Ann Thorac Surg 2009;87:957–9
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Fig 2. (A) Pathologic findings with hematoxylin and eosin staining showed well-demarcated, multi-cystic tumors in the low power field (original magnification, ⫻4). (B) Cells with substantially round nuclei and columnar cytoplasms growing in a tubular manner in the high-power field (original magnification, ⫻200).
References 1. Yousem SA, Hochholzer L. Alvelar adenoma. Hum Pathol 1986;17:1066 –71. 2. Semeraro D, Gibbs AR. Pulmonary adenoma: a variant of sclerosing haemangioma of lung? J Clin Pathol 1989;42:1222–7. 3. Bruke LM, Rush WI, Khoor A, et al. Alveolar adnoma: a histochemical, immunohistochemical, and ultrastructural analysis of 17 cases. Hum Pathol 1999;30:158 – 67. 4. Cakan A, Samancilar O, Nart D, Cagirici U. Alveolar adenoma:an usual lung tumor. Interact Cardiovasc Thorac Surg 2003;2:345–7. 5. Hartman MS, Epstein DM, Geyer SJ, Keenan RJ. Alveolar adenoma. Ann Thorac Surg 2004;78:1842–3. 6. Sak SD, Koseoglu RD, Demirag F, Akbulut H, Gungor A. Alveolar adenoma of the lung. Immunohistochemical and flow cytometric characteristics of two new case and review of the literature. APMIS 2007;115:1443–9. © 2009 by The Society of Thoracic Surgeons Published by Elsevier Inc
Testicular Metastasis From Adenocarcinoma of the Esophagus Sonja Gillen, MD, Marcus Feith, MD, Ralf Gertler, MD, Rupert Langer, MD, Jörg Massmann, MD, Mario Sarbia, MD, and Helmut Friess, MD Departments of Surgery and Pathology, Technische Universität München, Klinikum Rechts der Isar and Gemeinschaftspraxis Pathologie/Lachnerstrasse, Munich, Germany
We report the case of a 61-year-old patient, operated on for adenocarcinoma of the esophagus in 2002, who presented in 2007 with a hydrocele and palpable mass of the right testis. Operative exploration and orchiectomy were performed. Histopathology revealed a testicular and epididymidal metastasis from the esophageal adenocarcinoma. Only a few testicular metastases have been reported from gastrointestinal cancers. No case of testicular metastasis from esophageal cancer, including Barrett’s carcinoma has been reported. In most cases, the testicular tumor was accompanied by a hydrocele. Therefore, cancerous and metastatic lesions should be considered in the management of hydrocele and testicular masses. (Ann Thorac Surg 2009;87:957–9) © 2009 by The Society of Thoracic Surgeons
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sophageal carcinoma is the sixth leading cause of cancer death worldwide. The incidence of esophageal adenocarcinoma has increased more rapidly than any other cancer type [1]. Overall the prognosis of Barrett=s carcinoma is poor with a 5-year overall survival rate of approximately 14% [1]. Usually gastrointestinal adenocarcinomas metastasize predominately in local and distant lymph nodes [2], and later hematogenic to the liver, peritoneum, lung, and bone. Genital metastasis like Krukenberg tumor has not been published from esophageal cancer yet. We report a 61-year-old man who was diagnosed with adenocarcinoma of the distal esophagus (Barrett’s carci-
Accepted for publication July 25, 2008. Address correspondence to Dr Feith, Department of Surgery, Technische Universität München, Klinikum rechts der Isar, Ismaninger Str. 22, Munich, 81675, Germany; e-mail:
[email protected].
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of nuclear division or cell necrosis [1– 4, 6]. Differential diagnoses such as sclerosing hemangioma and papillary adenoma for cases of benign tumor or atypical adenomatous hyperplasia and bronchio-alveolar carcinoma for cases of malignancy are often problematic. In particular, sclerosing hemangioma is characterized by a polymorphic histologic appearance with a mixture of solid, vascular, and sclerotic and papillary patterns [6], which is different from the progression of AA that occurs along the alveolar structure. In addition, the expression of thyroid transcriptase factor-1, which was positive in our case, has also been reported to be important for differentiation purposes [6]. A preoperative diagnosis and intraoperative pathologic diagnosis of AA are often difficult [5], and a diagnosis can often only be confirmed after a resection, but fortunately, there have been no cases of recurrence after a resection [1, 3, 4, 6]. Although the progression of AA is very similar to those of atypical adenomatous hyperplasia or bronchio-alveoral carcinoma, the cell images are clearly different, because AA showed a diploid cell pattern based on the findings of a flow cytometry analysis and the mindbomb homolog-1 (MIB-1) positive rate is less than 1% [6]. The AA has thus far been considered to be a rare disease, but it is possible that the number of detected cases will gradually increase as progress is made in diagnostic imaging, and we believe there is a need to be aware of its characteristics as a benign tumor.
