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Metastatic adenocarcinoma arising from a small bowel duplication cyst
CASE REPORTS It is useful in the initial stage to send the purulent aspirate or drainage for both culture and cytology. In our second case, the cytologist was able to pick up the presence of atypical squames in a background of acute inflammation. The use of fine needle aspiration for lumps to diagnose squamous cell carcinoma has been suggested.5 Those breast abscesses that are to be treated conservatively in the post-menopausal female should undergo this in order to exclude the possibility of an underlying carcinoma. The presence of a haemopurulent aspirate should alert the primary surgeon as this was noticed in both our patients at the initial drainage. It is also necessary to send the wall of the abscess for histological analysis and either a trucut biopsy or specimen obtained at drainage is adequate. The surgical management of primary squamous cell carcinoma of the breast is similar to that for invasive ductal carcinoma. Both our patients underwent mastectomy because of the large initial size of the lesion. Wide resection can be carried out for smaller lesions with adequate margins. More importantly, axillary dissection should be carried out since squamous carcinomas have a propensity for lymph node metastasis.4 This is not surprising as other sites of squamous cell carcinoma have similar tumour biology of locally aggressive disease and lymph node involvement.
93
CONCLUSION Patients that present acutely with breast abscesses may harbour an underlying squamous cell carcinoma of the breast. Fine needle aspirates of the pus or biopsy of the abscess wall should be performed for all cases. This will ensure that the diagnosis is not missed and realized only at a more advanced stage. The subsequent treatment of such patients is similar to other invasive breast cancers and involves multidisciplinary cancer care.
REFERENCES 1. Wrightson WR, Edwards MJ, McMasters KM. Primary squamous cell carcinoma of the breast presenting as a breast abscess. Am Surg 1999; 65: 1153–5. 2. Habif DV, Perzin KH, Lipton R, Lattes R. Subareolar abscesses associated with squamous metaplasia of the lactiferous ducts. Am J Surg 1970; 119: 523–6. 3. Schaefer FV, Custer RP, Sorof S. Squamous metaplasia in human culture: induction by cyclic adenine nucleotide and prostaglandins and influence of menstrual cycle. Cancer Res 1983; 43: 279–86. 4. Stevenson JT, Graham DJ, Khiyami A, Mansour EG. Squamous cell carcinoma of the breast: A clinical approach. Ann Surg Oncol 1996; 3: 367–74. 5. Chen KTK. Fine needle aspiration cytology of squamous cell carcinoma of the breast. Acta Cytol 1990; 34: 664–8. Accepted for publication 9 April 2001
doi:10.1053/ejso.2001.1154, available online at http://www.idealibrary.com.on
Metastatic adenocarcinoma arising from a small bowel duplication cyst D. J. Fletcher, P. B. Goodfellow and D. Bardsley Department of Surgery, Chesterfield and North Derbyshire NHS Trust, Royal Hospital, Calow, Chesterfield, Derbyshire, UK
A case report of adenocarcinoma arising from a small bowel mesenteric cyst is presented. A discussion and review of the relevant literature then follows. 2002 Harcourt Publishers Ltd Key words: mesenteric cyst; adenocarcinoma; case history.
CASE REPORT A previously fit and well 28-year-old man was referred as an outpatient to the urologists with a history of cloudy urine and mild supra-pubic pain. He was not on any regular medications and there was no family history Correspondence to: D. J. Fletcher, Department of Surgery, Chesterfield and North Derbyshire NHS Trust, Royal Hospital, Calow, Chesterfield, Derbyshire, S44 5BL, UK.
