Literature DB >> 33780490

Referral pattern of oral and maxillofacial surgery cases in Sudan: A retrospective age-and sex-specific analysis of 3,478 patients over four years.

Musadak Ali Karrar Osman1, Mohammed Hassan Ibrahem Aljezoli2, Mohamed Alfatih Mohamed Alsadig3, Ahmed Mohamed Suliman4.   

Abstract

Oral and maxillofacial surgery (OMFS) is a specialty widening in its scope. An objective analysis of the referral pattern can provide essential information to improve healthcare. This four-year retrospective study was implemented in Khartoum Teaching Dental Hospital. Data (age, sex, diagnosis, and type of treatment) were collected from patient records. Disease frequency, as well as the effect of sex and age, were analyzed for each group. The frequency of treatment types was also assessed. Data were collected from a total of 3,478 patients over the four-year study period. There was a male predominance with the third decade of life being the most common age group. Pathological diseases were the most common (37%) reason for referral, followed by trauma (31%). Temporomandibular joint (TMJ) disorders and dentoalveolar extraction were the least frequently observed. Open reduction and internal fixation (ORIF) was the most commonly performed procedure (28%). These data represent the epidemiology of oral and maxillofacial diseases in Sudan. Given that the third decade of life is the most represented age group, it is beneficial to learn the long-term consequences of these diseases in these young patients and to use modern surgical techniques to improve their lives.

Entities:  

Year:  2021        PMID: 33780490      PMCID: PMC8007037          DOI: 10.1371/journal.pone.0249140

Source DB:  PubMed          Journal:  PLoS One        ISSN: 1932-6203            Impact factor:   3.240


Introduction

Oral and maxillofacial surgery (OMFS) is a specialty that is expanding in scope [1]. Despite the high number of services offered by OMFS specialists, few reports explore the overall characteristics of patients undergoing oral and maxillofacial procedures[2]. Most published articles subjectively assess the scope of OMFS practices [3] and focus on a particular type of service [2,4]. Overall, the lack of knowledge in this area limits health institutions’ ability to provide better services and to improve their infrastructure. Thus, the importance of an epidemiological study to enhance the health care system is evident [5]. The department of OMFS in Khartoum Teaching Dental Hospital (KTDH) receives referrals from all parts of Sudan and treats a nondefined population. A lack of epidemiological research in oral and maxillofacial diseases in Sudan makes it a favorable location for such a study. Not only is the literature deficient, but little is known about the regional variations in the epidemiology of surgical diseases [6]. The present study aims to assess the prevalence of diseases diagnosed in a cohort of patients visiting an OMFS service in Sudan over four years, the pattern and distribution of OMFS diagnoses in relation to age and sex, and the type of treatments provided. The prevalence of specific disease groups can help to quantify the scope of OMFS surgery in Sudan, thus increasing awareness of the training needed to competently perform surgery.

Material and methods

This is a retrospective study of the OMFS unit in KTDH. Patients attended the outpatient clinic between January 1, 2011, and December 31, 2014. Data were collected anonymously using patient records. All patients were categorized based on their age, sex, and diagnosis, as well as type of treatment. The diagnoses were coded according to the International Classification of Diseases (ICD-10). Then, the diagnosis was classified into seven major groups: pathology, trauma, infection, developmental defect, Temporomandibular joint (TMJ) disorders, abnormalities of teeth, and other. Other included diseases where nonsurgical treatment is the primary or only approach. Then, and whenever possible, Neville BW et al.’s textbook of Oral and Maxillofacial Pathology [7] nomenclature was adopted for individual diagnoses as well as for classification and subclassification, with minor modifications as necessary to avoid overlap (Table 1). Guided by the type of intervention, the treatment type was classified into 11 clinical categories.
Table 1

The diagnosis groups, classification and subclassification of the study.