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Fig 1. Primary adenocarcinoma of the esophagus composed of variously sized tubular glands (⫻200).
FEATURE ARTICLES
noma) in 2002. After induction chemotherapy, total esophagectomy and gastrectomy with systematic lymphadenectomy was performed in August 2002. Histopathology revealed a poorly differentiated adenocarcinoma of the esophagus with local lymph node metastasis in a long-segment Barrett’s esophagus. The International Union Against Cancer (UICC) classification was determined as ypT3, pN1 (2 of 10), G3, R0. After reconstruction of the gastrointestinal passage in 2003, one follow-up was performed without signs of tumor recurrence. In 2004, a palpable mass in the right paramedian abdominal wall was revealed, which was resected. Histologic assessment approved a cutaneous metastasis (G3, R0) of Barrett’s adenocarcinoma. As there was no evidence of further tumor manifestations, no additional oncological therapy was applied and close follow-up was continued until 2007, without signs of recurrent disease. In 2006, the patient had undergone a right inguinal hernia repair after a tumor free follow-up, including a urological assessment. A year later, the patient presented with a swelling of the right scrotum again. A symptomatic hydrocele and a chronic epididymitis with palpable mass in the right testis was found. The ultrasound showed a homogenic echostructure. Operative exploration was performed. The intraoperative situs showed a typical hydrocele, hiding a suspicious mass originating from the epididymic region. Intraoperative histopathologic examination showed neoplastic tissue. Therefore, a radical orchiectomy was performed. Macroscopic pathologic examination of the operation specimen revealed a 2.5 ⫻ 2.2 cm measured mass involving the epididymis, the spermatic cord, and the adjacent testicular tissue. Histologically, the tumor was classified as a poorly differentiated adenocarcinoma. A comparative histologic review of the tumor specimen of the esophagus, the subcutaneous tissue, and the right testis was performed. This analysis showed a highly congruent histologic picture in all tumor manifestations (Figs 1 and 2) being composed of variously sized tubular glands embedded in a densely sclerotic stroma. The light
Ann Thorac Surg 2009;87:957–9
Fig 2. Similar histologic appearance of tumor glands were present in the testicular metastasis. In addition, the atrophic seminiferous tubules can be depicted (⫻200).
microscopic view revealed concordance of the three tumor manifestations. Moreover, all tumor manifestations showed an identical immunophenotype with a high expression of CDX2 (Fig 3) and cytokeratin 7. The tumors did not show expression of cytokeratin 20. In addition, the testicular tumor showed no expression of CD30 and prostate specific antigen. Thus, an embryonal carcinoma and a metastasis of prostate cancer could be excluded. We herein report the first case of an epididymal and testicular metastasis from Barrett’s carcinoma. Additional staging examinations showed no evidence of further tumor lesions. Because the testicular and paratesticular metastasis was completely resected (R0), no further oncological treatment was performed, and close follow-up was recommended.
Comment We believe that the reported case is the first description of testicular and epididymal metastasis from esophageal
Fig 3. Strong nuclear expression (brown reaction product) of the intestinal transcription factor CDX2 was present in the tumor cells of the testicular metastasis (⫻200).
cancer. Approximately 200 cases of testicular metastases have been published so far [3, 4], most commonly arising from the prostate, lung, or colon, and rather seldom from the stomach, kidney, and malignant melanoma [4]. Secondary spread of malignant tumors to the testis is mainly associated with diffuse systemic disease and poor prognosis [5]. In the current case, histologic examination of the testicular tumor did not resemble the typical gland forming tumor of the testis, such as embryonal carcinoma and yolk sac tumor. No evidence for a teratoma (with somatic type of adenocarcinoma) was found. Primary adenocarcinoma of the epididymis was also considered, and this entity is probably even rarer than testicular metastasis and the typical centration was not evident in our case. The strong expression of CDX2 in all samples makes a metastatic spread of Barretts’s carcinoma highly likely. The CDX2 is a cloned homeobox gene that encodes an intestine-specific transcription factor expressed in the epithelial cells throughout the gastrointestinal tract [6]. Although CDX2 expression has been observed in extraintestinal adenocarcinomas (eg, in the ovaries), it is considered as specific and highly sensitive for intestinal differentiation and is retained in the majority of adenocarcinomas of the gastrointestinal tract. As metastases to the testis are rare, the metastatic route is not well defined, and several hypotheses have been postulated. Although some authors discuss hematogenic [5] or lymphogenous spread [7], others favor the concept of tumor cell spread either “per continuitatem” (eg, peritoneal carcinomatosis) or intraperitoneal seeding (eg, Krukenberg tumor) [8]. Intraperitoneal tumor cells might also pass over retrograde to the testes through a patent processus vaginalis [4]. Another way of tumor spread might be the direct extension though the retroperitoneum. In our case, a hematogenic metastasis is most likely, as there is no evidence of peritoneal carcinomatosis or direct tumor extension from the abdomen. Gastrointestinal adenocarcinomas usually develop lymphatic spread to local or distant lymph nodes [2] later on through hematogenic metastasization mainly to the liver and lung. However, we believe that testicular and epididymal metastases have not yet been reported for Barrett’s carcinoma. This presented case and systematic review of literature shows that metastatic spread to the testes and epididymis is rare, but it should be included in the differential diagnosis in patients with a history of gastrointestinal cancer presenting with a hydrocele or testicular mass. In this constellation, histopathologic clarification by biopsy or operative exploration should be considered.
