LEGACY

NEW WORLD CRANIAL DEFORMATION PRACTICES: HISTORICAL IMPLICATIONS FOR PATHOPHYSIOLOGY OF COGNITIVE IMPAIRMENT IN DEFORMATIONAL PLAGIOCEPHALY
Gregory P. Lekovic, M.D., Ph.D., J.D.
Division of Neurological Surgery, Barrow Neurological Institute, St. Josephs Hospital and Medical Center, Phoenix, Arizona

Brenda J. Baker, Ph.D.

Department of Anthropology, Center for Bioarchaeological Research, School of Human Evolution and Social Change, Arizona State University, Tempe, Arizona

Jill M. Lekovic, M.D.

Department of Pediatrics, St. Josephs Hospital and Medical Center, Phoenix, Arizona

Mark C. Preul, M.D.

Division of Neurological Surgery, Barrow Neurological Institute, St. Josephs Hospital and Medical Center, Phoenix, Arizona Reprint requests: Mark C. Preul, M.D., c/o Neurosurgery Research, Barrow Neurological Institute, 350 W. Thomas Road, Phoenix, AZ 85013. Email: neuropub@chw.edu Received, September 14, 2006. Accepted, January 31, 2007.

INTRODUCTION: Throughout history, prehistoric and even some contemporary civilizations have practiced various forms of intentional and unintentional cranial deformation. Plagiocephaly can be the result of craniosynostosis, infant positioning, or other unintentional or intentional deformation. MATERIALS: We reviewed the medical and anthropological literature and utilized the anthropological collections of Arizona State University and the San Diego Museum of Man for evidence of cranial deformation and its possible physiological and cognitive side effects. Evidence of cranial shaping was also sought among art or stone work from representative cultures. RESULTS: The anthropological record and literature attest to the presence of much more severe forms of deformation than that seen as a result of contemporary infant positioning. Despite this evidence, there is no anthropological evidence as to the possible cognitive effects that such deformation may have, although some evidence is reviewed that suggests a possible physiological mechanism for the same. CONCLUSION: Because we can only view these cultures through the relics of time, any conclusions one might draw from the anthropological and historical record regarding the cognitive effects of head deformation can only be inferred through generalized observations and are tenuous. Nevertheless, there does not seem to be any obvious evidence of negative effect on the societies that have practiced even very severe forms of intentional cranial deformation (e.g., the Olmec and Maya). On the other hand, the physical anthropology and the contemporary developmental literature suggest possible mechanisms for such an effect.
KEY WORDS: Cognitive impairment, Cranial deformation, Craniosynostosis, Neurosurgical history, Plagiocephaly
Neurosurgery 60:11371147, 2007
DOI: 10.1227/01.NEU.0000255462.99516.B0

www.neurosurgery-online.com

lagiocephaly can be caused by craniosynostosis or deformation and has been associated with impaired cognition (2226). This issue has become more pressing because the incidence of deformational plagiocephaly has increased since the American Academy of Pediatrics adopted the Back to Sleep campaign (50). However, whether or not plagiocephaly per se plays a causative role in such impairment remains controversial. To gain perspective on this issue, we examined the historical and archeological records of cultures that routinely practiced severe forms of cranial deformation. These practices created deformational

plagiocephaly that was far more acute than what is typically encountered by practitioners of pediatric neurosurgery today. Clearly, the effects of these practices on cognition can only be surmised. Nonetheless, such a survey may offer insights relevant to the current concerns about positional plagiocephaly. We examined human skeletal remains from the collections of Arizona State University and the San Diego Museum of Man and reviewed the anthropological literature on the intentional deformational practices among prehistoric Native Americans to examine whether or not the possible effects on cognition could be

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inferred from the anthropological record. Several reviews describe the general practices of intentional cranial deformation in many parts of the world (5, 1921, 38, 51). None, however, concentrate on its possible effects on cognition. Current thought on the treatment of head deformation in pediatrics and neurosurgery is a convenient context for this historical examination. It is impossible to assess the cognitive abilities of individuals or groups in cultures that once practiced cranial deformation. However, their remains, coupled with cultural evidence, may suggest implications relevant to the contemporary debate on the effect of positional deformational plagiocephaly on cognition.

MATERIALS AND METHODS

The anthropological, neurosurgical, and pediatric literature on cranial deformation was reviewed, specically from readily available New World cultural evidence. Representative examples of cranial deformation in archaeological collections of human skeletal remains, mainly from ancient cultures of the Southwest United States, from the School of Human Evolution and Social Change at Arizona State University in Phoenix and from the San Diego Museum of Man were used to demonstrate types of alteration in past peoples and for purposes of illustration. Evidence of cranial shaping was sought among art or stone work from representative ancient cultures of the New World, including the Maya, Olmec, and Inca, and various ancient Indian cultures of North America.

The Problem: Does Deformational Plagiocephaly Cause Cognitive Impairment?

Interestingly, the debate about the impact of articial head shaping on cranial and brain development and cognition intrigued even some of the most prominent of mid-19th century neuroscientists such as Rudolf Virchow and Pierre-Paul Broca (16). Likewise, in the late 1950s, nearly the same discussions on potential impacts of cranial deformation were taking place at the Johns Hopkins Hospital between George Anderson, then assistant professor of pediatrics, Earl Walker, chief of neurosurgery, and the noted anthropologist, anatomist, and scientist, Ashley Montagu. Their discussions and letters document interest in better treatment for plagiocephaly or other malformations of the cranium, and were related to a large National Institute of Neurological Disease and Blindness collaborative project into identifying the etiology of neurological disease and defect in children (44). The Back to Sleep recommendation of the National Institute of Child Health and Human Development and the American Academy of Pediatrics was adopted in 1992. This campaign was an attempt to reduce the incidence of sudden infant death syndrome, which has been linked in epidemiological studies to infants sleeping in the prone position. Since the adoption of the campaign, the rate of sudden infant death syndrome has dropped by an impressive 40%. However, an unintended consequence has been an increase in the incidence of plagio-

