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International Journal of Osteoarchaeology Int. J. Osteoarchaeol. 9: 369–373 (1999) SHORT REPORT A Case of Diffuse Idiopathic Skeletal Hyperostosis (DISH) from a Medieval Necropolis in Southern Italy BRUNA REALE, DAMIANO MARCHI AND SILVANA M. BORGOGNINI TARLI* Universita ` degli Studi di Pisa, Dpt. Di Etologia, Ecologia ed Evoluzione, Unita ` di Antropologia, Via S. Maria 55 -56126 Pisa, Italy Key words: idiopathic hyperostosis; medieval abbey; southern Italy Introduction The subject described here was found in a single burial (No. 272) of the cemetery pertaining to the S. Angelo Abbey, located in Mon- tescaglioso, 30 km southwest of Matera (Basili- cata, southern Italy). The site was excavated in 1996 under the direction of Dr G. Canosa, the then Director of the Museo Nazionale D. Ri- dola of Matera. The burial was archaeologically dated be- tween 1100 and 1400 AD and appears as a ditch covered by irregular stones lying under the southern corridor leading to the A Cloister (Canosa, personal communication). Both the corridor and the A Cloister were built after 1400 during work for the expansion of the perimeter of the abbey, thus suggesting that the tombs were originally located outside the boundaries (Venezia, personal communication). Unfortunately, there is a lack of historical docu- mentation about the use of the cemetery by the monks and/or by the local population. The skeleton is fairly complete and in a good state of preservation, as only part of the skull, the atlas, axis, sternebrae, most ribs and part of hand and foot bones are missing. Sex diagnosis (male) was performed by mor- phological methods (Phenice, 1969; Ferembach et al., 1977), age at death (25–35 years) was estimated by the degree of cranial suture closure (Meindl & Lovejoy, 1985) and by the modifica- tions of the pubic symphysis (Meindl et al., 1985). Stature (167.9 9 2.99 cm) was evaluated by Trotter & Gleser’s (1952) equations for white males. Palaeopathological findings Skull A vermicular aspect was observed on the inner side along the sagittal suture. Signs of porotic hyperostosis were present on the outer table near the lambdoid suture. Vertebral column This was the most affected skeletal region, with massive new bone formation on the vertebral bodies, but without involvement of the articular surfaces or the intervertebral spaces. In the tho- racic section (T6–T12) flowing ossifications were located on the right side of the anterior longitudinal ligament, appearing as a dense and continuous line of bumps (Figure 1). In the * Correspondence to: Universita ` degli Studi di Pisa, Dpt. Di Etologia, Ecologia ed Evoluzione, Unita ` di Antropologia, Via S. Maria 55-56126 Pisa, Italy. E-mail: [email protected] CCC 1047–482X/99/050369 – 05$17.50 Copyright © 1999 John Wiley & Sons, Ltd. Received 26 November 1998 Revised 26 January 1999 Accepted 30 April 1999

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International Journal of OsteoarchaeologyInt. J. Osteoarchaeol. 9: 369–373 (1999)

SHORT REPORT

A Case of Diffuse Idiopathic SkeletalHyperostosis (DISH) from a MedievalNecropolis in Southern ItalyBRUNA REALE, DAMIANO MARCHI AND SILVANAM. BORGOGNINI TARLI*Universita degli Studi di Pisa, Dpt. Di Etologia, Ecologia ed Evoluzione,Unita di Antropologia, Via S. Maria 55-56126 Pisa, Italy

Key words: idiopathic hyperostosis; medieval abbey; southern Italy

Introduction

The subject described here was found in a singleburial (No. 272) of the cemetery pertaining tothe S. Angelo Abbey, located in Mon-tescaglioso, 30 km southwest of Matera (Basili-cata, southern Italy). The site was excavated in1996 under the direction of Dr G. Canosa, thethen Director of the Museo Nazionale D. Ri-dola of Matera.

The burial was archaeologically dated be-tween 1100 and 1400 AD and appears as a ditchcovered by irregular stones lying under thesouthern corridor leading to the A Cloister(Canosa, personal communication). Both thecorridor and the A Cloister were built after1400 during work for the expansion of theperimeter of the abbey, thus suggesting that thetombs were originally located outside theboundaries (Venezia, personal communication).Unfortunately, there is a lack of historical docu-mentation about the use of the cemetery by themonks and/or by the local population.

The skeleton is fairly complete and in a goodstate of preservation, as only part of the skull,the atlas, axis, sternebrae, most ribs and part ofhand and foot bones are missing.

Sex diagnosis (male) was performed by mor-phological methods (Phenice, 1969; Ferembachet al., 1977), age at death (25–35 years) wasestimated by the degree of cranial suture closure(Meindl & Lovejoy, 1985) and by the modifica-tions of the pubic symphysis (Meindl et al.,1985). Stature (167.992.99 cm) was evaluatedby Trotter & Gleser’s (1952) equations for whitemales.

Palaeopathological findings

Skull

A vermicular aspect was observed on the innerside along the sagittal suture. Signs of porotichyperostosis were present on the outer tablenear the lambdoid suture.

