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Cranial dysraphism mistaken for trephination.

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Cranial Dysraphism Mistaken for Trephination
T. D. STEWART
Division of Physical Anthropology, National Museum of Natural History,
Washington, D . C. 20560
K E Y WORDS Skull. Dysraphism . Trephination . New England
Peru Paleopathology.
’
ABSTRACT
Attention is called to a disputed diagnosis of trephination
reported for a perforated skull of a prehistoric New England Indian. The perforation, surrounded by a saucer-like depression, is located exactly in the
midline just in front of bregma. The finding of a similar lesion in a prehistoric
Indian skull from the North Coast of Peru -where, as in New England, good
evidence of the practice of trephination is lacking - provides support for a
more reasonable diagnosis
for both cases: congenital cranial dysraphism,
specifically encephalocele. Information about dysraphic states from modern
clinical experience is summarized.
In 1970 Bernard W. Powell of Wilton,
Connecticut, published a report in Science
entitled, “Aboriginal trephination: Case
from southern New England?” Note the
question mark. The skull with the putative trephination (fig. 1, left) is part of a
skeleton encountered and partly disturbed
by laborers working on a road at a known
site of Indian occupancy on a glacial terrace bordering Long Island Sound at Norwalk, Connecticut. Powell’s subsequent
investigation revealed that this individual
-a
young adult, probably male- had
been buried semiflexed in a deep layer of
loam during the Woodland Period. He
named the site Spruce Swamp for reasons
given in another publication (Powell, ’65)
and designated the skeleton Spruce Swamp
burial 1 (B.1).
In his report Powell summarized the
opinions of seven persons (including the
writer) with whom he had discussed the
nature of the cranial perforation. Four
unnamed pathologists “were flatly of the
opinion that it was a trephination and not
meningioma or other disease.” Milton Helpern, the New York City Medical Examiner, “suggested that a downward invasion
of a basal-cell tumor of the scalp could
cause the condition, but he declined ultimately to say definitely what the lesion
represented.” I “suggested that [it] might
be from a cyst or tumor . . . [but] conceded that [I] had never seen anything
quite like this specimen.” And lastly, H.
AM. J. PHYS. ANTHROP.,42: 435-438.
Jaffee of the Hospital for Joint Diseases
in New York City “raised the possibility
that the anomaly might result from a longstanding lesion which prompted attempts
to scrape and treat it.”
The article in Science attracted the attention of Detroit-neurosurgeon H. Harvey
Gass and led him to write a letter to the
Editor giving his diagnosis of the case
(Gass, ’71). The following extract conveys
the point of the letter:
The skull may become “saucerized” by benign tumors within it and growing out of it,
or resting upon it, especially in young individuals. When this process is present exactly in the midline and an aperture pierces
the skull at the bottom of the “saucer,” a
diagnosis of congenital cranial dysraphism
seems mandatory.
I suspect that many physical anthropologists missed Dr. Gass’ letter, or, if they
saw it, failed to grasp its full significance.
For this reason, I a m using my chance
discovery of a second prehistoric skull with
a comparable lesion a s an excuse to put
both cases on record in a n anthropological
journal and to summarize what is known
about cranial dysraphism. Hopefully, fuller understanding of what appears to be a
rare manifestation of a congenital state
will prevent human paleopathologists from
repeating a diagnostic error.
The new specimen is a n isolated skull
without lower jaw bearing the catalogue
number 264,629 in the National Collection
(fig. 1, right); it is one of some 600 skulls
435
436
T. D. STEWART
Fig. 1 Left: Skull B.1 from the Spruce Swamp site at Norwalk, Connecticut (Courtesy of
Bernard W . Powell). Right: Skull U.S.N.M. No. 264,629 from the Chicama Valley on the
North Coast of Peru. Both skulls show signs of a herniated sac - encephalocele - in the
midline a t or j u s t in front of bregma.
picked up by HrdliCka in 1910 from the
surface of unrecorded sites in the Chicama
Valley on the North Coast of Peru. A note
in unidentified handwriting on the catalogue card for this specimen reads: “Trephining? at bregma.” I judge the skull to
be a young-adult female.
The lesion in this Peruvian skull is remarkably similar in appearance to the
one in the New England skull as pictured
in Science. The main difference is that in
the Peruvian skull both the perforation
and the surrounding “saucer” are smaller
and situated just at bregma, rather than
a centimeter or so anterior to bregma. To
the extent that the “saucers” are indicative of tumor sizes, neither one can be
considered large. The rim of each “saucer” is prominent anteriorly and fades
away on either side, somewhat like the
bow-waves of a ship. Dr. Gass explained
the raised rim in his letter as follows:
Brain pulsations transmitted through the
aperture [in the skull] meeting against the
bone edge cause the bone to “build up” a t
this edge, receiving the pulsations maximally.
