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PALEOBIOS ,13 / 2004 / Lyon-France ISSN 0294-12/Relationship between Porotic Hyperostosis and Cribra Orbitalia ? Relationship between Porotic Hyperostosis and Cribra Orbitalia ? Bruce M. Rothschild (1), Frank J.Rühli (2), Jeno Sebes (3), Virginia Naples (4) & Michel Billard (5) ( 1): Professor of Medicine at Northeastern Ohio Universities College of Medicine (NEOUCOM);Adjunct Professor of Biomedical Engineering, University of Akron; Research Associate at the Carnegie and University of Kansas Museums of Natural History and Director of The Arthritis Center of Northeast Ohio, Youngstown, OH 44512. (2): Assistant at Institut of Anatomy, research fellow at Institute for the History of Medicine, University of Zurich, Winterthurstr.190, 8057 Zurich, Switzerland (3): Professor of Radiology at University of Tennessee, Memphis, TN 38119, USA. (4): Professor of Biology at Northern Illinois University, De Kalb, IL 60115, USA. (5): Research fellow at Laboratory of Anatomical Anthropology and Paleopathology, University of Lyon, Rockefeller Av.8, 69373 Lyon, France Reprint requests to: B. M. Rothschild, Arthritis center of Northeast Ohio, 5500 Market Street, Suite 119, Youngstown, Ohio 44512, USA. Tel: 330-783-5900; Fax: 330-783-5350; e-mail : bmr@neoucom.edu ABSTRACT Objective : To assess the relationship of porotic hyperostosis and cribra orbitalia. Previous analysis has been compromised by the untested assumption that these two cranial phenomenon share pathophysiology, that they are anemia-derived. Methods : Skulls from New World and Old World archeologic sites were macroscopically examined. Results : An inverse relationship between cribra orbitalia and porotic hyperostosis was documented. Conclusion : While cribra orbitalia and porotic hyperostosis may occasionally occur in the same individual, they are clearly manifestations of disparate processes. Running title: Cribra orbitalia & porotic hyperostosis Key words: Anemia, cribra orbitalia, disease, paleopathology, pathology, porotic hyperostosis, RESUME L'examen macroscopique de crânes de l'Ancien et du Nouveau Monde montre une relation inverse entre cribra orbitalia et hyperostose porotique: ces deux atteintes n'apparaissant seulement que de temps en temps sur un même individu, elles sont clairement la manifestation de processus distincts, alors que les études précédentes sont compromises par l'hypothèse non vérifée que ces deux phénomènes crâniens partagent physio-pathologiquement une même origine (l'anémie). Titre: Relation entre hyperostose porotique & cribra orbitalia Mots clés : Anémie, cribra orbitalia, maladie, paléopathologie, pathologie, hyperostose porotique 1 - INTRODUCTION Porotic hyperostosis (expansion of the diploic space) seems clearly documented as a manifestation of marrow hyperplasia (Angel, 1978; Hill & Armelagos, 1990; Salvadei et al., 2001; Stuart-Macadam, 1985, 1989). Review of the physical anthropology literature additionally reveals frequent lumping of cribra orbitalia (disruption of the orbital roof) first named by Welcker in 1888 (1888) and porotic hyperostosis (Mensforth et al., 1978; Salvadei et al., 2001; Williams, 1929). Such lumping is predicated, however, on the untested assumption that they are related. Variation in the gross and histologic appearance of cribra orbitalia (Schultz, 1993; Smay & Armelagos, 2000) suggests that it might actually represent more than one entity/etiology, rather than simply a severity gradation as suggested by Hengen (1971). Does cribra orbitalia actually define a specific entity/etiology or are there several phenomenon which cause supraorbital lesions? One such hypothesis has been anemia (Angel, 1978; Mensforth et al., 1978; Stuart-Macadam, 1987, 1989), predicated upon association with porotic hyperostosis. Data-based approach (Amin, 2002) to that question requires examination as to whether such an association actually exists. This is analogous to the alleged inverse relationship of osteoarthritis to loss of bone density (osteoporosis). Skulls from North American and Asian sites, ranging from Archaic to protohistoric, were therefore examined to assess whether any correlation actually exists between cribra orbitalia and porotic hyperostosis. 2 - METHODS Skulls from North American, European and Asian sites were examined for macroscopic signs of porotic hyperostosis and cribra orbitalia. The groups studied (Table 1) included Wedda, Altern, Blue Blanket Point, the Bonnell Site, Greenville, Lake Bronson, the McCutchan-McLaughlin site, Morrison's Island, Peter Lee Mound, Pindi Pueblo, Seh Gabi, Warner Mound, Gandaillat, Gerzat Chantemerle and Sarlieve (Cybulski, 1992; Kaufmann, 1977; Lewis, 1998; Myster & O'Connell, 1997; Pfeiffer, 1977; Powell & Rogers, 1980; Sarasin & Sarasin, 1892/3; Stodder, 1999; Waddell, 1994; Williams, 1997).