cephaly attributable to infant head positioning (3, 50). In a community-based practice environment, for example, more than 15% of patients measured prospectively showed evidence of at least mild positional deformation, whereas 1.5% had a severe deformity associated with concomitant facial and cranial base deformation (32). Recognizing that a supine sleep position may contribute to deformational plagiocephaly, the American Academy of Pediatrics Task Force on Infant Sleep Position and Sudden Death Syndrome recommended tummy time (i.e., prone positioning when awake and observed) for infants to promote upper shoulder girdle strength and to avoid plagiocephaly. The Task Force also recommended alternating head position weekly to prevent children from developing a preference for one side over the other (26). Recently, the use of dynamic orthotic cranioplasty has garnered publicity as an alternative treatment to changing infant head position. In uncontrolled studies, the apparatus restored normal head shape better than standard repositioning techniques (26). Whether or not the mildly superior performance of orthotic cranioplasty over repositioning justifies the expense of the orthosis depends partially on the indications for treatment. Supine-sleeping infants were found to have gross motor milestones later than prone-sleeping children (15). This difference has been attributed to relative weakness in upper shoulder girdle strength among infants sleeping supine and disappears after 18 months of age (14). Overall, these initial concerns that supine sleeping may be associated with adverse developmental effects have received little support. However, several studies have provided evidence of adverse cognitive effects from plagiocephaly caused by positional occipital deformation. Indeed, increases in the incidence of learning disabilities among patients with persistent deformational plagiocephaly may be comparable to those found in patients with craniosynostosis. Miller and Clarren (35) found that the rate of learning disabilities among 63 patients with persistent deformational plagiocephaly was 39.7% compared with 7.7% of the siblings of affected children who served as controls. Parents were interviewed about their childs school performance and participation in an early intervention program, special education classes, or other special aid. Compared with the controls, signicantly more children with deformational plagiocephaly required such services. Among 35 patients ranging from 4 to 48 months of age with pure deformational plagiocephaly (i.e., excluding patients with any diagnosis of genetic disorder, prematurity, or syndrome), Habal et al. (28) found a 51% incidence of language delays or disorders. Between 1997 and 1999, Panchal et al. (43) used the Bayley Scales of Infant Development to assess 42 consecutive infants with deformational plagiocephaly. These infants had a significantly elevated incidence of severe developmental delay (8.7%). Differences in auditory-evoked responses predictive of later cognitive functioning have been studied in infants with plagiocephaly (9, 36). Balan et al. (4) evaluated auditory-evoked

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potentials in 15 patients with plagiocephaly, including 10 with positional plagiocephaly, two with lambdoid synostosis, and three with anterior plagiocephaly. Irrespective of the cause of plagiocephaly, these infants had smaller amplitude P150 and N250 responses than normal controls. The ndings support a common etiology of cognitive impairment in craniosynostosis and deformational plagiocephaly. The causal relationship between head deformation and adverse cognitive sequelae is, however, difficult to establish because many comorbid conditions may predispose a child to develop deformational plagiocephaly. Hypotonic children, such as those with mental retardation, chromosomal abnormalities, and cerebral palsy, or children with torticollis, are at risk for deformational plagiocephaly because they develop a preference for one head position over the other. Still, a strong circumstantial argument can be made against the likelihood of deformational plagiocephaly, per se, having a significant effect on childrens cognitive development, namely that head deformation, whether unintentional or by design, is a widespread cultural phenomenon among prehistoric and indigenous peoples.

Anthropological and Historical Contexts of Deformational Plagiocephaly

Cranial deformation practices, whether intentional or unintentional, have been a common feature in many ancient societies. Indeed, in many parts of the world such practices continue today.

Unintentional Cranial Deformation

Unintentional shaping of an infants cranium is the consequence of habitual positioning. Many historic and prehistoric Native American groups, particularly in the Southwest and Plains, used cradleboards to contain an infant while it was carried or sleeping, or while the mother was engaged in other activities. The use of cradleboards attens the occiput, much like the deformation seen in contemporary American infants who are habitually placed on their backs to sleep (Fig. 1A). As in contemporary infants, the deformation was often asymmetrical as a result of an infants preference for looking toward one side or another (Fig. 1B). Positional plagiocephaly is common in prehistoric Southwestern skeletal samples and extends across a considerable length of time. Cradleboards have been found from the Basketmaker II period (c. 100 BCAD 400). Occipital deformation was also common in the Adena and Hopewell people and at some Mississippian and protohistoric sites in the Southeast and Midwest (40). These people, however, did not use a written language. Thus, the learning disorders thought by some (4, 28, 50) to be associated with deformational plagiocephaly may have been too subtle to be recognized by people in societies in which cranial deformation was common.

also widely practiced in the past and continues today. Dingwalls (16) 1931 book, Artificial Cranial Deformation: A Contribution to the Study of Ethnic Mutilations, remains unequaled for its breadth and detail of the topic, surveying the history of the practice around the world. Gerszten and Gerszten (20) and Goodrich and Tutino (21) reviewed information on intentional cranial deformation as it relates to the history of neurosurgery and to the development of craniofacial surgery from various parts of the world. Anatomists and others (17, 27, 29, 34, 47) have been fascinated with artificial cranial deformation even before the birth of physical anthropology as a formal discipline, as exemplified by works like Samuel Mortons (37) Crania Americana; or a Comparative View of the Skulls of Various Aboriginal Nations of North and South America, published in 1839. This volume contains 72 plates, principally of skulls from North and South America and the Caribbean. Extreme examples are best known from the Northwest Coast, Peruvian cultures, and the ancient Olmec, Toltec and Maya in Mesoamerica. Intentional deformation was also common in the southwestern and southeastern United States (12, 40, 48). Known European cases date from the Neolithic period, and have been found in Britain (7), among the Huns of eastern and central Europe (49), and in ancient Cyprus (33). In antiquity, head shaping was practiced on every inhabited continent, with evidence from Iraq as early as 45,000 BC (42) and in fossilized remains of early Homo sapiens from Australia (2). Devices used to shape heads include cloth bindings wrapped around the infants head, anteroposterior compression via boards or stones bound together and placed around the head, and cradleboard attachments that compress the forehead. In some cultures, mothers molded and squeezed the heads of their infants between their hands while nursing, changing them from breast to breast so that a desired symmetrical appearance could be obtained (16). Most cultures used cranial modication as a marker of ethnic identity or social status. It was also done to appear ferocious in battle and for cultural or racial imitation. Deformation and sex also correlate,

