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The New Zealand Medical Journal

 Journal of the New Zealand Medical Association, 15-July-2005, Vol 118 No 1218

Inflammatory breast cancer in a male
Michel Choueiri, Zaher Otrock, Ayman Tawil, Ihab El-Hajj, Nagi El Saghir
Abstract
Male breast cancer is very rare, especially inflammatory breast cancer, which is an aggressive, rapidly proliferating manifestation of primary breast carcinoma. We present a case report of a 56-year-old man in Lebanon who died 8 months after being diagnosed with inflammatory breast cancer.

Male breast cancer affects 1500 men each year worldwide.1 Inflammatory breast cancer (IBC) is an aggressive, rapidly proliferating manifestation of primary breast carcinoma.2 Male IBC is very rare and our review of the English literature yielded only six reports of eight cases.1,3–7 Here, we present the case of a man diagnosed with IBC after he presented with cervical lymphadenopathy, swelling, and redness of the right anterior chest. A biopsy of the breast showed infiltration of dermal lymphatics by tumour cells. His disease was aggressive and he died 8 months after diagnosis.

Case report

A 56-year-old man presented to our hospital (American University of Beirut Medical Center, Beirut, Lebanon) with neck swelling, oedema, and warmth of the right anterior chest wall, and gynaecomastia of 2 months duration. He was a 75 pack-years tobacco smoker. He reported that an enlarged right cervical mass appeared 2 months prior to his presentation. This mass was soon complicated by generalised, painless neck swelling that rapidly spread involving the right arm and chest wall with erythema and warmth. The right breast became painful and swollen. He then developed a feeling of suffocation, myalgias, arthralgias, and dyspnoea on exertion. He had no nipple discharge or bone pain.
On physical examination, the patient had a sensitive and painful thickened skin with redness and warmth over the right chest wall, upper abdomen, and upper arm. Both breasts were tender (especially on the right side) but no masses could be felt. Oedema involved the right breast and right anterior chest, and it extended to the right axilla. His right nipple was retracted. He had generalised neck lymphadenopathy with a decrease in the range of motion of the neck. He had no palpable axillary or inguinal lymph nodes.
His complete blood count, liver function tests, and urinalysis were normal. Erythroid sedimentation rate was 97 mm/hour (reference range 0–15 mm/hour). The mammogram showed severe skin-thickening and was suspicious of a malignancy. Biopsy of the breast skin revealed plugs of poorly differentiated adenocarcinoma within dermal lymphovascular channels. The dermal stroma and overlying epidermis were free of tumour cell (Figure 1). At that point, the diagnosis of an inflammatory breast carcinoma was made.

Figure 1. Breast skin with tumour thrombi within lymphovascular channels in the dermis (arrow); haemotoxylin and oesinophil stain; original magnification (x40). Inset shows a higher magnification (x100) of the arrowed position

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Immunohistochemical stains showed tumour cells to be negative for oestrogen and progesterone receptors. They showed no HER2/neu overexpression. Almost all tumour-cell nuclei were positive for p53. Work-up was positive for metastatic disease to the lungs and the left frontal lobe of the brain.
Systemic chemotherapy using 5-Fluorouracil, Adriamycin, and Cyclophosphamide was administered. However, he showed no response to chemotherapy and refused further treatment. He was lost to follow-up for 3 months after which time he presented with generalised weakness, weight loss, and respiratory failure—and he died 8 months after diagnosis.

Discussion

The definition of IBC is somewhat controversial. Clinically, the triad of erythema, peau d’orange, and rapid onset makes the diagnosis of IBC.8 Pathologically, IBC is diagnosed as involvement of the dermal lymphatics by an infiltrating carcinoma,9 causing oedema and vascular congestion in the upper dermis and giving the clinical appearance of erysipelas.3
Our patient did not present with a breast mass but, rather, with skin changes that were clinically consistent with IBC. Pathologically, the carcinoma infiltrated the dermal lymphatics. Thus, when a male presents with inflammatory changes of the chest wall, the possibility of inflammatory breast cancer should be entertained even in the absence of a breast mass.
IBC has been reported to account for 1%–6% of all breast cancers (including males and females); IBC accounts for only about 1% of breast cancers in males however. Therefore, male IBC is extremely rare.4 Our review of the English literature yielded six reports of only eight cases of male IBC from 1953 to 2001.1,3–7
Inflammatory carcinoma is the most aggressive variant of breast cancer with a very poor outcome,10 and the survival period from the onset of clinical symptoms ranges from 6 to 33 months.4 In our case, the period from the appearance of clinical symptoms to death was 8 months.
Author information: Michel B Choueiri, Resident, Department of Internal Medicine; Zaher K Otrock, Research Fellow, Department of Internal Medicine; Ayman N Tawil, Associate Professor, Department of Pathology and Laboratory Medicine; Ihab I El-Hajj, Fellow, Department of Internal Medicine; Nagi S El Saghir, Clinical Associate Professor, Department of Internal Medicine, American University of Beirut Medical Center, Beirut, Lebanon
Acknowledgement: Informed consent for publication was given by the patient’s family.
Correspondence: Nagi El Saghir, MD, FACP, Clinical Associate Professor of Medicine, Division of Hematology/Oncology, Department of Internal Medicine, American University of Beirut Medical Center, PO Box 113-6044, Beirut, Lebanon. Fax: +961 1 744464; email: nagi.saghir@aub.edu.lb
References:
  1. Spigel JJ, Evans WP, Grant MD, et al. Male inflammatory breast cancer. Clin Breast Cancer. 2001;2:153–5.
  2. Cristofanilli M, Buzdar AU, Hortobagyi GN. Update on the management of inflammatory breast cancer. Oncologist. 2003;8:141–8.
  3. Sina B, Samorodin CS. Bilateral inflammatory carcinoma of the male breast. Cutis. 1984;33:501–2.
  4. Yamamoto T, Iriyama K, Araki T. Male inflammatory breast cancer. Surg Today. 1997;27:669–71.
  5. Abner A, Kaufman M, Pories S, Gauvin G. Unusual presentations of malignancy. Case 1. Male inflammatory (?) breast cancer. J Clin Oncol. 2001;19:3288–9.
  6. Treves N. Inflammatory carcinoma of the breast in the male patient. Surgery. 1953;34:810–20.
  7. Jeelani G, Beg MA, Hussain A, Bhan B. Simultaneous bilateral inflammatory medullary carcinoma of the male breast. Int Surg. 1977;62:147–8.
  8. Singletary SE, Ames FC, Buzdar AU. Management of inflammatory breast cancer. World J Surg. 1994;18:87–92.
  9. Rosai J. Inflammatory Carcinoma of Breast. In: Ackerman’s Surgical Pathology. 6th ed. vol 2. St Louis: Mosby; 1981, p1115.
  10. Amparo RS, Angel CD, Ana LH, et al. Inflammatory breast carcinoma: pathological or clinical entity? Breast Cancer Res Tr. 2000;64:269–73.


     
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