Clinical Anatomy 21:611–618 (2008)

A GLIMPSE OF OUR PAST

Intraneural Ganglion Cyst: A 200-Year-Old

Mystery Solved
ROBERT J. SPINNER,1* JEAN-FRANÇOIS VINCENT,2
ALEXANDRA P. WOLANSKYJ,3 AND BERND W. SCHEITHAUER4
1
Mayo Clinic, Department of Neurologic Surgery, Rochester, Minnesota
2
Conservateur—Chef du Service d’Histoire de la Médecine, Bibliothèque Interuniversitaire
de Médecine et d’ Odontologie, Paris, Cedex, France
3
Mayo Clinic, Division of Hematology, Rochester, Minnesota
4
Mayo Clinic, Department of Laboratory Medicine and Pathology, Rochester, Minnesota

We describe the first reported case of an intraneural ganglion cyst, an ulnar

(‘‘cubital’’) intraneural cyst, which, on literature review, dated to 1810. For
over 80 years, its original brief description by Beauchêne was wrongly attrib-
uted to Duchenne, effectively making the reference and specimen inaccessible
to scrutiny. Fortunately, the intact cyst had been safely housed in the Musée
Dupuytren, Paris, France, thus permitting its examination. Although originally
described as a ‘‘serous’’ cyst, our present understanding of the anatomy of the
ulnar nerve and of peripheral nerve pathology allowed us to reinterpret it as a
mucin-filled, elbow-level, ulnar intraneural ganglion cyst. In addition to its
description as a fusiform cystic enlargement of the nerve, we documented sim-
ilar enlargement of a lumen-bearing branch, the articular branch at the level
of the elbow. Based on our assessment of the specimen and with a modern
perspective, we concluded that the origin of the cyst was from the postero-
medial aspect of the elbow joint and that its fluid content, having dissected
through a capsular defect, followed the path of the articular branch into the
parent ulnar nerve. The purpose of this report is to clarify historical miscon-
ceptions regarding the pathogenesis of this controversial entity. Clin. Anat.
21:611–618, 2008. V 2008 Wiley-Liss, Inc.
C

Key words: medical history; intraneural ganglion cyst; unified theory; articular
nerve branch

INTRODUCTION Furthermore, it confirmed the general applicability of

A search for the first documented specimen of an
intraneural ganglion cyst was undertaken in that
such a specimen, aside from its obvious historic in-
terest, could potentially shed light on the controver-
sies long surrounding its pathogenesis. Having found
a 200-year-old citation documenting an ulnar nerve
example, we successfully located and examined the
specimen which heretofore had only been catalogued
and remained unillustrated. Clearly, its pathogenesis
was not understood. The current concept regarding
the pathogenesis of intraneural ganglion cysts per-
mitted a reinterpretation of its pathologic anatomy.
the unified articular (synovial) theory regarding their
formation.
*Correspondence to: Robert J. Spinner, Mayo Clinic, Gonda 8S-
208, Rochester, MN 55905, USA.
E-mail: spinner.robert@mayo.edu
Received 19 July 2008; Revised 2 August 2008; Accepted 4
August 2008
Published online 12 September 2008 in Wiley InterScience (www.
interscience.wiley.com). DOI 10.1002/ca.20709

C 2008
V Wiley-Liss, Inc.
612 Spinner et al.

MATERIALS AND METHODS

Herein, we describe our search for the earliest ref-
erence to intraneural cyst, setting in motion a chain
of events: verifying the description (part I), retriev-
ing original citations and source materials (part II),
locating and examining the specimen (part III), and
finally in the results section, interpreting its anatomic
features in light of contemporary concepts of intra-
neural ganglion cyst formation.

