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Double gallbladder
Double Gallbladder DONALD
Vesica fellea duplex (true double gallbladder) is a rare and unique congenital anomaly of the biliary tree. (Fig. 1.) Fifty-one cases have been reported in the literature since Pliny [I] first recorded the finding of a double gallbladder in a sacrificial victim at Emperor Augustus’ victory over Antony and Cleopatra at Actium in 31 B.C. The following case is the forty-second operative report, bringing the total of operative and autopsy cases to fiftytwo. This is the first case report presenting photographs of the specimen in situ demonstrating the cystic ducts entering the common duct, as well as presenting a photograph of the removed specimen. Case
Report
Nine months prior
to admission, the patient year old white woman, had pain in the right upper quadrant and nausea and vomiting which persisted for twentyfour hours while she was in the seventh month of her third pregnancy. She gave birth to a normal child at term without any further difficulty. Seven months after the first attack, the pain recurred and scleral icterus was noted. The jaundice subsided. A cholecystogram was obtained and there was good visualization with a small filling defect in the neck of the gallbladder compatible with a stone. The patient was referred to the Tulane Surgical Service at Charity Hospital in New Orleans. Roentgenogram of the chest, complete blood count, urinalysis, and determination of bilirubin, serum glutamic oxaloand alkaline phosphatase acetic transaminase, all gave normal results at this time. Elective cholecystectomy was carried out on October 11, 1966. At operation there appeared to be one gallbladder with two cystic ducts entering the common duct separately. (Fig. 2 and 3.) Removal of a common peritoneal sheath revealed two separate
(M.J.), a twenty-three
From the Department of Surgery, Tulane University School of Medicine, and the Charity Hospital of Louisiana, New Orleans, Louisiana. * Present address: Maj. D. J. Palmisano. USAF, MC Chief of Surgery, Ellsworth Air Force Base Hospital, South Dakota 57706.
Vol.
118, September
1969
J. PALMISANO,
M.D.,*
New Orleans,
Louisiana
gallbladders. (Fig. 4.) Exploration of the common bile duct yielded a small amount of sludge, but the dilators entered the duodenum with no difficulty, and no stone was found. An operative cholangiogram showed nothing abnormal. Both gallbladders were opened and one had hemorrhagic mucosa. No stones or polyps were found. The pathologist reported chronic cholecystitis in both specimens after microscopic examination. The patient had an uncomplicated recovery. A T tube cholangiogram showed nothing abnormal prior to removal of the tube. The patient was discharged and has remained well.
Comments The true double gallbladder, vesica fellea duplex (two separate gallbladders and two separate cystic ducts), is subdivided into (1) a Y-shaped type, in which two cystic duets unite into one prior to entering the common duct, and (2) a ductular type, in which two cystic ducts enter the common duct separately. The gallbladders may have a common peritoneal covering [2]. The theory of development of a double gallbladder has been reviewed by Boyden [Cl and Martenstyn [3]. At one stage in its development, it is bilobed and failure of these buds to fuse would result in a double gallbladder. Also a rudimentary duct bud may fail to regress, resulting in a double gallbladder. Blasius [4], in 1674, reported the first ductular type in a two year old male cadaver. The first Y-shaped type was discovered in an infant cadaver and described by Huber 151 in 1734. The first operative case was reported in 1911 by Sherren [6]. In 1926 Nichols [7] reported the first confirmed case demonstrated by roentgenogram. The incidence in human subjects was noted by Boyden [2] to be two in 9,221 autopsied patients and three in 9,970 patients in whom the gallbladder was visualized. The most common type of double gallbladder has been the ductular type with the two gallbladders side by side with a common peri463
Palmisano
Fig. 4. Specimen with common peritoneal covering removed demonstrating two separate gallbladders and cystic ducts.
Fig. 1. Double gal/bladder
with common
peritoneal
covering.