of note. Urine culture, full blood count, urea and electrolytes and urodynamics were all normal at this time. However a bladder scan suggested a significant residual volume of urine. A subsequent cystoscopy was normal but an abdominal/pelvic ultra-sound scan showed a thin-walled 20-cm diameter cyst separate from the bladder and related to bowel. The working diagnosis was a mesenteric cyst and he was therefore referred to the general surgeons. When seen in the general surgery clinic a mass had 2002 Harcourt Publishers Ltd
94 become palpable in the right iliac fossa and he was listed for elective laparotomy and excision of mesenteric cyst. At laparotomy a 25 cm by 15 cm cyst containing 3 litres of fluid was found in the mesentery of the small bowel adjacent and attached to the proximal jejunum. The rest of the laparotomy appeared normal. It was not possible to separate the cyst from the adjacent jejunum and therefore approximately 30 cm of jejunum was resected along with the cyst and an end-to-end anastomosis performed. Post-operative recovery was unremarkable and the patient went home 8 days later. Histology showed a duplication cyst lined with columnar epithelium with areas of atypical papillary tufts which had excessive mitotic activity and nuclear pleomorphism. A specialist second opinion from a national centre confirmed a duplication cyst with dysplasia but also found an area of possible early invasion. Complete excision was thought to have been achieved and therefore no further action was felt to be necessary. The patient was re-admitted as an emergency 3 weeks after discharge with small bowel obstruction, thought to be secondary to adhesions. His symptoms failed to settle and he went back to theatre for an emergency laparotomy. At his second laparotomy a solid mass of presumed adhesions was found stuck to the back of the abdominal wall. A further small cyst at the base of the mesentery was also found. Attempts to separate the adhesions failed and therefore the mass and small bowel were resected and a jejuno-ileal end to side anastomosis was performed. Post-operative recovery was slow but he was discharged 18 days later. Histology of the adherent mass showed it to be a large deposit of metastatic adenocarcinoma. Histology of the second small cyst confirmed a further duplication cyst with areas of dysplastic columnar epithelium. However an area of infiltrating adenocarcinoma was also seen and this was thought to represent the primary tumour. Unfortunately the patient was re-admitted within 2 weeks of his discharge having generally deteriorated. Ultrasound scan and spiral CT at this time showed widespread liver metastases. He died 3 weeks following his re-admission.
DISCUSSION The Florentine anatomist Beneviene first described mesenteric cysts in 1507 after performing an autopsy on an 8-year-old boy. Telow performed the first successful surgical removal in 1889. The reported incidence of mesenteric cysts varies from 1/150 000 to 1/250 000 hospital admissions.1 Duplication cysts are one of the rare types of mesenteric cysts. Duplication cysts are congenital and there are various theories regarding their aetiology in the embryo.2 Theories include: a) a diverticulum of the developing intestine; b) that there is incomplete resolution of the solid phase of the gut; c) the persistence of the neuro-
CASE REPORTS enteric canal; and d) possible cordal twining during development. Duplication cysts can occur anywhere from the mouth to the anus but are most commonly found in the mesentery of the ileum. They are second most commonly found in the oesophagus. They can vary from long tubular duplications to small spherical cysts. Their walls must contain a double layer of smooth muscle and be lined with gastrointestinal epithelium (by Ladds’ definition (1937)) that may be different from that of the adjacent bowel (heterotopic). Muscle within the wall of the cyst and that of the adjacent bowel usually share a common blood supply which is relevant when considering surgical excision.2 Benign duplication cysts usually present in infancy or childhood with vague abdominal pain and distension being the commonest symptoms. They can also present as a painless abdominal mass, as an incidental finding or as an acute abdomen with obstruction, rupture or haemorrhage. Ultrasound and computed tomography (CT) are generally thought to be the most useful investigations. Radionucleotide scanning can be used to demonstrate the presence of heterotopic acid secreting gastric mucosa. Malignant change in duplication cysts appears to be exceedingly rare. A literature search found 20 cases (16 adenocarcinoma and four squamous cell carcinoma). Malignancy was not suspected in the great majority of these cases pre-operatively. However pre-operative carcinoembryonic antigen levels were found to be high in two cases.3 All cases of adenocarcinoma, in duplication cysts that have been reported, have been in adults aged 26–65 years. This is in stark contrast to the usual presentation of benign cysts, which tend to present in childhood. Although one of the least common sites of benign duplication cysts, the colon appears to be the commonest site for malignant duplication cysts.3 Prognosis is poor.4 Excision of a duplication cyst may be contemplated in childhood to relieve symptoms or complications and for the purposes of diagnosis. Resection of the adjacent bowel is usually required. Biopsy has been suggested for adult duplication cysts by some authors,2 but we would recommend that all adult duplication cysts are excised where possible in view of the possibility of malignant change.