1. Pathologya. Benign    Odontogenic    Salivary gland    Soft tissue    Bone pathology    Hematologyb. Malignant    Odontogenic    Salivary gland    Epithelium    Soft tissue    Bone pathology    Hematologyc. Cyst    Odontogenic    Salivary glandN    onodontogenicd. Inflammatory
2. Trauma
3. Infection. Odontogenic infection. Nonodontogenic infection
4. Developmental defect. Cleft deformity. Skeletal deformity. Inherited and syndromic disorder. Soft tissue deformity
5. Temporomandibular joint disorders
6. Abnormalities of teeth
7. Other. Inflammatory. Autoimmune disorder. Facial pain and neuromuscular diseases. Salivary gland. Epithelium. Bone pathology
Descriptive statistics were analyzed using SPSS version 24 (SPSS Inc., Chicago, IL, USA). The prevalence of disease groups, as well as the effect of age and sex, were evaluated. For comparison, the relevant existing literature was reviewed. Ethics approval was obtained from the KTDH research committee.

Result

Diseases

A total of 3,478 cases met the inclusion criteria. Of these, 63.1% (n = 2,196 cases) were male and 36.9% (n = 1,282) were female, with a ratio of 1.7:1. Age ranged from days to 95 years, with a mean age of 30.8 ± 20.2. The most common age group represented were those in the third decade of life 23.2% (n = 808), followed by the second decade 18.4% (n = 641) (Table 2). The mean age of males was 30.6 years and 31.2 for females, with no statistical significance difference (p = 0.4).
Table 2

Distribution of oral and maxillofacial diseases by age (years).

Disease groupsAgeTotal
0–910–1920–2930–3940–4950–5960–69≥ 70
Trauma611814032381116035191108
Developmental defect287124472010220492
Pathology1072332031501501441221471256
Temporomandibular joint disorders1440247541196
Infection1737754935221920274
Other721292728303122195
Abnormalities of teeth052715631057
Total493 (14.2%)641 (18.4%)808 (23.2%)506 (14.5%)345 (9.9%)265 (7.6%)211 (6.1%)209 (6.0%)3478

Data are shown as frequency (percentage).

P-value = 0.001.

Data are shown as frequency (percentage). P-value = 0.001. In accordance with ICD-10, there were 11 different clinical categories (Table 3).
Table 3

Diagnosis distribution by ICD-10 classification.

ICD-10 classificationFrequencyPercentage
1. Injury, poisoning and certain other consequences of external causes111432.0%
2. Diseases of the digestive system76021.9%
3. Congenital malformations, deformations and chromosomal abnormalities47213.6%
4. Benign neoplasms43812.6%
5. Malignant neoplasms40511.6%
6. Diseases of the musculoskeletal system and connective tissue1163.3%
7. Diseases of the nervous system651.9%
8. Diseases of the skin and subcutaneous tissue511.5%
9. Certain infectious and parasitic diseases280.8%
10. Diseases of the respiratory system220.6%
11. External causes of morbidity and mortality70.2%
Total3478100.0%
The frequency of diagnosis groups is indicated in Fig 1. Except for the group “other” and “abnormality of teeth” males were more affected than females. Moreover, the discrepancy was obvious in the trauma group (Fig 2).
Fig 1

Distribution (number and percentage) of the diagnosis groups.

Fig 2

Distribution (number) of disease groups by sex.

Regarding age, all diseases occurred at the third decade of life or earlier, except those grouped as “other,” for which the sixth decade of life was most prevalent (Table 2).

Pathologies

Diagnosed pathologies were the most common reason for patient visits, 36.1% (n = 1,256) (Fig 1), with the classification, including subclasses and diseases diagnosed are indicated in Fig 3 and Table 4, respectively.
Fig 3

Distribution (number and percentage) of pathological disease classification.

Table 4

Frequency of 1256 pathological diseases presented as class, subclass, and individual diagnosis.