Intentional Cranial Deformation: Headshaping

Intentional shaping of infants heads, resulting in more extreme deformation of the cranium than cradleboarding, was

FIGURE 1. Example of unintentional cranial deformation among the Southwest Native Americans. Lateral (A) and basilar (B) views with occipital attening caused by cradleboarding showing plagiocephaly similar to the positional plagiocephaly that results from positioning infants supine. Photographs courtesy of Charles Merbs, Ph.D., with permission.

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with males usually exhibiting a more extreme appearance, although in a few cultures, females were more likely to show such deformation (16, 19, 21, 49). In Mesoamerica, early Olmec (1400 BC400 AD) gurines have a characteristic elongated head (Fig. 2). Such cranial deformations are thought to have been common throughout the Olmec civilization. The Olmecs, the forerunners of civilizations in Mexico, are considered to be a dynamic culture with mathematics, a calendar, a well-developed system of writing, art, farming, and relatively large-scale engineering projects (11, 39, 54).

As many as 90% of Mayan skeletal remains exhibit evidence of intentional, decorative, or cosmetic cranial deformation practices (48). Patterns of deformation varied according to geographical regions and periods. However, the prevalence of cranial deformation remained constant throughout the Preclassical and Postclassical periods of Mayan civilization (100 BC1521 AD), during which time there was a stable progression of Mayan arts and sciences. Unlike the unintentional cranial deformation that occurred as a consequence of cradleboarding, deformation in Mayan crania is much more severe. Our ability to conclude that deformational plagiocephaly did not adversely affect the cognition of Native Americans who practiced cradleboarding is hampered by the absence of a written language. Mayan culture, however, did practice writing. Presumably, adverse effects of head shaping among the literate elite would have been obvious to others, as cognitive impairment would have hampered reading and writing capabilities (Fig. 3). Although it is impossible to assess the effects of Mayan cranial deformational practices on cognition, the anatomic and archaeological records provide a circumstantial argument against a negative consequence (Fig. 4). It is logical to assume that cultures would associate changes in cognition or deleterious effects on a childs health with severe head shaping. Such practices would likely have died out quickly, especially among the Mayans or Peruvians, for example, where severe intentional head shaping was comparatively widespread. Nevertheless, the practice seems to have been most common among ruling, religious, or elite classes. The consequential cognitive sequelae of severe cranial deformation may have been culturally accepted and identied with these social classes of unique standing, and thus continued. Or, perhaps the sequelae were not behaviorly obvious. Among anthropologists of the early to mid-19th century, it was thought that the skulls of ancient Peruvians were naturally elongated with retreating foreheads. The idea of the Peruvian skull as being naturally at head . . . was discussed in the report of the Council of the American Antiquarian Association in 1855. . . . It is not difcult to understand why the earlier inquirers failed to grasp the fact of Peruvian articial deformation. The custom was so widespread, and the sites which attracted early attention provided such a mass of specimens, that the theory that such conformations were natural was easy of acceptance (16). Among 500 skulls unearthed from Peru (ancient Inca) in the mid-19th century housed in Paris and another similar number in the University Museum at Rome, only 10% did not show extraordinary skull deformations (16). Furthermore, 19th century studies from France on living subjects found no evidence of brain injury or cognitive impairment associated with severe head shaping (52).

FIGURE 2. A, carved jade gurine of a seated Olmec ruler in a ritual pose displaying a cone-like cranial shaping, Mexico state of Puebla, San Martn Texmelucan, Highland Olmec culture, c. 900500 BC. Photograph courtesy of the Dallas Museum of Art, with permission. B, ceramic gurine showing cranial deformation in Olmec artwork. Note the lateral concave shaping. Photograph courtesy of Jorge Prez de Lara, with permission. Frontal (C) and side (D) views of a skull from Mortons Crania Americana showing a deformation common to the Natchez tribe of the southcentral United States, which was similar to that of the Olmec. The Natchez may have had origins in or close associations with cultures in Mexico. From, Morton SG: Crania Americana; or a Comparative View of the Skulls of Various Aboriginal Nations of North and South America. Philadelphia, J Dodson, 1839 (37).

Impact of Deformation on Cranial Growth and Development

In the anthropological literature, the application of devices to shape the head have usually been interpreted to act primarily

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FIGURE 3. A, Mayan codex vesC sel from the Middle Classic Period (AD 400600) depicting the elongated head of the Maize God being born from a seed. B and C, two of a set of three Yaxchilan limestone lintels, Numbers 24 and 26, (Late Classic Mayan Period, AD 600900), each of which formed the ceiling of a deep doorway to one of the Yaxchilan temples three separate rooms. As worshipers entered, they looked up to see the scene overhead. The lintels were commissioned by Lady Xoc and feature her performing crucial duties during the reign of her husband. The stonework shows the attening of the forehead and elongated, conical head of a famous Mayan king and queen. In royal Mayan courts, women lled important civic and religious roles denied them in many Mesoamerican cultures. On the rst of these lintels (B), Lintel 24, commemorating a blood-letting rite in AD 709, Shield Jaguar stands holding a aming torch with Lady Xoc kneeling in front of him. As she runs a rope studded with thorns or spines through her tongue, spots of blood collect on the paper in the basket in front of her. In Lintel 26 (C), commemorating a ritual in AD 725, Lady Xoc hands a jaguar helmet to Shield Jaguar, securing his place in the line of kings, or perhaps on his way to battle. Photographs courtesy of Jorge Prez de Lara, with permission.

on the outer table and secondarily on cerebrally influenced regions (1). Cheverud et al. (10) performed nite-element scaling on samples of deformed skulls from Peru and the Northwest Coast Salish to investigate the effects of fronto-occipital shaping. They found that physical reshaping of the neurocranial vault results in a series of compensatory bony changes required for normal neural growth, so that the size of the neurocranial vault remains the same. In this case, the compensatory growth involved the mediolateral aspect of the cranium. Thus, a 5% decrease in the anteroposterior dimensions of the face corresponded to a 5% increase in the width of the cranial base.