Porotic hyperostosis was recognized on the basis of scattered fine to large foramina, linkage of such foramina to form a trabecular structure or outgrowth of such a structure from the outer bone table (Stuart-Macadam, 1985). This is easily distinguished from hemorrhagic phenomenon (e.g., subdural or epidural hematoma) and inflammatory conditions (e.g., meningitis) which are endocranial in distribution, sparing the ectocranium or outer bone table (Resnick, 2002). Cribra orbitalia was recognized on the basis of isolated or conglomerate small or large pores or accretion of trabecular bone in the superior aspect of the orbit (Nathan & Haas, 1966; Stuart-Macadam, 1985). 3 - RESULTS Both porotic hyperostosis and cribra orbitalis were found in the Bonnell, Greenville, Seh Gabi and Wedda samples, but were mutually exclusive among affected individuals (Table 2). Cribra orbitalia was present as the sole pathology in skulls from Altern, Peter Lee Mound, Pindi Pueblo, Gandaillat, Sarlieve and Gerzat Chantemerle. This contrasted with the Blue Blanket Point, Lake Bronson, McCutchan-McLaughlin, Morrison's Island and Warner Mound sites, where only porotic hyperostosis was found. Porotic hyperostosis was predominantly parietal in distribution. As no individual with porotic hyperostosis had cribra orbitalia (Table 2), the character and severity in the various individuals is not pertinent to the primary question: Occurrence of cribra orbitalia and porotic hyperostosis is statistically segregated (Chi square = 3.98, P < 0.05).
4 - DISCUSSION Complete and statistically significant segregation of porotic hyperostosis and cribra orbitalia in multiple sites documents the two phenomena should not be lumped as manifestations of the same disorder. As the sites represent such geographically and chronologically disparate proveniences as protohistoric and Archaic in both the New (North America) and Old (Asia) World, more general extrapolation seems appropriate. Why has this not been previously recognized? After all, Stuart-Macadam (1989, p. 187) considered the two related and having 'a common etiology.' However, she also reported orbital lesions as the sole finding in 173 individuals, vault lesions as the sole finding in 7 individuals and both orbital and vault lesions in 50 individuals, among the 546 Poundbury skulls she examined. Chi square analysis (Chi square = 57.88) of her data (not reported in the 1989 article) revealed P < 0.0001 a statistically significant lack of correlation. This current study, and indeed Stuart-Macadam's 1989 study, clearly refute the assumption that porotic hyperostosis and cribra orbitalia are directly relatable phenomenon. Until this matter is clarified, they should be recorded separately. Equally important will be establishment of the cause of various 'components' of cribra orbitalia (e.g., resorptive and proliferative lesions). Schultz' (1993) exquisite histologic studies illustrate the spectrum of supraorbital pathologies, but awaits correlation with clinical cases in which diagnosis were made in life.We await rapproachment of anatomy and pathology, enabling histologic analysis of individuals actually diagnosed with anemia or inflammatory eye disease in life (evidence-based analysis). ACKNOWLEDGEMENTS We thank Dr. Felix Wiedemeyer of the Natural History Museum of Basel for assistance with this study and Jerome Rose for cogent input to the subject. 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PALEOBIOS ,13 / 2004 / Lyon-France ISSN 0294-12/Relationship between Porotic Hyperostosis and Cribra Orbitalia ? |