REFERENCES 1. Kurtz R, Heimann T, Beck A, Holt J. Mesenteric and retroperitoneal cysts. Am Surg 1986; 203: 109–12. 2. Shamberger R. Duplications of the gastrointestinal tract and mesenteric cysts. In: Morris PJ, Wood WC (eds). Oxford Textbook of Surgery 2nd edn. 42.5: 2649–2654. 3. Inoue Y, Nakamura H. Adenocarcinoma arising in colonic duplication cysts with calcification: CT findings of two cases. Abdom Imag 1998; 23: 135–7. 4. Adair H, Trowell J. Squamous cell carcinoma arising in a duplication of the small bowel. J Pathol 1981; 133: 25–31. 5. Lee M, Jenson E, Kwak S, Larson R. Metastatic adenocarcinoma arising in a congenital foregut cyst of the esophagus. A case report with review of the literature. Am J Clin Oncol 1998; 21: 64–6.
93
CONCLUSION Patients that present acutely with breast abscesses may harbour an underlying squamous cell carcinoma of the breast. Fine needle aspirates of the pus or biopsy of the abscess wall should be performed for all cases. This will ensure that the diagnosis is not missed and realized only at a more advanced stage. The subsequent treatment of such patients is similar to other invasive breast cancers and involves multidisciplinary cancer care.
REFERENCES 1. Wrightson WR, Edwards MJ, McMasters KM. Primary squamous cell carcinoma of the breast presenting as a breast abscess. Am Surg 1999; 65: 1153–5. 2. Habif DV, Perzin KH, Lipton R, Lattes R. Subareolar abscesses associated with squamous metaplasia of the lactiferous ducts. Am J Surg 1970; 119: 523–6. 3. Schaefer FV, Custer RP, Sorof S. Squamous metaplasia in human culture: induction by cyclic adenine nucleotide and prostaglandins and influence of menstrual cycle. Cancer Res 1983; 43: 279–86. 4. Stevenson JT, Graham DJ, Khiyami A, Mansour EG. Squamous cell carcinoma of the breast: A clinical approach. Ann Surg Oncol 1996; 3: 367–74. 5. Chen KTK. Fine needle aspiration cytology of squamous cell carcinoma of the breast. Acta Cytol 1990; 34: 664–8. Accepted for publication 9 April 2001
doi:10.1053/ejso.2001.1154, available online at http://www.idealibrary.com.on
Metastatic adenocarcinoma arising from a small bowel duplication cyst D. J. Fletcher, P. B. Goodfellow and D. Bardsley Department of Surgery, Chesterfield and North Derbyshire NHS Trust, Royal Hospital, Calow, Chesterfield, Derbyshire, UK
A case report of adenocarcinoma arising from a small bowel mesenteric cyst is presented. A discussion and review of the relevant literature then follows. 2002 Harcourt Publishers Ltd Key words: mesenteric cyst; adenocarcinoma; case history.
CASE REPORT A previously fit and well 28-year-old man was referred as an outpatient to the urologists with a history of cloudy urine and mild supra-pubic pain. He was not on any regular medications and there was no family history Correspondence to: D. J. Fletcher, Department of Surgery, Chesterfield and North Derbyshire NHS Trust, Royal Hospital, Calow, Chesterfield, Derbyshire, S44 5BL, UK.
of note. Urine culture, full blood count, urea and electrolytes and urodynamics were all normal at this time. However a bladder scan suggested a significant residual volume of urine. A subsequent cystoscopy was normal but an abdominal/pelvic ultra-sound scan showed a thin-walled 20-cm diameter cyst separate from the bladder and related to bowel. The working diagnosis was a mesenteric cyst and he was therefore referred to the general surgeons. When seen in the general surgery clinic a mass had 2002 Harcourt Publishers Ltd
94 become palpable in the right iliac fossa and he was listed for elective laparotomy and excision of mesenteric cyst. At laparotomy a 25 cm by 15 cm cyst containing 3 litres of fluid was found in the mesentery of the small bowel adjacent and attached to the proximal jejunum. The rest of the laparotomy appeared normal. It was not possible to separate the cyst from the adjacent jejunum and therefore approximately 30 cm of jejunum was resected along with the cyst and an end-to-end anastomosis performed. Post-operative recovery was unremarkable and the patient went home 8 days later. Histology showed a duplication cyst lined with columnar epithelium with areas of atypical papillary tufts which had excessive mitotic activity and nuclear pleomorphism. A specialist second opinion from a national centre confirmed a duplication cyst with dysplasia but also found an area of possible early invasion. Complete excision was thought to have been achieved and therefore no further action was felt to be necessary. The patient was re-admitted as an emergency 3 weeks after discharge with small bowel obstruction, thought to be secondary to adhesions. His symptoms failed to settle and he went back to theatre for an emergency laparotomy. At his second laparotomy a solid mass of presumed adhesions was found stuck to the back of the abdominal wall. A further small cyst at the base of the mesentery was also found. Attempts to separate the adhesions failed and therefore the mass and small bowel were resected and a jejuno-ileal end to side anastomosis was performed. Post-operative recovery was slow but he was discharged 18 days later. Histology of the adherent mass showed it to be a large deposit of metastatic adenocarcinoma. Histology of the second small cyst confirmed a further duplication cyst with areas of dysplastic columnar epithelium. However an area of infiltrating adenocarcinoma was also seen and this was thought to represent the primary tumour. Unfortunately the patient was re-admitted within 2 weeks of his discharge having generally deteriorated. Ultrasound scan and spiral CT at this time showed widespread liver metastases. He died 3 weeks following his re-admission.
