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El examen histolgico revel la pared de un quiste dermoide con un colculo tisular compuesto
de piel y tejido tiroideo. Los folculos tiroideos fueron infiltradas por clulas linfoides que forman
folculos linfoides y centros reactivos (Figuras 1 y 2). El diagnstico final fue struma ovarii con
tiroiditis de Hashimoto.
Como resultado de este diagnstico inusual, el paciente se coloc bajo el cuidado
endocrinolgico 8 meses despus de la ciruga. Ella no haba tenido trastornos de la glndula
tiroides y la funcin tiroidea no se haba evaluado antes de la ciruga. El paciente tuvo sntomas
noncharacteristic como las siguientes: ganar 10 kilos de peso dentro de 4 aos, el estreimiento,
el cabello y las uas quebradizas, lo que podra indicar hipotiroidismo. Evaluacin de la glndula
tiroides realiz 8 meses despus de la ciruga mostr caractersticas de tiroiditis autoinmune.
Ultrasonido revel tiroides simtrica con un volumen de 18.4ml y una estructura irregular lbulo
hipoecoica. Los estudios de laboratorio revelaron aumento de la concentracin de anticuerpos de
tiroglobulina (211,6 UI / ml, rango de referencia <100). Anticuerpos contra la peroxidasa tiroidea
y TSH receptores anticuerpos estaban dentro de los rangos de referencia. Inicialmente, la funcin
de la glndula tiroides era normal con TSH, T4 libre, los valores sricos de T3L, respectivamente,
1.534 mU / ml (0,34-4,94); 14,64 pmol / l (9.01- 19.05); 5,63 pmol / l (2,63 a 5,7). Terapia de
sustitucin de tiroxina no se administr inicialmente. Once meses despus de la ciruga, cuando
la concentracin de TSH en suero aument a 3,76 mU / ml (con fT4-14.64 pmol / l), la terapia de
reemplazo de tiroxina se inici a una dosis de 25 mg / 24 h. Despus de un ao de TSH terapia
fue de 3,0 mU / ml luego dosis diaria tiroxina se elev a 37,5 mg y posteriormente a 50 mg.
Despus de 2 aos de tratamiento, los sntomas del paciente mejorado: su peso era de 75 kilos,
no tena problemas con el pelo y la piel, deposiciones eran regulares. Los estudios de control
revelaron: TSH 1,62 mU / ml con fT4 10,9 pmol / l en dosis de tiroxina 50 mg / 24 h y la tiroides
ecografa mostr una disminucin en el volumen de la glndula a 13,4 ml. Imagen abdominal no
ha demostrado la recurrencia de malignidad ovrica hasta la fecha.
Discusin
Al igual que en nuestro paciente, la mayora de los casos de bocio ovarii son diagnosticados en la
quinta o sexta dcada de la vida y con la presentacin clnica de una asintomtica, por cierto
encontraron masa patolgica en la pelvis [1,6,7]. En las imgenes, ovarii struma se describe
como un gran multilocular, tumor suave delineada, varios centmetros de dimetro no infiltrante
los tejidos circundantes [8]. La ascitis es coincidente con estruma ovrico en 30% de los casos y
desaparece tras la extirpacin del tumor [6,9]. Otros sntomas como dolor abdominal, orinar o
defecar disturbios y sangrado vaginal patolgica son muy poco frecuentes [1].