Part I: Verifying the Earliest

Described Reference
The task of cross-referencing the existing litera-
ture, which included more than 300 articles, and the
use of common internet search engines regarding
intraneural ganglion cysts and intraneural cysts was
undertaken. Although many authors have attributed
the first description of an intraneural ganglion cyst to
the publication of Hartwell (1901), we found histori-
cal citations and well documented descriptions pre-
dating this one, some to the early 19th century, the
earliest being attributed to Duchenne in 1810 and to
Houel in 1811 (Moreau and Van Bogaert, 1923). All
attempts to locate or confirm the existence of these
two citations were unsuccessful, despite multiple ex-
haustive library searches.

Part II: Retrieving the Original Citations

and Source Materials
Attempts to obtain original sources resulted in
correspondence with the curator (JFV) at the Biblio-
thèque Interuniversitaire de Médecine et d’Odonto-
logie of Paris, France, who both developed and oper-
ates a web portal for the history of medicine
(URL:http://www.bium.univ-paris5fr/histmed). Cor-
respondence related to the thesis of Bertrand
(1837), a document stored at the University of Paris,
led to retrieval of the original, subsequently refer-
enced source materials dating to 1810, that being
the observation of a ‘‘serous’’ cyst of the ulnar (‘‘cu-
bital’’) nerve by Beauchêne. The limited description
in the Bulletin de la Faculté de Médecine de Paris
(1810) (Fig. 1A) is provided in Table 1a (Fig. 1B). Fig. 1. A, Cover page and B, specimen description
This initial description could be corroborated else- by Beauchêne (1810).
where including its inclusion in the 2-volume 1st, or
1857, edition (Table 1b) and a 5-volume 2nd, or
1878, edition of the catalogue of 6,000 specimens
(Fig. 2) (Table 1c) housed in the Musée Dupuytren
and prepared by its curator Charles-Nicolas Houel. 75006) revealed that this catalogued specimen was
This specimen was not included in an earlier, 1842 fortunately not only still part of its collection but was
catalogue of the library (Museum d’Anatomie Patho- displayed on its shelves. The Musée Dupuytren,
logique de la Faculté de Paris, ou Musée Dupuytren, founded in 1835 at the bequest of the famed anato-
1842) which described exclusively bony lesions. A mist and surgeon Guillaume Dupuytren (1777-1835)
subsequently published medical encyclopedia of the (Barsky, 1984), has long been a repository of
time also correctly described the case ‘‘le névrome numerous important anatomic specimens. At its
kystique’’ (Nouveau Dictionnaire de Médecine et de founding, the museum had inherited original speci-
Chirurgie Pratiques, 1877). mens from faculty members in Paris and elsewhere.
Unfortunately, funding issues over the years led to
Part III: Locating and Examining its closure in 1937, placement of specimens into
the Specimen storage, and the subsequent loss of many speci-
mens. In 1967, the museum was resurrected and
An inquiry to the Musée Dupuytren (15–21, rue refurbished. Today, it contains many notable speci-
de l’école de médecine, Les Cordeliers, Paris, France mens, including the brains of aphasic patients stud-

