Foot polydactyly and bipartite medial cuneiform: A case of co-occurrence in a Celtic skeleton from Verona (Italy).

G Model JCHB-25383; No. of Pages 13

ARTICLE IN PRESS

HOMO - Journal of Comparative Human Biology xxx (2015) xxx–xxx

Contents lists available at ScienceDirect

HOMO - Journal of Comparative Human Biology journal homepage: www.elsevier.com/locate/jchb

Foot polydactyly and bipartite medial cuneiform: A case of co-occurrence in a Celtic skeleton from Verona (Italy) Z. Laffranchi a, J.S. Martín Flórez a, S.A. Jiménez Brobeil a,∗, V. Castellani b a Departamento de Medicina Legal, Toxicología y Antropología Física, Facultad de Medicina, Avda. de Madrid 11, 18071 Granada, Spain b U.O.C. Radiologia ULSS 20 Verona, Dipartimento Interdirezionale per l’Area Sanitaria, Ospedale G. Fracastoro, Via Circonvallazione 1, 37047 San Bonifacio, Verona, Italy

a r t i c l e

i n f o

Article history: Received 15 October 2014 Accepted 14 January 2015 Available online xxx

a b s t r a c t We report a case of bilateral foot polydactyly and bipartite medial cuneiform in a male individual buried in a Celtic/Roman necropolis (3rd to 1st century BCE) in the city of Verona (Italy). During the construction of an underground garage in the main courtyard of the Bishop’s Seminary at Verona between 2005 and 2010, archaeologists uncovered the remains of 174 individuals (108 non-adults and 66 adults). It is thought that these graves could belong to some of the first inhabitants of the urban area of Verona. The individual presented here (US 2807) is a middle-aged male (40–50 years) in a good state of preservation. His estimated stature is 1756 mm (±32.1 mm). This male presents congenital anomalies in the feet and dental agenesis. We believe this to be the only known archaeological case of bilateral postaxial polydactyly with forked (Y) shape, in which both fifth metatarsals are associated with complete bipartition of the left medial cuneiform and partial bipartition of the

∗ Corresponding author. Tel.: +34 958246340. E-mail address: [email protected] (S.A. Jiménez Brobeil). http://dx.doi.org/10.1016/j.jchb.2015.01.003 0018-442X/© 2015 Elsevier GmbH. All rights reserved.

Please cite this article in press as: Laffranchi, Z., et al., Foot polydactyly and bipartite medial cuneiform: A case of co-occurrence in a Celtic skeleton from Verona (Italy). HOMO - J. Comp. Hum. Biol. (2015), http://dx.doi.org/10.1016/j.jchb.2015.01.003

G Model JCHB-25383; No. of Pages 13 2

ARTICLE IN PRESS

Z. Laffranchi et al. / HOMO - Journal of Comparative Human Biology xxx (2015) xxx–xxx

right one. Polydactyly is fairly common in modern clinical cases but bipartite medial cuneiform is relatively rare; neither of these congenital conditions is well documented archaeologically. © 2015 Elsevier GmbH. All rights reserved.

r i a s s u n t o Si presenta lo studio di un caso archeologico di polidattilia e di bipartizione dell’osso cuneiforme mediale in entrambi i piedi di un individuo di sesso maschile. L’inumato è stato rinvenuto durante lo scavo di una necropoli Gallo-Romana di III-I secolo a.C., tenutosi tra il 2005 e il 2010 nella città di Verona (Italia). Durante i lavori di costruzione di un parcheggio sotterraneo nel Cortile Maggiore del Seminario Vescovile di Verona, gli archeologi hanno rinvenuto i resti scheletrici di 174 individui (108 infanti e 66 adulti). L’idea iniziale è che queste sepolture possano appartenere ai primi abitanti dell’area urbana di Verona. Il caso ivi descritto (US 2807) corrisponde a un uomo maturo di 40–50 anni che si presenta in un buon stato di conservazione. La statura stimata è di circa 1756 mm (±32.1 mm). L’individuo mostra anomalie congenite nei piedi e agenesie dentarie. Si ritiene che si tratti dell’unico caso archeologico noto di polidattilia bilaterale post-assiale con biforcazione (forma di Y) del quinto metatarso associato con la completa bipartizione del cuneiforme mediale sinistro e la parziale bipartizione del destro. La polidattilia sembra essere una condizione piuttosto comune nella casistica clinica moderna mentre la bipartizione dell’osso cuneiforme mediale risulta, d’altra parte, relativamente rara; nessuna di queste anomalie genetiche sembra essere ben documentata nel registro archeologico. © 2015 Elsevier GmbH. All rights reserved.

Introduction The construction of an underground garage in the main courtyard of the Bishop’s Seminary at Verona (Italy) between 2005 and 2010 led to the discovery of a large native/Roman necropolis (3rd to 1st century BCE) of a Roman-influenced Celtic culture (Galli Cenomani). The site contains approximately 163 simple burial graves with a minimum of 174 skeletons in a good state of preservation. The majority of individuals are non-adults (108 individuals), and there are 66 adults. The burials are currently under study and results have not been published. This short report describes the only individual from this necropolis (US 2807) showing the co-presence of unusual congenital anomalies: foot polydactyly, bipartite medial cuneiform and dental agenesis. Archaeological context and general description of individual US 2807 In the “Seminario Vescovile” necropolis of Verona, individuals are buried in single graves, with the body laid out in supine position. This pattern differs from that of other La Tène cemeteries, where funerary rituals usually included both inhumations and, more frequently, cremations. Grave goods include animal bones, ceramics, rare weapons and decorations. These ritual objects are documented in many but not all of the adult and non-adult burials. The chronology of the burials is associated with a period of cultural and political transition during which Celtic populations (late Iron Age groups) lived alongside the first Roman communities in the region (3rd to 1st century BCE). Archaeologists first considered that this necropolis might belong to some of the first inhabitants of urban Verona (Cavalieri Manasse and Salzani, unpublished results; Thompson and Bersani, unpublished results). Please cite this article in press as: Laffranchi, Z., et al., Foot polydactyly and bipartite medial cuneiform: A case of co-occurrence in a Celtic skeleton from Verona (Italy). HOMO - J. Comp. Hum. Biol. (2015), http://dx.doi.org/10.1016/j.jchb.2015.01.003


ARTICLE IN PRESS


3

Fig. 1. Seminario Vescovile of Verona: the burial of US 2807 (black arrow) in proximity to a female burial (US 2808), with his grave goods (Thompson and Bersani, unpublished results).

