Recurrence of unilateral angioedema of the tongue: A case report

Table 1 Results of laboratory examinations on admission

Parameter	Recorded value	Standard value
White blood cell count (µL)	5560	4000-9000
Neutrophils (%)	91.1	40.0-71.9
Lymphocytes (%)	7.4	26.0-46.6
Monocytes (%)	1.1	2.3-7.7
Eosinocytes (%)	0.4	0.2-6.8
Hemoglobin (g/dL)	13.7	11.3-15.2
Platelet count (µL)	9.1 × 104	15.0-35.0 × 104
Prothrombin time/activity (%)	69.7	80-120
International normalized ratio	1.25	0.90-1.20
Activated partial thromboplastin time (s)	37.0	26.0-35.0
C-reactive protein (mg/dL)	0.16	< 0.30
Total protein (g/dL)	6.9	6.7-8.3
Albumin (g/dL)	4.0	3.9-4.9
Total bilirubin (mg/dL)	1.0	0.2-1.2
Aspartate aminotransferase (IU/L)	64	13-33
Alanine aminotransferase (IU/L)	49	8-42
Lactase dehydrogenase (IU/L)	253	124-222
Creatine kinase (IU/L)	213	45-163
Blood urea nitrogen (mg/dL)	14.4	8.0-20.0
Creatinine (mg/dL)	0.52	0.40-0.80
Sodium (mEq/L)	143.6	135.0-147.0
Potassium (mEq/L)	3.8	3.4-4.8
Chloride (mEq/L)	107	98-110
Glucose (mg/dL)	126	70-109
Hemoglobin A1c (%)	5.0	4.6-6.2
Ferritin (ng/mL)	116.5	5.0-157.0
Total cholesterol (mg/dL)	157	130-219
Triglyceride (mg/dL)	43	30-150
High-density lipoprotein cholesterol (mg/dL)	76	48-103
Low-density lipoprotein cholesterol (mg/dL)	73	70-139

Table 2 Additional tests performed to identify the cause of angioedema

Parameter	Recorded value	Standard value
C1 inhibitor activity (%)	73	70-130
C3 (mg/dL)	75	86-160
C4 (mg/dL)	10	17-45
CH50 (U/mL)	26.7	25.0-48.0
C1q (mg/dL)	12.6	8.8-15.3
Nonspecific IgE (IU/mL)	71.81	< 170
Antinuclear antibody	< 40 ×	< 40 ×
Estradiol	16.8	< 47.0 pg/mL (postmenopausal)

IgE: Immunoglobulin E.

Table 3 Clinical features of cases of unilateral angioedema of the tongue in the literature