Benign: 441 (35.1%)FrequencyPercentage within subclass
Odontogenic: 146 cases
Ameloblastoma11075%
Odontoma1913%
Odontogenic myxoma107%
Calcifying epithelial odontogenic tumor43%
Ameloblastic fibroma11%
Squamous odontogenic tumor11%
Ameloblastic fibro-odontoma11%
Salivary gland: 80 cases
Pleomorphic adenoma7695%
monomorphic adenoma34%
Myoepithelioma11%
Soft tissue: 104 cases
Hemangioma2524%
Pyogenic granuloma1918%
Lipoma1615%
Vascular malformation1514%
Lymphangioma1010%
Fibroma66%
Neurofibroma55%
Fibroepithelial polyp22%
Peripheral giant cell granuloma11%
Schwannoma11%
Angiofibroma11%
Fibromatosis11%
Solitary fibrous tumor22%
Bone pathology: 110 cases
Ossifying fibroma5045%
Florid cemento-osseous dysplasia2018%
Fibrous dysplasia2018%
Central giant cell granuloma1312%
Chondroma55%
Brown tumor22%
Hematology: 1 case
Eosinophilic granuloma1100%
Malignant: 405(32.2%)
Odontogenic: 4 cases
Malignant ameloblastoma125%
Ghost cell odontogenic carcinoma125%
Ameloblastic carcinoma125%
Malignant odontogenic myxoma125%
Salivary gland:61 cases
Mucoepidermoid carcinoma2338%
Adenoid cystic carcinoma1626%
Adenocarcinoma, not otherwise specified (NOS)1525%
Acinic cell adenocarcinoma58%
Basal cell adenocarcinoma12%
Myoepithelial carcinoma12%
Epithelium: 304 cases
Squamous cell carcinoma27490%
Verrucous carcinoma134%
Clear cell carcinoma52%
Malignant melanoma52%
Basal cell carcinoma41%
Nasopharyngeal carcinoma31%
Soft tissue: 16 cases
Fibrosarcoma744%
Metastatic lesion319%
Rhabdomyosarcoma213%
Malignant fibrous histiocytoma16%
Hemangioendothelioma16%
Liposarcoma16%
Angiosarcoma16%
Bone pathology: 8 cases
Ewing’s sarcoma450%
Osteosarcoma225%
Chondrosarcoma225%
Hematology: 12 cases
Lymphoma1192%
Leukemia18%
Cyst: 372(29.6%)
Odontogenic: 171 cases
Dentigerous cyst6639%
Radicular cyst5432%
Odontogenic keratocyst4426%
Lateral periodontal cyst42%
Calcifying odontogenic cyst32%
Salivary gland:129 cases
Ranula11690%
Mucocele1310%
Nonodontogenic: 72 cases
Nasopalatine cyst4360%
Dermoid/epidermoid cyst1115%
Globulomaxillary cyst710%
Thyroglossal cyst57%
Median palatine cyst34%
Aneurysmal bone cyst23%
Lymphoepithelial cyst11%
Inflammatory: 29(2.3%)
Salivary gland stone2897%
Necrotizing sialometaplasia13%
Premalignant diseases: 9 (0.7%)
Leukoplakia/ Erythroplakia9100%
Both sex and age groups had a significant relationship in terms of classification distribution (Table 5). Males were more affected than females, and the second decade of life was the most affected age group. Regarding sex, males had more malignant diseases, while females had more benign diseases. Regarding age groups, the most common pathology was cystic diseases in the first or second decade of life. In contrast, the third and fourth decades of life featured a higher frequency of benign neoplasms. From the fifth decade of life and onwards, the most common diseases were malignancies (Table 5).
Table 5

Pathological disease groups distribution by age (years) and sex.

Pathological diseases groupsAgeSexTotal
0–910–1920–2930–3940–4950–5960–69≥ 70MaleFemale
Benign33921017553421926197244441
Malignant10102425538587111244161405
Inflammatory040511612151429
Cystic6412777453110117191181372
Premalignant diseases00102141729
Total107 (8.5%)233 (18.6%)203 (16.2%)150 (11.9%)150 (11.9%)144 (11.5%)122 (9.7%)147 (11.7%)654 (52.1%)602 (47.9%)1256

Data are shown as frequency (percentage).

P-value = 0.001.

