The patient was admitted to the hospital on October 7, 2013 with “dizziness and weakness for 5 years, hair loss and arthralgia for 1 year, and breast enlargement for 5 months”. The dizziness and weakness appeared 5 years ago without any obvious cause, and 4 years ago the dizziness worsened with dark urine color, hemoglobin 73g/L was investigated outside the hospital, hemoglobin electrophoresis and thalassemia gene test showed a-thalassemia (intermediate type), direct Coomb test (+), combined with autoimmune hemolytic anemia was considered, prednisone 60mg/d and symptomatic treatment was given, 1 month later the hemoglobin increased to 105g/L. One month later, hemoglobin rose to 105g/L, and the hormone was gradually reduced to 5mg/d for maintenance. One year later, ANA3.3S/CO, anti-dsDNA antibody, anti-ENA antibody (-), complement C30.499g/L and C40.121g/L were detected, so he stopped hormone use and changed to traditional Chinese medicine treatment. 1 year ago, he felt arthralgia and hair loss, searched outside hospital for glistening 74g/L, direct Coombs test (+), ANA1:320 (+), anti-dsDNA antibody 598.5U/ml, C30.536g/L, C40.117g/L, diagnosed as SLE, given prednisone 50mg/d + cyclosporine A 100mg/d treatment. After 2 weeks, the hormone dose was reduced by 5mg per week to 10mg/d and maintained: 9 months ago, unplanned pregnancy was detected and rechecked with hemoglobin 89g/L, ANA 1.643, anti-dsDNA antibody 3.30, C30.641g/L, C40.155g/L. Cyclosporine A was discontinued and replaced by prednisone 10mg/d + hydroxychloroquine 0.4g/d treatment, 5 months ago (14 weeks of pregnancy>Self-perceived breast swelling and pain with obvious itching of breast skin, no fever and nipple overflow, consulted the dermatology department for contact dermatitis and treated with anti-allergic treatment with poor results, and developed progressive breast enlargement with an average monthly increase of 10cm in breast circumference. Blood prolactin >200ug/L was checked at 28 weeks of pregnancy, breast ultrasound showed bilateral mega-breasts, thickened skin of both breasts, tissue interstitial edema, large hypoechoic areas in both breasts. Multiple cystic hypoechoic areas were seen in the subcutaneous glands, and no enlarged lymph nodes were seen in the axillae bilaterally. At 37 weeks of gestation, her chest circumference was 137 cm, the maximum circumference of the right breast was 74 cm, and the maximum circumference of the left breast was 63 cm. After 15 d, the breast shrunk significantly and the pain decreased, so the bromocriptine was discontinued. Since the onset of the disease, there was no photosensitivity, facial erythema, oral ulcers, no hematuria or foamy urine, no dry mouth or eyes, no vision loss or visual field defects, no fear of cold or facial swelling, no loss of appetite or skin mucous membrane pigmentation, no axillary or pubic hair thinning or decreased libido, no polyuria or pale urine. She had two previous pregnancies, both of which were aborted at about 8 weeks of gestation, and there was no breast enlargement during pregnancy. There was no family history of breast cancer. Physical examination: BMI 25.6 kg/m2, anemic appearance, chest circumference 132 cm, significant bilateral breast enlargement, maximum circumference of the right breast 70 cm, the lower edge reaching the level of the anterior superior iliac spine in the standing position; maximum circumference of the left breast 57 cm, the lower edge reaching the level of the umbilicus in the standing position. Superficial varicose veins in the breasts were obvious, the nipples were centered, without obvious depressions.1 The skin was not broken or retracted, both breasts were tough and nodular, no tenderness, no nipple overflow, and no enlarged lymph nodes were palpated in the supraclavicular and axillary areas. Laboratory tests: hemoglobin 67g/L, reticulocytes 152.4x107L, direct Coombs test (+), total bilirubin 18.8umol/L, indirect bilirubin 15 (junol/UANA0.835S/CO value, anti-dsDNA antibody 2.205, C3.C4 normal; prolactin 255ug/L, follicle stimulating hormone 5.75U/L, luteinizing hormone 2.05U/L, estradiol 14.0ng/L, free testosterone 0.15pg/ml, dehydroepiandrosterone sulfate 33.96ng/ml, androstenedione 0.35ng/ml, free T34.73pmol/L, thyroid stimulating hormone 2.657mll/L, cortisol (8:00) 115.77nmol /L, growth hormone 0.375ng/ml; carcinoembryonic antigen and methemoglobin were normal. Pituitary magnetic resonance imaging (MRI) scan + enhancement showed a fuller pituitary gland and a possible small adenoma in the left lower part of the anterior lobe. Breast color Doppler ultrasound showed: significant bilateral breast gland thickening with multiple cystic lesions within (max. about 35 mmx23.4 mm, Figure 1), no obvious solid occupancy; bilateral axillary lymph node sonograms (max. about 9.5 mmx5.0 mm). Part II: Diagnosis and discussion Diagnosis: SLE combined with gestational macromastia was considered and treated with prednisone 30mg/d + hydroxychloroquine + methotrexate + bromocriptine + hydrochlorothiazide. After 1 month of treatment, the patient’s breasts were significantly smaller than before, with a maximum circumference of 43 cm on the right side and 39 cm on the left side. Ultrasound color Doppler of the breast indicated that there was still significant fluid accumulation, so the fluid was withdrawn under ultrasound guidance, and a total of 70 ml of yellow transparent fluid was withdrawn. The patient is still being followed up and is scheduled for elective breast surgery after stabilization and improvement of anemia. Discussion Gigantomastia is an enlargement of the breast volume more than twice the normal size or at least 1.5 kg unilateral breast resection, which can cause breast pain, ulceration, infection, postural balance problems, respiratory distress in the lying position, back pain and chronic strain injury to the 4th-6th intercostal