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AUTOTRANSPLANTATION OF SPLEEN: SPLENOSIS

CASE REPORT AND PRELINIINARY REPORT OF AN EXPERIMIENTAL STUIDY IN


REVASCULARIZATION OF THE HEART*
JOSEIH J. GARAMELLA, M.D. AND LYLE J. HAY, MI.D.
MINNEAPOLIS, MINN.
FROM TIlE DEPARTMENT OF SURGERY OF THE VETERANS ADMIINISTRATION HOSPITAL, NIINNEAPOLIS, MINNESOTA

THE ENTITY OF autotransplantation of the Splenosis. The term splenosis, autotrans-


spleen, splenosis, is sufficiently rare to plantation of splenic tissue, was first intro-
warrant publication of a case. This paper duced by Buchbinder and Lipkoff3 in 1939.
will present a case report and a preliminary They described the first case in this coun-
report of an allied experimental study. try. Microscopic examination of splenic im-
History. The first known reference to the plants show that the blood vessels do not
condition of autotransplantation of splenic enter through a hilus as is true of accessory
tissue following rupture of the spleen was spleens, but rather penetrate the capsule
made by von Kuttner" in 1910. He re- around its circumference. The histological
garded these as accessory spleens and not pattern is essentially the same as normal
as implants. Faltin6 was the first to suggest spleen.
that these splenic nodules were actually Parenchymatous cells of no other abdom-
splenic implants. inal organ exhibit similar characteristics
Albrecht,1 in 1896, and Schilling,16 in except possibly those of uterine endo-
1907, reported cases with splenic foci, 400 metrium.8
and 42 respectively, dispersed throughout For reviews of the literature, the reader
the peritoneal cavity. Although reported as is referred to the reports of Shaw and
cases representing accessory spleens, the Shafi,17 Buchbinder and Lipkoff,3 Hamrick
distribution and number are more consist- and Bush, 8 Waugh,2' and recently by
ent with autotransplantation of splenic tis- Storsteen and ReMine,19 who collected 22
sue. A history of trauma was lacking in each cases from the literature and added one of
report. their own. The case herein reported will be
Accessory Spleens. Shaw and Shafi17 dis- the twenty-fourth.
tinguished splenic implants from accessory Waugh's case is the first report of auto-
spleens (spleniculi) by their greater num- transplantation in an adult following rup-
ber, widespread location and history of ture of the spleen and splenectomy. The
trauma. Accessory spleens are usually lo- case reported by Roettig, Nusbaum and
cated in the gastrolienal ligament, but may Curtis';' is of interest since intestinal ob-
be found about the pancreas and rarely in struction was attributed to splenic implants.
the omentum. They are usually two to six The case report of Stobie18 deserves com-
in number, but as many as 40 have been ment. Although reported as a case of recur-
found. Accessory spleens are common con- rent congenital hemolytic anemia due to
genital anomalies. splenosis following splenectomy, several
aspects warrant analysis. That is to say, the
* Subnmitted for publication November, 1953. etiology of the patient's jaundice, anemia
107
GARAMELLA AND HAY Annals of Surgery
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FIG. 1. Photograph of small intestine at time