Data are shown as frequency (percentage). P-value = 0.001.

Benign

The most common class was benign diseases 35.1% (n = 441). The odontogenic tumor was the most common frequent subgroup 33.3% (n = 147), followed by bone pathology in the form of nonodontogenic tumor 24.9% (n = 110 cases). Ameloblastoma was the most common individual diagnosis 75% (n = 110) (Table 4). Pleomorphic adenoma was the second most common individual diagnosis with 76 cases, representing 95% of benign salivary gland neoplasms (Table 4). The third decade of life was the most involved age group 22.9% (n = 101), and females were the predominant sex 55.3% (n = 244) (Table 5).

Malignant

Malignant diseases were the second most common pathological diagnosis 32.2% (n = 405). Squamous cell carcinoma was the most common individual diagnosis with 272 cases, representing 90.1% of malignant epithelial neoplasms. Mucoepidermoid carcinoma was the second most common individual malignant diseases 38% (n = 23) regarding malignant salivary gland neoplasms (Table 4). This type of neoplasm rarely occurs before the fifth decade of life, but it drastically increases in incidence during the subsequent decade, and males are the predominant sex 60.2% (n = 244) (Table 5).

Cystic

A total of 372 cases (29.6%) were diagnosed with cysts. Of these, 45.9% (n = 171) were odontogenic cysts, 34.6% 9 (n = 129) were sialoceles, and 19.3% (n = 72) were nonodontogenic cysts. Among odontogenic cysts, dentigerous cysts were the most common 39% (n = 66), while nasopalatine cysts were the most common nonodontogenic cyst 60% (n = 43) (Table 4). This type of pathology occurs more frequently during the first three decades of life and rarely occurs in older age groups. Males show slightly higher frequency 51.3% (n = 191) 51.3% (Table 5) The frequency of premalignant diseases, as well as their distribution among age groups and sex, are indicated in Tables 4 and 5, respectively.

Trauma

Trauma was the second most common class 31.8% (n = 1108) (Fig 1). Males were significantly more involved 85.8% (n = 951) with the third decade of life being the most represented age group 36.4% (n = 403) (Table 2).

Developmental defects

Developmental defects represented 14.1% (n = 492) of the study sample (Fig 1). The most common defect was clefts 86.6% (n = 426), with a predominance of cleft lips noted 46.4% (n = 197), followed by cleft lips and palates 28.2% (n = 120) and cleft palates 25.4% (n = 108), (Table 6).
Table 6

Frequency of 492 developmental defects.

Cleft deformity: 426 casesFrequencyPercentage within subclass
Cleft lip19846%
Cleft lip and palate12028%
Cleft palate10825%
Skeletal deformity: 24 cases
Class II malocclusion938%
Condylar hyperplasia729%
Class III mal occlusion313%
Hemifacial hyperplasia28%
Severe skeletal anterior open bite28%
Mandible retrognathia with bifid tongue and cleft mandible14%
Inherited and syndromic disorder: 10 cases
Osteopetrosis330%
Cherubism220%
Gorlin syndrome110%
Melkersson–Rosenthal syndrome110%
Osteogenesis imperfecta110%
Ectodermal dysplasia110%
Xeroderma pigmentosa110%
Soft tissue deformity: 32 cases
Ankyloglossia2888%
Double lip39%
Geographic tongue13%
Among all cleft cases, only 61% (n = 260) were treated during the first decade of life (Table 7). Cleft palates were more frequent in females 52.8% (n = 57), while cleft lips and palates and cleft lips were more predominant in males 54.2% (n = 65), and 60.1% (n = 19) respectively (Table 7).
Table 7

Frequency and cleft subclassification Distribution by age group and sex.

Cleft subclassificationAgeSexTotal
0–910–1920–2930–3940–4950–5960–69MaleFemale
Cleft lip1135715841011979198
Cleft Lip and palate8523723006555120
Cleft Palate62281321115157108
Total260 (61.0%)108 (25.4%)35 (8.2%)12 (2.8%)8 (1.9%)2 (0.5%)1 (0.2%)235 (55.2%)191 (44.8%)426

Data are shown as frequency (percentage).

