Risk factors and comorbidities in Brazilian patients with orofacial clefts

Silva, Heglayne Pereira Vital da; Arruda, Thaynnan Thómaz Silva; Souza, Karla Simone Costa de; Bezerra, João Felipe; Leite, Gisele Correia Pacheco; Brito, Maria Edinilma Felinto de; Lima, Valéria Morgiana Gualberto Duarte Moreira; Luchessi, André Ducati; Bortolin, Raul Hernandes; Ururahy, Marcela Abbott Galvão; Rezende, Adriana Augusto de

doi:10.1590/1807-3107bor-2018.vol32.0024

Original Research, Community Dental Health • Braz. oral. res. 32 • 2018 • https://doi.org/10.1590/1807-3107bor-2018.vol32.0024 linkcopy

Risk factors and comorbidities in Brazilian patients with orofacial clefts

Authorship SCIMAGO INSTITUTIONS RANKINGS

Considering that environmental risk factors substantially contribute to the etiology of orofacial clefts and that knowledge about the characteristics and comorbidities associated with oral clefts is fundamental to promoting better quality of life, this study aimed to describe the risk factors, main characteristics, and comorbidities of a group of patients with cleft lip and/or cleft palate (CL/P) from Rio Grande do Norte (RN), Brazil. Data were obtained from 173 patients with CL/P using a form from the Brazilian database on Orofacial Clefts. Most patients were male with cleft lip and palate and had a normal size and weight at birth; presented few neonatal intercurrent events; and had anemia and respiratory and cardiovascular diseases as main associated comorbidities. They also required timely surgical rehabilitation and multidisciplinary care to stimulate their neuropsychomotor development. In addition, a high frequency of familial recurrence and of parental consanguinity was evidenced in the studied population, especially for the cleft lip and cleft palate type. Other relevant findings were the considerable maternal exposure to alcohol, infections, smoking, and hypertension, as well as low supplementation with vitamins and minerals and deliberate consumption of analgesics, antibiotics, and antihypertensives during pregnancy. Characterization of the CL/P patient profile is essential for the planning of health services and integration among the health professionals involved in the diagnosis and treatment of these malformations. Our results reinforce the need for additional research to confirm the association between environmental factors and the development of orofacial clefts.

Keywords:
Cleft Lip; Cleft Palate; Comorbidity; Risk Factors; Environmental Exposure

Variable			n = 173	%	p-value
Type of cleft
	Cleft lip and palate (CLP)		81	46.8	< 0.001
	Cleft palate (CP)		61	35.2
	Cleft lip (CL)		31	17.9
Severity (CLP and CL)
	Unilateral		81	72.3	< 0.001
	Bilateral		30	26.8
	Midline		1	0.9
Gender
	Male		104	60.1	0.008
	Female		69	39.9	0.008
Birth weight
	< 2,500 g		26	15.0	< 0.001
	25,00 – 3,999 g		122	70.5
	≥ 4,000 g		13	7.5
	Missing data		12	6.9
Birth length
	Boys
		< 46.1 cm	7	6.7	< 0.001
		46.1 – 53.7 cm	76	73.1
		> 53.7 cm	3	2.9
		Missing data	18	17.3
	Girls
		< 45.4 cm	11	15.9	< 0.001
		45.4 – 52.9 cm	41	59.4
		> 52.9 cm	6	8.7
		Missing data	11	15.9
Head circumference at birth
	Boys
		< 31.9 cm	4	3.8	< 0.001
		31.9 – 37.0 cm	54	51.9
		> 37.0 cm	1	0.9
		Missing data	45	43.2
	Girls
		< 31.5 cm	5	7.2	< 0.001
		31.5 – 36.2 cm	32	46.4
		> 36.2 cm	4	5.8
		Missing data	28	40.6
Neonatal intercurrent events			43a	24.9a
	Icterus		25	14.5	< 0.001
	Respiratory distress		23	13.3
	Intubation and mechanical ventilation		9	5.2
	Infections		5	2.9
	Other		13	7.5
	None		93	53.8
	Missing data		37	21.4
Comorbidities			79b	45.7b
	Anemia		28	16.2	< 0.001
	Respiratory diseases		21	12.1
	Cardiovascular diseases		17	9.8
	Neurological diseases		14	8.1
	Otitis		13	7.5
	Hearing loss		10	5.8
	Pneumonia		9	5.2
	Gastrointestinal diseases		6	3.5
	Other		20	11.6
	None		70	40.5
	Missing data		24	13.9
Neuropsychomotor development
	Normal for age		82	47.4	< 0.001
	Motor, speech and behavioral delays and NPMDc		14	8.1
	Speech delay only		12	6.9
	Motor and speech delay		5	2.9
	Motor delay only		4	2.3
	Speech and behavior delay		3	1.7
	Behavioral delay only		1	0.6
	Not applicable		27	15.6
	Missing data		25	14.5
Therapy
	No		87	50.3	0.002
	Yes		51	29.5
	Missing data		35	20.2
Type of therapy
	Speech Therapy		41	23.7	< 0.001
	Physical therapy		8	4.6
	Psychology		5	2.9
	Occupational therapy		4	2.3
	Other		4	2.3
Syndrome
	Nonsyndromic		119	68.8	< 0.001
	Syndromic		47	27.2
	Not classified		7	4.0