I can add that in the Peruvian skull
the aperture appears circular only when
viewed frontally and just over the rim of
the “saucer.” Endocranially the posterior margin of the perforation is slightly
grooved. Apparently, therefore, the tumor
was connected with the endocranial structures by a stalk or pedicle directed anteroposteriorly. Information is lacking on this
point in the skull from New England.
For further enlightenment on dysraphic
states Dr. Gass (personal communication,
December 18, ’73) kindly referred me to
some text-books on neurology. In one of
them (Dekaban, ’59) I learned that dysraphism embraces two related conditions
commonly called spina bifida and cranium
bifidum. The term is descriptive in that it
CRANIAL DYSRAPHISM MISTAKEN FOR TREPHINATION
437
refers to the embryological stage of closure pends mostly upon the amount of brain
of the neural canal and in particular to damage prior to repair, in pre-surgical
defects along the line of closure (Dys-, times the prognosis for survival to adultGr. 8vu bad; raphe, Gr. pa+< seam; -ism, hood would have been favorable only for
Gr. L(TFOI,expressing the thing done). In small meningoceles.
Dekaban's words (p. 177),
This information from modern clinical
experience accords so well with the evi. . . the neural canal begins to form at about
the nineteenth day of gestation and is comdence in the two prehistoric specimens
pletely closed in both cephalic and caudal
as to rule out an exotic explanation such
regions by the end of the first month of intraas trephination. It is true that Pedro Weiss
uterine life . , .
A noxious factor or occasionally a ge('58) claims to have found evidence in the
netic lesion may disturb the development
form of supra-inion depressions for proof a segment of the neural axis as well a s
phylactic
trephination having been practhe overlying primitive mesenchymal tissue.
ticed in the Chicama region. But I have
This can lead to severe malformation and
to a defect i n the d u r a mater and cranial
explained these depressions in more nator spinal bones.
ural and reasonable ways (Stewart, '71;
Judging from recently collected cases, manuscript). Besides, Chicama is located
cranial dysraphism is six and one-half well away from that part of Peru where
times less common than spinal dysra- the established type of trephination was
phism. In the present U . S. population, practiced (Stewart, '58). As for New Engaccording to Dekaban's estimate, the for- land, there does not appear to be any
mer occurs about once in 6,500 births. other kind of evidence that the Indians
As seen in the head of a living child, the there had developed a surgical capability.
condition consists of a pulsating tumor of
LITERATURE CITED
variable size located usually in the midDekaban,
A
.
1959 Neurology of Infancy. Willine, most commonly in the occipital region
liams and Wilkins. Baltimore.
and next most commonly in the naso-fron- Gass, H. H. 1971 Skull from Spruce Swamp:
tal region. It is not clear from the termiCase of cranial dysraphism? Science, 171 : 1268.
nology used in the tabulation whether the Powell, B. W . 1965 Spruce Swamp: A partially
drowned coastal midden in Connecticut. Am.
region of bregma is included in the nasoAntiq.. 30: 4 6 0 4 6 9 .
frontal region, or in the less-susceptible
1970 Aboriginal trephination: Case from
parietal region.
southern New England? Science, 170: 732-734.
Pulsation is due to the tumor being a Stewart, T. D. 1958 Stone age skull surgery:
A general review, with emphasis on the New
sac that has herniated through a defect
World. Smithsonian Rep. for 1957, pp. 4 6 9 4 9 1 .
in the skull. When the sac contains only
1971 Pseudo-trephination. Am. J. Phys.
fluid and strands of leptomeninges with
Anthrop., 35: 29&297 (abstract).
(Manuscript) Are supra-inion depressions
a few blood vessels, it is called a cranial
evidence of prophylactic trephination?
meningocele; when in addition it contains Sublett,
A. J., and C. F. Wray 1970 Some exa portion of the brain or cerebellum, it is
amples of accidental and deliberate h u m a n skelcalled an encephalomeningocele. There are
etal modification in the Northeast. Bull. N. Y.
State Archaeol. Assn., 50: 14-26.
other still more severe grades that need
Pedro 1958 Osteologia cultural, praticas
not concern us here. Considering that in Weiss,
cefalicas. la. parte: Cabezas trofeos, trepanacithis age of miraculous surgery the progones, cauterizaciones. An. Facultad. Med. Univ.
nosis for the two less-severe grades deNac. Mayor San Marcos, Lima, 41 (4): 5054355.
Note added in proof: After the above was accepted for publication I discovered
in the literature another case of cranial dysraphism mistaken for
trephination (Sublett and Wray, '70). The illustration accompanying the
description (fig. 14, p. 20) shows the top anterior half of the skull of a
middle-aged female Seneca Indian from Boughton Hill, New York (16701687 A.D.). Exactly at bregma is a perforation in a saucer-like depression. This perforation appears to have been enlarged through breakage.
In any case, the sloping borders of the perforation are surrounded,
except posteriorly, by a low rounded ridge. A s should now be recognized, all of these features are characteristic of a herniated encephalocele.
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