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Reduplication of the gallbladder and cystic ducts
Reduplication of the Gallbladder and Cystic Ducts ROBERT N . WHITTENBERGER, M .D ., Grand Rapids, Michigan
From the Surgical Service of Butterwortb Hospital, Grand Rapids, Michigan .
recognition of reduplication anomalies T of the extrahepatic biliary system occurred at least as early as ancient Hebrew times when HE
certain variations were described in relation to the religious codes of the time . Yet as late as 1926, when Boyden' reviewed the reports of supernumerary abnormalities of the biliary tract, only twenty human cases had been collected . Since 1926 less than fifty additional cases have been reported . This is a report of another case of reduplication of the gallbladder and cystic ducts . That this condition is rarely encountered in the human is pointedly shown above . The paucity of attention focused on this type of developmental anomaly in standard texts on pathology and in the literature further attests to its infrequent occurrence . Indeed, only one text reference could be found .' In one collected series of over 19,000 cases, including cadaver dissections and cholecystograms, only five reduplications of some form were found (an incidence of 0 .02 per cent) . In other mammals the incidence ranges from 12 .5 per cent in domestic cats to o.5 per cent in pigs . With an increasing amount of surgery being carried out on the biliary system the importance of this type of pathologic condition, even though rarely encountered, should become more and more apparent, both in regard to the technical aspects of the surgery and the diagnosis of complaints referable to the biliary tract . Boyden' describes five congenitally distinct types of accessory vesicles, as follows : (i) Arising from a pancreatic anlage Pancreatic bladders : first found by DeGraaf in 1664 ; found in domestic cats only (2) Arising from a hepatobiliary anlage Cleft gallbladders : originate by initial 926
subdivision of the embryologic primary cystic diverticuIum ; chiefly characteristic in cats but present in some human cases Diverticular bladders : vesicles or lobes arising as buds from the neck of the embryonic gallbladder ; chiefly characteristic of ungulates Ductular bladders : supernumerary vesicles derived from hepatic, cystic or common bile ducts ; chiefly characteristic of humans but also present in cats ; this can be further subdivided into the Y-shaped type (the two gallbladders usually adherent and occupying the same fossa with the two cystic ducts uniting before entering the common duct), and the ductular type (the two gallbladders usually separate, sometimes occupying different lobes, and with the two cystic ducts entering the common duct separately) Trabecular bladders : vesicular outgrowths of the liver trabeculae bordering the fossa ; rare ; found only in cattle, sheep and possibly cats Our case is actually a combination of the Y and ductular types of ductular bladders in that the two gallbladders were adherent and occupied the same fossa with the two cystic ducts entering the common duct separately . CASE REPORT
E. N . (No. 34625), a forty-eight year old white woman, para iv, was first seen in a physician's office in January, 1 953, complaining primarily of suprapubic pain with menses and with early menopausal symptoms . During the interview it was determined that the patient avoided fatty foods, cabbage and apples . Occasionally after dietary indiscretion the patient experienced mild right upper quadrant distress
Reduplication of Gallbladder and Cystic Ducts and flatulence . The patient had had an anterior colporrhaphy and appendectomy twenty-one years before, and prior to that had had a tonsillectomy . Physical examination at this visit showed essentially : a mild essential hypertension (i 50%90), right upper quadrant, subcostal tenderness, and a lacerated, eroded and cystic cervix . A cholecystogram obtained on February 6, 1953, was reported as showing "multiple biliary calculi in an otherwise normally functioning gallbladder ." The patient was admitted to Butterworth Hospital on February 18th, with the history as recorded . Positive physical findings were the same . The serology was negative ; urinalysis was within normal limits ; red blood cell count was 4 .15 million with 82 per cent hemoglobin (11 .8 gm .) ; white blood cell count was 8,200 with 5 i per cent neutrophils and 49 per cent lymphocytes . Diagnoses of (i) essential hypertension, mild ; (2) chronic cystic cervicitis and (3) cholelithiasis were made . The patient was operated upon February i9, 1953 . At operation dilatation and curettage with wide conization of the cervix was first performed . Following this, through a right upper quadrant right rectus incision, exploration
FIG . i . Reduplication of gallbladder and cystic ducts .