Variable			n = 173	%	p-value
Age at first surgical repair
	Cheiloplasty				0.001
		< 6 months old	18	26.9
		6–12 months old	36	53.7
		13 month–5 years old	13	19.4
		> 5 years old	0	0.0
		Total	67	100
	Palatoplasty				< 0.001
		< 1 year old	6	12.2
		1–2 years old	23	46.9
		2–5 years old	16	32.7
		> 5 years old	4	8.2
		Total	49	100
Delay in surgical repair
	Cheiloplasty				0.009
		Waiting for surgery and on time	9	27.3
		Waiting for surgery and delay	24	72.7
		Total	33	100
	Palatoplasty				0.113
		Waiting for surgery and on time	32	41.0
		Waiting for surgery and delay	46	59.0
		Total	78	100

Variable		n = 173	%	p-value
Paternal age at pregnancy (years)
	< 20	12	6.9	< 0.001
	20-30	80	46.2
	31-35	31	17.9
	> 35	28	16.2
	No information	22	12.7
Maternal age at pregnancy (years)
	< 20	28	16.2	< 0.001
	20–30	95	54.9
	31–35	32	18.5
	> 35	18	10.4
Paternal education level
	Illiterate	9	5.2	< 0.001
	Literate	4	2.3
	Elementary School	42	24.3
	Middle School	42	24.3
	High School	48	27.7
	College	9	5.2
	Missing data	19	11.0
Maternal education level
	Illiterate	2	1.2	< 0.001
	Literate	2	1.2
	Elementary School	40	23.1
	Middle School	46	26.6
	High School	69	39.9
	College	14	8.1
Maternal occupation during pregnancy
	Homemaker	89	51.4	< 0.001
	Salesperson	16	9.2
	Housekeeper	15	8.7
	Teacher	9	5.2
	Farmer	7	4.0
	Cook or waitress	7	4.0
	Student	6	3.5
	Industrial worker	5	2.9
	Administrative assistants	3	1.7
	Other	16	9.2
Parental consanguinity
	No	159	91.9	< 0.001
	Yes	14	8.1	< 0.001
History of familial cleft
	No	105	60.7	0.005
	Yes	68	39.3	0.005