P-value (age) = 0.23.

P-value (sex) = 0.09.

Data are shown as frequency (percentage). P-value (age) = 0.23. P-value (sex) = 0.09.

Infections

Over the study period, a total of 7.9% (n = 274) of patients were affected by orofacial infections. Of these, 78.8% (n = 216) and 21.1% (n = 58) were odontogenic and nonodontogenic in origin, respectively (Table 8). The most affected age group were those in their third decade of life with 28.2% (n = 61) and 24.1% (n = 14) being odontogenic and nonodontogenic, respectively (p-value = 0.03).
Table 8

Frequency of 274 infections.

Odontogenic infection: 216FrequencyPercentage within subclass
Single space5325%
Osteomyelitis5023%
multiple/deep space4923%
Ludwig’s angina4521%
Necrotizing fasciitis199%
Nonodontogenic infection: 58 cases
Infected plate1831%
Tuberculous cervical lymphadenitis610%
Mumps59%
Leishmaniasis47%
Aspergillosis47%
Cancrum oris35%
Osteoradionecrosis35%
Tuberculous osteomyelitis23%
Candidiasis23%
Actinomycosis23%
Carbuncle12%
Viral warts12%
HIV12%
Histoplasmosis12%
Herpetic gingivostomatitis12%
Oral warts12%
Syphilitic ulcer12%
Squamous papilloma12%
Shingle12%
In our sample, females represented 50.5% (n = 109) of odontogenic infections and males represented 55.1% (n = 32) of nonodontogenic infections; however, the sex difference was not statistically significant (p-value = 0.27).

TMJ disorders

Fig 4 illustrates the frequency of individual TMJ disorders, corresponding to a total of 2.8% (n = 96) of the study sample. The most frequent diagnosis was ankylosis 82.3% (n = 79).
Fig 4

Distribution (number and percentage) of temporomandibular joint disorders.

The second decade of life was the most represented age group for ankylosis as well as dislocation deformities with 45.6% (n = 36) and 28.5% (n = 4), respectively (p-value = 0.001). There was a female predominance in dislocation 57.1% (n = 8) and internal derangement 100% (n = 3), whereas ankylosis demonstrated a male predominance 68.3% (n = 54); p-value = 0.015.

Abnormalities of teeth

A 1.6% (n = 57) accounted for impacted teeth which indicated for extraction under general anesthesia (Fig 1). The effects of sex and age are indicated in Fig 2 and Table 2.

Other

Table 9 shows the distribution of the “other oral diseases” class. The effects of sex and age are indicated in Table 10.
Table 9

Frequency of 195 other class diseases.

Facial Pain and Neuromuscular Diseases: 92 casesFrequencyPercentage
Trigeminal neuralgia3538%
Myofascial pain2325%
Facial palsy1820%
Atypical facial pain1011%
Massetric hypertrophy33%
Burning mouth syndrome33%
Autoimmune disorder: 59 cases
Erythema multiform1729%
Vesiculobullous lesions1627%
Sjögren syndrome915%
Aphthous ulcer814%
Lichen planus58%
Stevens–Johnson syndrome23%
Angioedema12%
Erythema bullosa haemorrhagica12%
Salivary gland: 30 cases
Sialadenitis2273%
Mucositis13%
Cheilitis glandularis13%
Xerostomia310%
Sialorrhea13%
Frey’s syndrome13%
Sialosis13%
Epithelium: 13 cases
Heck’s disease646%
Eosinophilic traumatic ulcer431%
Squamous hyperplasia18%
Naevous18%
Frictional keratosis18%
Bone pathology: 1 case
Paget’s disease1100%
Table 10

Sex and age effect on other class.