of laparotomy showing multiple splenic nodules of
varying sizes on antemesenteric borders of
jejunum.
and splenomegaly is not entirely clear. FIG. 2. Photomicrograph (Magnification x 4.6)
From the data given, one may conclude of one of the splenic nodules removed at surgery.
Note the outer capsule (A), Trabecula (B), Fol-
that the patient had cholelithiasis, acute licle (C), Red Pulp (D).
cholecystitis, choledocholithiasis, and a FIG. 3. Photomicrograph (x 23) of same splenic
nodule showing follicles and red pulp in more
stricture of the common duct which re- detail.
quired a secondary operation. The diag-
nosis of congenital hemolytic anemia from Rectal examination disclosed marked tenderness
the data given is not supported by sphero- on the right side.
cytosis, fragility tests, nor by a positive fam- Laboratory Data: Hemoglobin, 15.3 Gm.;
ily history. The contention, therefore, of W.B.C., 15,600, 82 per cent neutrophils, 17 per
cent lymphocytes, 1 per cent eosinophils. Urinaly-
those who maintain that splenosis does not sis was normal; chest roentgen ray was also normal.
follow rupture of a diseased spleen,' 11, 17, 21 The clinical diagnosis was acute appendicitis and
requires clarification and may remain laparotomy was performed. The appendix was
tenable. found to be acutely inflamed. In addition there
were multiple, reddish-brown nodules of various
CASE REPORT sizes on the antemesenteric border of the jejunum,
on the right lateral peritoneal
A 22-year-old white male (R#94170) was meso-appendix. Appendectomywall,
and one on the
was performed and
admitted on August 15, 1949, with a 6-day history two nodules were removed for histologic
of pain in the right lower quadrant associated with study.
Pathologic Report. Gross Appearance: Speci-
nausea and vomiting for 36 hours prior to admis- men No. 1. Consists of an
sion. He took castor oil with no relief. The day length to which is attached a appendix
7 cm. in
firm, matted
prior to admission, the pain became severe and he appendix. On cut section the appendix is found meso-
vomited 3 to 4 times. Splenectomy had been pre- to be kinked at the distal end and the distal
viously performed on an undisclosed date for rup- tion is dilated and filled with purulent material. por-
ture of the spleen following a fall from a horse. The proximal end shows no
The patient weighed 140 pounds on admission. lumen, but the serosal surface isdilatation
involved
of the
in the
He was not in acute distress. Physical examination inflammatory process.
disclosed: T, 37.2° C; P, 82; R, 18; B.P., 110/70. Specimen No. 2. Consists
Pertinent findings were the presence of pain, ten- pieces of tissue, one measuring 2ofx two lobulated
1.6 x 0.5 cm.;
derness and spasm in the right lower quadrant. the second measuring 9 mm. in greatest
diameter.
108
'olujiie 1 10 AUTOTRANSPLANTATION OF SPLEEN: SPLENOSIS
Nurnhber I

On cut section, the tissue is firm and has a lightly


mottled, brownish color.
Microscopic Appearance: Specimen No. 1. The
mucosa is absent at the tip of the appendix. The
lumen is packed with polymorphonuclear cells.
The acute inflammatory exudate shows extensive
infiltration throughout all layers of the appendix,
but is most marked in the region of the meso-
appendix.
Specimen No. 2. Sections through the nodule
reveals an outer capsule with trabeculae extend-
ing from this into the underlying parenchymal tis-
sue. The parenchyma contains scattered lymph
follicles separated by tissue having the histological
appearance of the red pulp of the spleen with cords
of Billroth separating sinusoids that are filled with
erythrocytes.
AMicroscopic Diagnoses. The microscopic diag-
nosis was (1) acute suppurative appendicitis; (2)
splenic implants.

Experimental Splenosis. Experimentally,


splenic tissue may be implanted and readily
survives. Von Stubenrauch20 removed the
spleen in several dogs and seeded the
crushed pulp throughout the peritoneal cav-
ities. Sacrifice of the animals 1 to 3 months jlE.'~~~~~~~~~~~..... .tF;
... .....

later showed multiple implants throughout FIG. 4. Photograplh of heart-lung specimen of