TABLE 1. Original Quotations and Translations
Original quote in French
a. Bulletin de la Faculté de médecine de Paris, et de
la Société établié dans son sein (1810) [p. 35] ‘‘15
mars. M. Beauchêne présente vingt-cinq pièces
d’anatomie pathologique dont le détail suit.’’
[p. 36] ‘‘9.8 Kyste séreux formé dans l’épaisseur du
nerf cubital, sur un homme d’environ quarante ans.’’
[p. 39] ‘‘toutes ces pièces numérotées et
étiquetées ont été déposées dans les cabinets de
la Faculté, où on pourra les consulter.’’
b. Houel (1857) ‘‘72. Kyste séreux formé dans
l’épaisseur du nerf cubital, chez un homme de
quarante ans environ (Beauchêne, Bull de la Fac.,
1810, p. 36).’’
c. Houel (1878a) ‘‘n8 137. Portion du nerf cubital;
kyste séreux cette pièce provident d’un home de
40 ans environ. Le nerf cubital a acquis un volume
considérable; il est d’aspect fusiforme et dans son
intérieur, existait un kyste séreux. Cette pièce est
malheureusement sans renseignements (M.
Beauchêne, Bull de la Faculté de Médecine de
Paris, 1810, p. 36).’’
d. Hand-labelled specimen in Musée Dupuytren (Fig.
3A) ‘‘n 137. Portion du nerf cubital; kyste séreux
chez un homme de 40 ans. Mr. Beauchêne. Bull de
la fac. 1810, p. 36.’’
e. Moreau and Van Bogaert (1923) ‘‘devant la Societé
de Chirurgie de Paris proclaimait que ‘quand les
tumeurs des nerfs atteignent un certain volume on
peut y rencontret des cavités kystiques a contenu
séreux (Mem Soc Chir Paris I. III, p 260).’ ’’
f. Houle (1878b, p. 259) ‘‘lorsque la tumeur acquiert
un gros volume, on y trouvé des cavités ou petits
kystes contenant un liquide séreux.’’
ied by Paul Pierre Broca, specimens critical in the
localization of brain functions, in addition to wax an-
atomical models, books, and photographs.
The specimen of note is in a container which is
hand-labeled (Fig. 3A) (Table 1d). The information
and numbering of the specimen (#137) corre-
sponded to Houel’s 2nd compendium (1878a).
Bathed in formaldehyde, the ulnar nerve specimen
was suspended by strings. Using loupe magnifica-
tion, the remarkably well-preserved specimen was
readily examined. The *30-cm long segment of
nerve was displayed in w-shape configuration to
accommodate its length (Fig. 3B). Thus, it was not
anatomically oriented. The described, fusiform,
cystic enlargement was apparent in the mid-portion
of the specimen; measuring 7 cm in length and 12
mm in diameter, it appeared ‘‘balloon-like,’’ its fas-
cicles being eccentrically displaced by small cystic
blebs situated on the medial aspect of the cyst.
Tapering cyst extended *1 cm above the fusiform
expansion and *3 cm distal to it (Fig. 3C). At the
distal extent, a medially projecting cystic nerve
branch was noted and seen to possess a minute but
distinct lumen (Fig. 3D and 3E). Further distally, short
remnants of several other branches could be identified
that did not contain lumens and were not enlarged by
cyst. We were not permitted to remove the specimen
from its container for direct examination.
Intraneural Ganglion Cyst 613
Translated quote in English
March 15. Mr. Beauchêne presents 25 anatomic
pathology specimens with the following details.
Number 9. A serous cyst which has formed within
the ulnar nerve in a man of about 40 years of age.
All these numbered and tagged specimens have
been stored in the cabinets of the university where
one should be able to consult them.
Number 72. Serous cyst which has formed within the
ulnar nerve in a man about 40 years of age
(Beauchêne, Bull de la Fac., 1810, p. 36).
Number 137. Portion of the ulnar nerve: serous cyst.
This specimen is from a man about 40 years of age.
The ulnar nerve has acquired a large size. It was
fusiform and within there was a serous cyst.
Unfortunately, this specimen has little additional
information (M. Beauchêne, Bull de la Faculté de
Médecine de Paris, 1810, p. 36).
Number 137. Portion of ulnar nerve: serous cyst in a
man 40 years of age. Mr. Beauchêne. Bull de la fac.
1810, p. 36.
Before the Surgical Society of Paris, he proclaimed
that ‘‘when the tumors of nerve attain a certain
volume, one can find within them cystic cavities with
serous contents (Mem Soc Chir Paris I. III, p 260).’’
When the tumor becomes large in size, one can find
within them cavities or little cysts containing a
serous liquid.
RESULTS
Reinterpreting the Specimen with a
Modern Perspective
Despite the lack of surrounding anatomic land-
marks and operative and modern imaging data, as
well as our inability to probe the lumen of the articu-
lar branch, image or to histologically examine the
specimen, it was nonetheless possible to make a
number of conclusions, particularly with respect to
the pathogenesis of the cyst.
