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Other risk factors for CAM have been described, including obstruction of the ipsilateral lymphatic drainage,
bulky tumor in the breast, extensive tumor burden in the axilla, and as mentioned before, previous surgery
[3,7]
and/or radiotherapy .
There are challenges in the clinical scenario of CAM. Contralateral spread from the original tumor is not
the only possibility. An occult second primary in the contralateral breast and axillary metastasis from the
[8]
extrammamary site should be considered . Although it is possible to have certainty of the tumor origin
with a biopsy of the axillary node, to discard an occult second primary in the contralateral breast is not
always an easy task. MRI has been proposed to be a good technique to identify occult primary breast
[9]
carcinoma (OPBC). In a prospective analysis carried out by Buchanan et al. , 69 patients with occult OPBC,
underwent breast MRI, identifying 80% (55 of 69) of suspicious lesions and proved to be breast carcinoma
in 49% of them (34 of 69). Although MRI has its limitations, due to false positive results, the authors of this
study support that OPBC is a primary indication for the use of MRI.

CAM is an uncommon event in breast cancer and has a poor prognosis. AJCC 8th edition classifies CAM
as stage IV disease because contralateral axillary lymph nodes are not considered a regional extent of the
[10]
disease, but a distant metastasis . However, several publications have questioned this affirmation and
proposed that treatment of CAM should be aggressive and multidisciplinary as it is in the case of locally
advanced breast cancer, rather than treated as a distant metastasis [3,4,8] . A systematic review analyzed 48
patients with CAM, 23 had complete follow up data during a mean time of 50.3 months with an overall
survival of 82.6%. An important fact is that 92.1% of patients received surgical treatment and 88.9% systemic
[11]
treatment .
The patient of the case had many risks factors, like conservative surgery, radiotherapy in the left breast and
an ipsilateral recurrence treated with mastectomy and SLNB. After completing ALND, 15 lymph nodes were
negative for metastasis. However, CAM was identified two years later.

History of HL has been described as a risk factor for breast cancer, with an excess risk of 40% even after 40
years. The main risk factor is mantle radiotherapy, especially in young women in the “peri-pubertal” period,
corresponding as the highest breast radio sensitivity period [12,13] . The patient in our case was treated with
chemotherapy exclusively and after menopause, thus we cannot assume a specific risk association.

It is important to mention that even though this patient received aggressive surgical treatment, chemotherapy
and radiotherapy, the disease progressed as a systemic disease in a short period of time. This reflects the
complexity of the case and that different approaches should be taken into account when treating these patients.

In conclusion, CAM are an unusual form of metastasis in unilateral breast cancers. The assumption of
a disseminated disease prompt to systemic therapies rather than an aggressive surgical treatment. There
are many controversies regarding the appropriate management and the prognostic influence on these
patients. Unfortunately, there is poor evidence for the adequate treatment, due to the rarity of the event.
An individualized and multidisciplinary approach is encouraged for these patients, so a positive impact on
survival is achieved.

DECLARATIONS
Authours’ contributions
Conception or design of the work: Giménez MJ, Patrón JM
Data collection: Giménez MJ, Patrón JM, Vento G, Ferrer R, Asensi J, Gavila J
Drafting, critical revision of the article, final approval of the version to be published: Giménez MJ, Patrón
JM, Vento G, Bayón A, Maisto V, Bolumar I, Ferrer R, Asensi J, Gavila J, Estevan R