Metastatic signet ring cell adenocarcinoma of bone marrow with bilateral ovarian masses: a case report
© Jain et al; licensee BioMed Central Ltd. 2008
Received: 25 July 2008
Accepted: 19 November 2008
Published: 19 November 2008
We present a case of metastatic signet ring cell adenocarcinoma of bone marrow with radiologically proven bilateral ovarian masses in a 50 year old Asian Indian female. Even after thorough search no extraovarian primary site could be found. Based on overall clinicopathologic correlation, a diagnosis of metastatic signet ring cell adenocarcinoma of bone marrow with uncertain primary was established.
Previously reported patients of Krukenberg carcinoma with bone and marrow metastases
Site of metastasis
Type of Krukenberg tumor
Simecek A 
Lowman and Kushlan 
Zeigerman JH 
Osteoplastic metastases to spine, pelvis, ribs, clavicle, scapulae and upper humeri
Engeler et al 
Osteoplastic metastases and necrosis of the marrow.
Joshi VV 
Ribs, pelvis, spine and greater trochanter of left Femur along with marrow dissemination.
Metz et al 
1980 (Two cases)
1. 40/F 2. 29/F
1. Skull, spine, ribs and hip with marrow involvement 2. Skull and spine
Osteolytic lumbar vertebrae with bone marrow involvement
From review of clinical and autopsy material, the most common occurrence of marrow metastases in nonhematologic malignancies is in patients with carcinomas arising in the prostate, breast, lung, and neuroblastoma . Epithelial tumors of the ovary especially mucinous carcinomas rarely metastasize to the marrow . In the present case we found radiologically proven bilateral ovarian masses with signet ring cell adenocarcinoma in the bone marrow. Unfortunately, due to disseminated disease and early demise of the patient histopathology of ovarian mass could not be done. Autopsy was also not obtained. Even after extensive investigations including endoscopic examination of GIT, mammography and cervical biopsy, we were unable to find any extraovarian lesion. Therefore it was assumed that bone marrow metastasis would have been occurred either due to occult primary site in the GIT or to poorly differentiated mucinous carcinoma or primary Krukenberg tumor of the ovary. Poorly differentiated mucinous carcinomas show foci of typical signet ring cells, and the lesion may in parts resemble a Krukenberg tumor. However in the bone marrow we observed diffuse presence of signet ring cells. Diagnosis of primary krukenberg tumor requires survival for 5 years or longer or the presence of a detailed autopsy examination to rule out any other primary site. Both of these criteria were not met in our case, thus excluding primary Krukenberg tumor. Although histologic examination of ovarian masses could not be performed, radiologic features were suggestive of Krukenberg tumor. With the features described in this case, the ovaries are of metastatic origin until proven otherwise. It is not uncommon for a secondary Krukenberg tumor of the ovary to present first before the true extra-ovarian primary tumor is discovered. The majority of those are of gastric origin. Sometimes, this can require an exhaustive search. All tumors with microscopic features of the Krukenberg tumor in the ovary are metastatic, very rare examples may be primary. Joshi  accepted as primary 18 reported cases, including 11 in which autopsy examination revealed no evidence of an extraovarian source, and 7 in which the patient survived for 5 to 13 years after the removal of the ovarian tumor. To the best of our knowledge only few cases of Krukenberg carcinoma with bone and bone marrow metastases have been described in the literature. Zeigerman  described a case of primary Krukenberg tumor with osteoplastic metastases predominantly to axial skeleton. Joshi  reported a 33 year old Negro female with right ovarian tumor and metastatic lesions in the spine and greater trochanter of the femur. Even after thorough post mortem examination he could not find any extraovarian primary site; therefore considered it as a primary Krukenberg carcinoma. Subsequently Engeler et al  reviewed primary Krukenberg tumours in pregnancy and they found one patient with osteoplastic metastases and necrosis of the marrow. In 1980 Metz et al  described two patients of secondary Krukenberg carcinoma with bone marrow metastases. In both the cases primary tumor was present in gastric antrum. All the previously reported patients died due to disseminated disease, as was seen in the index patient. It seems likely that impairment of marrow function by tumor contributed significantly to the rapid deterioration of these patients. As seen in the present case and from review, the disease is a virulent one. The presence of ascitis is an ominous sign. The prognosis of primary Krukenberg carcinoma is less bleak than that of secondary Krukenberg tumor .
The present case adds to the literature a rare case of metastatic signet ring carcinoma of marrow and possibly secondary Krukenberg carcinoma of the ovary with an uncertain primary site.
A fully informed written consent was obtained from the patient family for the publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
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