dog. Splenic tissue implanted 23 days prior to ex-
the peritoneal cavity. Kreuter'0 performed ploration. Note layer of splenic tissue growing be-
the same experiments in monkeys, obtain- tween epicardium and pericardium (A). The peri-
cardium and epicardium at the apex are fused by a
ing similar results. Jarcho and Anderson9 layer of splenic tissue (B). Isolated splenic implants
reported cases of dogs that had sustained as (C) and (D) are also present.
FIG. 5. Photomicrograph (x 16) showing layer
severe abdominal trauma showing multiple of splenic tissue (A) implanted on pericardium
splenic implants at autopsy. They stated (B).
that Griffini and Tizzoni,7 as early as 1883,
noted spleen-like nodules in the peritoneum ture, if successfully implanted upon the
following partial splenectomy in dogs. Ma- pericardium, might be effective in protect-
rine and Manley,12 Roettig, Nusbaum and ing the heart with coronary artery insuffi-
Curtis,'5 and Williams,22 successfully im- ciency following the "blood bath" principle
planted splenic tissue in rabbits, the graft of Beek.2
sites being the subcutaneous tissues of the It was the object of this study, therefore,
abdomen, peritoneum and ears, respec- to determine whether or not splenic tissue
tively. Perla13 transplanted splenic tissue in could be successfully implanted upon the
the abdominal wall of albino rats. Put- pericardium of experimental animals.
schar14 transplanted splenic tissue in the Alethod. Dogs not older than one year
peritoneuim of rats while Calder' graftedl were used as experimental subjects. Anes-
splenic tissue in the omlenitiiml o)f wivite Imlice thlesia consisted of intravenous nembutal,
ained albino rats. 14 mg. per pound of body weight. An endo-
Experitmietital Stuid,. Sinice splenic tissue tracheal tube was employed and connecte(d
is rich in vascularity and serves as blood to a mechanical r espirator. Thoracotomy
a

reservoir, it was conjectured that this struc- was performed through the seventh inter-
109
GARAMELLA AND HAY Annals of Surgery
J ul y 1 9 5 4

A-
c-

.B
FIG. 6 FiG. 7
FIc. 6. Photomicrograph (x 16) showing layer of splenic tissue (A) implanted on iliyo-
cardiuim (B).
FIG. 7. Photomicrograph (x 10) of biopsy done 28 days after implantation of splenic tissue
in pericardial sac. Note bridging of pericardium (A) and myocardiumii (B) by splenic tissues
(C). Also note the vascularity of the extra pericardial fat (D).

costal interspace on the left, facilitating increase in fibrous tissue elements. Autopsy
access to the diaphragm and the pericar- of dog number 188 showed bilateral hydro-
dium at the same time. Transdiaphragmatic thorax, bilateral atelectasis, compression
splenectomy was performed and the re- type, pericardial effusion, and congestion of
moved spleen was subsequently cut into the liver. Histological study disclosed an
fine particles, 1 to 2 mm. The pericardium acute inflammatory reaction of the splenic
was incised vertically for a distance of 3 to tissue in the pericardium. It is felt that
4 cm., and approximately half of the finely this animal developed an infection of the
cut splenic tissue was introduced into the pericardium and succumbed to heart fail-
pericardial sac. The pericardium was ure secondarv to cardiac tamponade.
closed only tightly enough to prevent es-
cape of splenic tissue. Penicillin, 500,000 DISCUSSION
uinits, was introduced into the hemithorax
and the chest closed. The pericardium was One can only speculate as to the practical
explored three to four weeks later. application of splenic tissue implanted
Comment. From the data in Table I, it uipon the epicardium and pericardium as a
seems clear that splenic tissue can be suc- means of revascularizing the heart. The
cessfully and consistently implanted upon vascular character of splenic tissue and its
the epicardium and pericardium of dogs. reservoir function are well recognized, and
The coalescence of implants occurs at the it would seem that an actively growing
dependent portion of the pericardial sac, structture such as this, bridging the epicar-
overlying the left ventricle, principally. dium and pericardium, diverting the extra
Deliberate attempts to produce a more coronary blood supply, may be more effec-
diffuse implantation of splenic tissue and tive than the granulation tissue of surgically
bridging of the epicardium and pericar- indtuce(d adhesive pericarditis, the natural
dium by employing abrasion, thrombin history of which is retraction and scar for-
solution and gel foam met withl only a slight mationi. A method to induce a diffuse and
measure of success in these few experi- uniform implantation of splenic tissue with
ments. The splenic tissue in these latter bridging of the epicardium and pericar-
instances histologically showed a significant dium would be desirable. An artificial
110
Volunme I1
Number 1 AUTOTRANSPLANTATION OF SPLEEN: SPLENOSIS