FIGURE 4. A and B, detail of Mayan stone panels found in Palenque's Temple XIX, carved in the early 8th century AD. The images show King Ahkal Mo' Nahb III (K'inich Ahkal Mo' Nahb), who was born on September 16, 678 and died after August 22, 731; he was grandson of the famous Mayan king, K'inich Hanab Pakal. Note the oblique tabular cranial shaping in which the forehead becomes slanted and elongated. Also, Ahkal Mo' Nahb is typically depicted with what appears to be a nose-forehead prosthetic that accentuated the angle of cranial shaping. The curved line of the nose represents the true prole, and the prosthetic is seen above the nose. Photographs courtesy of Jorge Prez de Lara, with permission.

The relationship between cerebrally derived pressure on the cranial vault and sutural fusion, as evidenced by the tendency to promote premature sutural fusion in shunted hydrocephalus, has led some to posit a causal relationship between

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externally applied forces and sutural fusion. Graham et al. (2226) and Koskinen-Moffett et al. (31) have argued that persistent deformation in utero may promote premature fusion. According to this view, deformational plagiocephaly exists on a continuum with synostosis per se. Although this hypothesis is widely cited, experimental models of sutural constraint in rabbits and sheep have failed to demonstrate such an association (6, 18). The persistence of sutures in mechanically deformed skulls is evident in the anthropological literature. Several anthropological studies have focused on the impact of artificial cranial deformation on sutural complexity. An increase in the frequency of wormian bones (sutural ossicles) in the lambdoidal suture has been suggested (16, 41, 45, 46). Pucciarelli (45) concluded that higher frequencies of wormian bones were found in experimentally deformed rat crania and that such experimental deformation may be an extragenetic expression of wormian bones. He inferred that this evidence may be relevant to studies on racial differences based on discontinuous cranial traits (16, 46). Konigsberg et al. (30), however, found that the frequency of coronal ossicles increases regardless of the type of deformation. They observed that ossicles typically occur near the position of pads or a wrapping device. Their study suggests that biomechanical forces operating on the skull as it is shaped promote compensatory growth at the sutures where force is applied. This idea is supported by Konigsberg et al.s (30) statement that nonmetric traits that develop prenatally are unaffected by deformation. White (53) found a strong association between sagittal synostosis and lambdoidal wormian bones among Mayans at Lamanai, Belize. The high frequency of synostosis in undeformed skulls suggested an underlying heritability. However, the increased frequency of wormian bones in individuals with fronto-occipital deformation reinforces the importance of sutural development in maintaining overall skull size. Environmental factors seem to affect posterior sutures more than anterior sutures, perhaps a result of the rapid growth of the posterior cranium immediately after birth (41). However, none of these studies demonstrated a signicant increase in sutural fusion in deformed skulls, irrespective of the extent of deformation. Investigating endocranial vascular changes related to both synostosis and articial cranial deformation, OLoughlin (41) found signicant alterations in the patterns of venous sinus drainage between normal, synostosed, and deformed skulls. Both artificial deformation and craniosynostosis altered endocranial vasculature, but the patterns differed. Venous sinus impressions are shallower under the areas of greatest deformational stress; those further away from the stress show compensatory enlargement. In synostosis, however, the sinuses are wider and deeper under the fused sutures. Moreover, 71% of occipitally deformed skulls demonstrated enlarged occipital and marginal sinus impressions, and the frequency of left transverse sinus dominance increased in right-sided asymmetrically deformed skulls. This nding demonstrates both developmental plasticity of venous outow channels and also testies to the ability of external compressive forces to affect them. It could be

the basis for a pathophysiological mechanism common to both craniosynostosis and external deformation, namely, regional venous outflow obstruction and subsequent consequential increases in local intracranial pressure. Using silicone casts in rats, Burrows et al. (8) compared the changes in endocranial vascular channels in experimental conditions of immobilization and synostosis. The dimensions of the posterior dural venous sinus were significantly reduced in synostosed rats compared with normal or immobilized rats. They concluded that the endocranial events were secondary phenomena that reflected regionally altered sutural dynamics, including increased intracranial pressure, local reductions in intracranial pressure, or accumulations of cerebrovascular fluid. Imaging studies seem to point toward a decrease in cerebral glucose metabolism in the occipital cortex in children with synostosis. Using positron emission tomographic imaging of children with single suture synostosis (metopic, coronal, or sagittal), David et al (13) showed regionally variable signals for glucose metabolism, whereas there was a consistent decrease in the posterior occipital region, the area of visual development and visual spatial coordination. After surgical release of the suture, there were signicant increases in maximal and average glucose metabolism in the occipital cortex. None of these studies has focused on cognition or been able to link events surrounding cranial or brain malformation or vascular alteration to effects on cognition.

