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Metastatic adenocarcinoma within a functioning adrenal adenoma: a case report.
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MedLine Citation:
PMID:  19830028     Owner:  NLM     Status:  PubMed-not-MEDLINE    
Abstract/OtherAbstract:
We present the case of a 54-year-old woman who underwent right adrenalectomy for palliation of Cushing's symptoms. She had recently been diagnosed with lung adenocarcinoma. Pathologic findings revealed a 5 cm adrenal adenoma with a metastatic adenocarcinoma deposit. The occurrence of tumor-to-tumor metastasis is rare, and the finding of a metastasis within a functional adrenal adenoma exceptionally so. Previously reported incidences of this finding in patients with lung cancer range from 0.14% to 0.63%. We review the literature regarding this unusual finding.
Authors:
Jeremiah T Martin; Fuad Alkhoury; Scott Helton; Paul Fiedler; Olga Sakharova; Steven Yood
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Publication Detail:
Type:  Journal Article     Date:  2009-07-02
Journal Detail:
Title:  Cases journal     Volume:  2     ISSN:  1757-1626     ISO Abbreviation:  Cases J     Publication Date:  2009  
Date Detail:
Created Date:  2009-10-15     Completed Date:  2011-01-07     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  101474272     Medline TA:  Cases J     Country:  England    
Other Details:
Languages:  eng     Pagination:  7965     Citation Subset:  -    
Affiliation:
Department of Surgery, Hospital of St. Raphael 1450 Chapel Street, New Haven, CT 06511 USA. jerrymartin@eircom.net
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Journal Information
Journal ID (nlm-ta): Cases J
ISSN: 1757-1626
Publisher: Cases Network Ltd
Article Information
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© 2009 Martin et al.; licensee Cases Network Ltd.
open-access:
Received Day: 26 Month: 5 Year: 2009
Accepted Day: 16 Month: 6 Year: 2009
Electronic publication date: Day: 02 Month: 7 Year: 2009
collection publication date: Year: 2009
Volume: 2E-location ID: 7965
PubMed Id: 19830028
DOI: 10.4076/1757-1626-2-7965
Publisher Id: 7965

Metastatic adenocarcinoma within a functioning adrenal adenoma: a case report
Jeremiah T Martinaff1 Email: jerrymartin@eircom.net
Fuad Alkhouryaff1 Email: falkhoury@gmail.com
Scott Heltonaff1 Email: shelton@srhs.org
Paul Fiedleraff1 Email: pfiedler@srhs.org
Olga Sakharovaaff1 Email: osakharova@srhs.org
Steven Yoodaff1 Email: syood@srhs.org
Department of Surgery, Hospital of St. Raphael1450 Chapel Street, New Haven, CT 06511USA

Introduction

Tumor-to-tumor metastasis is an infrequent occurrence. Here we report on the finding of metastatic lung adenocarcinoma within a functioning adrenal adenoma which was excised for palliation of Cushing’s symptoms in a 54-year-old female.


Case presentation

A 54-year-old Caucasian American female who previously had a hiatal hernia repair was referred for abdominal CT scan after operative findings of hepatomegaly were noted. On CT (Figure 1) and subsequent MRI a 4 cm right adrenal adenoma with benign radiologic characteristics was noted. Over the preceeding two years the patient had symptoms consistent with Cushing’s syndrome with complaints of weight gain, lower extremity swelling and muscle weakness. Workup revealed an elevated free urinary cortisol (195 µg/day), suppressed ACTH (<5 pg/mL) consistent with adrenal Cushing’s syndrome. She was scheduled for laparoscopic adrenalectomy, however a liver lesion was found at laparoscopy which, on biopsy, was positive for metastatic adenocarcinoma of pulmonary origin. The procedure was terminated at this point for further patient counseling. Upon the patient’s insistence, she was again scheduled for adrenalectomy to control her Cushing’s symptoms prior to undergoing therapy for her lung cancer.

At laparoscopy, a 5 × 4 × 4 cm, 45 g right adrenal mass was identified and removed without complication (Figure 1). The patient recovered well and was commenced on supplementary corticosteroids. Final pathology revealed an adrenal cortical adenoma with a focus of adenocarcinoma, positive for cytokeratin 7 and TTF-1, an immunophenotype consistent with lung primary (Figures 2-6).


Discussion

The occurrence of adrenal metastasis in the setting of non-small cell lung cancer (NSCLC) is not uncommon. In patients with NSCLC, the incidence has been reported from 25-40% during the course of the disease [1,2]. Adrenal metastases likely develop via lymphatic spread in early disease and via hematogenous spread in more advanced disease. This is evidenced by a greater propensity towards ipsilateral metastasis early in the disease course, with contralateral or bilateral metastases more likely to occur with advanced disease [2].

Despite the relative frequency of adrenal metastasis, this finding in the setting of an adrenal adenoma is rare. This phenomenon has been studied by Moriya et. al who noted a 0.63% incidence of metastasis to an adrenal adenoma in a review of lung cancer autopsy cases [3]. This compares with an incidence of 0.14% found by Onuigbo et al. in a review of 7232 lung cancers [4]. The statistical analysis of Moriya’s group indicated that there was a propensity for lung cancer to metastasize to an adrenal adenoma if present.