Other class subclassificationAgeSexTotal
0–910–1920–2930–3940–4950–5960–69≥70MaleFemale
Inflammatory15443403131124
Autoimmune disorder11067131192253459
Facial pain and neuromuscular diseases33161511141713434992
Salivary gland01111011336
Epithelium2220013310313
Bone pathology00000010011
Total7 (3.6%)21 (10.8%)29 (14.9%)27 (13.8%)28 (14.4%)30 (15.4%)31 (15.9%)22 (11.3%)94 (48.2%)101 (51.8%)195

p-value (age) = 0.205.

p-value (sex) = 0.264.

p-value (age) = 0.205. p-value (sex) = 0.264.

Type of treatment

The most commonly performed surgery was open reduction and internal fixation (ORIF; 779 cases: 22.4%), followed by tumor excision ± neck dissection (694 cases: 20%). In fact, 83 neck dissections were recorded either alone or as part of another operation. Table 11 shows the frequency of each type of treatment. The category “others” includes surgeries performed in less than 1% of patient visits, namely the surgical removal of teeth, soft tissue repair, palate removal, and rarely orthognathic surgery, coronoidectomy, bone shaving, and relocation of the submandibular ducts. Only two patients refused treatment.
Table 11

Type of treatment.

ProcedureFrequencyPercentage
    1. Open Reduction and internal Fixation (ORIF)77922.4
    2. Tumor excision ± neck dissection69420.0
    3. Cleft lip/palate repair42612.2
    4. Conservative management/follow up3028.7
    5. Enucleation2547.3
    6. Incision & drainage/debridement/sequestrectomy2507.2
    7. Other1644.7
    8. Salivary gland excision2146.2
    9. Close reduction1915.5
    10. Temporomandibular arthroplasty962.8
    11. Biopsy1083.1
Total3478100