Individual US 2807 (Fig. 1) was buried in a single earth-cut grave in a supine position with his arms aligned to the body and left hand resting on his left femur. His body was oriented North–South and his legs were straight and slightly turned towards the West. The cadaver had decomposed in a closed environment, and the remains were in a good state of preservation. Preliminary absolute dating results and an initial study of the grave goods date this individual in the 3rd or 2nd century BCE. The grave goods of US 2807 comprise a type of ovoid pottery vase very common in burials of this epoch and a wooden container found next to the head of the individual. The remains of the container comprised two parallel iron circles joined by vertical transects, with traces of wooden planks visible on the inner surface of the circles. Archaeologists initially assumed that the wooden container was a small tub, but a wooden tub of this shape (circular or troncoconical) has not been found in other late Italian La Tène cemeteries, and the object remains under investigation (Cavalieri Manasse and Salzani, personal communication). The grave was in close proximity to the tomb of a middle-aged female (US 2808) with no grave goods. Preliminary absolute dating results based on their bone collagen indicate that the two individuals were contemporaneous, with dates of 2201–2137 cal BP for US 2807 and 2197–2131 cal BP for US 2808. The fact that the grave of US 2808 female does not impinge on the neighbouring tomb of US 2807 suggests that the latter may have been visible or indicated when she was buried. The results of osteological analysis reveal no pathologies, congenital anomalies, or cranial/post-cranial anatomical variants that may indicate a possible kinship between US 2808 and US 2807. Preliminary stable isotope data (␦15 N and ␦13 C) suggest that their diets were different. A highly negative ␦13 C value was found for US 2807 (≈−20‰) but a more positive value for US 2808 (≈−12‰), broadly in line with preliminary carbon data findings for the other adult females at the site. The isotopic analysis of palaeodietary data of individuals from this necropolis has already began and the results will be reported in a future publication. Anatomical description of individual US 2807 including anomalies Individual US 2807 was 40–50 years old at the time of death (age estimated using known methods based on observations of the pubic bone, auricular surface of the iliac bone and 4th left rib) (Brooks and Suchey, 1990; Isçan et al., 1984; Lovejoy et al., 1985; Todd, 1921). His sex was determined from the set of morphometric characters of the skull and pelvis given by Ferembach et al. (1979). His height was estimated based on the maximum length of the left femur, 1756 ± 32.1 mm (Ruff et al., 2012). Please cite this article in press as: Laffranchi, Z., et al., Foot polydactyly and bipartite medial cuneiform: A case of co-occurrence in a Celtic skeleton from Verona (Italy). HOMO - J. Comp. Hum. Biol. (2015), http://dx.doi.org/10.1016/j.jchb.2015.01.003


ARTICLE IN PRESS


Fig. 2. US 2807: bones of the feet of US 2807 evidencing type A bilateral polydactyly.

He is a tall and strong individual, with very marked muscle attachments in the upper and lower extremities. The right leg is generally less robust than the left one, and the left femur shows stronger muscle development (i.e., more marked linea aspera) than the right one; the popliteal line (soleus muscle insertion) is also more evident in the left versus right tibia. The shafts of the metatarsals are much less robust in the right versus left foot, and spurs are evident on the left heel. In addition, a slight asymmetry in the sizes of femora and tibiae can be noted. The femur and tibia are shorter and midshaft diameters and circumferences smaller on the right versus left side. The right fibula is highly fragmented and could not be measured. We ruled out a possible relationship between these features and the congenital anomalies described below. Individual US 2807 evidences the following foot anomalies: bilateral postaxial polydactyly type A with bifid (Y-shaped) fifth metatarsals (Figs. 2 and 3), bipartition of the left medial cuneiform into dorsal and plantar segments (bipartite medial cuneiform) and partial bipartition of the right medial cuneiform (Fig. 4a and b). Examples of similar anomalies are given by Barlow (1942), Burnett and Case (2011), Case et al. (2006), Temtamy and McKusick (1978) and Venn Watson (1976). The intermediate or second cuneiform in the right foot is missing, either because it has been lost during the excavation or, more likely, because it has not been preserved. The two halves of the left bipartite medial cuneiform articulate with each other but also articulate with the first metatarsal and with the navicular, which shows four distal facets rather than the normal three. In each foot, the first metatarsal presents two distinct facets rather than the more normal single facet (Fig. 5). The navicular of the right foot shows the usual three facets, probably because the right medial cuneiform presents only a partial bipartition. Another congenital anomaly is documented in the teeth, i.e., agenesis of the two lower third molars (M3) and the right lower central incisor (I1) (Fig. 6). Radiographic study of the mandible and maxilla confirms that these were not ante-mortem losses (Fig. 7). No alveolar resorption or evidence of deciduous tooth retention is observed in the mandible. However, M3 agenesis cannot be fully confirmed, because one of the upper M3 teeth is present and had erupted, whereas the X-ray of the jawbone clearly shows that the other upper M3 is absent in the socket (Fig. 8). The dental agenesis rate in the Please cite this article in press as: Laffranchi, Z., et al., Foot polydactyly and bipartite medial cuneiform: A case of co-occurrence in a Celtic skeleton from Verona (Italy). HOMO - J. Comp. Hum. Biol. (2015), http://dx.doi.org/10.1016/j.jchb.2015.01.003


ARTICLE IN PRESS


5

Fig. 3. US 2807: close-up view of the bifid (Y-shaped) fifth metatarsals.