DISCUSSION The Florentine anatomist Beneviene first described mesenteric cysts in 1507 after performing an autopsy on an 8-year-old boy. Telow performed the first successful surgical removal in 1889. The reported incidence of mesenteric cysts varies from 1/150 000 to 1/250 000 hospital admissions.1 Duplication cysts are one of the rare types of mesenteric cysts. Duplication cysts are congenital and there are various theories regarding their aetiology in the embryo.2 Theories include: a) a diverticulum of the developing intestine; b) that there is incomplete resolution of the solid phase of the gut; c) the persistence of the neuro-
CASE REPORTS enteric canal; and d) possible cordal twining during development. Duplication cysts can occur anywhere from the mouth to the anus but are most commonly found in the mesentery of the ileum. They are second most commonly found in the oesophagus. They can vary from long tubular duplications to small spherical cysts. Their walls must contain a double layer of smooth muscle and be lined with gastrointestinal epithelium (by Ladds’ definition (1937)) that may be different from that of the adjacent bowel (heterotopic). Muscle within the wall of the cyst and that of the adjacent bowel usually share a common blood supply which is relevant when considering surgical excision.2 Benign duplication cysts usually present in infancy or childhood with vague abdominal pain and distension being the commonest symptoms. They can also present as a painless abdominal mass, as an incidental finding or as an acute abdomen with obstruction, rupture or haemorrhage. Ultrasound and computed tomography (CT) are generally thought to be the most useful investigations. Radionucleotide scanning can be used to demonstrate the presence of heterotopic acid secreting gastric mucosa. Malignant change in duplication cysts appears to be exceedingly rare. A literature search found 20 cases (16 adenocarcinoma and four squamous cell carcinoma). Malignancy was not suspected in the great majority of these cases pre-operatively. However pre-operative carcinoembryonic antigen levels were found to be high in two cases.3 All cases of adenocarcinoma, in duplication cysts that have been reported, have been in adults aged 26–65 years. This is in stark contrast to the usual presentation of benign cysts, which tend to present in childhood. Although one of the least common sites of benign duplication cysts, the colon appears to be the commonest site for malignant duplication cysts.3 Prognosis is poor.4 Excision of a duplication cyst may be contemplated in childhood to relieve symptoms or complications and for the purposes of diagnosis. Resection of the adjacent bowel is usually required. Biopsy has been suggested for adult duplication cysts by some authors,2 but we would recommend that all adult duplication cysts are excised where possible in view of the possibility of malignant change.
REFERENCES 1. Kurtz R, Heimann T, Beck A, Holt J. Mesenteric and retroperitoneal cysts. Am Surg 1986; 203: 109–12. 2. Shamberger R. Duplications of the gastrointestinal tract and mesenteric cysts. In: Morris PJ, Wood WC (eds). Oxford Textbook of Surgery 2nd edn. 42.5: 2649–2654. 3. Inoue Y, Nakamura H. Adenocarcinoma arising in colonic duplication cysts with calcification: CT findings of two cases. Abdom Imag 1998; 23: 135–7. 4. Adair H, Trowell J. Squamous cell carcinoma arising in a duplication of the small bowel. J Pathol 1981; 133: 25–31. 5. Lee M, Jenson E, Kwak S, Larson R. Metastatic adenocarcinoma arising in a congenital foregut cyst of the esophagus. A case report with review of the literature. Am J Clin Oncol 1998; 21: 64–6.