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J. Perinat. Med. 38 (2010) 247253 Copyright by Walter de Gruyter Berlin New York. DOI 10.1515/JPM.2010.043

From monster to twin reversed arterial perfusion: a history of acardiac twins

Michael Obladen*
Department of Neonatology, Charite University Medicine, Berlin, Germany

Antique enigmatic people and medieval monsters

In the fifth century BC, Herodotus speculated w15x: In that country wNorth Africa near the Red Sea coastx are the large snakes and the lions, and the elephants and bears and asps, the horned asses, the dog-headed wkynekephaloix and the headless wakephaloix men that have their eyes in their chests, as the Libyans say, and the wild men and women besides many other creatures not fabulous. Pliny the elder, who lived from 24 to 79 AD, located the enigmatic people to Ethiopia w31x: The Blemmyes are reported to have no heads, their mouth and eyes being attached to their chests. Throughout the middle ages, this mixture of poor observation, superstition, and ignorance persisted and was repeated by the Dominican Thomas of Cantimpre in his 1263 Liber de Natura Rerum w6x and depicted by Nuremberg encyclopedist Hartmann Schedel in 1493 w39x and by Conrad Lycosthenes in 1557 w22x (Figure 1A, B). The term moon-calf, coined in the 16th century to denote severe malformation, became popular by treatises of Luther and Melanchthon and by its use in Shakespeares Tempest w42x. In scientific publications, the term headless monster was used.

Abstract
A human being born without heart and head, i.e., the acardius/acranius malformation, has been described since antiquity. Superstition and fear made it a mystical disorder, a sign of Gods wrath. The inquisition ruled that acranic infants should not be baptized and located the soul in the brain. Acardia was not associated with twin gestation until the reports of Mery in 1720 and Winslow in 1740. In 1850, Meckel identified the pathogenetic mechanism as reversed perfusion due to large arterio-arterial and veno-venous anastomoses; he believed the heart would fail to develop or arrest during development, and the acardiac fetus would be maintained by arterial perfusion from the pump twin. In 1859, Claudius articulated that after normal initial development, the heart degenerates when reversed flow in the aorta leads to thrombosis. Today, it is assumed that both mechanisms may exist. With the advent of prenatal ultrasound diagnosis and radiofrequency ablation of the acardiac twins circulation, it became possible to save the pump twin. Keywords: Acardius; acranius; history; placental anastomoses; TRAP sequence; twin reversed arterial perfusion.

Gods wrath, imagination, and the baptism dilemma

The headless monster was always accompanied by a normally shaped infant (the pump twin), what excluded its explanation as a mole. This is illustrated by two printed leaflets, the popular press of the Renaissance w17x (Figure 1C): On the fourth of December, 1551, the Friday after St. Andrews day, three children were born in the new hospital at Breslaw named All Saints, to a butchers wife named Ursula Walter Hosperg. One was a small boy who died soon after the baptism. The other two were girls and one was imperfect wimmaturex and stillborn. The third, which is depicted here, was of wondrously rare and frightening shape. For it had neither head nor hands and arms, only the trunk and the feet could be discerned. But it was imperfect wimmaturex and stillborn. Because such frightening miracles and monsters usually without doubt indicate Gods wrath, all Christian souls should sigh and pray to God, our dear Lord Jesus Christs eternal father, to avert his penalty and anger Observation met fantasy quite easily in this century of superstition and pietism w43x:

Introduction
A malformation as frightening as acardius-acranius did not escape the interest of ancient historians. Roman writers adopted the Greek habit of imagining the stillborn or moribund malformed neonate as a living adult, and incorporated the malformation into their mythology. In the case of acranic individuals even a mystique population in a remote area was imagined.
*Corresponding author: Prof. Dr. Michael Obladen Department of Neonatology Charite University Medicine Berlin Augustenburger Platz 1 13353 Berlin Germany Tel.: q49 30 (0) 30 450566122 Fax: q49 30 (0) 30 450566922 E-mail: michael.obladen@charite.de

2010/205

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248 Obladen, History of twin reversed perfusion

Figure 1 Pre-scientific accounts of acranius as a moral warning. (A) Headless blemmye from Schedels Weltchronik 1493 w39x; (B) Brustbutzen from Lycosthenes marvelous wonders 1557 w22x; (C) Frightening prodigy from a Breslau leaflet 1551, see text for details w17x.