adult sample of Verona is only 5.9%, including agenesis of upper lateral incisors, lower central incisors, upper canines and third molars. Classification and clinical review of polydactyly and other congenital anomalies Skeleton US 2807 is the only published archaeological case of foot polydactyly, bipartite medial cuneiform and hypodontia in the same individual. There have been very few reports of additional skeletal defects in individuals with bipartite medial cuneiform, including one case of bilateral os intermetatarseum (an accessory bone of the foot) associated with bilateral partial bipartition of the medial cuneiform (Case et al., 1998) and two cases of accessory navicular with bipartite medial cuneiform (Dwight, 1907; Fulwadhva and Parker, 2007). This paucity of reports suggests either that these cases are very rare or that researchers paid little attention to them. Bipartite medial cuneiform is also easily missed in the clinical setting and is often not detected on plain radiographs (Azurza and Sakellariou, 2013). Estimates by anatomical studies on the incidence of bipartite medial cuneiform have ranged from 0.27 to 0.31% based on an anatomical sample of European adults (Gruber, 1877; Pfitzner, 1896), to 2.4% based on a sample of European embryos (Trolle, 1948 after Burnett and Case, 2011). This congenital defect is not a fracture but can cause midfoot pain and has been treated with different techniques, including surgery (Elias et al., 2008). The term “polydactyly” refers to a category of developmental defects characterized by the presence of extra fingers or toes. These supernumerary digits can be a small mass of soft tissue without osseous structure or a fully developed extra digit, such as a sixth metacarpal or metatarsal (Barnes, 1994; Temtamy and McKusick, 1978). Polydactyly is usually represented by a poorly developed digit, which would be difficult to identify archaeologically (Case et al., 2006). The state of preservation of skeletons is known to have a major impact on reports on the frequency of postaxial polydactyly detection (Case et al., 2006), which may explain the small number of archaeological cases. Osseous polydactyly that involves branching from a metatarsal, as in the present individual, is uncommon in the general Please cite this article in press as: Laffranchi, Z., et al., Foot polydactyly and bipartite medial cuneiform: A case of co-occurrence in a Celtic skeleton from Verona (Italy). HOMO - J. Comp. Hum. Biol. (2015), http://dx.doi.org/10.1016/j.jchb.2015.01.003


ARTICLE IN PRESS


population. Watanabe et al. (1992) reported that duplication of the fifth toe is most frequent at the level of the intermediate phalanx, followed by the fifth metatarsal and proximal phalanx, with complete duplication (sixth metatarsal with duplication of distal phalanx) being the least frequent type (Case et al., 2006). The fact that phalangeal polydactyly is more commonly found in the clinical literature than metatarsal polydactyly suggests that phalangeal polydactyly would also be more frequently observed in archaeological samples. Polydactyly is usually localized on the medial or lateral digit of the hands or feet and is classified as preaxial when the extra digit is on the first digit and postaxial when on the fifth digit (Temtamy and McKusick, 1978). Postaxial polydactyly appears to be more frequent and is the only type reported from ancient times (Barnes, 1994; Hill and Case, 1996; Regan et al., 1996). The preaxial form, which presents a marked preference for the hands, is generally characterized by an osseous component and is therefore identifiable in archaeological remains (Bingle and Niswander, 1975). Polydactyly can be divided into two genetic subtypes (Temtamy and McKusick, 1978). Type A is responsible for a well-formed digit that articulates with the fifth metacarpal or metatarsal or the phalanges of the fifth digit or for a completely separate sixth digit; it is more frequently related to the foot than to the hand (Castilla et al., 1973) and includes numerous fifth-digit morphologies. In this study, we observed Y-shaped or bifid metatarsals. Type B is associated with a poorly differentiated digit, usually a simple piece of skin attached to the fifth digit with no osseous component, virtually ruling out its identification in archaeological remains (Bingle and Niswander, 1975; Temtamy and McKusick, 1978). Other primary types are central, mixed and crossed polydactyly. Central polydactyly is a very rare condition that can affect the second, third or fourth digit. Mixed polydactyly indicates both preaxial and postaxial involvement in the same person (Meltzer, 1987). Finally, crossed polydactyly is a variety of mixed polydactyly in which one form of polydactyly is observed in the hands and the other in the feet (Ishikiriyama et al., 1991). The frequency of polydactyly varies widely among populations. In a study of 120,000 live births, Frazier (1960) reported an overall incidence of 1.7/1000 births (McCarthy et al., 1995). Another more detailed study revealed that postaxial polydactyly is much more frequent than preaxial polydactyly

Fig. 4. (a) US 2807: left bipartite medial cuneiform and right medial cuneiform with partial bipartition (medial view). (b) US 2807: right medial cuneiform with partial bipartition (above, proximal view) and left bipartite medial cuneiform (below, medial view).



ARTICLE IN PRESS


7

Fig. 5. US 2807: left first metatarsal proximally shows two distinct facets.

in African-American and European-American populations, with a similar incidence of preaxial polydactyly (0.08 per 1000) in both but an 11-fold higher frequency of postaxial polydactyly in the African origin population (13.53/1000 vs. 1.24/1000) (Woolf and Myrianthopoulos, 1973). The highest frequencies have been recorded in African and Asian populations (Murphy, 1999), although the precise human embryological parameters underlying this variant are not known.

Fig. 6. US 2807: agenesis of the two lower M3 teeth (white arrows) and right lower I1 (black arrow).



ARTICLE IN PRESS


Fig. 7. US 2807: X-ray image of maxilla and mandible.

Fig. 8. US 2807: X-ray image of maxilla showing agenesis of upper left M3 (white arrow).