First, basic anatomic landmarks permitted us to
orient the specimen. The larger diameter of the
nerve (right side of Fig. 3B) corresponded to the
proximal ulnar nerve, wherein it is ‘‘a nerve of pas-
sage,’’ not having any branches. The distal portion
of the continuous specimen was of smaller diameter
(left side of Fig. 3B) and exhibited several branches
corresponding to the segment in the proximal fore-
arm. The most proximal of these, the articular
branch to the elbow, typically arises at the level of
the medial epicondyle (Bekler et al., 2008). The
additional branches would be muscular branches to
the flexor carpi ulnaris and flexor digitorum profun-
dus. The posteromedially directed take-off of the
articular branch would suggest the specimen was
from the right arm. Further aids to localization are
614 Spinner et al.
Fig. 2. A, Cover page and B, specimen description
in Houel’s 2nd compendium (1878a).
the cystic blebs within the medial aspect of the par-
ent nerve, a pattern typical of proximal fluid propa-
gation.
Secondly, general orientation of the specimen
having been achieved, our current understanding of
the pathologic anatomy and of the mechanism of
intraneural ganglion cyst formation permits us to
firmly state that this specimen represents such a
cyst involving the ulnar nerve, and that it is derived
from the medial portion of the right elbow joint. The
mechanism of cyst formation and propagation is
illustrated in Figure 4. With respect to our specimen,
the articular branch served as the conduit, a path of
least resistance for joint fluid to pass from the joint
into the parent ulnar nerve. The fusiform configura-
tion of the cyst is characteristic of this lesion, akin to
the shape of an inflated long, thin circus balloon
(i.e., the ‘‘balloon sign’’), a reflection of hydro-
dynamic principles (Spinner and Amrami, 2006).
Preferential cyst extension in a proximal direction
(‘‘ascent") rather than in a distal direction
(‘‘descent") is also typical (Spinner et al., 2008). It
would seem that, due to pressure differences in
overlying tissues, the balloon-like expansion oc-
curred proximal to the region of the cubital tunnel.
The pathologic finding of a definite, hollow lumen in a
cystic dilated branch of a parent nerve also indicates it
is the articular branch, the conduit leading from the
joint into the parent nerve. No such lumen was seen
within the muscular branches. Finally, the eccentric,
lateral displacement of nerve fascicles by the cyst
explains the ‘‘signet ring’’ sign so well seen on MRI
(Spinner et al., 2006), and a manifestation of the
tendency of proximal dissection to occur in the medial
portion of the nerve.
DISCUSSION
Historical Clarification
For 200 years, this historic specimen has received
little attention, its very nature being misunderstood.
In many previous reports, cross-referencing of origi-
nal sources led to perpetuated errors of citation. For
more than 80 years, the existence of the specimen
described herein and its contribution to medicine
was attributed to Duchenne (occasionally to Duch-
êne) by Moreau and Van Bogaert (1923) on pages
866 and 873, respectively. These probably incor-
rectly referred to the noted French neurologist
(1806–1875) with a similar sounding name.
To our knowledge, Beauchêne himself was never
accurately referenced in the many papers on the sub-
ject. Indeed, the very next known case of intraneural
cyst, included in an 1837 thesis by Bertrand, failed to
acknowledge Beauchêne’s description. In the extant
references, he was variably referred to as Beauchêne
(Houel, 1857; Nouveau Dictionnaire de Médecine et
de Chirurgie Pratiques, 1877), M. Beauchêne (Bull de
la Faculté de Médecine de Paris, 1810), Mr. Beau-
chêne (original labeled specimen, Fig. 3A), and as M.
Beauchêne fils (1809). We now know more about
Beauchêne than before. He was the son of Edmé-
Pierre Chauvot de Beauchêne, one of the physicians
of Louis XVIII, and a member of the French Royal So-
ciety of Medicine. At the University of Paris, Beau-
chêne fils (1809) wrote a prize-winning thesis on cat-
aracts. He also worked as a prosector in the Ecole de
Médecine at that time and later served as deputy
head surgeon at the Hospital in Rue Saint-Antoine.
Lastly, in 1818, he recognized the occurrence of air
emboli during surgery in the neck (Gross, 1857).
Similar errors in citations related to our specimen
have been equally problematic. Charles-Nicolas
Houel (1815–1881) was credited for a quote in
1811, fully 4 years before his birth! (Moreau and Van
Bogaert, 1923) (Table 1e). In fact, the actual quote
is found in a different reference (Houle, 1878b,
p.259) (Table 1f).