Vertebral columnThis was the most affected skeletal region, withmassive new bone formation on the vertebralbodies, but without involvement of the articularsurfaces or the intervertebral spaces. In the tho-racic section (T6–T12) flowing ossificationswere located on the right side of the anteriorlongitudinal ligament, appearing as a dense andcontinuous line of bumps (Figure 1). In the

* Correspondence to: Universita degli Studi di Pisa, Dpt. DiEtologia, Ecologia ed Evoluzione, Unita di Antropologia, Via S.Maria 55-56126 Pisa, Italy. E-mail: [email protected]

CCC 1047–482X/99/050369–05$17.50Copyright © 1999 John Wiley & Sons, Ltd.

Received 26 November 1998Revised 26 January 1999

Accepted 30 April 1999

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lumbar section (L1–L5), a discontinuous ossifi-cation of the anterior longitudinal ligament wasbilateral and showed a candle flame shape. Thecervical section (C3–C7) did not show modifi-cations. Ankylosis of the apophyseal and cos-tovertebral joints was not present.

Shoulder girdle and ribsOn the two scapulae ossification of the cora-coacromial ligament was bilateral with involve-

ment of both the acromion and the coracoidprocess. The whole shoulder girdle showedarthropic changes. Manubrium sterni showed,on the left side, calcification of the sternocostalcartilage. Four of the first ribs on the right sideshowed signs of a healed fracture at the level ofthe costal angle. Arthropic changes were alsoobserved at the costovertebral joints.

Upper limbsThe right ulna showed a huge bony spur (14mm high) located on the olecranon at theinsertion of the triceps brachii muscle (Figure2). The left ulna had no such formation. Theright radius showed hyperostotic changes onthe radial tuberosity, while the left humerus hadan osteophyte on the medial epicondyle, corre-sponding to the insertion of the collateral ulnarligament. The preserved hand bones (carpal andmetacarpal bones and phalanges) did not showsigns of enthesopathies.

PelvisNew bone formation with definite and normaltrabeculae, most probably of traumatic origin,was observed on the right innominate boneextending for more than half the insertion areaof the gluteus minimus muscle. Bilateral hyper-ostosis was present at the level of the iliolumbarligament (height of the bone spur is 5 mm onthe right side and 7 mm on the left), of thesacroiliac ligament (spur height 3 mm right and2 mm left), of the sacrospinous ligament (spurheight 7 mm right and not measurable left), andof the sacrotuberous ligament (spur height 4mm right and left). Marked exostoses werepresent on the iliac crest, while the ischialtuberosity had a tufted and wispy aspect(Resnick et al., 1975).

Lower limbsThe femur showed bilateral changes in the hy-pertrochanteric fossa (insertion of the obturatorexternus muscle, spur height 5 mm on the leftand not measurable on the right side), of thelesser trochanter (insertion of the iliopsoas mus-cle), and of the linea aspera.

The patella had small osteophytes at theinsertion of the quadriceps femoris muscle. Thetibia showed bilateral enthesopathies on the

Figure 1. Lateral view (right side) of fused thoracic vertebrae(T6–T12) of the male subject from Tomb No. 272 of Mon-tescaglioso Abbey (Matera, southern Italy, 1100–1400 AD).Note the ossification of the anterior longitudinal ligament andthe intact intervertebral space (white arrows) (scale 1:0.8).

Copyright © 1999 John Wiley & Sons, Ltd. Int. J. Osteoarchaeol. 9: 369–373 (1999)

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Figure 2. Proximal portion of right (left side of the graph) and left ulna of Montescaglioso S. Angelo adult male from Tomb No.272. Note the bony spur on the right olecranon (white arrow) (scale 1:0.7).

proximal articular surface corresponding to theinsertion of the cruciate ligaments and hyperos-totic changes on the lateral intercondylar tuber-cle. The tibial tuberosity and the interosseouscrests had a normal aspect.

The calcaneum had strong bony spurs at theinsertion of the Achilles tendon (height 10 mmon the right, 8 mm on the left side). Signs ofplantar bursitis were also present.

The talus, tarsal and metatarsal bones andphalanges showed slight arthropic changes.

Conclusions

The changes caused by diffuse idiopathic skele-tal hyperostosis (DISH) have the greatest simi-larity to the alterations observed in this case,especially considering the typical flowing ossifi-cation of the anterior longitudinal ligament ofthe thoracic vertebrae (Crubezy, 1990), and theossification of the iliolumbar, sacroiliac,sacrospinous and sacrotuberous ligaments (Fig-ure 3). In the long bones of the limbs, thecharacteristic bilateral symmetry of the alter-ations is generally respected, with the exception

of the forearm, where only the right radius andulna are affected. The knee, usually involved intypical DISH cases, showed in this subject onlysome changes at the level of the patella. Finally,the age at death, if not underestimated by themethods applicable to the skeleton under study,is relatively young for a typical DISH case. Inspite of these seeming discrepancies, we con-sider that a diagnosis of DISH is the mostprobable for the male skeleton from tomb No.272 of the S. Angelo abbey.