Polydactyly can be present without causing dysfunction, and the main reason for patients to seek surgery are shoe-fitting problems (Galois et al., 2002). Nonsyndromic polydactyly (in hand or in foot) is considered an autosomal dominant trait with variable penetrance (Castilla et al., 1973). Genetic studies have suggested that each type of polydactyly results from a different autosomal dominant gene with irregular modifiers (Castilla et al., 1973; Temtamy and McKusick, 1978). The heritability of both preaxial and postaxial polydactyly has been demonstrated (Case, 1996; Watanabe et al., 1992), and some authors have observed differences in their frequency among populations with different geographic ancestries (Bingle and Niswander, 1975; Castilla et al., 1973). Thus, the highest frequency of preaxial polydactyly of the hand was reported to be among East Asians and the highest frequency of postaxial polydactyly of the hand among Africans (Case, 2003). Please cite this article in press as: Laffranchi, Z., et al., Foot polydactyly and bipartite medial cuneiform: A case of co-occurrence in a Celtic skeleton from Verona (Italy). HOMO - J. Comp. Hum. Biol. (2015), http://dx.doi.org/10.1016/j.jchb.2015.01.003


ARTICLE IN PRESS


9

Polydactyly is rarely associated with other congenital anomalies except in recognized syndromes (Castilla et al., 1998). Castilla’s study on the combination of polydactyly with other limb defects showed syndactyly to be the most frequently associated anomaly. In particular, postaxial polydactyly was often combined with three types of syndactyly: toes 2–3, toes 4–5, and other (Castilla et al., 1998). Polydactyly is observed in genetic syndromes such as trisomy 13 (Patau syndrome), trisomy 18 (Edward’s syndrome), trisomy 21 (Down syndrome) and Meckel–Gruber syndrome. However, Down syndrome is not only strongly associated with first digit duplication but is also negatively associated with postaxial polydactyly (Castilla et al., 1998; Haber et al., 2007). The co-occurrence of polydactyly, bipartite medial cuneiform and hypodontia has not been reported in any of these syndromes with the exception of Down syndrome, whose clinical features include preaxial polydactyly and hypodontia (Källén et al., 1996; Roizen and Patterson, 2003). Thus, dental hypodontia is also sometimes associated with congenital disorders such as ectodermal dysplasia or Down syndrome (De Coster et al., 2009). Hypodontia refers to the congenital absence (agenesis) or extreme microdontia of a tooth, most frequently the third molar (Case, 2003). Other commonly affected teeth are the maxillary lateral incisor, mandibular central incisor and mandibular and maxillary second premolars (Alt and Türp, 1998; Case, 2003). Agenesis apparently occurs when the tooth bud fails to form. Inheritance of agenesis has been demonstrated for mandibular central incisors (Svinhufvud et al., 1988), maxillary lateral incisors (Alvesalo and Portin, 1969; Markovic, 1982; Svinhufvud et al., 1988), mandibular second premolars (Markovic, 1982), maxillary second premolars (Markovic, 1982; Svinhufvud et al., 1988) and third molars (Grahnen, 1956). The frequency of hypodontia (excluding the third molar) appears to vary worldwide, with lower frequencies (3–5% range) in Americans, British, Germans, Swiss and Hungarians (Davis, 1987; Graber, 1978) and higher frequencies in Scandinavians (6–8%) and East Asians (6–7%) (Case, 2003). The polydactyly, bipartite medial cuneiform and dental agenesis in the present individual may be congenital conditions or anomalies arising during early foetal development from the blastemal stage to the commencement of ossification rather than being of genetic origin. The independent occurrence of a gene for polydactyly and a gene for bipartite medial cuneiform in the same person appears unlikely, given the low frequency of these defects in the osteo-archaeological literature. We found no evidence for the heritability of either defect in the 174 skeletons from this Verona site, which included no other individual with these skeletal defects. Some authors have supported a possible hereditary tendency for bipartite medial cuneiform (Burnett and Case, 2011; Kjellström, 2004), but we found no other individual (adult or non-adult) with this anomaly from the same necropolis. However, it is more difficult to find congenital anomalies of the foot such as bipartite medial cuneiform in non-adult skeletons because of their poor state of preservation. Bones of the feet and hands of infants are generally very frail and are easily lost during excavations or damaged by post-depositional events. The co-occurrence of multiple minor variants in an individual has been associated with major birth defect syndromes (Cohen, 1997) and some forms of cancer (Anbazhagan and Raman, 1997; Mehes, 2000), while other authors have suggested that they may evidence developmental instability (Burnett, 2005; Hoyme, 1994; Mehes, 2000). The latter proposition is supported in the present case, because US 2807 shows no characteristic skeletal features of a genetic syndrome and lived to the age of 40–50 years. Genetic disorders such as trisomy 13, trisomy 18 and Meckel syndrome are lethal, and most infants die in the first few days postpartum, although some survive until the first year of life (Taylor, 1968). Hence, the co-presence in US 2807 of foot polydactyly and bipartite medial cuneiform with dental agenesis may suggest developmental instability or some type of developmental syndrome.

Archaeological skeletal evidence of polydactyly and bipartite medial cuneiform There are no frequency studies from archaeological samples in the anthropological literature, only some reports of single discoveries (Barnes, 1994; Case et al., 2006; Han et al., 1986; Murphy, 1999; Reed, 1981; Regan et al., 1996). Few archaeological cases of foot polydactyly have been reported in the literature (Barnes, 1994, 2008; Case et al., 2006; Murphy, 1999; Reed, 1981; Regan et al., 1996; Wrobel et al., 2012). Most are postaxial type A, and there is only one report of a preaxial case in a non-adult skeleton from the Iron Age (1100–1500 CE) site of Simbusenga in Zambia (Murphy, 1999). Please cite this article in press as: Laffranchi, Z., et al., Foot polydactyly and bipartite medial cuneiform: A case of co-occurrence in a Celtic skeleton from Verona (Italy). HOMO - J. Comp. Hum. Biol. (2015), http://dx.doi.org/10.1016/j.jchb.2015.01.003