Discussion

Hospital-based studies are the closest to clinical practice and represent its standards [8]. The current study evaluates the scope of OMFS in Sudan. Pathological diseases accounted for most of the diagnoses (36.1%), followed by traumatic events (31.9%). The frequency of the diseases differs between studies; however, it fluctuates between trauma and pathological diseases [8,9], which depends on the scope of focus for each hospital as well as the study population. While hospitals with an elective procedural scope tend to have more developmental deformities and pathological diseases, trauma is more common in hospitals possessing an emergency scope [2,8,10]. Furthermore, studies focused on outpatient populations may differ compared to inpatient focused studies [11]. The OMFS department in KTDH is the biggest in Sudan and is a highly equipped maxillofacial center. Furthermore, it is the only department in Sudan that receives referrals for a nondefined population, distinguishing it from military and police hospitals that focus on their personnel or employees. Most patients were male, which aligns with previous reports [8,9,12]). Higher prevalence of tobacco use with subsequent malignancy and involvement in trauma may exhibit a sex norm pattern for males. Sex norms are defined as socially constructed roles, behaviors, activities, and attributes that a given society considers appropriate for men and women [13]. In contrast, Bezerra et al. [2] and Brennan et al. [4] report that patients are predominantly female. Age ranges from days to 95 years, with the mean age of 30.84 ± 20.17 years. The second and third decades of life were the most common age groups represented. The frequency of different diseases is somewhat determined by the age distribution of patients seen. While developmental defects and odontogenic tumors and cysts are more prevalent in younger age groups, squamous cell carcinoma is more prevalent in the elderly. Moreover, Ibikunle et al. [9] noted the skew distribution of age, and explained by inclusion of minor surgeries, for instances routine dental extraction. The most common pathological diagnoses were benign diseases, representing 33.9% of all diagnoses. Ameloblastoma was the most frequent diagnosis. This finding is in agreement with reports by Adebayo et al. [9] and Siriwardena et al [14], where benign diseases were assessed along with benign and odontogenic tumors, respectively, and they were found to be the most common among both categories. Pleomorphic adenoma was the second most common benign neoplasm and the most common among benign salivary gland neoplasms. This finding is consistent with Vargas et al.’s [15]. In terms of malignant diseases, oral squamous cell carcinoma (OSCC) is the most common diagnosis, with 272 cases (67%). In fact, if individual diagnoses are considered, OSCC represents the most common presentation after trauma. This high frequency rate can be explained by the results of a recent metanalysis findings, where OSCC significantly correlated to “toombak,” a local name of Sudanese smokeless tobacco [16]. Furthermore, in vitro, research indicates that toomback damages human oral epithelium DNA and leads to malignant transformation [17]. Knowing that, and the fact that approximately 45% of men aged 40 years or older use toombak [18], there is an urgent need for programs addressing early detection and prevention. Interestingly, there is no statistical relationship between oral cancer and snus, the Swedish smokeless tobacco. This disparity is correlated to higher concentration (100-fold) of N-nitrosamines, the most abundant carcinogens, in Sudanese toombak [18]. Moreover, snus has been suggested to play a harm-reduction role by providing an alternative to cigarettes and reducing the smoking incidence as well as the related mortality and morbidity [19,20]. Such a strategy may be considered as one preventive method. In this study, dentigerous cysts were the most observed odontogenic cysts, which is consistent with Butt et al [21]. In contrast, the most frequent odontogenic cyst reported in the literature is radicular cyst [22-24], which can be attributed to the fact that most patients treated for periapical cysts are treated as outpatients in minor-surgery departments and are rarely sent for histopathological examination. Furthermore, many opt for a nonsurgical endodontics approach and leave surgical intervention as the last option [25], and we may experience a low volume of such cases in the future. Although sialoceles are the most common individual diagnosis among cystic diseases in the present study, the authors have no justification for that, and it should be considered in further research. Maxillofacial injuries usually constitute a high workload for oral and maxillofacial surgeons [8]. In the present study, maxillofacial injury comprised 31.8% of all patients. A clear predominance (86%) of males is noted, and the third decade of life is the most represented age group. The same trend is indicated in other studies [26,27]. However, the sex difference is narrower in developed countries when compared to developing countries [28]. The fact that males are more likely to be involved in traffic accidents and interpersonal violence, this helps explain the higher frequency of trauma among males. Also, reckless driving behaviors and more physically active hobbies make young adults more likely to be involved in trauma [29,30]. Developmental defects represent 13.9% of all cases, with cleft deformity being the most common (87.6%). This prevalence confirms previous reports that the most common orofacial deformity is a cleft deformity [31]. In contrast, when including asymptomatic defects, fissured tongue, the cleft deformity frequency may decrease to 45% [11]. Generally, cleft lip with or without cleft palate (CL ± P) is believed to be etiologically related, while cleft palate (CP) is considered as a different category [7]. The importance of clinical presentations of the cleft lies in their inference regarding associated or potential defects. Many subjects with cleft lip and palate (CLP) had isolated defects, while most subjects with CP only had additional associated congenital anomalies [32]. Furthermore, CLP has the potential to develop skeletal malocclusion with a severity associated with the number of previous surgeries [33]. The pattern of cleft lip (CL; 46.4%), CLP (28.2%) and CP (25.4%) identified in the present study differs when compared to reports from Ethiopia, Hawaii, and Nigeria, where CLP occurred more frequently [32,34,35]. All reports indicate that CP occurs least often. However, the cleft deformity frequency reports from Sudan show inconsistent data. Ali et al. [36] reported the same trend as in the present study, and was a hospital-based study estimating that CLP was most frequent, and CL was the least frequent cleft deformity [37]. It is important to note that the lack of a national registry program for clefts makes pattern evaluation difficult in Sudan. Notably, 39% of cleft patients were older than 10 years. The same trend was reported from other developing countries [38]. A possible cause is low socioeconomic status and poor access to health facilities. However, further investigation is needed to determine the relevance of factors such as delays in treatment-seeking behaviors. In general, a male prevalence was noted in cleft deformity (55.2%). The same pattern was observed in Nigeria, Ethiopia, and Poland [39-41]. The frequency of CL ± P in males is higher when compared to females, whereas the frequency of CP alone is higher in females, consistent with the findings of Martelli et al. [42]. Another interesting observation is the low frequency of maxillofacial syndromic patients, which can be attributed to a lack of awareness of the role of the maxillofacial surgery specialty among the population and the medical community in the treatment of such lesions. Odontogenic infections are important because of their high incidence and associated morbidity [43] when compared to nonodontogenic infections. This kind of infection can spread to facial and deep neck spaces, can lead to cavernous sinus thrombosis, necrotizing fasciitis, descending mediastinitis, and can progress to sepsis [44,45]. Moreover, the incidence of deep neck space infection is reported to be significantly higher in patients with odontogenic infections when compared to nonodontogenic infections [46]. In the present study, the majority of infections originated from odontogenic sources, which is consistent with findings reported by Igoumenakis et al. [47]. Of these infections, only 24.5% were single space infections, whereas the rest were complicated cases—namely, cases of multiple spaces, Ludwig’s angina, and cervicofacial necrotizing fasciitis. Knowing that the severity of infections is correlated to the number and the site of the infected spaces [48], it is crucial to further study this type of patient and the reasons for late presentation. The distribution of orofacial infection was higher in the third decade of life, with 55 cases (20.8%) and near equal disruption between the sexes. The same trend was noted in other developing countries [49]. Diseases requiring TMJ surgeries are relatively rare, and the current study is no exception, with only 2.8% diagnosed. Kamalkumar et al. reported 1.2% TMJ surgical cases, and Islam et al. reported 15.1% TMJ ankylosis cases [8]. Most TMJ disorders in the study were TMJ ankylosis (82.3%), and TMJ dislocation represents only 14.6% of cases. However, as acute dislocation is usually treated in the outpatient department, the current estimate reflects only those cases that required further treatment after a failed outpatient intervention. The results of this study are consistent with another study from Sudan [50], which reported that ankylosis more commonly occurred in adolescents. The associated deformities are more complicated. The technical difficulties in surgery and the high relapse rate [51] mandate a surgical protocol for future audit to arrive at best practices for Sudanese patients. The low frequency of extractions under general anesthesia demonstrated in the present study does not reflect their actual number, as most extractions are performed in minor surgical departments and are not included in the referral clinics record. The most common treatment modality was ORIF (768 cases: 22.1%), followed by tumor excision ± neck dissection (694 cases: 19.9%), including both benign and malignant diseases. Given that most patients with malignancy showed up late [52], neck dissection was performed in at least 83 cases. Although fracture may be treated quickly, bone and soft tissue resections may need full oral rehabilitation to improve quality of life. Likewise, CLP and TMJ ankylosis in young patients may necessitate additional surgeries in the future, such as orthognathic surgery and TMJ prosthesis.