Ms del 90% de los pacientes con tiroiditis ovarii tienen la funcin tiroidea normal y 5% a 15% de
los pacientes presentan sntomas de hipertiroidismo [10,11]. Sntomas de hipertiroidismo se
deben a la produccin de hormonas adicional por tejido tiroideo normal de estruma ovrico o
sobreproduccin de tiroxina por adenoma txico en estruma ovrico [10,11]. Tambin se ha
informado coexistencia de la enfermedad de Graves y tiroiditis ovarii en los pacientes. Chiofalo
et al. describe un paciente con enfermedad de Graves y el hipertiroidismo persistente a pesar
estrumectoma en el curso de estruma ovrico. Ella desarroll hipotiroidismo despus struma
escisin ovarii [12]. Ciccarelli et al. denunciado el caso de un paciente con recada
hipertiroidismo en el curso de bocio nodular causada por ovarii struma. Ella se convirti en
eutiroideo despus struma escisin ovarii [10]. Amareen et al. y Carvalho et al. describe una
mujer diagnosticada con la enfermedad de Hashimoto, que desarroll sntomas de hipotiroidismo
despus struma escisin ovarii [13,14]. Ambos autores interpretan eutiroidismo de los pacientes
antes de la ciruga debido a la produccin adicional de la hormona tiroidea por el tejido tiroideo
Introduction
Struma ovarii is a rare type of mature ovarian teratoma. Histopathological criterion of struma
ovarii is that thyroid tissue forms more than 50% of the teratomas tissue on microscopic
examination or is macroscopically recognizable as thyroid. Almost 20% of ovarian teratomas
contain thyroid tissue, but, according to the definition, struma ovarii is diagnosed in only 1% to
3% of them. Histopathological evaluation of ovarian teratoma in most cases shows normal
thyroid tissue. Occasionally, it may resemble nodular goitre. In rare cases, struma ovarii contains
follicular or papillary carcinoma foci (<1%) [4,5]. Hashimoto thyroiditis in struma ovarii has been
reported in only a few cases. Following our database search, we have found only six cases to
date. The authors present the case of struma ovarii with chronic lymphocytic inflammation in a
woman without previously diagnosed thyroid gland disease. Eight months after surgery, the
patient was diagnosed with Hashimoto disease and later thyroxin replacement therapy was
initiated.
Case report
A 52-year-old White woman (P2 by Caesarean section), had undergone routine gynecological
examination after a 3-year break. She had no complaints from the genitourinary tract and
menstruations were regular. Gynecological examination revealed right adnexal enlargement. It
was confirmed on CT scanning, showing a 4060mm pathological mass adjacent to the right
ovary. The same month, the patient was hospitalized in the Clinic of Gynecology in the Medical
University of Gdansk. Abdominal hysterectomy with bilateral salpingo-oophorectomy was
performed.
On macroscopic examination, the right adnexa consisted of a 6-cm long fallopian tube and a
large 66 cm creamcolored cystic tumor with a smooth surface. The cysts centre contained a
sebaceous mass. A 1.51 cm colliculus was enclosed in the inner lining of the cysts wall.
Histological examination revealed the wall of a dermoid cyst with a tissular colliculus composed
of skin and thyroid tissue. Thyroid follicles were infiltrated by lymphoid cells forming lymphoid
follicles and reactive centers (Figures 1 and 2). The final diagnosis was struma ovarii with
Hashimoto thyroiditis.
As a result of this unusual diagnosis, the patient was placed under endocrinological care 8
months after surgery. She had had no thyroid gland disorders and thyroid function hadnt been
assessed before surgery. Patient had noncharacteristic symptoms such as the following: gaining
10 kilos in weight within 4 years, constipation, brittle hair and nails, which could indicate
hypothyroidism. Thyroid gland evaluation performed 8 months after surgery showed features of
autoimmune thyroiditis. Ultrasound revealed symmetrical thyroid with a volume of 18.4ml and an
irregularly hypoechoic lobe structure. Laboratory studies revealed increased thyroglobulin
antibodies concentration (211.6 IU/ml, reference range <100). Thyroid peroxidase antibodies and
TSH receptors antibodies were within reference ranges. Initially, thyroid gland function was
normal with TSH, fT4, fT3 serum values, respectively, 1.534 U/ml (0.34-4.94); 14.64 pmol/l
(9.01- 19.05); 5.63 pmol/l (2.63-5.7). Thyroxin substitution therapy was not administered initially.
Eleven months after surgery, when TSH serum concentration increased to 3.76 U/ml (with fT4
14.64 pmol/l), thyroxin replacement therapy was started at a dose of 25 g/24 h. After a year of
therapy TSH was 3.0 U/ ml then daily thyroxin dose was raised to 37.5 g and later to 50 g.
After 2 years of treatment, patients symptoms improved: her weight was 75 kilos, she had no
problems with hair and skin, defecations were regular. Control studies revealed: TSH 1.62 U/ ml
with fT4 10.9 pmol/l on thyroxin dose 50 g/24 h and thyroid ultrasound showed a decrease in
the glands volume to 13.4 ml. Abdominal imaging has not shown recurrence of ovarian
malignancy to date.