nerve resulting in nipple sensory loss. The cause of breast enlargement is not known, but most of it occurs during adolescence and pregnancy and may be related to hormonal imbalance in the body. The incidence of gigantomastia during pregnancy is called gestational gigantomastia, which is rare clinically, with 4 cases reported in China and less than 100 cases reported in foreign literature. Idiopathic gigantomastia can be divided into three categories according to the etiology: idiopathic, endocrine hormone-related (mainly including adolescent and gestational gigantomastia), and pharmacogenic. The prognosis of idiopathic breast enlargement can be achieved by breast reduction surgery. Adolescent and gestational gigantomastia mostly present with progressive and uninterrupted breast enlargement and usually require multiple breast reduction treatments. In the literature [1, 9], penicillamine, aminothiourea, dexamethasone and cyclosporine A have been reported to cause mastocytosis. Cyclosporine A may increase prolactin levels through central and peripheral mechanisms, thereby promoting the growth of breast and adipose tissues Drug-induced breast enlargement does not increase further after discontinuation of the drug with or without breast retraction. There are also case reports of autoimmune diseases and malignancies manifesting as gigantomastia. Clinical attention should be paid to differentiate between lymphoma, breast malignancy and gigantomastia in patients with excessive breast enlargement. The pathogenesis of gestational breast enlargement is unclear and may be related to elevated hormone levels in the body during pregnancy and/or hypersensitivity of the breast tissue to physiological levels of hormones. The hormones involved in the pathogenesis include estrogen, progesterone, prolactin, testosterone and cortisol, of which estrogen and prolactin are the most important. In some patients, the administration of estrogen sphincters or bromocriptine can inhibit breast enlargement or even reduce breast size. In addition, some scholars believe that gestational gigantomastia is due to inflammatory hyperplasia of the breast caused by dramatic hormonal changes during pregnancy, and inflammatory cell infiltration can be seen in the diseased breast tissue, and the application of small doses of glucocorticoids is effective in some patients. In this case, color Doppler ultrasound of the breast indicated a large amount of exudate fluid, and laboratory tests indicated a significant increase in protein and a mild increase in total white blood cells, mainly lymphocytes, which were considered mild inflammation. However, conservative treatment only controls the growth of gigantomastia and makes it difficult to restore the breast to normal levels, so surgery is the primary treatment for gigantomastia. Mastectomy is the only definitive cure for gigantomastia. Delayed reconstruction bilateral mastectomy and breast reduction are the most common procedures, but the latter carries a risk of recurrence. It has been reported that patients with gestational breast enlargement who do not undergo a complete mastectomy have essentially a 100% risk of recurrence in another pregnancy. Therefore, the choice of surgical approach should be considered in the context of the patient’s overall health status and the need for a second pregnancy. In the present case, the SLE patient had progressive breast enlargement during pregnancy, more than twice the normal size, and multiple color Doppler ultrasound of the breast indicated thickened breast tissue with multiple cystic lesions within, which was consistent with the ultrasound diagnosis of gigantomastia, so the diagnosis of gestational gigantomastia was established. The patient’s pituitary MRI showed a suspicious small adenoma in the left lower pituitary gland, repeated menstrual irregularities and infertility, and a significant decrease in prolactin on the day of postpartum recheck. The patient had a suspicious small adenoma in the left lower pituitary gland, which may be related to pregnancy. In addition, the patient did not have any occupying lesions on color Doppler ultrasound of the abdomen and pelvis, and her prolactin was normal after 1 month of bromocriptine treatment. It is also unlikely that there is an occupying lesion in the body that causes autonomous secretion of prolactin. Discontinuation of CsA and reduction of mechanical stimulation of the breast may prevent a secondary rise in prolactin. The patient’s elevated blood estradiol and prolactin and decreased androgens may be associated with SLE in addition to pregnancy, which may also contribute to the development of macromastia. The patient had a history of thalassemia and hemoglobin had been fluctuating around 65 g/L after delivery, while the breast enlarged below the level of the umbilicus with obvious varicose veins in the breast, and the surgery might be invasive, long and with a lot of intraoperative blood loss, so conservative treatment was given first. Combined with the patient’s SLE condition and the large amount of exudate in the bilateral multiple cystic lesions of the breast on color Doppler ultrasound, the dose of prednisone was increased to 30 mg/d to enhance anti-inflammation and symptomatic treatment such as hydrochlorothiazide diuresis, breast pumping and hot compresses, while methotrexate was added to control the lupus condition and bromocriptine was continued to antagonize the secretion of lactogen. At the follow-up after 1 month of treatment, the patient’s breasts were significantly smaller than before and the symptoms were reduced, suggesting that active conservative treatment before surgery could significantly reduce breast edema exudation, which not only could make the breasts significantly smaller and relieve the symptoms, but also could reduce the surgical trauma and facilitate the surgery.