www.AJOG.
org
into the uterine cavity, and the membranes of the ovum could
be touched, as they can be sometimes during the last days of
gestation. . . according to our prognostications, abortion re-
sulted but a few weeks afterwards, from the inability of the
uterus to retain its contents.” Thus, it seems that Grant and
Harger have basis in crediting Gream with a description of
cervical insufficiency secondary to prior cervical surgery. Inas-
much as cervical insufficiency today remains one of “the great
obstetrical syndromes,” we do not wish to deny Gream credit
for his observation. However, we want to clarify that the orig-
inal sources contain a description of a patient with cervical
insufficiency, but not the term “cervical incompetence” or
“cervical insufficiency.”
We believe that these issues deserve further research by those
interested in the history of medicine. f
Roberto Romero, MD
Chief, Perinatology Research Branch
Program Director for Obstetrics and Perinatology
NICHD, NIH, DHHS
Bethesda, MD, and Detroit, MI
Center for Molecular Medicine and Genetics
Wayne State University
Detroit, MI
nichdprbchiefstaff@med.wayne.edu
Jimmy Espinoza, MD
Associate Professor
Wayne State University
Perinatology Research Branch
NICHD, NIH, DHHS
Bethesda, MD, and Detroit, MI
Wayne State University/Hutzel Hospital
Department of Obstetrics and Gynecology
Detroit, MI
Pathogenesis of amniotic band syndrome
TO THE EDITORS: I read with interest the article by Soldado
et al1 in which silk suture extremity ligation is proposed to
replicate annular constrictions of the limbs in utero, represent-
ing a model of amniotic band syndrome. Throughout the
manuscript, the authors describe amniotic band syndrome
solely as a result of mechanical effects with progressive stran-
gulation. The authors further propose that fetal surgery may be
an effective treatment of moderate forms of amniotic band
syndrome. It is unfortunate that the authors did not fully dis-
cuss alternative pathogenic mechanisms of amniotic band syn-
drome. Numerous studies have purported that human fetuses
with amniotic band syndrome contain anomalies of internal
organs, in addition to the manifestation of external limb de-
fects. These autopsy findings suggest a disturbance of normal
morphogenesis as the etiology of amniotic band.2,3 Specifi-
cally, limb-body wall complex associated with amniotic band
syndrome has been proposed to be a result of an intravascular
disruption sequence4 with necrosis and subsequent scarring of
Letters to the Editors
Offer Erez, MD
Wayne State University
Perinatology Research Branch
NICHD, NIH, DHHS
Bethesda, MD, and Detroit, MI
Sonia Hassan, MD
Assistant Professor
Wayne State University
Perinatology Research Branch
NICHD, NIH, DHHS
Bethesda, MD, and Detroit, MI
Wayne State University/Hutzel Hospital
Department of Obstetrics and Gynecology
Detroit, MI
REFERENCES
1. Romero R, Espinoza J, Erez O, Hassan S. The role of cervical cerclage
in obstetric practice: can the patient who could benefit from this proce-
dure be identified? Am J Obstet Gynecol 2006;194:1-9.
2. Althuisius SM, Dekker GA, van Geijn HP. Cervical Incompetence: A
reappraisal of an obstetric controversy. In: Althuisius SM. Cervical incom-
petence, you better believe it. Amsterdam: Vrije Universitet Medical Cen-
ter. 2001;70-85.
3. Culpeper N, Cole A, Rowland W, editors. Of Barrennefs: In The practice
of Phyfick (Being chiefly a tranflation of the works of that learned and
renowned Doctor Lazarus Riverius). London: George Sawbridge; 1655.
pp. 502-9.
4. Grant A. Cervical cerclage to prolong pregnancy. In: Chalmers I, Enkin
M, Keirse MJNC, editors. Effective care in pregnancy and childbirth. Vol.
1. New York (NY): Oxford University Press; 1989. pp. 633-46.
5. Harger JH. Cervical Cerclage: Patient Selection, Morbidity, and Suc-
cess Rates. Clin Perinatol 1983;10:321-41.
6. Gream GT. Dilatation or division of the cervix uteri. The Lancet 1865:
381.
