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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 6  |  Issue : 2  |  Page : 130-133

Ludwig's angina in pregnancy and puerperium: Case series in an academic hospital, Sokoto, Northwest Nigeria


1 Department of Dental and Maxillofacial Surgery, Usmanu Danfodiyo University Teaching Hospital, Sokoto, Nigeria
2 Department of Surgery/Dental and Maxillofacial Surgery, College of Health Sciences, Usmanu Danfodiyo University/Usmanu Danfodiyo University Teaching Hospital, Sokoto, Nigeria
3 Department of Obstetrics and Gynaecology, Usmanu Danfodiyo University/Usmanu Danfodiyo University Teaching Hospital, Sokoto, Nigeria

Date of Web Publication15-Sep-2017

Correspondence Address:
Ramat Oyebunmi Braimah
Department of Dental and Maxillofacial Surgery, Usmanu Danfodiyo University Teaching Hospital, Sokoto
Nigeria
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/sjhs.sjhs_17_17

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  Abstract 

Ludwig's angina (LA) otherwise known as angina ludovici is a potentially life-threatening diffuse cellulitis of the neck, the floor of the mouth and submandibular regions bilaterally leading to airway obstruction. This condition during pregnancy and puerperium is rare. Many physiological changes occur through pregnancy and puerperium that place the mother at a higher risk of infection or deteriorating condition once infected. This change includes, reduced immune response resulting in potential faster progression of an infection. We report two cases of LA in third trimester of pregnancy and two cases during peurperium within 5 months at the Departments of Dental and Maxillofacial Surgery and Obstetrics and Gynaecology, Usmanu Danfodiyo University Teaching Hospital, Sokoto, Nigeria. Treatment modalities and challenges involved in their management were also discussed. Overall, three patients survived the disease, two cases of LA in pregnancy and one case of LA in the puerperium, one patient in the puerperium died due to overwhelming sepsis. The babies of both patients in the puerperium survived, although they came in with very low birth weight and failure to thrive. They were managed in the special care baby unit of the hospital. Management of LA in a resource and personnel scarce setting is demanding especially in special group of patients such as pregnancy and puerperium. Multidisciplinary approach in management is essential, especially in the antenatal period of the patient.

Keywords: Antenatal, Ludwig's angina, nutrition, pregnancy, puerperium


How to cite this article:
Braimah RO, Ibikunle AA, Taiwo AO, Tunau K. Ludwig's angina in pregnancy and puerperium: Case series in an academic hospital, Sokoto, Northwest Nigeria. Saudi J Health Sci 2017;6:130-3

How to cite this URL:
Braimah RO, Ibikunle AA, Taiwo AO, Tunau K. Ludwig's angina in pregnancy and puerperium: Case series in an academic hospital, Sokoto, Northwest Nigeria. Saudi J Health Sci [serial online] 2017 [cited 2019 May 25];6:130-3. Available from: http://www.saudijhealthsci.org/text.asp?2017/6/2/130/214845


  Introduction Top


Ludwig's angina (LA) is a diffuse cellulites involving the submandibular, sublingual, and submental spaces bilaterally with increasing of the floor of the mouth leading to airway obstruction.[1] It is potentially life-threatening unless it is recognized early and aggressively treated.[1] LA results mainly from odontogenic sources, especially sequelae of caries and periodontal infections. This condition during pregnancy and puerperium is rare. Kathleen andMaryam [2] from Michigan, USA; in 2010 reported two cases of LA in pregnancy. In Northwest Nigeria, Osunde et al.[3] have reported 10 cases of LA in pregnancy within 2 years. To the best of our knowledge, no case of LA has been reported during puerperium. We report two cases of LA in the third trimester of pregnancy and two cases during peurperium.


  Case Reports Top


Ludwig's angina in pregnancy

Cases 1 and 2

A 31-year-old Gravida 5 Para 4 at 34 weeks gestation patient (case 1) and a 28-year-old housewife, Gravida 3 Para 1 at 30 weeks gestation (case 2), both unbooked, presented in our facility (3 months interval) with 1-week history of lower jaw swelling, associated difficulty in eating, breathing, and fever. There were previous 2 weeks and 1 week history of toothache, respectively, in the lower right quadrant. Both patients presented with a temperature >38°C. Examination in both patients revealed diffuse lower jaw swelling involving submandibular region bilaterally and the submental space [Figure 1] and [Figure 2], respectively]. The swelling was brawny hard, tense, tender with raised local temperature. A diagnosis of LA in pregnancy was made in both patients by supervising consultants. Packed cell volumes were 23% and 20%, respectively. They were immediately placed on intravenous (IV) ceftriazone 1 g 12 h, metronidazole 400 mg 8 h for 1 week, and IV paracetamol 600 mg 8 h for 3 days and IV hydrocortisone 100 mg 12 h for 3 days to improve their breathing.
Figure 1: Clinical photograph of patient (case 1) showing the pregnancy with bilateral submandibular swelling and raised floor of the mouth with limited mouth opening

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Figure 2: Clinical photograph of patient (case 2) with bilateral submandibular swelling and raised floor of the mouth with limited mouth opening

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Both patients had incision and decompression under local anesthesia. They were nursed in the Ante natal ward and both requested discharge before delivery.