Conclusion

This is the first study presenting the overall prevalence and scope of OMFS in Sudan. Given that malignant diseases and trauma are frequently encountered, both can be addressed better by implementing preventive methods. Delayed care-seeking behaviors need more attention to identify causative factors and to support patients and their families. Furthermore, it provides a guide to improve provider education and to appropriately allocate health resources. However, it carries the limitation of a retrospective study in terms of inaccurate or incomplete data. Given the difficulties encountered during data collection, the using of a predesigned chart templates with each disease category may help in future research and auditing to improve the quality of OMFS Department services.

Dataset.

(XLS) Click here for additional data file. 22 Jan 2021 PONE-D-20-35051 Referral pattern of oral and maxillofacial surgery cases in Sudan: A retrospective age- and sex-specific analysis of 3,478 patients over four years PLOS ONE Dear Dr.  Osman Thank you for submitting your manuscript to PLOS ONE. After careful consideration, we feel that it has merit but does not fully meet PLOS ONE’s publication criteria as it currently stands. Therefore, we invite you to submit a revised version of the manuscript that addresses the points raised during the review process. Please submit your revised manuscript by Mar 08 2021 11:59PM. If you will need more time than this to complete your revisions, please reply to this message or contact the journal office at plosone@plos.org. When you're ready to submit your revision, log on to https://www.editorialmanager.com/pone/ and select the 'Submissions Needing Revision' folder to locate your manuscript file. 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