ARTICLE IN PRESS


The vast majority of archaeological evidence of postaxial type A polydactyly comes from prehistoric settings in Southwest USA (Case et al., 2006). Two of these cases are non-adults, one an infant from the Tapia del Cerrito site and the other a juvenile from the Nuvakwewtaqa (Chavez Pass Ruin) site, which both exhibit a left fifth metatarsal with a lateral branch (Case et al., 2006). A further two cases of postaxial polydactyly have been reported, one from Pueblo Bonito in Chaco Canyon, New Mexico, and the other from Sand Canyon Pueblo, Southwest Colorado (Barnes, 1994; Kuckelman and Martin, 2007). A further case was found in Schoolhouse Point Mound, Arizona (Regan et al., 1996). Reed (1981) described a case of polydactyly with a double-headed fifth metatarsal from Pueblo de las Humanas, Gran Quivira National Monument in New Mexico (Case et al., 2006), which is similar to a recent finding in an adolescent skeleton from Trincheras burials in Northwest Mexico (Barnes, 2008). Evidence of postaxial polydactyly is also documented in a single specimen from Belize, where Wrobel et al. (2012) found a single right fifth metatarsal with a small deformity in the form of a small accessory digit among the commingled human remains of Tomb 1, burial 8 at the Peligroso site in Upper Macar River valley; they described it as the first archaeological example of type A postaxial polydactyly reported in Mesoamerica (Wrobel et al., 2012). No European archaeological cases have been published in the literature. There is an unpublished prehistoric case of preaxial foot polydactyly in a male adult from Homme Mort’s cave (France) found by Broca in 1874 and briefly reported by Bertrand (1874); the skeleton showed Y-shaped duplication of the distal phalanx of the first toe, but the chronology is not specified. Archaeological examples of bipartite medial cuneiform are also rare. The oldest case was identified in a Homo erectus from Dmanisi, Georgia (Jashashvili et al., 2010). There are documented cases in France, including a partial bipartition of medial cuneiform in a Palaeolithic skeleton (J. Dastugue personal communication in Anderson, 1987a,b) and a complete bipartition of medial cuneiform in a Merovingian individual (Volkov, 1904). A few cases from Britain have been reported by Anderson: one from sub-Roman Cannington, four from Roman Poundbury (R. Powers personal communication in Anderson, 1987b) and a possible case of partial bipartition from Brackmills, Northamptonshire (Anderson, 1998). Mediaeval cases of complete bipartition of medial cuneiforms have been described in Norway (Anderson, 1987a,b) and Sweden (Kjellström, 2004). Finally, a 19th century case (Ratliff, 1993) was reported in commingled remains excavated in Texas (Burnett and Case, 2011). Evidence of polydactyly in archaeological artefacts and in ancient literary sources Depictions of extra fingers and toes are often found in prehistoric rock art from the Southwest United States (Barnes, 2008), with examples from Texas (Jackson, 1938), New Mexico, Colorado (Barnes, 1994; Wellmann, 1979), Utah and Arizona (Castleton, 1979; Haury, 1945; Malotki and Weaver, 2002; Pilles, 1975; Wellmann, 1975). They have also been discovered in other parts of the USA, including Wyoming (Francis and Loendorf, 2002), Kansas (Richards and Richards, 1960), Montana, Missouri and Michigan (Wellmann, 1979). Other images of polydactyly in rock engravings have been found in Chihuahua, Mexico (Jackson, 1938) and in Argentina (Castilla et al., 1973). These rock art depictions testify that polydactyly was a condition familiar to American Indians of the prehistoric period (Case et al., 2006). There are several cases of polydactyly representations from the Roman Age or immediately beforehand in Europe, especially in France and Italy. Examples of polydactyly are known from several ex-votos from central Italy, including an example from Palestrina (Grmek and Gourevitch, 1998) and an Etruscan-Roman ex-voto from Rimini (Charlier, 2008). As in the Italian depictions, an anatomical ex-voto of a foot with six toes was also found in the Gallic-Roman temple of Essarois (Cote d’Or, France) (Curie et al., 2011). Other cases are occasionally mentioned in archaeological literature, but it has not been possible to establish with any certainty whether these truly represent cases of polydactyly or whether the presence of a sixth toe derives from a mistake made by the potter (Grmek and Gourevitch, 1998). Examples of polydactyly are also cited in Roman literature. In Naturalis historia, Pliny the Elder (Nat. hist. XI, 244) mentions the case of two sisters, daughters of the Roman patrician M. Coranus, who were known as “Sedigita” (literally six digits) because they had six fingers. He also reports that the grammarian Volcacius Sedigitus, who lived in the 2nd and 1st centuries BCE, had hand polydactyly Please cite this article in press as: Laffranchi, Z., et al., Foot polydactyly and bipartite medial cuneiform: A case of co-occurrence in a Celtic skeleton from Verona (Italy). HOMO - J. Comp. Hum. Biol. (2015), http://dx.doi.org/10.1016/j.jchb.2015.01.003