References 1. Tew WP, Kelsen DP, Ilson DH. Targeted therapies for esophageal cancer. Oncologist 2005;10:590 – 601. 2. Feith M, Stein HJ, Siewert JR. Pattern of lymphatic spread of Barrett’s cancer. World J Surg 2003;27:1052–7. 3. Dutt N, Bates AW, Baithun SI. Secondary neoplasms of the male genital tract with different patterns of involvement in adults and children. Histopathology 2000;37:323–31. © 2009 by The Society of Thoracic Surgeons Published by Elsevier Inc
CASE REPORT KAMIYOSHIHARA ET AL CHILAIDITI’S SIGN MIMICKING DIAPHRAGMATIC HERNIA
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4. Patel SR, Richardson RL, Kvols L. Metastatic cancer to the testes: a report of 20 cases and review of the literature. J Urol 1989;142:1003–5. 5. Pienkos EJ, Jablokow VR. Secondary testicular tumors. Cancer 1972;30:481–5. 6. Werling RW, Yaziji H, Bacchi CE, Gown AM. CDX2, a highly sensitive and specific marker of adenocarcinomas of intestinal origin: an immunohistochemical survey of 476 primary and metastatic carcinomas. Am J Surg Pathol 2003;27:303–10. 7. Eadie DG. Presentation of carcinoma of the stomach as a left testicular tumor. Br J Surg 1962;50:156 – 8. 8. Zuk RJ, Trotter SE, Baithun SI. “Krukenberg” tumour of the testis. Histopathology 1989;14:214 – 6.
Chilaiditi’s Sign Mimicking a Traumatic Diaphragmatic Hernia Mitsuhiro Kamiyoshihara, MD, PhD, Takashi Ibe, MD, and Izumi Takeyoshi, MD, PhD Department of General Thoracic Surgery, Maebashi Red Cross Hospital, and Division of Thoracic and Visceral Organ Surgery, Gunma University Graduate School of Medicine, Maebashi, Gunma, Japan
A 75-year-old man had bruising develop after a traffic accident. A chest roentgenogram revealed elevation of the right hemidiaphragm. A subsequent computed tomographic scan showed interposition of the colon between the liver and diaphragm in front of the liver. Initially, we suspected a traumatic diaphragmatic hernia. Urgent exploratory video-assisted thoracoscopic surgery showed no injury of the right diaphragm. With the benefit of hindsight, there was no rupture of the diaphragm on the image. Hepato-diaphragmatic interposition of the colon is a very rare anomaly. This case was “Chilaiditi’s sign” mimicking a traumatic diaphragmatic hernia. (Ann Thorac Surg 2009;87:959 – 61) © 2009 by The Society of Thoracic Surgeons
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hysicians engaged in critical care must address a wide variety of different types of trauma and disease and will occasionally encounter unanticipated cases. Chilaiditi’s sign is an anomaly in which the bowel is interposed between the liver and right hemidiaphragm. This anomaly, which was first described by Chilaiditi [1], occurs in 0.025% to 0.28% of the general population [2], and it is rarely presented in case studies or imaging reports, which are usually published in journals on gastroenterological medicine [2–5]. However, thoracic surgeons should be aware of Chilaiditi’s sign, because it may cause overestimation of the degree of trauma. Chilaiditi’s sign must be distinguished from Morgagni’s hernia, subdiaphragmatic abscess, and pneumoperitoneum. In this case, Chilaiditi’s syndrome mimicked a traumatic diaphragmatic hernia. We believe that this is
Accepted for publication July 9, 2008. Address correspondence to Dr Kamiyoshihara, Department of General Thoracic Surgery, Maebashi Red Cross Hospital, 3-21-36 Asahi-Cho, Maebashi, Gunma, 371-0014, Japan; e-mail:
[email protected].
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Ann Thorac Surg 2009;87:959 – 61