Fig. 3. Beauchêne’s original specimen as it appears
today in the Musée Dupuytren in Paris. A: Hand-written
label on the specimen container. The date 1810 can be
seen. Part of the label is not shown in this view due to the
curvature of the container. B: Anterior view. Specimen of
ulnar nerve cyst. * demonstrates the fusiform intraneural
cyst. Arrow, cyst dissection from joint; dashed arrow,
direction of proximal extension; arrowheads, muscular
Unfortunately but understandably, the few incor-
rect citations in the paper by Moreau and Van
Bogaert (1923) have been repeated by subsequent
authors over the ensuing decades, including a
recent report (Bonar et al., 2006). The widespread
problems surrounding such inaccuracies were the
focus of editorials (Spinner and Northouse, 2004;
Spinner et al., 2005). Still the excellent review by
Intraneural Ganglion Cyst 615
branches; P - proximal; D - distal. C: Close-up view of
(B). A cystic bleb within the ulnar nerve can be seen distal
to the fusiform enlargement (*). D: Posterior view. The
transparent nature of the cyst (*) and the displaced fas-
cicles of the ulnar nerve can be appreciated within the
fusiform enlargement. E: Close-up view of (D). The lumen
of the articular branch is well seen. Subtle evidence of
cyst distal to articular branch (*) suggests mild descent.
Moreau and Van Bogaert (1923) comprehensively
summarizes not only the early reports of intraneural
ganglion cysts, many reported in obscure journals
and in several languages, but also the unreported
cases as well. All credit to them; their paper pro-
vided us with useful clues permitting the tracking
down of original sources and, eventually, the actual
specimen.
616 Spinner et al.

