos de Europa, province of León (north-

western Spain) (Figure 1). It is delimited by the Cantabrian

Mountains. The weather is rainy Atlantic, with a harsh winter and

short mild summers.

Figure 1. Localization and landscape of the medieval necropolis of

Barrejo. Province of León in red. The star shows the localization of

the necropolis.

Sex estimation: Visual method using the human hip bone (Bruzek,

2002).

Age-at-death estimation: Morphological changes in the auricular

surface of the ilium (Lovejoy et al., 1985).

Stature estimation: Regression equations based on femur length

(Mendonça, 2000).

Paleopathological analysis: The skeleton was macroscopically

examined by the three authors at the same time and the bones

radiographed.

RESULTS

Conservation: The skeleton COR XVIII is relatively well-preserved

although its structure was affected by chemical diagenesis due to the

necropolis proximity to the River Cares. Only the left side of the

mandible is present. The proximal epiphyses of both fibulae were not

preserved.

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).

Cribra orbitalia was observed in both orbits.

Porotic hyperostosis was present in the supraorbital area.

Dental enamel hypoplasias were recorded in the upper anterior teeth.

Upper central incisors were lost post-mortem.

No lesions were found in either hand or foot bones.

Figure 2. Skull of COR XVIII.

Figure 3. Resorption of the anterior nasal spine and osteolysis of the

piriform aperture are noticeable (a). Lytic lesions are also identified in

the nostrils, which is indicative of an inflammatory condition,

culminating in the total resorption of the vomer and nasal conchae,

with the ethmoid also showing signs of intranasal destructive

alterations (b). These lesions are accompanied by pitting and

proliferation of woven bone in the lateral margins of the nasal cavity.

Figure 4. Hard palate presents evidence of pitting and abnormal

proliferative bone (a). Perforation in palatine transverse suture and

the floor of the nasal cavity are noted probably related to

inflammatory processes (b).

Figure 5. Remodelling and thinning of the anterior surface of the right

maxilla, which led to post-mortem fracture.

Figure 7. The radiographic analysis of the tibiae and fibulae shows

the reduction of the medullary cavities, particularly in tibiae, and

maintenance of cortical thickness. No tumours were found. No

radiolucent lines, suggestive of fractures, were identified.

Figure 6. Cortical periosteal reaction, bilateral and symmetrical,

recorded at the diaphyses of both tibiae (a, c) and fibulae (b). A post-

mortem fracture of the right fibulae allows for the observation of

subperiosteal bone reaction (d).

Acknowledgements. Dr. José Manuel Gonzalo Orden. Head of Radiology. Veterinary Clinic Hospital of University of León, Spain. Mr. Donal Savage for the English language revisionThis 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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