Anthropological Literature on Cognitive Ability

Although Dingwalls (16) compendium is rich in detail on the scope and means of cranial deformation (especially for New World cultures), there are only minor relevant comments associating it with cognitive ability one way or the other; indeed, this was not the object of the work. Dingwall, in referring to extant cultures of which he had direct contact stated, It does not seem that the practice of head deformation among the North West Coast tribes affects their mental faculties (16). In his 1839 Crania Americana: Or a Comparative View of the Skulls of Various Aboriginal Nations of North and South America, Morton (37) described the devices used for shaping heads in detail. Despite the deformation, he noted that the absolute internal capacity of the skull is not diminished, and, strange as it may seem, the intellectual faculties suffer nothing (37). He cited journals of travelers to the area to support this contention. Nineteenth-century American explorers, including Lewis and Clark, described the extreme cranial deformations of Native Americans in the Columbia River Basin. While visiting the Chinook, Dingwall noted that both sexes had their heads flattened in a most disgusting manner (16), whereas other explorers tended to discount the deformations as hidden by the hair. However, the explorer J.K. Townsend, in his 1839 Narrative of a journey across the Rocky Mountains to the Columbia River, recounts the observation of a young child from whom the deforming apparatus had just been removed. The entire frontal region was flattened, causing an enormous projection

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behind, and the childs protruding eyes were inflamed, and discolored as were the surrounding orbital areas. However, the child seemed to behave and react normally to its mother and others (16). Collectively known at the time as Flatheads, these groups practiced parallelofronto-occipital deformation by a board or bindings placed across the infants forehead while it was positioned in a cradleboard (Fig. 5). Lewis and Clark are quoted as nding these people inquisitive and loquacious, with understandings by no means decient in acuteness, and with very retentive memories.Every thing they see excites their attention and inquiriesall our inquiries they answered with great intelligencethey are keen, acute and intelligent (37). Morton had occasion to meet one full-blood Chenouk whose head was as much distorted by mechanical compression as any skull of his tribe in my possession, and presented the very counterpart to the Kalapooyah gured on the annexed plate (Fig. 5) (37). According to Morton, who gained the young mans consent to measure his skull, he appeared to me to possess more mental acuteness than any Indian I had seen (37). Although later derided for many of his racial categorizations and conclusions based on cranial measurements and phrenological observations of various cultures, Mortons information on cranial shaping among American continent peoples was unsurpassed in its day and remains a unique primary source of information on cranial deformation. He collected ancient skulls from North, Central, and South America, along with others from various extant tribes in the continent. He also interviewed many explorers of these regions and was familiar with writings from the age of the Spanish conquistadors. In fact, a central theme of Mortons work is that he could nd no evidence of cognitive deficits among peoples practicing severe cranial deformation. He theorized similar means by which the tribes on the Columbia river, ancient inhabitants of Venezuela, the Charibs of the Antilles, some tribes of Peruvians, and other peoples of North and Central America had foreheads compressed by art, the back and lateral parts of the cranium become proportionately expanded, in order to make room for the brain that has been displaced from the anterior chamber. (37). Furthermore, Morton wrote that many ancient Peruvians heads were as remarkable for their narrowness as for their length (37). He continued, when nature has denied an imaginary grace, art is called in to supply the deficiency; and even where there has been no such deficiency, human vanity prompts to extravagance.It would be natural to suppose that a people with heads so small and badly formed would occupy the lowest place in the scale of human intelligence. Such, however, was not the case as Morton quotes accounts of Conquistadors who described the fabulous architectural structures of pre-Incan and Incan civilization (37). Morton criticized others for their primitive view of the Inca:

FIGURE 5. A, frontispiece of Mortons Crania Americana. B, skull of a Kalapooyah Indian, a living counterpart to which Morton encountered among the Chinook who appeared to . . . possess more mental acuteness than any Indian [he] had seen (37). As documented by plates in Crania Americana, other tribes on the Columbia River that practiced cranial deformation included the Chinook, Cowalitsk, and Killemook. From, Morton SG: Crania Americana; or a Comparative View of the Skulls of Various Aboriginal Nations of North and South America. Philadelphia, J Dodson, 1839 (37).

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Thus the seemingly superhuman efforts of the Egyptians are at least equaled by those of the Peruvians.a people destitute of horses, oxen, or any beast of burden except the feeble lama; and yet they have left monuments which sufciently attest their great ingenuity and indomitable perseverance.The arts of the present day, with all the refinements of successive generations of ingenious minds, would perhaps be inadequate to achieve those remarkable ends which are common in the monuments of Peru (Fig. 6) (37). Morton also discussed Incas in the province of Chicuito who molded their childrens heads for the purpose of increasing the ferocity of countenance in warand adding to the health and strength of the body (37). For one Peruvian tribe, all their attention is bestowed on preserving a rm texture of the body, and on attening the forehead and hinder part of the head [in an upward direction] with a view of resemblingthe full moon, and of becoming the strongest and most valiant people in the world (37). The Inca Huyna Capac, through a passage from Garcilaso de la Vega in Mortons work, mentioned a culture that he conquered that achieved a remarkable appearance with severe cranial deformation: they deform the heads of their children by placing, at birth, a small board on the forehead and another on the occiput, and drawing them tighter day by day until the child has attained the age of four or ve years. By this process the head becomes broad from side to side, and narrow from back to front. Not satised with this deformity they shave the hair from the top of the head, and the nape of the neck, letting it grow on the sides only; and this not being combed or arranged, but rude and entangled, adds to the hideousness of their physiognomy (37). As among the Maya, Morton noted the custom of molding the cranium into articial forms is of great antiquity and prevalence in Peru. (37) In fact, it was so common that in 1585, the Ecclesiastical Court of Lima forbade parents, under specied penalties, to compress or distort the heads of their children in the various modes which were in vogue even at that late period (37). More recently, Ortner (42) stated that, the complications of cranial deformation are mainly cosmetic, while noting that compression could disrupt normal growth at the cranial sutures and give rise to minor abnormalities. Citing Mosss 1958 study indicating that the magnitude of growth is not reduced, Ortner concluded that the practice probably did not produce any serious health problems (42). In most societies in which intentional cranial deformation is practiced, deformed crania are typically associated with people of high status. Therefore, it seems unlikely that signicant cognitive impairment results from such shaping (Fig. 7). That far more extreme intentional cranial deformation than that found in modern American children has been practiced across cultures for long periods suggests that such adverse cognitive effects are unlikely. In his letter to George Anderson, Ashley Montagu replied,