Around Christmas of 1569 Caspar Hugens wife, from Matzingen/Thurgau, gave birth to two infants in one delivery. One had no deficiency or blemish, while the other had no head. Instead of a head it had a great black hole out of which it cried; it also had two goose feet and expired when it had emitted one or two cries. On April 6, 1701, the anatomist Alexis Littre reported to the Paris faculty of an acranic and two anencephalic fetuses and pondered on the function of the brain assumed since Aristotle w21x: The first two male fetuses, of seven and eight months gestation, were both large and fat whydropicx. That of seven months had neither head nor neck, and the upper part of the trunk was covered with skin This observation sheds doubt on the purpose ascribed to the brain or at least to make it suspect, that the spirits which its glands separate from the blood are as necessary as assumed for movement, nutrition, and other functions of the body Concerning the functions of the soul, it was barely apparent that this fetus, deprived entirely of the brain, exercised any imagination, judgment, or reasoning, even in an imperfect way The larger a humans brain, the more the functions of his soul are perfect, and the more he is able to carry them out.

Highly relevant in theological terms and in contrast to St. Augustine, acranic neonates forced the Catholic church to localize the infants soul in the head: the inquisitor F. Cangiamila, who was in addition to theology a graduate in medicine and law, was appointed to rule on baptism and decreed (1st ed. 1745) w5x: Therefore, the acranius, how many relicts of the human body he might have, should under no circumstances be baptized: because the head is missing, undoubtedly the primary seat of soul and reason.

A twin-specific disorder with abnormal umbilical vessels

Mauriceau, the man who established obstetrics as a science, wrote in his traite (1st ed. 1668) w23x: The moon-calves are nourished in the womb, to which they nearly always adhere at some place, and are maintained by the blood of which they are perfused like plants watered from the earth. Frequently besides the moon-calf there is another infant, of which it may be separated.

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In 1720, the Parisian surgeon Jean Mery reported to the Academie des Sciences w26x: Marie Guerlin, aged 30 years and pregnant for 6 months, delivered two little girls on September 10 of last year. Nothing was missing to the perfect body of the firstborn infant who lived for half an hour. But the dead wsecond bornx had a severely deformed trunk whose upper part ended with the first dorsal vertebra. This trunk had no head, neck, scapula, clavicula, nor arms For the two little girls there was only one placenta the membranes of which formed but one pouch enclosing them both, which is very rare. Of this fused placenta sorted but one cord, which in the middle of its length divided in two, which separately ended at their navels Having opened the body, we found no lungs nor heart, but in front of the spine there were two vessels in addition to those already described. Mery ordered drawings to be made during the dissection by Mr. Chatillon, dessinateur de lAcademie, however, these depictions could not be located. In 1740, the Dane Jacob Benignus Winslow, a Parisian professor of anatomy, reported a case sent to him without the placenta w49x: After the delivery of a well formed male infant there came another, but without head Before I started to dissect the body, I should have had the idea to inject the blood vessels through the umbilical cord Winslow found neither a heart nor an explanation for the fetal circulation without a heart wp. 835x: Perhaps the mechanism is so simple that, after more experience and further research one will be surprised not to have understood it earlier. In 1789, the Sommerfeld physician D. Kahler related the acardiac malformation to abnormal umbilical vessels in one of triplets w18x: The midwife wafter delivery of two stillborn infants at seven months gestation in June 1777x found that something was left in the uterus, which did not feel like a properly formed fruit It was larger and heavier than the twins, with imperfect genitalia, cleft lip, and a bundle of empty and thin vessels around the umbilicus The skull contained a rudimentary and imperfect brain, the chest abnormal remnants of the lung. The eyes, the heart with the large vessels, the trachea and the esophagus were entirely absent. Likewise all abdominal viscera lacked except small intestines, which by size and position were abnormal. Lacking a good artist, I could only obtain a rough drawing (Figure 2A). In 1812, the Halle anatomist Johann Friedrich Meckel wJuniorx compiled )50 cases of acrania from the world literature and included four of his own w24x. He stated, that the disorder is specific for twin gestation, but could not explain the pathogenesis. This feat was ultimately achieved by his nephew and foster son Heinrich Meckel, 38 years later w25x. In June of 1826, Etienne Geoffroy St. Hilaire verbally accosted a colleague of his in the Paris faculty, sarcastically remarking w11x: Mr. Moreau gave me the honor of informing me that he is going to present a human monster born this morning, a complete acephalus. Will you also present, I answered, his first-