Fig. 2. Operative specimen demonstrating common peritoneal covering which appears as a single gallbladder.
covering. In 1954, Moore and Hurley [8] found thirty-six cases of confirmed double gallbladder in the literature. Twenty-seven were noted at operation and nine at autopsy. Twenty-two were of the ductular type and seven were Y-shaped. An additional fifteen proved cases have been found in the literature since 1954 [3,9-,211. Including the case reported herein, ten of these sixteen cases since 1954 were of the ductular type. Separate locations have been reported. Wischnewsky [22] reported one gallbladder to the right of the falciform ligament and the other to the left. In an infant at autopsy, Mulla and Weintraub [11] found a second gallbladder connected to an accessory liver located in the right chest. toneal
fig. 3. Drawing and photograph showing two cystic ducts entering separately. Silk ligatures are looped around cystic ducts. 464
common
The
American
duct
Journal
of Surgery
Double Gallbladder
On occasions, both gallbladders have not been removed at operation. Moore and Hurley [8] found only one of the gallbladders inflamed and since they were separated, they left the normal one. Recht [23] did the same in a case of volvlus. Ingegno and D’Albora [24] examined a double gallbladder at coincidental laparotomy and left both. Carcinoma has also been reported in a case of double gallbladder [20]. Preoperative diagnosis of a double gallbladder and confirmation at operation has been possible in fifteen cases [25]. The radiologic diagnosis of a double gallbladder may be difficult. A kinked gallbladder with a phrygian cap deformity or cholecystitis glandularis proliferans may be the cause of bilocular appearance. The diagnosis of double gallbladder is permissible only if two cystic ducts can be shown. Stones outside of a visualized gallbladder also suggest double gallbladder. Certain diagnostic problems may arise with a double gallbladder. Visualization of the gallbladder may appear normal in a patient with acute cholecystitis because disease may affect only one of the gallbladders and the noninflamed gallbladder will visualize. The physician’s attention would then be directed away from the pathologic condition. An inflamed gallbladder may be left behind, as reported by Munson and Teixido [21]. Removal of a stone-filled gallbladder failed to relieve the patient’s symptoms and exploration revealed another gallbladder with stones. The symptomatic cases of double gallbladder have a high incidence of disease in both gallbladders, as might be expected 119,261. Double cholecystectomy has been recommended as the procedure of choice.
The true double gallbladder is two separate gallbladders and two cystic ducts. The fortysecond operative case is presented, bringing the total cases proved by operation or autopsy to fifty-two. Certain diagnostic problems may arise with a double gallbladder. Visualization of the gallbladder may appear normal in a patient with acute choleeystitis because disease may affect only one of the gallbladders, and the noninflamed gallbladder will visualize. Operative photographs, including the first published photograph demonstrating the two cys1969
the common
duct separately,
References 1. 2.
3. 4.
PLINY Natural History, vol. III, p. 555. Translated bv H. Rackham. Cambridre. Mass., 1949. Harvard University Press. BOYDEN, E. The accessory gallbladder: an embryological and comparative study of aberrant biliary vesicles occurring in man and the domestic mammals. Am. J. Amt., 38 : 1’77,1926. MARTENSTYN.H. R. L. and MACKENZIE.S. G. Congenital ’ duplication of the gallbladder. Nova Scotia M. Bull., 44: 171, lY65. BLASIUS,G. Observata Anatomica in Homine, Variisque Animalibus Aliis, p. 128, Amstelod, 1674. HUBER, J. J. Acta Phys. Med. AC. Cues. 9
: 382,1752.
SHERREN, J. A double gallbladder removed by operation. Ann. Surg., 54: 204,191l. NICHOLS, B. H. Double gallbladder. Radiology,
6 : 255,1926.
14.
MOORE,T. C. and HURLEY,A. D. Congenital duplication of the gallbladder. Surgem, 35: 283,1954. WHITTENBERGER, R. N. Reduplication of the gallbladder. Am. J. Surg., 81: 926,1954. CORCORAN.D. B. and WALLACE. K. K. Congenital ‘anomalies of the gallbladder. Am. Surgeon, 20 : 709,1954. MULLA, N. and WEINTRAUB, S. Accessory liver with double gallbladder. Arch. Surg., 71: 202,1955. BANTON, P. B. Double gallbladder with calculi in one. Med. Radiography Phot., 31: 127.1955. Ross, J. A. Double gallbladder with report of a case. Brit. J. Radial.. 29: 109. 1956. WILLIAMS, J. L. A ‘double ’ gallbladder.