ARTICLE IN PRESS


11

(Curie et al., 2011). Further testimony of polydactyly in the Roman Age is contained in the Digesta (or Pandectae). According to one of the laws described, dated to the beginning of the 3rd century CE (D.21, 1, 10, 2), the presence of polydactyly in the feet or hands was not a valid motive for a new owner to claim an actio redhibitoria (i.e., reimbursement) for the purchase of a slave, because the presence of more or fewer fingers or toes was considered irrelevant unless it impaired the slave’s ability to move (Alemán Monterreal, 2012). This law suggests a tolerance towards cases of polydactyly in the Roman Imperial Age. In the case of the site at Seminario Vescovile, the archaeological materials remain under study, but the grave goods of this necropolis appear to be generally uniform, with no initial evidence of major inequalities in the value of the objects buried with the deceased. We can speculate that US 2807 was probably well-integrated in his community, given the localization of his grave near the other tombs and the presence of grave goods of similar value to others at the site. Preliminary absolute dating of the individual suggests that his grave is among the most ancient in the necropolis, although only a few other C14-analyzed samples are available, and the archaeological material analysis of his grave goods remains in progress. Acknowledgements The authors thank Dr. Giuliana Cavalieri Manasse of the Soprintendenza Archeologica of Verona for the opportunity to study this skeletal collection and the archaeologists Simon Thompson and Marzia Bersani of Verona who excavated the necropolis of Seminario Vescovile for use of their archaeological data. They wish to express their gratitude to Dr. Francesca Fornasa for providing X-ray images of the mandible and maxilla of this individual and to Dr. Antonio Delgado Huertas for making possible the stable isotope and radiocarbon analyses of individuals US 2807 and US 2808. They are grateful to Prof. Troy Case and Prof. Scott Burnett for their advice and comments and valuable bibliographical information. The authors also thank Dr. Julien Curie and the other (anonymous) reviewer for their valuable insights and suggestions. References Alemán Monterreal, A., 2012. Precisiones terminológicas sobre ostentum D.50, 16, 38 (Ulpianus libro 25 ad edictum). In: Resina Sola, P. (Ed.), Fundamenta Iuris. Terminologia, Principios e Interpretacción. Editorial Universidad de Almeria, Almeria, pp. 49–64. Alt, K.W., Türp, J.C., 1998. Hereditary dental anomalies. In: Alt, K.W., Rösing, F.W., Teschler-Nicola, M. (Eds.), Dental Anthropology: Fundamentals, Limits, and Prospects. Springer, New York, pp. 95–128. Alvesalo, L., Portin, P., 1969. The inheritance pattern of missing, peg-shaped, and strongly mesio-distally reduced upper lateral incisors. Acta Odontol. Scand. 27, 563–575. Anbazhagan, R., Raman, V., 1997. Homeobox genes: molecular link between congenital anomalies and cancer. Eur. J. Cancer 33, 635–637. Anderson, T., (M.A. thesis) 1987a. Post-cranial Non-metric Variation: The Examination of a Neglected Subject. Sheffield University, Sheffield. Anderson, T., 1987b. A medieval bipartite cuneiform I with attempted unilateral fusion. Ossa 13, 39–48. Anderson, T., 1998. Two cases of hypoplastic hamulus from Iron Age and Roman Northamptonmshire. J. Paleopathol. 10, 31–35. Azurza, K., Sakellariou, A., 2013. ‘Osteosynthesis’ of a symptomatic bipartite medial cuneiform. Foot Ankle Int. 22, 499–501. Barlow, T.E., 1942. Os cuneiforme I bipartitum. Am. J. Phys. Anthropol. 29, 95–111. Barnes, E., 1994. Polydactyly in the Southwest. Kiva 59, 419–431. Barnes, E., 2008. Congenital anomalies. In: Pinhasi, R., Mays, S. (Eds.), Advances in Human Palaeopathology. John Wiley and Sons, Chichester, pp. 329–362. Bertrand, A., 1874. Celtes, Galois et Francs. Bull. Soc. Anthropol. Paris 9 (II), 108–113. Bingle, G.J., Niswander, J.D., 1975. Polydactyly in the American Indian. Am. J. Hum. Genet. 27, 91–99. Brooks, S.T., Suchey, J.M., 1990. Skeletal age determination based on the Os Pubis: a comparison of the Acsádi–Nemeskéri and Suchey–Brooks methods. Hum. Evol. 5, 227–238. Burnett, S.E., (Ph.D. dissertation) 2005. Developmental Variation in South African Bantu: Variant Co-occurrence and Skeletal Asymmetry. Arizona State University, Tempe. Burnett, S.E., Case, D.T., 2011. Bipartite medial cuneiform: new frequencies from skeletal collection and a meta-analysis of previous cases. Homo – J. Comp. Hum. Biol. 62, 109–125. Case, D.T., (M.A. thesis) 1996. Developmental Defects of the Hands and Feet in Paleopathology. Arizona State University, Tempe. Case, D.T., (Ph.D. dissertation) 2003. Who’s Related to Whom? Skeletal Kinship Analysis in Medieval Danish Cemeteries. Arizona State University, Tempe. Case, D.T., Ossenberg, N.S., Burnett, S.E., 1998. Os intermetatarseum: a heritable accessory bone of the human foot. Am. J. Phys. Anthropol. 107, 199–209.