TABLE I.
Presence of Bridging of Epicardium and
Dog No. Operation Exploration Implants Distribution Pericardium by Splenic Tissue
70 3- 4-53 4- 1-53 Yes Overlying left ventricle, dependent Yes. At site where pericardium in-
portion area 3.5 x 2.0 cm. Also cised initially. Confirmed by
scattered implants 0.5 + cm. biopsy.
62 3-11-53 4- 6-53 Yes Essentially same as Dog No. 70. Yes. Essentially same as Dog. No.
70. Confirmed by biopsy.
132 3-18-53 4-22-53 Yes Scattered implants on epicardium No.
and pericardium.
159 4-29-53 5-22-53 Yes Pancake layer of splenic tissue nmost Yes. Diffuse bridging overlying left
*1 dense over the left ventricle. ventricle.
188 5- 6-53 5-14-53 Yes Extensive. Yes. Diffuse.
*2 Died
146 5-13-53 6- 5-53 Yes Pancake of splenic tissue overlying Yes. Overlying left venitricle. See
*3 left ventricle. I Photograph.
162 5-20-53 6-11-53 Yes Layer overlying left ventricle with Yes.
*4 scattered nodules overlying peri-
cardium of right ventricle.
Attempting to induce diffuse implantation of splenic tissue, the operative procedure was modified in the last four experimenits.
*1. Pericardium abraded and sterile talc introduced.
*2. Epicardium and pericardium abraded with gauze. Thrcmbin solution introduced into pericardial Fac.
*3. Epicardium and pericardium abraded. Splenic tissue mixed with gel foam.
*4. Epicardium and pericardium abraded. Thrombin solution introduced into pericardial sac.

means of producing an effective hyper- 3 Buchbinder, J. H., and C. J. Lipkoff: Splenosis:


plasia of implanted splenic tissue may be Multiple Peritoneal Splenic Implants Follow-
ing Abdominal Injury. Surgery, 6: 927, 1939.
practical. The long term behavior of splenic 4 Calder, R. M.: Autoplastic Splenic Grafts:
tissue in the pericardial sac bears study. Their Use in the Study of the Growth of
The degree of protection afforded the myo- Splenic Tissue. Jour. Path. and Bact., 49:
cardium by implanted splenic tissue is 351, 1939.
being pursued. 5 Curtis, G. NI., and D. Movitz: The Surgical
Significance of the Accessory Spleen. Ann.
SUMMARY Surg., 123: 276, 1946.
1. The subject of splenosis is briefly re- 6 Faltin, R.: Milzartige Bildungen in Peritoneum,
viewed and a case report is presented. beobachtet ca. 6 Jahre nach einer Wegen
2. Instances of experimental splenosis Milzruptur vorgenonimenen Splenektomie.
Deutsche Ztschr. f. Chir., 110: 160, 1911.
are cited. 7 Griffini, L., and G. Tizzoni: Etude Experimen-
3. The preliminary report of the experi- tale sur la reproduction partielle de la rate.
mental study, transplantation of splenic Arch. Ital. de Biol., 4: 303, 1883.
tissue into the pericardial sac, is presented. s Hamrick, R. A., and J. D. Bush: Autoplastic
4. In all experimental animals studied Transplantation of Splenic Tissue in Nian,
Following Traumatic Rupture of the Spleen.
(seven), splenic tissue was successfully im- Ann. Surg., 115: 84, 1942.
planted. 9 Jarcho, Saul, and D. H. Andersen: Traumatic
5. The protection afforded the myocar- Autotransplantation of Splenic Tissue. Am.
dium by splenic tissue is being studied. J. Path., 15: 527, 1939.
10 Kreuter: Experimentelle Untersuchungen uber
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