15.
RYRBERG, C.
9.
10. 11. 12. 13.
Postgrad.
16. 17.
Summary
Vol. 118, September
tic ducts entering are presented.
18.
M. J., 33:
236, 1957.
H.
Gallbladder duplication. Acta Chir. seandinav., 119: 36, 1960. OWEN, R. A. and WALLACE-JONES,D. R. A case of double gallbladder with unusual features. Brit. J. krg., 49: 577, 1962. HURWITZ, A. Double gallbladder. J. Maine M. A., 55 : 79,1964. DUNKERLY, D. R. Double gallbladder. Proc. Row Sot. Med.. 57: 331.1964.
19. 20. 21. 22.
MA&E, D. B. Diagnosis and treatment of duplication of the gallbladder. Postgrad. Med. J., 42: 213, 1966. RAYMOND,W. and THRIFT, C. B. Carcinoma of a duplicated gallbladder. Illinois M. J., 110: 23$1956. MUNSON. C. L. and TEIXIDO, R. Congenital abnormality of the gallbladder. De‘iaware M. J., 31: 193,1959. WISCHNEWSKY, A. W. Doppelgullenblase Aufgedeckt Operation der Wahrend (Ektomie der erkrankten Supplementaren Blase). Arch. klin. Chir., 135: 779, 1925. 465
Palmisano 23. 24. 26.
466
RECHT, W. Torsion of a double gallbladder. Btit. J. Surg., 39 : 342,1952. INGEGNO,A. P. and D’ALBORA, J. B. Double gallbladder. Am. J. Roentgenol., 61: 671, 1949. GUYER, P. B. and MCLOUGHLIN, M. Con-
26.
genital double gallbladder. Brit. J. Radial., 40: 214,1967. GROSS, R. E. Congenital anomalies of the gallbladder: a review of one hundred and forty-eight cases with report of a double gallbladder. Arch. Surg., 32: 131, 1936.
The American Journal of Surgery
Vesica fellea duplex (true double gallbladder) is a rare and unique congenital anomaly of the biliary tree. (Fig. 1.) Fifty-one cases have been reported in the literature since Pliny [I] first recorded the finding of a double gallbladder in a sacrificial victim at Emperor Augustus’ victory over Antony and Cleopatra at Actium in 31 B.C. The following case is the forty-second operative report, bringing the total of operative and autopsy cases to fiftytwo. This is the first case report presenting photographs of the specimen in situ demonstrating the cystic ducts entering the common duct, as well as presenting a photograph of the removed specimen. Case
Report
Nine months prior
to admission, the patient year old white woman, had pain in the right upper quadrant and nausea and vomiting which persisted for twentyfour hours while she was in the seventh month of her third pregnancy. She gave birth to a normal child at term without any further difficulty. Seven months after the first attack, the pain recurred and scleral icterus was noted. The jaundice subsided. A cholecystogram was obtained and there was good visualization with a small filling defect in the neck of the gallbladder compatible with a stone. The patient was referred to the Tulane Surgical Service at Charity Hospital in New Orleans. Roentgenogram of the chest, complete blood count, urinalysis, and determination of bilirubin, serum glutamic oxaloand alkaline phosphatase acetic transaminase, all gave normal results at this time. Elective cholecystectomy was carried out on October 11, 1966. At operation there appeared to be one gallbladder with two cystic ducts entering the common duct separately. (Fig. 2 and 3.) Removal of a common peritoneal sheath revealed two separate
(M.J.), a twenty-three
From the Department of Surgery, Tulane University School of Medicine, and the Charity Hospital of Louisiana, New Orleans, Louisiana. * Present address: Maj. D. J. Palmisano. USAF, MC Chief of Surgery, Ellsworth Air Force Base Hospital, South Dakota 57706.
Vol.
