Correspondence

Cullen's Sign in Metastatic Thyroid Cancer

N Engl J Med 1999; 340:1767-1768June 3, 1999

Article

To the Editor:

Marinella (Jan. 14 issue)1 described a patient with “widely metastatic thyroid cancer” (the sites of these metastases and means of documenting them were not mentioned) in whom abdominal ultrasonography and abdominal computed tomography were said to reveal “multiple hepatic lesions, consistent with the occurrence of metastases” and “multiple, large metastatic foci within the liver,” respectively, and who died approximately two weeks after admission. The statement is made that “the liver parenchyma was extensively replaced by metastatic tumor,” but there is no indication as to whether this conclusion was based on the above imaging studies, whether it was confirmed microscopically through a liver biopsy or an autopsy, or even whether an autopsy was performed.

According to the author, this case may be the first recorded case of Cullen's sign due to metastatic thyroid cancer. Therefore, shouldn't microscopical confirmation of the presence, location, and thyroidal nature of the metastasis have been an absolute requirement for publication of this report, especially because of the extreme rarity of liver metastases by papillary carcinoma of the thyroid and the fact that primary hepatocellular carcinoma has been reported in association with Cullen's sign, as is acknowledged by the author? I should think so, especially since confirmation would have been easily achieved through a very limited autopsy (a “posthumous analysis,” so to speak), which would have allowed the sampling of a liver nodule through a minute laparotomy.2

Juan Rosai, M.D.
Memorial Sloan-Kettering Cancer Center, New York, NY 10021

2 References

1. 1

   Marinella MA. Cullen's sign associated with metastatic thyroid cancer. N Engl J Med 1999;340:149-150
   Full Text | Web of Science | Medline

2. 2

   Rosai J. The posthumous analysis (PHA): an alternative to the conventional autopsy. Am J Clin Pathol 1996;106:Suppl 1:S15-S17
   Web of Science | Medline

Author/Editor Response

Dr. Marinella replies:

To the Editor: Dr. Rosai is skeptical that the presence of Cullen's sign in this patient was related to metastatic thyroid cancer. As noted in the case report, there was no evidence of more common causes of Cullen's sign, and the novelty of the case is the unusual association of periumbilical ecchymosis with liver metastases. The patient initially presented after sustaining a pathologic femur fracture. A biopsy specimen from the area of the fracture was histologically consistent with the presence of papillary carcinoma of the thyroid. A whole-body scan with iodine-131 obtained at about that time revealed markedly abnormal uptake in several bones and the lungs, a finding consistent with widely metastatic disease. The patient did not seek follow-up care until several weeks later, when the hepatic metastases were noted on abdominal imaging.

Even though this patient had a secure diagnosis of metastatic thyroid cancer at the latest hospitalization, Dr. Rosai contends that histologic diagnosis in a patient with hepatic metastases is necessary to rule out other cancers, such as hepatoma. Although it is possible that these lesions may have been due to another type of cancer, it is very unlikely. Hepatic metastases, although uncommon with papillary carcinoma of the thyroid, do occur and are typically associated with a poor outcome,1 as was the case with my patient. A biopsy of the hepatic lesions was not performed, per the request of the family and because it was thought that the lesions were probably due to metastases. A request for a postmortem examination was declined.

Even though some patients may have competing disease processes that account for a particular clinical finding, it is useful to think of Occam's razor translated into modern medical language: “Don't explain the clinical picture with two different diagnoses where one will do.”2

Mark A. Marinella, M.D.
Wright State University School of Medicine, Dayton, OH 45429

2 References

1. 1

   Shah DH, Samuel AM. Metastasis to the liver in well-differentiated carcinoma of the thyroid. Thyroid 1996;6:607-611
   CrossRef | Web of Science | Medline

2. 2

   Sapira JD. On violating Occam's razor. South Med J 1991;84:766-766
   CrossRef | Web of Science | Medline

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