showed a thin-walled gallbladder containing several small calculi . The spleen was two times the usual size and the right ovary contained a serous cyst the size of an English walnut . The remaining abdominal and pelvic viscera were normal . Retrograde cholecystectomy was then begun . The cystic artery had its origin from
FIG. 2 . Cholecystogram showing double gallbladder with gallstones in one
compartment . 927
Reduplication of Gallbladder and Cystic Ducts the right hepatic artery posterior to the common duct and passed superolaterally to the gallbladder fossa, coursing anterior to the cystic duct. When the cystic duct was doubly clamped and transected, approximately o .5 cm . from the common duct, it was seen to have two lumens . The gallbladder, after excision, was opened and found to be divided into two compartments of equal size . Each compartment had a narrow cystic duct which coursed to the common duct . The posteromedial (non-functioning) compartment contained dark, viscid bile, mucus and several small, rough-surfaced stones . The cystic duct leading from this compartment was completely occluded approximately i .o cm . from the common duct by a thin septum . The anterolateral (functioning) compartment contained normal bile and communicated with the common duct by a narrow cystic duct . (Fig . i .) The pathologic report on the gallbladder was as follows : Grossly, the gallbladder was 7 cm . in length and 4 cm . in diameter . It was completely divided into two portions of equal size . Each of these portions had a separate cystic duct . One of the cystic ducts was completely occluded by a thin septum . The mucosa of the gallbladder was smooth . Both portions of the gallbladder were opened ; received separately 3 stones ranging in size from peppercorns to cherry pits . Microscopically, sections of both portions of the gallbladder showed some increase in fibrous tissue in the mucosa and the remainder of the wall, but were not remarkable
otherwise . Sections of both cystic ducts showed the architecture of this organ . Diagnosis : cholelithiasis ; double gallbladder . The postoperative course was uneventful and the patient was discharged from the hospital on her sixth postoperative day . At the time of writing the patient remains free of her gastrointestinal symptoms . The cholecystograms, when viewed with the aid of retrospection, clearly define two compartments as seen in Figure 2 . The two gallbladders overlap one another to some extent . This could conceivably be confused with a phrygian cap but it should be remembered that the phrygian cap septum usually occurs more distally in the gallbladder and the cap is smaller in size than the globular shadow shown . SUMMARY
Reduplication anomalies of the extrahepatic biliary system occur rarely . One series of 19,ooo cases showed an incidence of 0 .02 per cent . Five types of such anomalies have been described . Actually, combinations of these types also occur . A case of reduplication of the gallbladder and cystic ducts is presented . REFERENCES
i.
A . Accessory gall-bladder ; embryological and comparative study of aberrant biliary vesicles occurring in man and domestic mammals . Am . J.
BOYDEN, F .
Anat ., 38 : 177, 1926 . 2 . ANDERSON, W . A . D ., editor . Pathology, p . 904 . Louis, 1948 . C . V . Mosby Co.
928
St .
From the Surgical Service of Butterwortb Hospital, Grand Rapids, Michigan .
recognition of reduplication anomalies T of the extrahepatic biliary system occurred at least as early as ancient Hebrew times when HE
certain variations were described in relation to the religious codes of the time . Yet as late as 1926, when Boyden' reviewed the reports of supernumerary abnormalities of the biliary tract, only twenty human cases had been collected . Since 1926 less than fifty additional cases have been reported . This is a report of another case of reduplication of the gallbladder and cystic ducts . That this condition is rarely encountered in the human is pointedly shown above . The paucity of attention focused on this type of developmental anomaly in standard texts on pathology and in the literature further attests to its infrequent occurrence . Indeed, only one text reference could be found .' In one collected series of over 19,000 cases, including cadaver dissections and cholecystograms, only five reduplications of some form were found (an incidence of 0 .02 per cent) . In other mammals the incidence ranges from 12 .5 per cent in domestic cats to o.5 per cent in pigs . With an increasing amount of surgery being carried out on the biliary system the importance of this type of pathologic condition, even though rarely encountered, should become more and more apparent, both in regard to the technical aspects of the surgery and the diagnosis of complaints referable to the biliary tract . Boyden' describes five congenitally distinct types of accessory vesicles, as follows : (i) Arising from a pancreatic anlage Pancreatic bladders : first found by DeGraaf in 1664 ; found in domestic cats only (2) Arising from a hepatobiliary anlage Cleft gallbladders : originate by initial 926