118, September
1969
J. PALMISANO,
M.D.,*
New Orleans,
Louisiana
gallbladders. (Fig. 4.) Exploration of the common bile duct yielded a small amount of sludge, but the dilators entered the duodenum with no difficulty, and no stone was found. An operative cholangiogram showed nothing abnormal. Both gallbladders were opened and one had hemorrhagic mucosa. No stones or polyps were found. The pathologist reported chronic cholecystitis in both specimens after microscopic examination. The patient had an uncomplicated recovery. A T tube cholangiogram showed nothing abnormal prior to removal of the tube. The patient was discharged and has remained well.
Comments The true double gallbladder, vesica fellea duplex (two separate gallbladders and two separate cystic ducts), is subdivided into (1) a Y-shaped type, in which two cystic duets unite into one prior to entering the common duct, and (2) a ductular type, in which two cystic ducts enter the common duct separately. The gallbladders may have a common peritoneal covering [2]. The theory of development of a double gallbladder has been reviewed by Boyden [Cl and Martenstyn [3]. At one stage in its development, it is bilobed and failure of these buds to fuse would result in a double gallbladder. Also a rudimentary duct bud may fail to regress, resulting in a double gallbladder. Blasius [4], in 1674, reported the first ductular type in a two year old male cadaver. The first Y-shaped type was discovered in an infant cadaver and described by Huber 151 in 1734. The first operative case was reported in 1911 by Sherren [6]. In 1926 Nichols [7] reported the first confirmed case demonstrated by roentgenogram. The incidence in human subjects was noted by Boyden [2] to be two in 9,221 autopsied patients and three in 9,970 patients in whom the gallbladder was visualized. The most common type of double gallbladder has been the ductular type with the two gallbladders side by side with a common peri463
Palmisano
Fig. 4. Specimen with common peritoneal covering removed demonstrating two separate gallbladders and cystic ducts.
Fig. 1. Double gal/bladder
with common
peritoneal
covering.
Fig. 2. Operative specimen demonstrating common peritoneal covering which appears as a single gallbladder.
covering. In 1954, Moore and Hurley [8] found thirty-six cases of confirmed double gallbladder in the literature. Twenty-seven were noted at operation and nine at autopsy. Twenty-two were of the ductular type and seven were Y-shaped. An additional fifteen proved cases have been found in the literature since 1954 [3,9-,211. Including the case reported herein, ten of these sixteen cases since 1954 were of the ductular type. Separate locations have been reported. Wischnewsky [22] reported one gallbladder to the right of the falciform ligament and the other to the left. In an infant at autopsy, Mulla and Weintraub [11] found a second gallbladder connected to an accessory liver located in the right chest. toneal
fig. 3. Drawing and photograph showing two cystic ducts entering separately. Silk ligatures are looped around cystic ducts. 464
common
The
American
duct
Journal
of Surgery
Double Gallbladder
On occasions, both gallbladders have not been removed at operation. Moore and Hurley [8] found only one of the gallbladders inflamed and since they were separated, they left the normal one. Recht [23] did the same in a case of volvlus. Ingegno and D’Albora [24] examined a double gallbladder at coincidental laparotomy and left both. Carcinoma has also been reported in a case of double gallbladder [20]. Preoperative diagnosis of a double gallbladder and confirmation at operation has been possible in fifteen cases [25]. The radiologic diagnosis of a double gallbladder may be difficult. A kinked gallbladder with a phrygian cap deformity or cholecystitis glandularis proliferans may be the cause of bilocular appearance. The diagnosis of double gallbladder is permissible only if two cystic ducts can be shown. Stones outside of a visualized gallbladder also suggest double gallbladder. Certain diagnostic problems may arise with a double gallbladder. Visualization of the gallbladder may appear normal in a patient with acute cholecystitis because disease may affect only one of the gallbladders and the noninflamed gallbladder will visualize. The physician’s attention would then be directed away from the pathologic condition. An inflamed gallbladder may be left behind, as reported by Munson and Teixido [21]. Removal of a stone-filled gallbladder failed to relieve the patient’s symptoms and exploration revealed another gallbladder with stones. The symptomatic cases of double gallbladder have a high incidence of disease in both gallbladders, as might be expected 119,261. Double cholecystectomy has been recommended as the procedure of choice.