born twin and the placenta in common between the two individuals? The presence of a twin is necessarily linked with development of an acephalic monster Mr. Moreau was stricken by the unequal distribution wof the amniotic fluidx in the two pouches; abundant to the excess in the pouch containing the regular fetus, and scarce or nearly absent in that of the monstrous fetus. Geoffroy proceeded in mocking Winslow, the common believe of mis-watching as cause of malformation, and the frequent use of the scaffold: Sulsman notified Winslow that on the 14th April 1726, a human monster without head was born in Strasbourg. Winslow invited him to publish this observation in the Journal des Savans. Searching for the cause in reasons other than the existence of a co-twin he held the following remarkable conversation with the mother: Did you witness any execution during your pregnancy? No, not at all. Nevertheless, to give birth to an infant without a head, you must have watched the head of a criminal being cut-off, or at least have seen the neck of a hanged man dislocated

Reversed perfusion: etiology and classification

Extensive reports on acardiacs were published by Tiedemann in 1813 w44x, Beclard in 1817 w2x, Vrolik in 1834 w48x, and Otto in 1841 w29x. They classified different variants of the malformation and helped to distinguish it from anencephaly. Yet, they interpreted it as an arrest of development and did not approach an adequate explanation of its cause. In 1836, Thomas Hodgkin and Astley Cooper from Guys Hospital described anastomoses on the placental surface and concluded w16x: The heart wof the normal fetusx impelled the blood into the arteries of the placenta and funis. Meckel von Hemsbach understood in 1850 that the acardiac twin must be kept alive by the pump twin w25x: In fruits, whose own circulation is impossible due to malformation of the heart, a meagre life is supported by the other fetus, while simultaneously general hydrops of the acephalus results from circulatory failure. In his thoughtful booklet, the anatomist Claudius from Kiel w7x denied that the acardiac twin has a primary malformation and linked the problem exclusively to fetal circulation: Certain appending or turned-out skin parts occur where organs declined; at the site of the head, the tips of the extremities, the ventral surface of the trunk, never the dorsal. Sometimes a small retraction is found instead of an appendix One umbilical artery of the healthy wtwinx branches over half of the placenta, the other proceeds towards the second insertion, bends from the surface and enters the umbilical cord of the acardiac; the umbilical vein divides into two larger branches, which accompany both arteries; one of them proceeds towards the second insertion and becomes the acardiacs umbilical vein In the acardiacs umbilical artery the blood therefore flows in a reversed direction, from the pla-

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250 Obladen, History of twin reversed perfusion

Figure 2 (A) Hydropic acardius anceps larger and heavier than the accompanying twins, described by Kahler 1777 w18x, (B) acardius acranius with twisted cord depicted by Ahlfeld 1882 to illustrate the connection of the acardiacus with his twin brother and the placenta w1x.

centa into the body; the flow splits at the hypogastric division, one half goes down towards the art cruralis, the other half up towards the art iliaca communis, the aorta likewise receives a current in reversed direction, from bottom to top. Friedrich Schatz, whose extensive work on placental anastomoses was acknowledged in another article w28x, advanced the theory of Claudius by postulating a gradual taking over of both circulations by the stronger, better placed, and better nourished individual. This process subsequently led to the degeneration of the weaker fetus. He showed that interplacental anastomoses in acardia are arterioarterial and venovenous, in contrast to arteriovenous anastomoses in twin-twin transfusion syndrome. Schatz postulated a third circulation in the placenta and hypothesized that for the reversal of circulation w38x, an obstruction of the flow we.g., due to an omphalocelex in the umbilical vein of one twin is required. Das w8x classified four variants in 1902 (Figure 3) according to the development of allantois and placenta, a classification that is still valid today: 1. Acardius anceps is the least atrophied form, characterized by absence or non-development of face, the extreme anterior part of the body. There are rudiments of cranial bones and of the brain. More or less perfect trunk and extremities (Figure 2A). 2. Acardius acephalus is the most common type. Head wanting, or very rudimentary. Intestines and abdominal organs rudimentary, and the organs above the dia-

phragm represented by the merest trace. Shoulder girdle undeveloped (Figures 2B and 4A). 3. Acardius amorphus, the least developed form. Little more than a lump of connective tissue, covered by edematous skin. There may be some rudiments of visceral tissue. 4. Acadius acormus, the rarest. Head alone present, but never fully developed (Figure 4B).