subdivision of the embryologic primary cystic diverticuIum ; chiefly characteristic in cats but present in some human cases Diverticular bladders : vesicles or lobes arising as buds from the neck of the embryonic gallbladder ; chiefly characteristic of ungulates Ductular bladders : supernumerary vesicles derived from hepatic, cystic or common bile ducts ; chiefly characteristic of humans but also present in cats ; this can be further subdivided into the Y-shaped type (the two gallbladders usually adherent and occupying the same fossa with the two cystic ducts uniting before entering the common duct), and the ductular type (the two gallbladders usually separate, sometimes occupying different lobes, and with the two cystic ducts entering the common duct separately) Trabecular bladders : vesicular outgrowths of the liver trabeculae bordering the fossa ; rare ; found only in cattle, sheep and possibly cats Our case is actually a combination of the Y and ductular types of ductular bladders in that the two gallbladders were adherent and occupied the same fossa with the two cystic ducts entering the common duct separately . CASE REPORT
E. N . (No. 34625), a forty-eight year old white woman, para iv, was first seen in a physician's office in January, 1 953, complaining primarily of suprapubic pain with menses and with early menopausal symptoms . During the interview it was determined that the patient avoided fatty foods, cabbage and apples . Occasionally after dietary indiscretion the patient experienced mild right upper quadrant distress
Reduplication of Gallbladder and Cystic Ducts and flatulence . The patient had had an anterior colporrhaphy and appendectomy twenty-one years before, and prior to that had had a tonsillectomy . Physical examination at this visit showed essentially : a mild essential hypertension (i 50%90), right upper quadrant, subcostal tenderness, and a lacerated, eroded and cystic cervix . A cholecystogram obtained on February 6, 1953, was reported as showing "multiple biliary calculi in an otherwise normally functioning gallbladder ." The patient was admitted to Butterworth Hospital on February 18th, with the history as recorded . Positive physical findings were the same . The serology was negative ; urinalysis was within normal limits ; red blood cell count was 4 .15 million with 82 per cent hemoglobin (11 .8 gm .) ; white blood cell count was 8,200 with 5 i per cent neutrophils and 49 per cent lymphocytes . Diagnoses of (i) essential hypertension, mild ; (2) chronic cystic cervicitis and (3) cholelithiasis were made . The patient was operated upon February i9, 1953 . At operation dilatation and curettage with wide conization of the cervix was first performed . Following this, through a right upper quadrant right rectus incision, exploration
FIG . i . Reduplication of gallbladder and cystic ducts .
showed a thin-walled gallbladder containing several small calculi . The spleen was two times the usual size and the right ovary contained a serous cyst the size of an English walnut . The remaining abdominal and pelvic viscera were normal . Retrograde cholecystectomy was then begun . The cystic artery had its origin from
FIG. 2 . Cholecystogram showing double gallbladder with gallstones in one
compartment . 927
Reduplication of Gallbladder and Cystic Ducts the right hepatic artery posterior to the common duct and passed superolaterally to the gallbladder fossa, coursing anterior to the cystic duct. When the cystic duct was doubly clamped and transected, approximately o .5 cm . from the common duct, it was seen to have two lumens . The gallbladder, after excision, was opened and found to be divided into two compartments of equal size . Each compartment had a narrow cystic duct which coursed to the common duct . The posteromedial (non-functioning) compartment contained dark, viscid bile, mucus and several small, rough-surfaced stones . The cystic duct leading from this compartment was completely occluded approximately i .o cm . from the common duct by a thin septum . The anterolateral (functioning) compartment contained normal bile and communicated with the common duct by a narrow cystic duct . (Fig . i .) The pathologic report on the gallbladder was as follows : Grossly, the gallbladder was 7 cm . in length and 4 cm . in diameter . It was completely divided into two portions of equal size . Each of these portions had a separate cystic duct . One of the cystic ducts was completely occluded by a thin septum . The mucosa of the gallbladder was smooth . Both portions of the gallbladder were opened ; received separately 3 stones ranging in size from peppercorns to cherry pits . Microscopically, sections of both portions of the gallbladder showed some increase in fibrous tissue in the mucosa and the remainder of the wall, but were not remarkable
otherwise . Sections of both cystic ducts showed the architecture of this organ . Diagnosis : cholelithiasis ; double gallbladder . The postoperative course was uneventful and the patient was discharged from the hospital on her sixth postoperative day . At the time of writing the patient remains free of her gastrointestinal symptoms . The cholecystograms, when viewed with the aid of retrospection, clearly define two compartments as seen in Figure 2 . The two gallbladders overlap one another to some extent . This could conceivably be confused with a phrygian cap but it should be remembered that the phrygian cap septum usually occurs more distally in the gallbladder and the cap is smaller in size than the globular shadow shown . SUMMARY
Reduplication anomalies of the extrahepatic biliary system occur rarely . One series of 19,ooo cases showed an incidence of 0 .02 per cent . Five types of such anomalies have been described . Actually, combinations of these types also occur . A case of reduplication of the gallbladder and cystic ducts is presented . REFERENCES
i.
A . Accessory gall-bladder ; embryological and comparative study of aberrant biliary vesicles occurring in man and domestic mammals . Am . J.
BOYDEN, F .
Anat ., 38 : 177, 1926 . 2 . ANDERSON, W . A . D ., editor . Pathology, p . 904 . Louis, 1948 . C . V . Mosby Co.
928
St .