FIGURE 6. A, a Chinook cradleboard that Morton acquired from a friend. The neck of the child rested on the ridge to the right of the board (D). C is a grass pad that was drawn over the childs forehead. The other parts marked D were for straps to keep the childs body in place. The body rested on a mat of soft grass. E is a support for the cradle or a step to raise the cradle head up in the air for easier lifting. According to Morton, children reportedly remained in such a cradle from 4 to 8 months, or until the sutures of the skull have in some manner united, and the bone becomes solid and rm (37). Morton wrote that so highly is this deformity valued among the Columbia River tribes, that their slaves (who are for the most part derived from the adjacent tribes) are not allowed to practice it.[The] absolute internal capacity of the skull is not diminished, and, strange as it may seem, the intellectual facilities suffer nothing. The latter fact is proved by the concurrent testimony of all travelers who have written on the subject (37). From, Morton SG: Crania Americana; or a Comparative View of the Skulls of Various Aboriginal Nations of North and South America. Philadelphia, J Dodson, 1839 (37). B, illustration of a Chinook mother with an infant in the head molding apparatus. Dingwall writes, The old Spanish Franciscan Diego de Landa who dealt with the Maya and who was Bishop of Merida about 1572, described how the natives of his day deformed the heads of their children. Scarcely four or ve days after birth the child was stretched out upon a sort of little bed made of reeds or strips of other material, and then the head was placed between a couple of boards, one at the back and one at the front. These were then pressed together and fastened. For days at a time the child was thus left in sufferingand sometimes so much pain was caused that the children died, [or] had openings behind the ears, a condition of things whichwas not uncommon (16). From, Dingwall EJ: Articial Cranial Deformation: A Contribution to the Study of Ethnic Mutilations. London, John Bales, Sons & Danielson Ltd., 1931 (16).

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All the evidence we have indicates that no form of head binding has any untoward effect upon the growth of either the brain or the development of the mind.My experience with articially deformed crania strongly suggests that no alterations are produced except 1) alteration in the form of the bones and 2) pressure may approximate the squama temporalis toward the frontal bone, or rather, the other way round, if the pressure is occipito-frontal, or merely frontal. The sutures remain unaffected, that is to say, there is no premature synostosis, and this is an important point because it enables one to distinguish between those pathological cases in which there has been premature synostosis of endogenous origin from those cases which, as in articial cranial deformation, are of exogenous origin. I have never heard of a case of articially produced microcephaly as a result of articial cranial deformation. Anything can happen, but I think it extremely unlikely that such a result is likely to follow upon any of the large varieties of artificial cranial deformation. What sometimes happens is that the visceral layer of the dura mater tends to adhere to the cranial layer, and that the arachnoid may also be involved, and sometimes, also, the pia, in forming adhesions to the inner table of the skull. This is particularly the case in frontal forms of deformation. In such forms of deformation an exophthalmos is produced by the pressure-in certain parts considered muy simpatico! [sic] (44)

CONCLUSION
The anthropological record supports endocranial effects of head deformation in situations that parallel those of the positional plagiocephaly that has followed the adoption of the Back to Sleep campaign. However, the overwhelming prevalence of head shaping in ancient cultures seems to argue against cranial shaping exerting a significant adverse effect on cognition. Across history, cranial deformation has been much more severe than that currently encountered by neurosurgeons. Yet many cultures, including most of their populations or at least their leading citizens, have practiced incredible skull deformation, while still achieving marked cultural progression (e.g., producing architecturally complicated structures, systems of calendars, mathematics, and materials workmanship). Such cultural progression would likely have been precluded if cognitive deficits had been widespread among those of high status, much less across most of their populations. Some cultures, such as the Maya, achieved high levels of scientific and artistic sophistication despite their widespread practice of extreme forms of head shaping. Even if the archeological record reflects only a small subset of the Maya, such as the elites, those people presumably responsible for advancing and maintaining Maya culture, it would be logical to assume that at least gross deleterious effects would be noted rapidly and the practice halted. Nevertheless, some

form of cognitive effect (e.g., a learning disability) cannot be discounted entirely. However, the observations of cranial deformation with proptosis by Ashley Montagu and J.K. Townsend imply significant increases in intracranial pressure in these individuals. Around-the-head, or band-type, deformation procedures would seem to be more serious in not allowing the head to expand in compensatory ways. Over time, such procedures would cause brain injury with associated intellectual deterioration. The translation to a population scale may be that a cognitive effect, although not obvious, could be serious enough and culturally widespread so as to interfere with the ability of a culture to respond to certain challenges. The collapse of the southern Mayan culture remains enigmatic, attributed to warfare, invasion, migration, disease, over-farming, social class struggle, economic collapse, religious change, and climactic alteration. It is intriguing, however, to postulate whether or not neurological effects from artificial head deformation contributed to the scenario of cultural demise. Unfortunately, the record of severe head shaping found in some of the most notable ancient cultures in the New World does not lend itself to a direct demonstration of the effects of cranial deformation on cognition. There is simply no way to test cognition in cultures that precede us by centuries. As Ashley Montagu stated, the mystery is likely to go on as unfortunately microscopic examinations [on the brains under conditions of articial cranial deformation] have seldom been done. If present, however, such a cognitive effect may have been adapted to or contributed to the perceived world or Weltanshauung of these peoples in an interesting and meaningful, or even culturally harmful, way. Perhaps, considering the evidence from past cultures that practiced severe skull deformation may help shed perspective on our own medicocultural practices by which we assess and treat deformational plagiocephaly today.