ARTICLE IN PRESS


Case, D.T., Hill, R.J., Merbs, C.F., Fong, M., 2006. Polydactyly in the prehistoric American southwest. Int. J. Osteoarchaeol. 16, 221–235. Castilla, E., Paz, J., Mutchinick, O., Munoz, E., Giorgiutti, E., Gelman, Z., 1973. Polydactyly: a genetic study in South America. Am. J. Hum. Genet. 25, 405–412. Castilla, E.E., Lugarinho, R., Da Graça Dutra, M., Salgado, L.J., 1998. Associated anomalies in individuals with polydactyly. Am. J. Hum. Genet. 80, 459–465. Castleton, K.B., 1979. Petroglyphs and Pictographs of Utah, Vol. Two: The South, Central, West and Northwest. Utah Museum of Natural History, Salt Lake City. Cavalieri Manasse, G., Salzani, L., 2012. Verona nella fase della Romanizzazione, alla luce dei nuovi scavi. In: Podium presentation at the 36 Colloque International de l’AFEAF (Asociation Française por l’Etude de l’Âge du Fer), Les Celtes et le Nord de l’Italie, premier et second Âge du Fer, Verona (unpublished results). Charlier, Ph., 2008. Les monstres humains dans l’Antiquité: analyse paléopathologique, Nouvelles Études Historiques 66. Fayard, Paris. Cohen, M.M., 1997. The Child with Multiple Birth Defects. Oxford University Press, New York. Curie, J., Petit, C., Ponçon, P., Goguey, D., 2011. Découverte d’ un ex voto particulier sur le site culturel d’Essarois (Côte d’Or): un cas de polydactylie chez les Gallos Romains? Revue Archéologique de l’Est 60, 563–568. Davis, P.J., 1987. Hypodontia and hyperdontia of permanent teeth in Hong Kong schoolchildren. Community Dent. Oral Epidemiol. 15, 218–220. De Coster, P.J., Marks, L.A., Martens, L.C., Huysseune, A., 2009. Dental agenesis: genetic and clinical perspectives. J. Oral Pathol. Med. 38, 1–17. Dwight, T., 1907. Variations of the Bones of the Hands and Feet. J.B. Lippincott Company, Philadelphia. Elias, I., Dheer, S., Zoga, A.C., Raikin, S.M., Morrison, W.B., 2008. Magnetic resonance imaging findings in bipartite medical cuneiform – a potential pitfall in diagnosis of midfoot injuries: a case series. J. Med. Case Rep. 2, 272. Ferembach, D., Schwidetzky, I., Stloukal, M., 1979. Recommandations pour déterminer l‘âge et le sexe sur le squelette. Bull. Mém. Soc. Anthropol. Paris 6 (XIII), 7–45. Francis, J.E., Loendorf, L.L., 2002. Ancient Visions: Petroglyphs and Pictographs of the Wind River and Bighorn Country, Wyoming and Montana. University of Utah Press, Salt Lake City. Frazier, T.M., 1960. A note on race-specific congenital malformation rates. Am. J. Obstet. Gynecol. 80, 184–185. Fulwadhva, U., Parker, R.J., 2007. Symptomatic bipartite medial cuneiform. Appl. Radiol. 3, 42–44. Galois, L., Mainard, D., Delagoutte, J.P., 2002. Polydactyly of the foot, literature review and case presentations. Acta Orthop. Belg. 68, 376–380. Graber, L.W., 1978. Congenital absence of teeth: a review of the literature with emphasis on inheritance patterns. J. Am. Dent. Assoc. 96, 266–275. Grahnen, H., 1956. Hypodontia in the permanent dentition. Odontol. Revy Suppl. 7, 1100. Grmek, M., Gourevitch, D., 1998. Les Maladies dans l’Art Antique. Fayard, Paris. Gruber, W., 1877. Monographie uber das Zweigtheilte erste Keilbeinbeim Menschen. Mém. Acad. Imp. Sci. St. Pétersbourg 24, 1–35. Haber, L.L., Adams, H.B., Thompson, G.H., Duncan, L.S., Didomenico, L.A., McCluskey, W.P., 2007. Unique case of polydactyly and a new classification system. J. Pediatr. Orthop. 27, 326–328. Han, T.L., Collins, S.L., Clark, S.D., Garland, A., 1986. Moe Kau a Ho’oilo: Hawaiian Mortuary Practices at Keopu, Kona, Hawaii. Departmental Report Series No. 86-1. Bernice Pauahi Bishop Museum, Honolulu. Haury, E., 1945. Painted Cave, Northeastern Arizona. Amerind Foundation, Dragoon. Hill, R.J., Case, D.T., 1996. Bilateral postaxial polydactyly of the foot in a Salado infant. In: Poster Presented at the Annual Meeting of the Paleopathology Association, Durham, NC. Hoyme, H.E., 1994. Minor anomalies: diagnostic clues to aberrant human morphogenesis. In: Markow, T.A. (Ed.), Developmental Instability: Its Origins and Evolutionary Implications. Kluwer Academic Publishers, Dordrecht, pp. 309–317. Isçan, M.Y., Loth, R.S., Wright, R.K., 1984. Age estimation from the rib by phase analysis: white males. J. Forensic Sci. 32, 452–466. Ishikiriyama, S., Sawada, H., Nambu, H., Niikawa, N., 1991. Crossed polydactyly type I in a mother and son: an autosomal dominant trait? Am. J. Med. Genet. 40, 41–43. Jackson, A.T., 1938. Picture-writing of Texas Indians. University of Texas Press, Austin. Jashashvili, T., Ponce de León, M.S., Lordkipanidze, D., Zollikofer, C.P.E., 2010. First evidence of a bipartite medial cuneiform in the hominin fossil record: a case report from the Early Pleistocene site of Dmanisi. J. Anat. 216, 705–716. Källén, B., Mastroiacovo, P., Robert, E., 1996. Major congenital malformations in Down syndrome. Am. J. Med. Genet. 65, 160–166. Kjellström, A., 2004. A case study of os cuneiforme mediale bipartum from Sigtuna, Sweden. Int. J. Osteoarch. 14, 475–480. Kuckelman, K.A., Martin, D.L., 2007. Human skeletal remains. In: Kuckelman, K.A. (Ed.), The Archaeology of Sand Canyon Pueblo: Intensive Excavations at a Late-Thirteenth-Century Village in Southwestern Colorado. Crow Canyon Archaeological Center, Cortez. Lovejoy, C.O., Meindl, R.S., Pryzbeck, T.R., Mensforth, R.P., 1985. Chronological metamorphosis of the auricular surface of the ilium: a new method for the determination of adult skeletal age at death. Am. J. Phys. Anthropol. 68, 47–56. Malotki, E., Weaver, D.E., 2002. Stone Chisel and Yucca Brush: Colorado Plateau Rock Art. Kiva Publications, Walnut, CA. Markovic, M., 1982. Hypodontia in twins. Swed. Dent. J. Suppl. 15, 153–162. McCarthy, G.I., Lindaman, L., Stefan, M., 1995. Pedal polydactyly: an overview with case report. J. Foot Ankle Surg. 34, 577–582. Mehes, K., 2000. Mild errors of morphogenesis in malignancy: macroscopic manifestation of genetic instability? Med. Pediatr. Oncol. 34, 111–112. Meltzer, R.M., 1987. Polydactyly. Clin. Podiatr. Med. Surg. 4, 57–62. Murphy, K.A., 1999. A prehistoric example of polydactyly from the Iron Age site of Simbusenga, Zambia. Am. J. Phys. Anthropol. 108, 311–319. Pfitzner, W., 1896. Beitrage zur Kenntenis des Menschlichen Extremitatenskelets, VII. Die Variationen in Aufbau des Fussskelets. In: Schwalbe, G. (Ed.), Morphologische Arbeiten. Gustav Fisher, Jena, pp. 245–527.