Case	Ref.	Age (yr) /sex	Causes of Angioedema	Comorbidity	Type of ACEI and duration of use	Other medication	Treatment of angioedema	Time to symptom improvement	Recurrence
1	Mlynarek et al[3]	73/F	ACEI	Diabetes mellitus, hypertension, hypothyroidism, Bell's palsy, rectal surgery for rectal cancer	Enalapril, 3 yr	Nifedipine, lorazepam, clonidine, levothyroxine, aspirin, and hydrochlorothiazide	MPSL, diphenhydramine, and penicillin. Enalapril was not discontinued initially	Overnight	Symptoms recurred after 3 wk. Enalapril was discontinued subsequently. No further recurrence at the 1-, 3-, and 12-mo follow-ups
2	Kaptanoglu and Aytas[4]	44/M	Aspirin	Headache	None	None	Epinephrine subcutaneously, mPSL, chlorphenoxamine	2 h	No recurrence at the 10-d follow-up
3	Chan et al[5]	68/F	ACEI	Type 2 diabetes mellitus and hypertension	Benazepril, several mon	Humulin 70/30, amlodipine, and chlorthalidone	Intravenous diphenhydramine, mPSL, and famotidine. PSL, famotidine, and diphenhydramine	Within 24 h	No recurrence at the 2-mo follow-up
4	Ee et al[6]	71/M	ACEI, evacuation of the subdural hematoma	Subdural hemorrhage, hypertension, history of deep vein thrombosis and right temporal subdural hemorrhage secondary to warfarin therapy	Perindopril, 3 mo	Pantoprazole (40 mg once daily), one dose of intravenous ceftriaxone (2 g), and pre-operative prophylaxis (evacuation of subdural hematoma)	Intravenous dexamethasone. Perindopril was discontinued	48 h	No recurrence at the 2-wk follow-up
5	Kuhlen and Forcucci[7]	62/M	ACEI	Hypertension, type 2 diabetes, vascular dementia, end-stage renal disease after cadaveric renal transplant	Lisinopril, recently	Other medications were not mentioned. He had undergone a kidney transplant	Diphenhydramine, famotidine, and mPSL. Intubation	48 h	NA
6	Leung et al[8]	64/F	ACEI	Liver transplant	Lisinopril, 2 d	Mammalian target of rapamycin inhibitor for the liver transplant	Intravenous steroids and antihistamine. Lisinopril was discontinued	Within h	No recurrence at the 4-wk follow-up
7	Amey et al[9]	76/M	ACEI	Allergic rhinitis, hypertension, ischemic heart disease, and two percutaneous coronary interventions	Perindopril, 10 yr	Clopidogrel, aspirin, pravastatin, and diclofenac for a rotator cuff injury. Clopidogrel and atorvastatin	Intravenous steroids and antihistamine. Intubation. Perindopril was discontinued. Intubation. Perindopril was discontinued	48 h	No recurrence
8	Amey et al[9]	78/M	ACEI	Asthma with moderate airflow obstruction, percutaneous coronary interventions performed 7 yr prior, epileptic	Perindopril, 7 yr	Clopidogrel, atorvastatin, and phenytoin	Epinephrine nebulizations, steroids, and antihistamines. Perindopril was discontinued	24 h	No recurrence
9	Deepthi et al[10]	30/F	Acetaminophen	Tension headache	None	Acetaminophen	Acetaminophen was discontinued, intravenous dexamethasone, intravenous diphenhydramine	2 d	NA
10	Arts et al[11]	67/M	ACEI, rtPA, cerebrovascular ischemia of the left hemisphere	Right-sided hemiparesis, and cerebrovascular ischemia of the left hemisphere	NA	NA	Intubation, clemastine, and adrenaline	NA	NA
11	Imai et al[12]	69/F	ARB	Hypertension, diabetes mellitus, and dysarthria	None (ARB used)	Nicardipine, azilsartan, glimepiride, metformin, vildagliptin, and aspirin	Azilsartan was changed to a carvedilol. Aspirin was discontinued	NA	NA
12	Al-Hoqani et al[13]	78/F	ACEI	Hypertension, hyperlipidemia, ischemic heart disease, bilateral knee osteoarthritis, and urinary incontinence	Lisinopril, 2 yr	Amlodipine, rosuvastatin, aspirin, calcium with vitamin D, bisoprolol, diclofenac sodium, and a multivitamin	Chlorpheniramine maleate intramuscularly, Lisinopril was stopped	12 h	No recurrence at the 2-mo follow-up
13	Wollmach et al[14]	49/F	Acute ischemic stroke, rtPA	Pulmonary sarcoidosis and post liposarcoma resection. Angioedema after receiving NSAIDS	None	None	mPSL, clemastine, ranitidine, intubation, and icatibant	48 h	NA
14	Lee and Bryant[15]	80/M	ACEI	Hypertension	Benazepril, 4 yr	Amlodipine	mPSL, diphenhydramine, and famotidine	48 h	NA
15	Gil Braga et al[16]	55/M	ACEI	Hypertension	Enalapril, 3 yr	None	Observation, enalapril was discontinued	Several h	No recurrence at the follow-up a few mon later
16	This present case	65/F	Unknown	Hypertension, hyperlipidemia, obesity, NASH, and liver cirrhosis	Lisinopril, 32 yr	Amlodipine, doxazosin, pitavastatin, and polyene phosphatidylcholine	Epinephrine intramuscular injection, mPSL. Lisinopril was discontinued	17 h	Recurrences occurred 10 times at the 2-yr follow-up

NA: Not applicable; ACEI: Angiotensin-converting enzyme inhibitor; mPSL: Methylprednisolone; rtPA: Recombinant tissue-type plasminogen activator; NSAIDS: Non-steroidal anti-inflammatory drugs; NASH: Non-alcoholic steatohepatitis; ARB: Angiotensin receptor blocker.