Preterm neonate with a large congenital hemangioma on maxillofacial site causing thrombocytopenia and heart failure: A case report

Table 1 Timelines for the findings and treatment

Timeline	Findings	Treatment
Day 1: Admission	(1) A 7.1 cm × 5.3 cm × 3.1 cm tumor was located at the right maxillofacial region; (2) the laboratory test revealed that the level of Hb and PLT was reduced, the APPT, PT, and TT were extended and INR, FDP, and D-Dimer were increased; alpha angle, MA, and K were elevated. TSH level was significantly elevated; and (3) atrial septal defect, pulmonary hypertension (severe)	(1) Appropriate limit the intake of fluid to reduce the preload of heart; (2) stepwise infusion of 40 mL cells suspension (4 h); platelet infusion of 40 mL (1 h); plasma infusion of 40 mL (4 h); prednisone tablets (4 mg/kg/d, evenly divided two times daily); intended use for 6 wk; propranolol (2mg/kg/d, evenly divided two times daily); levothyroxine (7 μg/kg/d, once a day); the dose was adjusted based on the level of TSH; vitamin K1 (1 mg/time); milrinone (0.5 μg/kg/min for 24 h); and (3) protect the tumor by paying attention to the tension change, avoiding rupture bleeding
Day 2: Continuing attempt to elevate the platelet level	(1) The laboratory test showed that the level of Hb and PLT was not significantly increased, and the level of PT and TT was not improved; however, the level of FDR and D-Dimer were increased; alpha angle, MA, and K were elevated; and (2) total bilirubin level was increased (mainly indirect bilirubin)	Additional diagnosis: Neonatal hyperbilirubinemia; platelet infusion of 40 mL (1 h); plasma infusion of 40m L (4 h); vitamin K1 (1 mg/time); blue light irradiation
Day 3: Further workup	(1) Neck enhanced CT suggested that it was a subcutaneous tumor in right maxillofacial region, tortuous and thickened vascular shadow of right neck, considered as round vascular lesion, atypical hemangioma; (2) PLT continually decreased compared with previous day; we attributed the decrease to the consumption by the hemangioma; and (3) there was no change for TT, and FDP and D-Dimer were still higher	Platelet infusion of 40 mL (1 h)
Day 4: Blood test	PLT was slightly increased; MA was significantly decreased	No adjustment of therapy strategy
Day 7: Blood test and echocardiography	(1) PLT level was still low, however, not worsened; (2) BNP was increased; (3) bilirubin was slightly decreased; and (4) symmetrical lower extremity edema; the major pulmonary artery diameter was about 10 mm, the size of the right atrium was about 21 mm × 21 mm, the heart was enlarged, mainly the right heart. The echo separation at the oval fossa was 2.0 mm; atrial level left to right shunt, tricuspid regurgitation signal, area of 0.5 cm2, the maximum reflux velocity of 395 cm/s, Pg 62 mmHg, which suggests of the whole heart enlargement (right heart), patent foramen ovale, moderate tricuspid incompetence, and pulmonary hypertension (moderate to severe)	(1) Additional diagnosis: congestive heart failure; and (2) digoxin (0.01 mg/kg/d, evenly divided two times daily); milrinone (0.5 μg/kg/min for 24 h); furosemide (0.5 mg/kg/time, one or twice a day)
Day 10: Blood test and physical examination	PLT was in normal range; BNP was further decreased; low extremity edema improved	Continued previous treatment
Day 14: Blood test and echocardiography	(1) PLT was not further decreased; (2) BNP was further decreased, but still higher than normal; (3) TSH fell into the normal range; (4) measurement showed the tumor was 7 cm × 6.5 cm × 3 cm; and (5) echocardiography showed that the left heart was full; the tricuspid regurgitation signal was detected with area of 0.5 cm2, the maximum reflux velocity of 301 cm/s, Pg 36 mmHg, pulmonary artery pressure 41 mmHg, which suggests patent foramen ovale, moderate tricuspid incompetence, and pulmonary hypertension (mild)	Withdrawal of digoxin and milrinone
Day 21: Blood test	(1) PLT was increased, though not as high as normal; (2) BNP was not in normal range; and (3) echocardiography: Left heart was enlarged, mild tricuspid insufficiency and patent foramen ovale were identified	No adjustment of treatment strategy
Day 25: Blood test	There was fluctuation of Hb and PLT	No adjustment of treatment strategy
Day 29: Blood test	(1) The level of Hb and PLT was still decreased; and (2) echocardiography: left heart was enlarged, mild tricuspid insufficiency, patent foramen ovale, cardiac function was within the lower limit of normal function	(1) Platelet infusion of 60 mL (1 h); red cell suspension infusion of 45 mL (4 h); and (2) pay attention to anemia and bleeding
Day 30: Blood test	The level of Hb was in normal range; PLT was increased	Continue current treatment
Day 34: Blood test and measurement of the tumor	(1) PLT was not further decreased; (2) BNP was improved; (3) TSH was further decreased, but still higher than normal; (4) echocardiography: Left heart was enlarged, mild tricuspid insufficiency and patent foramen ovale were identified, cardiac function was within lower limit of normal range; and (5) size of tumor was 6.5 cm × 6 cm × 3 cm	Surgical resection scheduled for next day
Day 35: Operation	-	(1) Volume of bleeding was about 350 mL, blood transfusion was about 360 mL; (2) the patient was safely returned to the ward after the operation; respiratory support was offered, transfusion of red blood cell suspension, platelets, cold precipitation, plasma was performed to prevent infection; and (3) symptomatic treatment was conducted when necessary
Day 36: Blood test, pathological examination and determination of myocardial enzymes	(1) Hb returned to normal and PLT was increased; (2) CRP increased; and (3) pathological examination showed that it was CH with massive hemorrhage; local extramedullary hematopoietic and fibrous tissue hyperplasia were seen	(1) Limited intake of liquid with precondition of maintaining normal circulation; (2) human serum albumin: 5 mL/kg/time, once; furosemide: 0.5 mg/kg/time, twice; record of intake and output of the patient; cefepime: 30mg/kg/time, Q12H; and (3) compression bandage and care for surgical wound and disinfection
Day 40: Blood test	BNP and TSH returned to normal	No adjustment of treatment strategy
Day 44: Blood test	Monitor PLT, and CRP; indicators of liver function and myocardial enzymes stayed in the normal range	Cefepime was withdrawn
Day 51: Blood test and echocardiography	PLT, indicators of coagulation function, BNP, FT3, FT4 and TSH were in normal range	The patient was discharged

Hb: Hemoglobin; PLT: Platelet count; Pg: Pressure gradient; APPT: Activated thromboplastin time; PT: Prothrombin time; MA: Maximum amplitude; TT: Thrombin time; INR: International normalized ratio; FDP: Fibrin degradation product; TSH: Thyroid-stimulating hormone; K: The clot formation speed; CT: Computed tomography; BNP: Brain natriuretic peptide; CRP: C-reactive protein; FT3: Free triiodothyronine.