Elusive pathogenesis
Two hypotheses on pathogenesis persisted: (1) Meckels theory, that the heart fails to develop or arrests during development, and the acardiac fetus is maintained by the pump twin. This theory has been recently supported by Napolitani and Schreiber w27x. (2) The theory of Claudius assuming that the heart develops and then degenerates when reversed flow leads to thrombosis. This theory also has been recently supported by Fujikura and Wellings w10x. Van Allen et al. proposed the term twin reversed arterial perfusion (TRAP sequence) in 1983 to name the disorder w47x. However, it still seems plausible that both mechanisms are at work given that a purely circulatory explanation has several shortcomings: (1) When chromosomal analysis became possible, Rashad and Kerr in 1966 w35x and Turpin et al. in 1967 w46x described gross karyotype anomalies in acardiac twins, and systematic karyotyping revealed chromosomal anomalies in a third of acardiacs w19x. (2) As pointed out by Benirschke

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Obladen, History of twin reversed perfusion 251

Figure 3 Equal and unequal development in twins from one ovum, as classified by Das 1902 w8x. (A, B) Placental circulations; (C, D) simplified heart and circulatory apparatuses.

and Des Roches Harper w3x, acardiac twins frequently have heterogeneous, but typical patterns of human malformation, such as cleft lip or omphalocele, which are difficult to explain by degeneration alone. (3) Genotyping studies showed that human acardiac twinning including placental anastomosis is not, as heretofore believed, restricted to monochorionic placentas, but may occur in dichorionic monozygotic twins w9x or in triploidy arising from fertilization of a polar body of the normal twin w4x. On the other hand, it has been shown in experimental animals that proper folding of the cardiac tube depends on normal blood flows and pressures w37x. The cause of the formation of large anastomoses in the placentae of some twins, with or without primary malformation, remains poorly understood.

Antenatal diagnosis and treatment

Gillim and Hendricks w12x calculated the prevalence of TRAP as 1% of monozygotic twins (one in 35,000 deliveries). Analyzing 184 cases of acardia published from 1960 to 1991, Healey found a perinatal mortality of 35% for the pump twin w14x. The principal problems were congestive cardiac failure, polyhydramnios, and preterm labor. When ultrasound revolutionized prenatal diagnostics in the 1960s, rational management and intervention became possible in order to save the pump twin. Lehr and DiRe described the prenatal ultrasound appearance of an acardiac twin in 1978 w20x, and Pretorius et al. documented the reversed arterial perfusion pattern with color Doppler ultrasound in 1988 w33x.

Platt et al. described the antenatal diagnosis with ultrasound in 1983 w30x as: It is believed that these fetuses often develop congestive heart failure because of the cardiovascular demands placed on the normal twin. If this hypothesis is correct, then the proposed surgical procedure of clamping the cord of the acardiac fetus, either by exteriorizing the cord of the fetus and clamping it or by using a small operating laparoscope with a clip holder and clip, should prove useful The logical next step was to develop interventions aimed to rescue the pump twin. In 1989, Robie et al. selectively delivered a 710-g acardiac twin at 22 weeks by hysterotomy, with subsequent delivery of a surviving 2130 pump twin at 33 weeks w36x. In the same year, percutaneous cord occlusion by coil embolization was described by Hamada et al. w13x and two years later by Porreco et al. w32x and their co-workers. In 1994, Quintero et al. reported successful fetoscopic ligation of the umbilical cord w34x. In 1987, Seeds et al. described an unsuccessful attempt to obliterate the vessels within the acardiac twin with laser w40x, a technique successfully used by Sepulveda et al. in 1995 w41x. Using radiofrequency ablation to block the acardiac twins circulation, Tsao et al. reported 12 out of 13 pump twins surviving from 1998 to 2001 w45x.

Conclusions
TRAP will remain a rare event. Due to better understanding of its pathogenesis, it has lost its mythical associations, and prenatal diagnosis and interventions have increased the

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252 Obladen, History of twin reversed perfusion

Figure 4 Schatz studies of placenta anastomoses by injection technique 1899 w38x: (A) acardius acranius; (B) acardius acormus.

chances of the pump twins survival. However, large gaps remain in the understanding. Only better insight into the cause of anastomosis formation within the placenta could lead to prevention of this most severe human malformation.

Acknowledgements
The author is grateful to Gabi Volker, Department of Neonatology, Charite Berlin, for technical help with the figures; to Heike Breuer, Institut fur Geschichte der Medizin, and to Helene Konig-Ellsasser, Department of Obstetrics, both at Freiburg University, for the friendly and guided access to the ancient texts; to Sieghard Irrgang, Kassel, for help with Latin and to Sarah Lieber, University of Michigan Medical School, Ann Arbor, MI, for English language editing.

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