ARTICLE IN PRESS


13

Pilles, P.J., 1975. Petroglyphs of the Little Colorado, River valley, Arizona. In: Grove, S.T. (Ed.), American Indian Rock Art, Papers presented at the 1974 Rock Art Symposium. San Juan County Museum Association, Farmington, NM, pp. 1–26. Ratliff, E.A., (M.A. thesis) 1993. Life and Death in the Mexican Army: An Analysis of Skeletal Remains from the Battle of Resaca de la Palma. University of Texas, Austin. Reed, E.E., 1981. Human skeletal material. In: Hayes, A.C. (Ed.), Contributions to Gran Quivira Archaeology. National Park Service, Publications in Archeology 17, Washington, DC, pp. 75–118. Regan, M., Turner, C., Irish, J., 1996. Physical anthropology of the Schoolhouse Point Mound, U:824/13a. In: Lindauer, O. (Ed.), The Place of the Storehouses: Roosevelt Platform Mound Study, Report on the Schoolhouse Point Mound, Pinto Creek Complex, Part 2. Roosevelt Monograph Series 6, Anthropological Field Studies 35. Office of Cultural Resource Management, Arizona State University, Tempe, pp. 787–840. Richards, A., Richards, D., 1960. Petroglyphs of the Russell site. Sci. Man 1, 18–21. Roizen, N.J., Patterson, D., 2003. Down’s syndrome. Lancet 361, 1281–1289 (review). Ruff, C.B., Holt, B.M., Niskanen, M., Sladék, V., Berner, M., Garofalo, E., Garvin, H.M., Hora, M., Maijanen, H., Niinimäki, S., Salo, K., Schuplerová, E., Tompkins, D., 2012. Stature and body mass estimation from skeletal remains in the European Holocene. Am. J. Phys. Anthropol. 148, 601–617. Svinhufvud, E., Myllarniemi, S., Norio, R., 1988. Dominant inheritance of tooth malpositions and their association to hypodontia. Clin. Genet. 34, 373–381. Taylor, A.I., 1968. Autosomal trisomy syndromes: a detailed study of 27 cases of Edward’s syndrome and 27 cases of Patau’s syndrome. J. Med. Genet. 5, 227–252. Temtamy, S.A., McKusick, V.A., 1978. The Genetics of Hand Malformations. The National Foundation – March of Dimes Birth Defects Original Article Series. Alan R. Liss, New York. Thompson, S.R., Bersani, M., Unpublished results. Relazione Archeologica del Seminario Vescovile 2005–2009. Todd, T.W., 1921. Age changes in the pubic bone: I: the male white pubis. Am. J. Phys. Anthropol. 3, 285–334. Trolle, D., 1948. Accessory bones of the human foot: a radiological. In: Histo-embryological Comparative Anatomical, and Genetic Study. Munksgaard, Copenhagen. Venn Watson, E.A., 1976. Problems in polydactyly of the foot. Orthop. Clin. North Am. 7, 909–927. Volkov, T., 1904. Variation squelettiques du pied. Bull. Mém. Soc. Anthropol. Paris 5, 201–331. Watanabe, H., Fujita, S., Oka, I., 1992. Polydactyly of the foot: an analysis of 265 cases and a morphological classification. Plast. Reconstr. Surg. 89, 856–877. Wellmann, K.F., 1975. New Mexico’s mutilated hand: finger mutilation and polydactylism in North American Indian rock art. J. Am. Med. Assoc. 219, 1609–1610. Wellmann, K.F., 1979. A Survey of North American Indian Rock Art. Akademische Druck und Verlagsanstalt, Graz. Woolf, C.M., Myrianthopoulos, 1973. Polydactyly in American Negroes and whites. Am. J. Hum. Genet. 25, 397–404. Wrobel, G.D., Helmike, C., Nash, L., Awe, J.J., 2012. Polydactyly and the Maya: a review and a case from the site of Peligroso, upper Macal River valley, Belize. Anc. Mesoam. 23, 131–142.


Bipartite Medial Cuneiform: Case Report and Retrospective Review of 1000 Magnetic Resonance (MR) Imaging Studies.

Dorsal dislocation of the intermediate cuneiform with a medial cuneiform fracture: a case report and review of the literature.

Symptomatic bipartite medial cuneiform treated with fluoroscopic and ultrasound-guided injections.

Symptomatic Bipartite Medial Cuneiform: Report of Five Cases and Review of the Literature.

Dislocation Intermediate Cuneiform with Fracture Medial Cuneiform.

A rare presentation of foot pain: bilateral navicular-medial cuneiform coalition.

Preaxial polydactyly of the foot: variable expression of trisomy 13 in a case from central Africa.

Diabetic foot in a patient with polydactyly.

An irreducible medial cuneiform fracture-dislocation.

A case of hydrometrocolpos and polydactyly.

A rare anomaly of the foot presented as polydactyly.

Medial tibial stress syndrome: a skeleton from medieval Rhodes demonstrates the appearance of the bone surface--a case report.

Rare presentation of foot postaxial polydactyly.

Bilateral absence of the lesser trochanter in a late Epigravettian skeleton from Arene Candide (Italy).

Chondroblastoma of the medial cuneiform bone in a 32-year-old woman.

Congenital bipartite carpal lunate. A case report.

An unusual case of postaxial polydactyly of the foot treated by metatarsal transfer.

Symptomatic bilateral bipartite lunate: a case report.

Exertional compartment syndrome of the medial foot compartment--diagnosis and treatment: a case report.

A case of polydactyly with multiple thresholds in the mouse.

Unusual fracture combination in a paediatric acute ankle (combined medial talar compression fracture with medial malleolus fracture in an immature skeleton): a case report.

A case of brachymetatarsia from medieval Sardinia (Italy).

Cooccurrence of Darier's Disease and Epilepsy: A Pediatric Case Report and Review of the Literature.

Li-Fraumeni syndrome: a case report from Italy.