LETTER TO THE EDITOR

Osteosarcoma in a pregnant woman: case report

Rosana Rosa Miranda Correa • Ana Paula Espindula • Juliana Reis Machado •

Marina Carvalho Paschoini • Janaınna Grazielle Pacheco Olegario •

Laura Penna Rocha • Marlene Antonia dos Reis

Received: 26 January 2012 / Accepted: 26 June 2012 / Published online: 24 July 2012

� Springer-Verlag 2012

Dear Editor,

Osteosarcoma is the most common primary malignant bone

tumor and it mainly affects large bones. In pregnant

women it is a rare entity [1]. We presented a case of

26-years-old, 33-weeks pregnant, who had lost 8 kg after

the onset of pregnancy and had progressive dyspnea, was

admitted on 25th April 2007. The patient reported axillary

and right shoulder pain. A nodule of 20 cm in diameter,

with rapid growth during the last 6 months, was observed.

Chest X-ray: pulmonary nodules; ultrasound: a mass in the

right anterior axillary region extending towards the pos-

terior axillary line and the scapular region; biopsy was

performed. The patient developed respiratory failure and

cesarean was performed. Then, it evolved to hemodynamic

instability and death on 27th April 2007. Biopsy result after

death was of high-grade osteosarcoma (grade 3 of 4).

Anatomopathologic exam Osteosarcoma of the right

scapula, measuring 19 9 15 9 10 cm and 2,600 g. Bilateral

pulmonary metastases with multiple nodules. Micros-

copy: osteoblastic, chondroblastic and fibroblastic sub-

types of osteosarcoma in the bone matrix; pleomorphic

cells with atypical nuclei (Fig. 1a). There was accentuated

Lactoferrin immunolabelling found in the bone matrix,

nucleus and cytoplasm of neoplasic cells (Fig. 1b).

Although osteosarcomas usually affect long bones [2],

in this case the osteosarcoma affected the scapula, which is

a flat bone. The occurrence of this type of neoplasia in

pregnancy is extremely rare and the few cases reported in

the literature showed rapid progress [1], similar to the

aforementioned case. Delayed diagnosis may be due to the

lack of data collection of all of the signs and/or symptoms

during the gestation period [3].

Pregnancy can create conditions for the immune system

to react using mechanisms which are similar to those used

in tumor escape [4]. These data might explain why the

neoplasia was only diagnosed in the third quarter of

pregnancy, when immune system changes favor the

development of osteosarcoma and its signs and symptoms

become more evident. Immunological modulation during

pregnancy may also contribute to the development of

metastases [4]. In this case report, the patient had diffuse

lung metastases, which were responsible for obstructing the

bronchial light and death. During autopsy, other metastases

were found in sites such as the lymph nodes and in sites

considered to be less common by the literature [3], such as

the kidney, thus showing how severe this case was.

The immunohistochemistry demonstrated markedly

positive staining to Lactoferrin in the neoplasic tissue.

Lactoferrin can promote bone growth by stimulating the

proliferation and the differentiation of primary osteoblasts

and by inhibiting their apoptosis, supporting a significant

role for this protein in promoting bone formation and in

growth of neoplasic bone tissues in humans [5].

In this case the diagnosis was performed only after

death, and the pregnancy contributed to a difficult clinical

diagnosis and rapid progression of osteosarcoma. This

seems to be one of the fewest reports of osteosarcoma,

The study was conducted in Uberaba, Minas Gerais, Brazil.

R. R. M. Correa (&) � A. P. Espindula � J. R. Machado �J. G. Pacheco Olegario � L. P. Rocha � M. A. dos Reis

Discipline of General Pathology, Federal University

of Triangulo Mineiro, 30 Frei Paulino St, Uberaba,

Minas Gerais 38025-180, Brazil

e-mail: rosana@patge.uftm.edu.br

M. C. Paschoini

Gynecology and Obstetrics Departament, Federal University

of Triangulo Mineiro, Uberaba, Minas Gerais, Brazil

123

Arch Gynecol Obstet (2012) 286:1601–1602

DOI 10.1007/s00404-012-2483-3

connected with pregnancy, with emphasis on scapula

damage, and on the severity of the disease and its rapid

progression leading to the death of a 26-year-old pregnant

woman.

Acknowledgments This work was financially supported by Con-

selho Nacional de Desenvolvimento Cientıfico e Tecnologico

(CNPq), Coordenacao de Aperfeicoamento de Pessoal de Nıvel

Superior (CAPES), Fundacao de Amparo a Pesquisa do Estado de

Minas Gerais (FAPEMIG), Fundacao de Ensino e Pesquisa de

Uberaba (FUNEPU) and Reestruturacao e Expansao das Universid-

ades Federais (REUNI).

References

1. Nepal P, Singh GK, Singh MP et al (2005) Osteosarcoma in

pregnancy. J Nepal Med Assoc 44:100–101

2. Dhillon MS, Singh DP, Gill SS et al (1993) Primary bone

malignancies in pregnancy. A report of four cases. Orthopaedic

Rev 22:931–937

3. Antunes AA, Antunes AP (2004) Tumores malignos dos ossos

gnaticos: estudo retrospectivo e revisao da literatura. Revista

Brasileira de Cirurgia de Cabeca e Pescoco 33:93–98

4. Shakhar K, Valdimarsdottir HB, Bovbjerg DH (2007) Risk of

breast cancer following pregnancy: could lasting systemic immune

alterations contribute? Cancer Epidemiol Biomark Prev 16:

1082–1086

5. Ieni A, Barresi V, Grosso M et al (2009) Lactoferrin immuno-

expression in human normal and neoplastic bone tissue. J Bone

Miner Metab 27:364–371

Fig. 1 Osteosarcoma—common light microscopy: osteoblastic,

chondroblastic and fibroblastic subtypes of osteosarcoma in the bone

matrix; pleomorphic cells with atypical nuclei (HE, 9820) (a). There

was accentuated Lactoferrin immunolabelling in the osteosarcoma,

found in the bone matrix, nucleus and cytoplasm of neoplasic cells

(PAP, 91250) (b). PAP peroxidase anti-peroxidase method of

immunohistochemistry–antibody (2B8) to Lactoferrin, Abcam, 1:100

1602 Arch Gynecol Obstet (2012) 286:1601–1602

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