Variable		n = 173	%	p-value
Conception method
	Spontaneous	170	98.3	< 0.001
	Assisted	3	1.7	< 0.001
Type of Birth
	Vaginal	65	37.6	0.550
	Cesarean	72	41.6
	Missing data	36	20.8
Timing of birth
	Preterm	24	13.9	< 0.001
	Full-term	97	56.1
	Post-term	17	9.8
	Missing data	35	20.2
Number of pregnancies
	1	55	31.8	0.003
	2	56	32.4
	3	31	17.9
	> 03	31	17.9
Birth order
	1^st	70	40.5	< 0.001
	2^nd	51	29.5
	3^rd	26	15.0
	> 3^rd	26	15.0
Miscarriage
	No	143	82.7	< 0.001
	Yes	30	17.3	< 0.001
Termination of pregnancy attempts
	No	163	94.2	< 0.001
	Yes	8	4.6
	Missing data	2	1.2
Prenatal history
	Alcohol consumption	39	22.5	< 0.001
	Urinary tract infection	24	13.9
	Smoking	20	11.6
	Hypertension	19	11.0
	Bleeding	15	8.7
	Diabetes Mellitus	10	5.8
	Illegal drug use	7	4.0
	Other	30	17.3
Medications used during pregnancy
	Folic acid	42	24.3	< 0.001
	Iron	37	21.4
	Analgesic	30	17.3
	Antibiotic	29	16.8
	Antihypertensive	15	8.7
	Vitamin supplementation	12	6.9
	Anti-inflammatory	7	4.0
	Anti-abortion drug	6	3.5
	Antiemetic	4	2.3
	Hypoglycemic agent	3	1.7
	Other	22	12.7
	None	70	40.5
	Missing data	2	1.2

Variable		CLP	CP	CL	p-value
Gender
	Male	57 (32.9)	28 (16.2)	19 (11.0)	0.013
	Female	24 (13.9)	33 (19.1)	12 (6.9)	0.013
Paternal age at conception
	< 35 years old	55 (31.8)	37 (21.4)	24 (13.9)	0.476
	≥ 35 years old	16 (9.2)	15 (8.7)	5 (2.9)	0.476
Maternal age at conception
	< 35 years old	71 (41.0)	53 (30.6)	29 (16.8)	0.611
	≥ 35 years old	10 (5.8)	8 (4.6)	2 (1.2)	0.611
Parental consanguinity
	No	71 (41.0)	58 (3.5)	30 (17.3)	0.151
	Yes	10 (5.8)	3 (1.7)	1 (0.6)	0.151
Family history
	No	47 (27.2)	44 (25.4)	14 (8.1)	0.035
	Yes	34 (19.7)	17 (9.8)	17 (9.8)	0.035
Bleeding during pregnancy
	No	68 (39.3)	58 (33.5)	29 (16.8)	0.028
	Yes	12 (6.9)	2 (1.2)	1 (0.6)	0.028
Maternal alcohol consumption
	No	65 (37.6)	45 (26.0)	23 (13.3)	0.657
	Yes	16 (9.2)	16 (9.2)	7 (4.0)	0.657
Maternal smoking
	No	73 (42.2)	55 (31.8)	24 (13.9)	0.134
	Yes	8 (4.6)	6 (3.5)	6 (6.5)	0.134
Maternal illegal drug use
	No	77 (44.5)	59 (34.1)	29 (16.8)	0.862
	Yes	4 (2.3)	2 (1.2)	1 (0.6)	0.862
Gestational diabetes
	No	75 (43.4)	59 (34.1)	28 (16.2)	0.568
	Yes	6 (3.5)	2 (1.2)	2 (1.2)	0.568
Folic acid supplementation
	No	63 (36.4)	46 (26.6)	20 (11.6)	0.423
	Yes	17 (9.8)	15 (8.7)	10 (5.8)	0.423

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[TFN1] ^a
Total number of neonatal complications and frequency relative to the whole study group;

[TFN2] ^b
Total number of comorbidities and frequency relative to the whole study group;

[TFN3] ^c
NPMD, Neuropsychomotor developmental delay.