© 2007 Mosby, Inc. All rights reserved. doi: 10.1016/j.ajog.2007.03.078
the extremity, perhaps resulting in secondary skin tags and an
inflammatory fibrous response culminating in an amniotic
band. Although not a necessary requirement for the develop-
ment of limb-body wall complex, it remains possible that am-
nion rupture and subsequent formation of amnion bands may
represent the pathogenesis of some cases of limb amputation.
It is essential that prior to any human intervention trials, in-
vestigators develop means to differentiate a primary vascular
event causing limb necrosis and a secondary band, from a pri-
mary amnion effect resulting in a constrictive effect. For it is
only in the latter circumstance that potential early surgical in-
tervention may be of value. A more comprehensive and bal-
anced assessment of pathogenesis and the potential of their
model would be of value to all readers. f
Michael G. Ross, MD, MPH
Professor of Obstetrics-Gynecology and Public Health
David Geffen School of Medicine at UCLA
and UCLA School of Public Health
AUGUST 2007 American Journal of Obstetrics & Gynecology 219
Letters to the Editors
Chair, Department of Obstetrics-Gynecology
Harbor-UCLA Medical Center, Los Angeles, CA
mikeross@ucla.edu
REFERENCES
1. Soldado F, Peiro JL, Aguirre M, et al. Extremity amniotic band syn-
drome in fetal lamb. I: An experimental model of limb amputation. Am J
Obstet Gynecol 2006;195:1607-10.
REPLY
We truly appreciate the comments forwarded regarding our
study and the opportunity to respond.
Pathogenesis is the great incognita in amniotic band syn-
drome. Although the extrinsecal theory has been widely ac-
cepted, several incongruencies are found with the clinical data
in newborns with amniotic band syndrome.1 We agree with the
fact that internal organ malformations and many other associ-
ated features make Torpin’s theory not completely acceptable.2
Research aiming for pathogenesis of amniotic bands is very
important. In fact, we are carrying out an experimental study in
rabbits regarding this issue.
Our study has been based on the effect of the band. Tadmor
demonstrated that the effect of the band is a mechanical one,
with progressive strangulation. This fact seems to be indepen-
dent of the pathogenesis and its extrinsecal or intrinsecal the-
ories. Furthermore, our model did not aim at reproducing the
band itself but the effect of passive progressive strangulation
described by Tadmor.3 The ligature—as well as amniotic
bands—acts as an inelastic structure capable of progressive in-
jury on the limb with deformities and evolution parallel to the
human EAB.
220 American Journal of Obstetrics & Gynecology AUGUST 2007
www.AJOG.org
2. Hartwig NG, Vermeij-Keers C, De Vries HE, Kagie M, Kragt H. Limb
body wall malformation complex: an embryologic etiology? Hum Pathol
1989;20:1071-7.
3. Van Allen MI, Curry C, Gallagher L. Limb body wall complex: I. Patho-
genesis. Am J Med Genet 1987;28:529-48.
4. Moerman P, Fryns JP, Vandenberghe K, Lauweryns JM. Constrictive
amniotic bands, amniotic adhesions, and limb-body wall complex: dis-
crete disruption sequences with pathogenetic overlap. Am J Med Genet
1992;42:470-9.
© 2007 Mosby, Inc. All rights reserved. doi: 10.1016/j.ajog.2007.03.079
The very final purpose of our research is to establish a valid
model to study the feasibility of amniotic band fetal surgery. In
fact, the second part of the study is already finished and ready
for publication. f
F. Soldado-Carrera
Universitat Autónoma de Barcelona
Hospital Maternoinfantil
Hospital Universitari Vall d=Hebron
Passeig Vall D=Hebron 119-129
Barcelona 08035, Spain
34194fsc@comb.es
REFERENCES
1. Bronshtein M, Zimmer E. Do amniotic bands amputate fetal organs?
Ultrasound Obstet Gynecol 1997;10:310-1.
2. Torpin R. Amniochorionic mesoblastic fibrous strings and amniotic
bands. Am J Obstet Gynecol 1965;91:65.
3. Tadmor OP, Kreisberg GA, Achiron R, Porat S, Yagel S. Limb ampu-
tation in amniotic band syndrome: serial ultrasonographic and Doppler
observations. Ultrasound Obstet Gynecol 1997;10:312-5.
© 2007 Mosby, Inc. All rights reserved. doi: 10.1016/j.ajog.2007.03.080