Discussion
As in our patient, most cases of struma ovarii are diagnosed in the fifth or sixth decade of life
and with the clinical presentation of an asymptomatic, incidentally found pathological mass in
the pelvis [1,6,7]. In imaging, struma ovarii is described as a large multilocular, smooth-outlined
tumor, several centimeters in diameter not infiltrating the surrounding tissues [8]. Ascites is
coincident with struma ovarii in 30% of cases and disappears after tumor excision [6,9]. Other
symptoms like abdominal pain, urination or defecation disturbances and pathological vaginal
bleeding occur rarely [1].
Over 90% of patients with struma ovarii have normal thyroid function and 5% to 15% patients
present symptoms of hyperthyroidism [10,11]. Hyperthyroidism symptoms are due to additional
hormone production by normal thyroid tissue of struma ovarii or thyroxin overproduction by toxic
adenoma in struma ovarii [10,11]. Coexistence of Graves disease and struma ovarii in patients
has also been reported. Chiofalo et al. described a patient with Graves disease and persistent
hyperthyroidism despite strumectomy in the course of struma ovarii. She developed
hypothyroidism after struma ovarii excision [12]. Ciccarelli et al. reported the case of a patient
with relapsing hyperthyroidism in the course of nodular goitre caused by struma ovarii. She
became euthyroid after struma ovarii excision [10]. Amareen et al. and Carvalho et al. described
a woman diagnosed with Hashimoto disease, who developed hypothyroidism symptoms after
struma ovarii excision [13,14]. Both authors interpret the patients euthyroidism before surgery
due to additional thyroid hormone production by thyroid tissue in struma ovarii. In spite of the
diagnosis of Hashimoto disease, the authors do not mention any histological finding of chronic
lymphocytic thyroiditis in excised struma ovarii.
Development of hypothyroidism after struma ovarii excision in our patient can be explained by
two theories. First, hypothyroidism may be connected to a natural, progressive course of
Hashimoto disease regardless of struma ovarii surgery. Second, hypothesis is similar to Carvalho
and Amareen [13,14]. Development of hypothyroidism after surgery may be related to removal
of hormonal active ectopic thyroid tissue.
Hashimoto disease is one of the most frequent endocrine diseases, particularly in women. It is
surprising that chronic lymphocytic thyroiditis in struma ovarii is a rare discovery. The first
histological finding of Hashimoto thyroiditis in bilateral benign ovarian teratomas was reported in
the literature in 1965 by Eres [15]. In this case, struma ovarii excision was performed 7 years
after subtotal thyroidectomy due to chronic thyroiditis which was not diagnosed as Hashimoto
disease. In 1972, Watson reported the case of a 31-year-old woman after hysterectomy and
bilateral adnexectomy due to menorrhagia [16]. Histological examination of a left ovarian cystic
tumor revealed struma ovarii with chronic lymphocytic thyroiditis. Just like our patient, she had
had no thyroid disorders before surgery. Immunological tests performed 1 year after the surgical
procedure showed no laboratory evidence of Hashimoto disease. Two following histological
findings of Hashimoto disease have been reported by Nielsen and Farrel [17,18]. In 2002,
Bonadio reported the case of a woman with the histological finding of Hashimoto thyroiditis in
struma ovarii. This patient had no symptoms of thyroid gland disease but immunological study
showed positive antithyroid antibodies [19]. In 2010, DAntonio et al. described a 38-year-old
woman with symptoms of hyperthyroidism and positive antithyroid antibodies but with normal
thyroid on ultrasound [2]. Additional examination revealed bilateral ovarian tumors which were
excised and histologically diagnosed as struma ovarii with Hashimoto thyroiditis. One month
after surgical treatment, the patient was euthyroid and antithyroid antibodies were within normal
ranges.
Conclusions
The reported case describes unusual finding of Hashimoto thyroiditis in struma ovarii. This
diagnosis enabled further studies and led to a final diagnosis of Hashimoto thyroiditis in early
stage of disease. It induced thyroxin treatment and enabled to reduce hypothyroidism
symptoms.
Ttulo.
Autor (es).
Ao de publicacin.
2011
Lugar de realizacin
Gdansk - Polonia
Tipo de investigacin y
metodologa.
Resultados
Conclusiones
Glosario de trminos