Among the hypotheses so far advanced onthe aetiology of DISH, Hajkova et al. (1965)mention a correlation between diabetes mellitusand DISH, although the disease does not showa different manifestation in diabetic or non-dia-betic patients. According to other authors(Julkunen et al., 1971), DISH would be associ-ated with obesity and hyperglycaemia. In partic-ular, for a medieval case, Waldron (1985)suggests a relationship between DISH and reli-gious life, considering the type and amount offood intake in monks as compared with com-mon people. A similar association betweenmonastic life, obesity and degenerative jointdisease is reported for three Basque skeletons

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(de la Rua & Orue, 1993), and supported byhistorical data. Unfortunately, our case, al-though from an abbey’s cemetery, cannot beattributed with certainty to a monk.

Julkunen et al. (1971) note a high prevalenceof DISH among farmers, pointing to the role ofphysical strain as a correlate of the disease.Other authors (Crubezy, 1990) observe an asso-ciation between DISH and osteoarthritis. Morerecently, Arlet & Mazieres (1985) suggested thata metabolic imbalance of vitamin A or of itsbinding protein might be among the causes ofidiopathic hyperostosis.

On the basis of osteological studies most ofthe above hypotheses cannot be supported ordenied, but bone robustness and the develop-ment of muscular insertions can give some infor-mation about physical stress during life. As amatter of fact, our male subject showed thesigns of an intense muscular activity, also con-

sidering his relatively young age at death. Theskeletal changes indicative of an initial stage ofthe illness (see, for example, the absence ofchanges in the cervical section of the spine, thelow number of extraspinal hyperostoses and theunilateral manifestation in the forearm), wouldbe in agreement with the estimate of the rela-tively young age at death, since the pathologyrequires a long lapse of time to become stronglyevident. The case under study also points to theassociation between DISH and osteoarthritis.

Acknowledgements

Thanks are due to Dr M.G. Canosa, formerDirector of the Museo Nazionale D. Ridola ofMatera for entrusting this study, to Drs A.Canci and V. Formicola for useful discussionand to Drs M.G. Canosa and P. Venezia forhistorical and archaeological data.

References

Arlet, J. and Mazieres, B. (1985) La maladie hyperos-tosique. Revue de Medecine Interne, 5: 553–564.

Crubezy, E. (1990) Diffuse idiopathic skeletal hyper-ostosis: diagnosis and importance in paleopathol-ogy. Journal of Paleopathology, 3: 107–118.

de la Rua, C. and Orue, J.M. (1993) Health condi-tions in a monastic community of the BasqueCountry (16th and 17th centuries). Journal of Pale-opathology, 4: 193–200.

Ferembach, D., Schwidetzky, I. and Stloukal, M.(1977) Raccomandazioni per la determinazionedell’eta e del sesso sullo scheletro. Rivista di Antro-pologia, 60: 5–51.

Hajkova, Z., Streda, A. and S& krha, F. (1965) Hyper-ostotic spondylosis and diabetes mellitus. Annals ofthe Rheumatic Diseases, 24: 536–542.

Julkunen, H., Heinonen, O.P. and Pyorala, K. (1971)Hyperostosis of the spine in an adult population.Its relation to hyperglycaemia and obesity. Annalsof the Rheumatic Diseases, 30: 605–612.

Meindl, R.S. and Lovejoy, C.O. (1985) Ectocranialsuture closure: a revised method for the determina-tion of skeletal age at death based on the lateral–anterior sutures. American Journal of PhysicalAnthropology, 68: 57–66.

Meindl, R.S., Lovejoy, C.O., Mensforth, R.P. andWalker, R.A. (1985) A revised method of age

Figure 3. General distribution of bone and joint involvement inthe adult male subject from Tomb No. 272 of the Mon-tescaglioso S. Angelo abbey (Matera, Southern Italy).

Copyright © 1999 John Wiley & Sons, Ltd. Int. J. Osteoarchaeol. 9: 369–373 (1999)

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determination using the os pubis, with a reviewand tests of accuracy of other current methods ofpubic symphyseal aging. American Journal of PhysicalAnthropology, 68: 29–45.

Phenice, T.W. (1969) A newly developed visualmethod of sexing the os pubis. American Journal ofPhysical Anthropology, 30: 297–302.

Resnick, D., Shaul, S.R. and Robins, J.M. (1975)Diffuse idiopathic skeletal hyperostosis (DISH):

Forestier’s disease with extraspinal manifestations.Radiology, 115: 513–524.

Trotter, M. and Gleser, G.C. (1952) Estimation ofstature from long limb bones of American Whitesand Negroes. American Journal of Physical Anthropol-ogy, 10: 463–514.

Waldron, T. (1985) DISH at Merton Priory: evi-dence for a ‘new’ occupational disease? British Med-ical Journal, 291: 1762–1763.

Copyright © 1999 John Wiley & Sons, Ltd. Int. J. Osteoarchaeol. 9: 369–373 (1999)