Tumor-to-tumor metastasis is an uncommon occurrence, and has only been documented in sporadic case reports and series since 1902 [5]. Fewer than 100 cases have been reported in the literature. Our institution previously reported a case of a colonic adenocarcinoma metastasizing to a thyroid adenoma [6]. The phenomenon generally involves metastasis from a donor (malignant) tumor to either a benign or malignant recipient tumor. Lung cancer is the most common primary tumor involved in up to 50% of the reported cases [7]. Other donor tumors which have been reported include breast, gastrointestinal, prostate and thyroid malignancies. The most frequent benign recipient is meningioma [8] with renal cell carcinoma being the most common malignant recipient [9].

Generally, the presence of an adrenal metastasis in the setting of lung cancer classifies the patient as having Stage IV disease and therefore benefiting most from chemotherapy. There is some evidence that patients with otherwise surgically resectable lung cancer and documented isolated adrenal metastasis may benefit from simultaneous resection [10]. This diagnosis can be difficult to determine preoperatively as it is far more likely for a screening CT scan to reveal an incidental adenoma. That said, thorough inspection of any adenoma with chemical shift imaging on MRI can with some confidence determine whether or not a mass is completely benign [11]. Given the high incidence of adrenal metastasis over the course of NSCLC, regular screening should include careful follow up of any adrenal masses. Metachronous adrenal masses should be resected when there is suspicion of adrenal metastasis in the setting of adrenal enlargement or cytologic confirmation [12].

The authors are not aware of other cases of NSCLC metastasis to a functioning adrenal adenoma. Tumor-to-tumor metastasis has been reported in the setting of a non-functioning adenoma [11]. The primary indication for adrenalectomy in our patient was to palliate her from Cushing’s symptoms. Following resection of her tumor she underwent adjuvant therapy for her lung cancer.

Early identification of adrenal metastases is essential as adrenalectomy may improve survival in selected patients. To this end, physicians caring for patients with lung cancer must maintain a high index of suspicion to detect these lesions.


Notes
Consent

Written informed consent was obtained from the patient for 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.

Competing interests

The author(s) declare that they have no competing interests.

Authors’ contributions

JM, FA, SH and SY were major contributors in writing the manuscript. PF performed histological examination, conducted literature review, and was a significant contributor to the manuscript. JM, SY and SH were the surgical team involved in this case. OS conducted the initial endocrinology workup and was involved in manuscript preparation. All authors read and approved the final manuscript.


References
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Karolyi P. Do adrenal metastases from lung cancer develop by lymphogenous or hematogenous route?J Surg OncolYear: 199043154156231410210.1002/jso.2930430306
Moriya T,Manabe T,Yamashita K,Arita S. Lung cancer metastasis to adrenocortical adenomas. A chance occurrence or a predilected phenomenon?Arch Pathol Lab MedYear: 19881122862892830864
Onuigbo WI. Lung Cancer Metastasis to Adrenal Cortical AdenomasJ Pathol BacteriolYear: 1963865415431406896610.1002/path.1700860232
Ricketts R,Tamboli P,Czerniak B,Guo CC. Tumor-to-tumor metastasis: report of 2 cases of metastatic carcinoma to angiomyolipoma of the kidneyArch Pathol Lab MedYear: 20081321016102018517262
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Petraki C,Vaslamatzis M,Argyrakos T,Petraki K,Strataki M,Alexopoulos C,Sotsiou F. Tumor to tumor metastasis: report of two cases and review of the literatureInt J Surg PatholYear: 2003111271351275463510.1177/106689690301100214
Honma K,Hara K,Sawai T. Tumour-to-tumour metastasis. A report of two unusual autopsy casesVirchows Arch A Pathol Anat HistopatholYear: 1989416153157251274310.1007/BF01606320
Mohammad Hossein Rahimi-Rad BI,Mohammad Khaled Rezaei. Tumor-to-Tumor Metastases from Lung Carcinoma to Soft Tissue Fibrous HistiocytomaTanaffosYear: 200877375
Beitler AL,Urschel JD,Velagapudi SR,Takita H. Surgical management of adrenal metastases from lung cancerJ Surg OncolYear: 19986954579762893
Shifrin RY,Bechtold RE,Scharling ES. Metastatic adenocarcinoma within an adrenal adenoma: detection with chemical shift imagingAJR Am J RoentgenolYear: 19961678918928819376
Lucchi M,Dini P,Ambrogi MC,Berti P,Materazzi G,Miccoli P,Mussi A. Metachronous adrenal masses in resected non-small cell lung cancer patients: therapeutic implications of laparoscopic adrenalectomyEur J Cardiothorac SurgYear: 2005277537561584830910.1016/j.ejcts.2005.01.047

Abbreviations
ACTH Adrenocorticotrophic hormone
NSCLC Non small-cell lung cancer
TTF-1 Transcription termination factor 1

Figures

[Figure ID: fig-001]
Figure 1. 

Abdominal CT scan, arrow depicts right adrenal mass.



[Figure ID: fig-002]
Figure 2. 

Gross Photo shows yellow adrenal cortical adenoma with small, gray focus slightly left of center representing metastatic adenocarcinoma.



[Figure ID: fig-003]
Figure 3. 

Photomicrograph shows metastatic adenocarcinoma on the left and adrenal cortical adenoma on the right (H&E stain - Low Power).



[Figure ID: fig-004]
Figure 4. 

Photomicrograph shows malignant glands (adenocarcinoma) intermixed with foamy adrenal cortical adenoma cells (H&E stain - High Power).



[Figure ID: fig-005]
Figure 5. 

TTF-1 immunostain demonstrates nuclear positivity of adenocarcinoma, consitent with lung origin.



[Figure ID: fig-006]
Figure 6. 

Cytokeratin 7 immunostain demonstrates cytoplasmic positivity of adenocarcinoma, consistent with lung origin.



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