The true double gallbladder is two separate gallbladders and two cystic ducts. The fortysecond operative case is presented, bringing the total cases proved by operation or autopsy to fifty-two. Certain diagnostic problems may arise with a double gallbladder. Visualization of the gallbladder may appear normal in a patient with acute choleeystitis because disease may affect only one of the gallbladders, and the noninflamed gallbladder will visualize. Operative photographs, including the first published photograph demonstrating the two cys1969
the common
duct separately,
References 1. 2.
3. 4.
PLINY Natural History, vol. III, p. 555. Translated bv H. Rackham. Cambridre. Mass., 1949. Harvard University Press. BOYDEN, E. The accessory gallbladder: an embryological and comparative study of aberrant biliary vesicles occurring in man and the domestic mammals. Am. J. Amt., 38 : 1’77,1926. MARTENSTYN.H. R. L. and MACKENZIE.S. G. Congenital ’ duplication of the gallbladder. Nova Scotia M. Bull., 44: 171, lY65. BLASIUS,G. Observata Anatomica in Homine, Variisque Animalibus Aliis, p. 128, Amstelod, 1674. HUBER, J. J. Acta Phys. Med. AC. Cues. 9
: 382,1752.
SHERREN, J. A double gallbladder removed by operation. Ann. Surg., 54: 204,191l. NICHOLS, B. H. Double gallbladder. Radiology,
6 : 255,1926.
14.
MOORE,T. C. and HURLEY,A. D. Congenital duplication of the gallbladder. Surgem, 35: 283,1954. WHITTENBERGER, R. N. Reduplication of the gallbladder. Am. J. Surg., 81: 926,1954. CORCORAN.D. B. and WALLACE. K. K. Congenital ‘anomalies of the gallbladder. Am. Surgeon, 20 : 709,1954. MULLA, N. and WEINTRAUB, S. Accessory liver with double gallbladder. Arch. Surg., 71: 202,1955. BANTON, P. B. Double gallbladder with calculi in one. Med. Radiography Phot., 31: 127.1955. Ross, J. A. Double gallbladder with report of a case. Brit. J. Radial.. 29: 109. 1956. WILLIAMS, J. L. A ‘double ’ gallbladder.
15.
RYRBERG, C.
9.
10. 11. 12. 13.
Postgrad.
16. 17.
Summary
Vol. 118, September
tic ducts entering are presented.
18.
M. J., 33:
236, 1957.
H.
Gallbladder duplication. Acta Chir. seandinav., 119: 36, 1960. OWEN, R. A. and WALLACE-JONES,D. R. A case of double gallbladder with unusual features. Brit. J. krg., 49: 577, 1962. HURWITZ, A. Double gallbladder. J. Maine M. A., 55 : 79,1964. DUNKERLY, D. R. Double gallbladder. Proc. Row Sot. Med.. 57: 331.1964.
19. 20. 21. 22.
MA&E, D. B. Diagnosis and treatment of duplication of the gallbladder. Postgrad. Med. J., 42: 213, 1966. RAYMOND,W. and THRIFT, C. B. Carcinoma of a duplicated gallbladder. Illinois M. J., 110: 23$1956. MUNSON. C. L. and TEIXIDO, R. Congenital abnormality of the gallbladder. De‘iaware M. J., 31: 193,1959. WISCHNEWSKY, A. W. Doppelgullenblase Aufgedeckt Operation der Wahrend (Ektomie der erkrankten Supplementaren Blase). Arch. klin. Chir., 135: 779, 1925. 465
Palmisano 23. 24. 26.
466
RECHT, W. Torsion of a double gallbladder. Btit. J. Surg., 39 : 342,1952. INGEGNO,A. P. and D’ALBORA, J. B. Double gallbladder. Am. J. Roentgenol., 61: 671, 1949. GUYER, P. B. and MCLOUGHLIN, M. Con-
26.
genital double gallbladder. Brit. J. Radial., 40: 214,1967. GROSS, R. E. Congenital anomalies of the gallbladder: a review of one hundred and forty-eight cases with report of a double gallbladder. Arch. Surg., 32: 131, 1936.
The American Journal of Surgery