REFERENCES
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10. Cheverud JM, Kohn LA, Konigsberg LW, Leigh SR: Effects of fronto-occipital articial cranial vault modication on the cranial base and face. Am J Phys Anthropol 88:323345, 1992. 11. Coe MD: Mexico: From the Olmecs to the Aztecs. London, Thames and Hudson, 2002. 12. Cybulski JS: Skeletal Variability in British Columbia Coastal Populations: A Descriptive and Comparative Assessment of Cranial Morphology. Ottawa, Archaeological Survey of Canada Mercury Series No. 30, Canadian Museum of Man, 1975. 13. David LR, Genecov DG, Camastra AA, Wilson JA, Argenta LC: Positron emission tomography studies conrm the need for early surgical intervention in patients with single-suture craniosynostosis. J Craniofac Surg 10:3842, 1999. 14. Davis BE, Moon RY, Sachs HC, Ottolini MC: Effects of sleep position on infant motor development. Pediatrics 102:11351140, 1998. 15. Dewey C, Fleming P, Golding J: Does the supine sleeping position have any adverse effects on the child? II. Development in the rst 18 months.ALSPAC Study Team. Pediatrics 101:E5, 1998. 16. Dingwall EJ: Articial Cranial Deformation: A Contribution to the Study of Ethnic Mutilations. London, John Bales, Sons & Danielson Ltd., 1931. 17. Flower HH: Fashion in Deformity, as Illustrated in the Customs of Barbarous and Civilized Races. Humboldt Library of Popular Science Literature. New York, Fitzgerald, 1882, vol 2. 18. Foley WJ, Kokich VG: The effects of mechanical immobilization on sutural development in the growing rabbit. J Neurosurg 53:794801, 1980. 19. Gerszten PC: An investigation into the practice of cranial deformation among the pre-Columbian peoples of northern Chile. Int J Osteoarcheology 3:8798, 1993. 20. Gerszten PC, Gerszten E: Intentional cranial deformation: A disappearing form of self-mutilation. Neurosurgery 37:374382, 1995. 21. Goodrich JT, Tutino M: An annotated history of craniofacial surgery and intentional cranial deformation. Neurosurg Clin N Am 12:4568, viii, 2001. 22. Graham JM Jr: Craniostenosis: A new approach to management. Pediatr Ann 10:2735, 1981. 23. Graham JM Jr: Alterations in head shape as a consequence of fetal head constraint. Semin Perinatol 7:257269, 1983. 24. Graham JM Jr, Badura RJ, Smith DW: Coronal craniostenosis: Fetal head constraint as one possible cause. Pediatrics 65:995999, 1980. 25. Graham JM Jr, deSaxe M, Smith DW: Sagittal craniostenosis: Fetal head constraint as one possible cause. J Pediatr 95:747750, 1979. 26. Graham JM Jr, Gomez M, Halberg A, Earl DL, Kreutzman JT, Cui J, Guo X: Management of deformational plagiocephaly: Repositioning versus orthotic therapy. J Pediatr 146:258262, 2005. 27. Grosse LA: Essai surles deformations articielles du crane [in French]. Paris, JB Balliere, 1855. 28. Habal MB, Leimkuehler T, Chambers C, Scheuerle J, Guilford AM: Avoiding the sequela associated with deformational plagiocephaly. J Craniofac Surg 14:430437, 2003. 29. Hrdlicka A: Physiological and medical observations among the Indians of Southwestern United States and Mexico. Washington, D.C., US Government Printing Ofce, 1908. 30. Konigsberg LW, Kohn LA, Cheverud JM: Cranial deformation and nonmetric trait variation. Am J Phys Anthropol 90:3548, 1993. 31. Koskinen-Moffett LK, Moffett BC Jr, Graham JM Jr: Cranial synostosis and intra-uterine compression: A developmental study of human sutures. Prog Clin Biol Res 101:365378, 1982. 32. Littleeld TR, Saba NM, Kelly KM: On the current incidence of deformational plagiocephaly: An estimation based on prospective registration at a single center. Semin Pediatr Neurol 11:301304, 2004. 33. Lorentz KO: Cultures of physical modifications: Child bodies in ancient Cyprus. Stanford J Archaeol 2:117, 2003. 34. Mason OT: Cradles of the American Aborigines. Annual Report of the Smithsonian Institute. Washington, D.C., US Government Printing Ofce, 1889. 35. Miller RI, Clarren SK: Long-term developmental outcomes in patients with deformational plagiocephaly. Pediatrics 105:E26, 2000. 36. Molfese DL, Molfese VJ: Discrimination of language skills at ve years of age using event-related potential recorded at birth. Dev Neuropsychol 13:135156, 1997. 37. Morton SG: Crania Americana; or a Comparative View of the Skulls of Various Aboriginal Nations of North and South America. Philadelphia, J Dodson, 1839.

38. Moss ML: The pathogenesis of artificial cranial deformation. Am J Phys Anthropol 16:269286, 1958. 39. National Science Foundation: Scientists nd earliest new world writings in Mexico. http://www.nsf.gov/od/lpa/news/02/pr0297.htm. Accessed 2006. 40. Neumann GK: Types of cranial deformation in the eastern United States. Am Antiquity 7:306310, 1942. 41. OLoughlin VD: Effects of different kinds of cranial deformation on the incidence of wormian bones. Am J Phys Anthropol 123:146155, 2004. 42. Ortner DJ: Identication of Pathological Conditions in Human Skeletal Remains. San Diego, Academic Press, 2003, ed 2. 43. Panchal J, Amirsheybani H, Gurwitch R, Cook V, Francel P, Neas B, Levine N: Neurodevelopment in children with single-suture craniosynostosis and plagiocephaly without synostosis. Plast Reconstr Surg 108:14921500, 2001. 44. Preul MC: Letters: George W. Anderson to M.F. Ashley Montagu, January 17, 1958; Ashley Montagu to George W. Anderson, January 23, 1958; George W. Anderson to Ashley Montgu, January 27, 1958. Private Collection of Mark C. Preul, M.D., 2006. 45. Pucciarelli HM: The inuence of experimental deformation on neurocranial wormian bones in rats. Am J Phys Anthropol 47:2938, 1974. 46. Pucciarelli HM: The inuence of experimental deformation on craniofacial development in rats. Am J Phys Anthropol 48:455461, 1978. 47. Sommerring ST: Vom baue des menschlichen Korpers [in German]. Leipzig, Voss, 1800. 48. Tiesler V: Head shaping and dental decoration among the ancient Maya: Archaeological and cultural aspects. Presented at the 64th Annual Meeting of the Society for American Archaeology, Chicago, March 2428, 1999. 49. Torres-Rouff C: Shaping Identity: Cranial Vault Modification in the PreColumbian Andes. Santa Barbara, University of California, 2003 (dissertation). 50. Turk AE, McCarthy JG, Thorne CH, Wisoff JH: The back to sleep campaign and deformational plagiocephaly: Is there cause for concern? J Craniofac Surg 7:1218, 1996. 51. von Winning H: Process of head deformation shown by mesoamerican gurines. Masterkey 42:5358, 1968. 52. Wells C: Aricial Interference. London, Thames and Hudson, 1964. 53. White CD: Sutural effects of fronto-occipital cranial modication. Am J Phys Anthropol 100:397410, 1996. 54. Wilford JN: Mother Culture, or Only a Sister? New York Times, March 15, 2005.