Fig. 4. Artist drawings corresponding to the original specimen. Interpretation

and proposed mechanism of Beauchêne’s ulnar intraneural cyst formation from the
elbow joint and propagation. 1 - articular branch of ulnar nerve with hollow lumen;
2 and 3 - distal and proximal portions of ‘‘balloon’’-like expansion; FCU - flexor carpi
ulnaris (by permission, Mayo Foundation, 2008).

Scientific Clarification intraneural ganglion cyst should be distinguished

The features of Beauchêne’s gross specimen are
typical of surgically and histologically proven intra-
neural ganglion cysts occurring at this and other
anatomic sites. They most often affect the fibular
(peroneal) nerve, arising there from the superior
tibiofibular joint, but examples have been reported
to involve the ulnar nerve at the elbow (Zum Busch,
1895; Jardini, 1907; Tanaka et al., 1969; Hori et al.,
1986; Allieu et al., 1987; Hansis et al., 1988; Allieu
and Cenac, 1989; Ferlic and Ries, 1990; Inhofe and
Moneim, 1996; Harway, 1997; Uetani et al., 1998;
Choi et al., 1999; Sharma et al., 2000; Chick et al.,
2001; Chan et al., 2003; Rezzouk and Durandeau,
2004; Boursinos and Dimitriou, 2007; Lohmeyer et
al., 2007). These latter cases in every way resemble
the case reported herein. However, joint connections
from articular branches have only rarely been
described in ulnar examples occurring at the elbow
(Hori et al., 1986, Rezzouk and Durandeau, 2004).
We maintain that joint connections in the remaining
cases were unrecognized. We have previously dem-
onstrated that failure to identify joint connections is
not only common but also a frequent cause of intra-
neural cyst recurrence (Spinner et al., 2003a,b).
Early accounts of tumors and mass lesions of
nerve did not distinguish between intraneural cysts
and other cystic lesions of nerve now well character-
ized. Although a distinctive form of neural cyst, the
intraneural ganglion cyst has been variously termed
as pseudocyst, mucoid pseudocyst, synovial cyst,
and cystic myxoid degeneration. The viscosity of the
cyst content, appears serous (as described by Beau-
chêne) in early and mucinous in long-standing
examples. With respect to differential diagnosis, an
from other cystic lesions of nerve, most particularly,
nerve sheath tumors. The latter are not unreason-
able considerations. For example, we have encoun-
tered remarkably thin-walled, nearly transparent
cystic schwannomas.
Our finding of this surviving cadaveric specimen
with a cystic articular branch provides compelling
evidence in support of our unified articular (synovial)
theory (Spinner et al., 2003b; Spinner et al., 2007).
We had previously attempted to examine two cases
dating to 1884 and 1902 both loosely referred to by
Moreau and Van Bogaert (1923); these consisted of
an intraneural ganglion cyst of the ulnar nerve in the
hand arising from an unspecified wrist joint and
another involving the internal popliteal nerve (tibial
nerve) with a ‘‘knee joint’’ (possibly the superior
tibiofibular joint) connection from an inferior internal
articular branch. These two specimens were housed
at the pathological museum of St. Bartholomew’s
Hospital Medical College, London, England (A De-
scriptive Catalogue of the Pathological Museum of St.
Bartholomew’s Hospital Medical College, 1929), but
could not be located and are thought to be lost. One
must wonder if the earlier description by Cheselden
(1741) of a similar case of a cystic ‘‘cubital’’ (ulnar)
nerve tumor in a similar location as Beauchêne’s -
and long thought to be the first description of a
nerve sheath tumor, might also have represented an
intraneural ganglion cyst. This specimen similarly
cannot be found. We are unaware of other examples
of intraneural ganglion cyst currently languishing in
museums but would welcome the opportunity to
examine them.
Our experience with the three aforementioned
cadaveric specimens, replete with past or present

descriptions of identified joint connections, further
expand and confirm our clinical and experimental ex-
perience, which underlies the unified articular (syno-
vial) theory. We believe that their joint relationship
is fundamental to intraneural cyst formation and that
propagation of joint fluid within epineurium along
path(s) of least resistance may be modulated by
dynamic pressure fluxes (Spinner et al., 2007).
Knowledge about the pathogenesis of intraneural
ganglion cysts has simplified the surgical treatment
now consisting of performing limited cyst drainage
procedures instead of cyst resection and disconnect-
ing the articular branch. The former decreases post-
operative neurologic deficits, and the latter prevents
once-frequent recurrences. This simple therapeutic
approach has markedly improved outcomes (Spinner
et al., 2003a).
CONCLUSION
Beauchêne’s intraneural cyst of the ulnar nerve is
an important specimen. Its brief description was pre-
viously hidden in a catalogue and long miscited. The
specimen itself, dormant on a cluttered museum
shelf, was only retrieved with great effort. It is now
on display for study. As Beauchêne originally
intended (1810, line 39), this specimen is now a val-
uable resource for researchers in the future. Its
availability has allowed us to establish it as a bona
fide example of an intraneural ganglion cyst and to
confirm its articular orgin. A 200-year-old medical
and nosologic mystery has been solved.
ACKNOWLEDGMENTS
The authors acknowledge the generous coopera-
tion of the Musée Duyputren, Paris, France, which
permitted examination and photography of the spec-
imen as well as its permission to reproduce the fig-
ures. Dr. Patrice Josset, the museum’s conservateur,
and Patrick Conan, its conservateur adjoint, were
most gracious and helpful. The assistance of Profes-
sor Peter Hicks of the Foundation Napoléan was also
invaluable. We also acknowledge the initial library
searches of Mrs. Pat Erwin and the persistent
searches for additional information by Huan Wang,
M.D. and Diana Angius, M.D., of Rochester, Minne-
sota and Mr. Steven Moore of London, England.
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