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Journal of Clinical Oncology, Vol 22, No 2 (January 15), 2004: pp. 373-374
© 2004 American Society of Clinical Oncology.
DOI: 10.1200/JCO.2004.02.131

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DIAGNOSIS IN ONCOLOGY

Complications of Malignancy

CASE 2. Infectious Tissue Pneumatosis As a Result of Colon Cancer Perforation in a Survivor of Hodgkin's Disease

Roderich E. Schwarz, Roger K. Strair

Divisions of Surgical Oncology and Medical Oncology, the Cancer Institute of New Jersey, New Brunswick, NJ

A 59-year-old man presented with acute distress and exacerbating abdominal pain that had started 2 weeks earlier. He had a history of mediastinal Hodgkin's disease, diagnosed 5 years prior. Initial therapy with doxorubicin, bleomycin, vinblastine, and dacarbazine led to a complete response. A mediastinal recurrence was treated with radiation therapy. A subsequent intraabdominal relapse was treated with doxorubicin, vinblastine, mechlorethamine, etoposide, vincristine, bleomycin, and prednisone (Stanford V), followed by high-dose chemotherapy (carmustine, etoposide, cytarabine, and melphalan) with autologous stem cell rescue, and subsequent consolidative radiotherapy to para-aortic and pelvic lymph nodes and spleen. The patient remained disease-free for 2 years. Six weeks before presentation, new liver and lung nodules were seen on computed tomography; a biopsy revealed metastatic adenocarcinoma. Physical exam revealed tachypnoea, tachycardia, and hypotension. His right abdominal wall and flank showed a violaceous discoloration of the skin (Fig 1). He had a tense abdomen with rebound tenderness. The WBC count was 11.7 (x 109/L), with 81% segmented neutrophils, 8% band neutrophils, and 4% lymphocytes. Abdominal computed tomography demonstrated massive intrahepatic (Fig 2A) and gluteal soft tissue air (Fig 2B) in addition to a smaller amount of free intraperitoneal air (Fig 2A, arrow), and intravenous air in the right common iliac vein (Fig 2B, arrow). Despite the low probability to survive an infection as a result of gas-forming organisms, the patient desired aggressive care. He underwent a laparotomy, ileocolectomy, ileostomy, and transverse colon mucous fistula. Intraoperative findings were consistent with a cecal adenocarcinoma that had perforated into the retroperitoneal soft tissues and into the peritoneal cavity. Pathologic evaluation showed a 9 cm poorly differentiated pT3N2M1 (Stage 4) adenocarcinoma. Despite aggressive support, the patient died from progressive septic shock within 24 hours.



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Fig 1.

 


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Fig 2.

 
Increased risks to develop overwhelming infections or secondary cancers have been well documented for patients with Hodgkin's disease [1-4]. Overwhelming sepsis, however, remains uncommon, and is primarily encountered after previous splenectomy [1,5]; it has been observed after doxorubicin, bleomycin, vinblastine, and dacarbazine therapy in the setting of recurrent disease [6]. The infectious hazards after splenic irradiation are less clear, but have been postulated [7,8]. An increased incidence of secondary neoplasms, both hematologic and solid, has been observed after all forms of therapy for Hodgkin's disease [9]; combination therapy leads to greater risks than monotherapy [10]. Age greater than 40 at the time of the initial Hodgkin's diagnosis appears to be a risk factor [11,12]. Secondary solid tumors are predominantly cancers of lung, breast, or colon, with an average latency period of 8 years [13]. Our patient presented with metastatic colon cancer, and an infection with gas-forming organisms involving soft tissue at the perforated primary tumor and metastatic sites within the liver. There was no evidence for recurrence of lymphoma. Despite the unusual presentation, this case serves as an important reminder of the significance and possible complexity of neoplastic and infectious hazards that can be encountered in survivors of Hodgkin's disease.

Authors' Disclosures of Potential Conflicts of Interest

The authors indicated no potential conflicts of interest.

REFERENCES

1. Jockovich, M. Mendenhall NP, Sombeck MD, et al: Long-term complications of laparotomy in Hodgkin's disease. Ann Surg 219:615-621, 1994[Medline]

2. Swerdlow AJ, Douglas AJ, Vaughan Hudson G, et al: Risk of second primary cancer after Hodgkin's disease in patients in the British National Lymphoma Investigation: Relationships to host factors, histology and stage of Hodgkin's disease, and splenectomy. Br J Cancer 68:1006-1011, 1993[Medline]

3. Bhatia S, Robison LL, Oberlin O, et al: Breast cancer and other second neoplasms after childhood Hodgkin's disease. N Engl J Med 334:745-751, 1996[Abstract/Free Full Text]

4. Foss Abrahamsen A, Andersen A, Nome O, et al: Long-term risk of second malignancy after treatment of Hodgkin's disease: The influence of treatment, age and follow-up time. Ann Oncol 13:1786-1791, 2002[Abstract/Free Full Text]

5. Baccarani M, Fiacchini M, Galieni P, et al: Meningitis and septicaemia in adults splenectomized for Hodgkin's disease. Scand J Haematol 36:492-498, 1986[Medline]

6. Fryer CJ, Hutchinson RJ, Krailo M, et al: Efficacy and toxicity of 12 courses of ABVD chemotherapy followed by low-dose regional radiation in advanced Hodgkin's disease in children: A report from the Children's Cancer Study Group. J Clin Oncol 8:1971-1980, 1990[Abstract]

7. Selby C, Hart S, Ispahani P, et al: Bacteraemia in adults after splenectomy or splenic irradiation. Q J Med 63:523-530, 1987[Medline]

8. Coleman CN, McDougall IR, Dailey MO, et al: Functional hyposplenia after splenic irradiation for Hodgkin's disease. Ann Intern Med 96:44-47, 1982

9. Maurizi Enrici R, Anselmo AP, Osti MF, et al: Analysis of the risk of solid tumor following Hodgkin's disease. Haematologica 82:57-63, 1997[Abstract/Free Full Text]

10. Ng AK, Bernardo MV, Weller E, et al: Second malignancy after Hodgkin disease treated with radiation therapy with or without chemotherapy: Long-term risks and risk factors. Blood 100:1989-1996, 2002[Abstract/Free Full Text]

11. Andre M, Henry-Amar M, Blaise D, et al: Treatment-related deaths and second cancer risk after autologous stem-cell transplantion for Hodgkin's disease. Blood 92:1933-1940, 1998[Abstract/Free Full Text]

12. Dietrich PY, Henry-Amar M, Cosset JM, et al: Second primary cancers in patients continuously disease-free from Hodgkin's disease: A protective role for the spleen? Blood 84:1209-1215, 1994[Abstract/Free Full Text]

13. Varady E, Deak B, Molnar ZS, et al: Second malignancies after treatment for Hodgkin's disease. Leuk Lymphoma 42:1275-1281, 2001[CrossRef][Medline]





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