Acknowledgments
Gregory P. Lekovic, M.D., Ph.D., received the Vesalius Prize from the History of Neurological Surgery Section of the American Association of Neurological Surgeons for this study, which was presented in part at the 73rd Annual Meeting of the American Association of Neurological Surgeons in New Orleans April 1621, 2005. We thank Charles Merbs, Ph.D., for his photographs from the collections of Arizona State University and the Museum of Man, Jorge Perez de Lara for photographs of Olmec and Mayan antiquities, and the Dallas Museum of Art, Dallas, TX, for the photograph of the Olmec gurine.

COMMENTS

he authors have performed a most interesting review of cranial deformation and its practice in ancient cultures. The report was originally sparked by a series of recent publications that suggest there is a higher incidence of developmental delays in children with craniosynostosis and, more importantly, in the population of children with deformational plagiocephaly. In reviewing some of the recent work on this subject, the increasing numbers of children with developmental issues is somewhat alarming. Keeping that concept in mind, the authors were interested in whether the practice of cranial deformation, intentionally performed in most cases, caused a similar incidence of developmental delay. In a careful analysis of the early literature from explorers and conquistadors, it became clear that intentional cranial deformation apparently did not cause a higher incidence of developmental delay. The images the authors have attached to the paper clearly point out some of the severe cranial deformations that were obtained with these practices. I remember a lec-

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ture by William Feindel, MD, in which he advocated that the possible demise of the Mayans during the late classic period (circa 800900 A.D.) might be caused by overly severe intentional cranial deformation. He argued that this practice, typically only allowed among the nobility, might have led to a higher incidence of epilepsy and developmental issues. Its a provocative thought, but no one has an answer; hence the speculations are most interesting to read. The only question that remains in my mind is the recent disturbing issues being raised regarding developmental delays in children with deformational plagiocephaly. If this is true, then we have a serious problem, because pediatric neurosurgeons and neurologists are observing an epidemic of this disorder. James T. Goodrich Bronx, New York

positional plagiocephaly. As the authors note, the incidence of this condition is increasing, making current dialog regarding the topic particularly relevant. Although careful to point out the lack of any direct evidence relating intentional cranial deformation and cognition, the authors consideration of the advancement and productivity of the historical civilizations in which this practice was prevalent makes for an intriguing argument. Considering the growing number of children who are developing plagiocephaly, along with their concerned parents, the historical context presented here will make for interesting discussion. Lissa C. Baird Michael L. Levy San Diego, California

his fascinating article is the product of a careful review of the scientic literature as well as the anthropological collections of two large institutions to examine the evidence for any adverse consequences on cognition associated with the practice of intentional cranial deformation. The authors focused their research on the practice in the Americas, although intentional cranial deformation occurred at different times by different cultures on every inhabited continent. The question stated by the authors for this study is whether plagiocephaly resulting from craniosynostosis causes cognitive impairment. They surmise that, given the severity of the deformational plagiocephaly caused by intentional cranial deformation, if the practice did not result in any cognitive decit, then the much milder cases of positional cranial molding encountered by physicians today would be even less likely to result in cognitive impairment. The authors conclude from their investigation that there were likely no adverse consequences on cognition associated with even the most extreme forms of intentional cranial deformation as practiced in the Americas. Otherwise, the practice would have likely not been so widely practiced throughout human history. An important question is, what were the origins of this mutilation that was practiced by cultures widely separated by time and space? Enrique Gerszten Richmond, Virginia Peter C. Gerszten Pittsburgh, Pennsylvania

n recent decades, paleoanthropology has undergone impressive advances from a merely descriptive and historical discipline to a dynamic interpretation of the variability of the human cranium in terms of the functional and structural relationship of its different components. The anatomical variations induced by volumetric changes of the brain are those that most attracted the scientists attention. Consequently, the changing human cranial phenotype is presently interpreted as an integrated functional structure rather than a simple coexistence of different and variable features. Craniosynostosis can be regarded as a kind of disorder that interrupts the harmony of the progressive change of the calvarium and face during human evolution. The therapeutic efforts are indeed aimed at reestablishing an ideal phenotype, namely, the perfect shape of the face and the cranium. Cranial molding, and in particular, positional posterior plagiocephaly, represents an additional puzzling phenomenon for functional craniology, as the deformed calvarium seems to not be associated with signicant functional disturbances. There are several examples of populations that seem to have associated articially deformed craniums with privilege. Although cumulative experience suggests minimal or absent neurofunctional impact of molded craniums, the reason for such an attitude continues to remain very intriguing. Lekovic et al. stimulate the debate with this elegant and very exciting review that uses an original source of information to question the possible impacts of cranial molding on neurodevelopment. Concezio Di Rocco Rome, Italy

his article contains a fascinating description of historical cranial deformation along with a relevant discussion of contemporary thought on

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