Ludwig's angina in puerperium

Cases 3 and 4

A 30-year-old Gravida 5 Para 3 homemaker with a 4-day-old baby was referred from Federal Medical Centre on account of 1 week history of facial swelling. There was the previous history of toothache and swelling throughout the history of pregnancy. She had spontenous vagianal delivering at home. The weight of the baby was 1.5 kg (case 1) [Figure 3]a. Another patient, a 35-year-old Gravida 4 para 4 homemaker with a 1-week-old neonate presented (2 months after case 1) with 5 days history of facial and neck swelling that was preceded by 1-week history of toothache. The pain was associated with a headache and fever. She went into labour during this period of a toothache and neck swelling and put to bed at home through spontaneous vaginal delivery. The weight of the baby at presentation was 1.8 kg (case 2) [Figure 4]a. Both babies were transferred to the special care baby unit (SCBU) for management. Both mothers presented with respiratory rates >28 cpm and temperature >38°C. There was bilateral submandibular and submental diffuse swelling in both patients with left infra-temporal space extension in Case 2. Packed cell volumes were 20% and 25%, respectively. Incision and decompression with the extraction of offending teeth were done in both patients under local anesthesia. Both patients were placed on IV ceftraizone 1 g 12 h and IV metronidazole 500 mg 8 h for 5 days. The disease condition was complicated by necrotizing fasciitis (NF) [Figure 3]b with worsening clinical condition and bilateral pitting pedal edema up to the knee in case 3. Medication was subsequently changed to rocephin 2 g 12 h. However, patient did not procure them due to financial reasons. She passed away 5 days postadmission. Case 4 also had NF [Figure 4]b and daily dressing continued with no further events.
Figure 3:(a) Clinical photograph of patient (case 3) with bilateral submandibular swelling with raised tongue. (b) Clinical photograph of patient (case 3) with necrotizing fasciitis

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Figure 4: (a) Clinical photograph of patient (case 4) with bilateral submandibular swelling and infraorbital spread. (b) Clinical photograph of patient (case 4) with necrotizing fasciitis

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  Discussion Top


Many physiological changes occur through pregnancy and puerperium that place the mother at a higher risk of infection or deteriorating condition once infected. The peuperium has been defined by the WHO as the period between when the woman immediately delivers and the return of the uterus to its normal size lasting approximately 6 weeks. It has also been described as the most critical and yet the most neglected phase in the lives of mothers and babies; most deaths occur during this period.[4] This change include reduced immune response resulting in potential faster progression of an infection.[5] Furthermore, there is reduced neutrophil chemotaxis, cell mediated immunity, and natural killer cell activity.[5] It has been reported that approximately 50% of pregnant women complain of dyspnea by 19 weeks of conception with depletion in the oxygen reserve.[5] This lower oxygen reserve could increase fetal hypoxia during periods of hypoventilation.[5] A pregnant patient with LA further worsens this hypoxic condition. Maxillofacial infection in pregnancy or immediate delivery period requires special attention because of the physiologic changes that occur that put the patient at increased risk. Such changes include increased sodium retention, dilutional anemia, decreased colloid pressure, and capillary engorgement of the upper airway and subsequent edema of the nasal passages, larynx, oropharynx, and trachea.[2] It has also been reported that preterm birth, low birth weight, fetal growth restriction, preeclampsia, and perinatal mortality are associated with periodontal disease.[2] Two of our cases in the puerperiun had low birth weight (case 2, 2 kg and case 4, 1.8 kg) babies with failure to thrive. The two babies were managed in the SCBU of our facility. The etiology of LA in all our cases was pericoronitis with sequelae of caries on the last molars. This combination insult has been identified as the main etiology of LA.[6] In Northwest Nigeria, tooth mortality from dental caries and its sequelae is higher than that reported from other regions in Nigeria.[7] This is due to low dental public awareness, poor accessibility to limited dental facilities from the rural communities and lack of preventive oral health measures.[7]

Both the physiologic changes of pregnancy and the perinatal effects of the treatment must be considered in the management of LA. Our management protocol includes incision and drainage under local anesthesia, empirical antibiotics with ceftriazone and metronidazole. IV hydrocortisone 100 mg 12 h was added to improve respiration. Opioid analgesics and other agents with sedative effects are avoided due to the potential risk to the fetus; therefore, only IV paracetamol 600 mg was given. None of the patients required tracheostomy or any form of a surgical airway. This is in agreement with reported cases of LA in pregnancy.[2],[3],[5] All our patients had surgical decompression under local anesthesia with cervical plexus block and inferior alveoli nerve block using 2% lignocaine!: 100,000 adrenaline. This technique has been reported to have low complication rate and high patient acceptance in the drainage of submandibular and submental abscesses.[8]

The two cases in the peurperium were also managed in cardiac position to prevent airway compromise. We observed that the patients in the peurperium had NF as a complication. The reason for this is still obscure; however, we opined that they have just gone through the process of childbirth which will further compromise their immunity. Furthermore, harmful cultural practices could also have contributed to low immunity. It has been reported that poor nutrition, heavy work, inability to make decisions, incorrect information on health services, and inadequate service delivery are some of the social factors which make women at risks, especially in pregnancy and peurperium.[9],[10] Similarly, there are some cultural norms that exert a strong influence on the nutritional intake of women in the pregnancy and postdelivery period.[9] One patient in this series (case 3) died due to severe sepsis that failed to respond to medication. Another possible cause of mortality, especially in pregnant patient is anemia. Osunde et al.[3] from their series have identified a significant difference between the mean basal hematocrit value in pregnant patients who survived LA and those that died.


  Conclusion Top


Management of LA in a resource and personnel scarce setting is demanding, especially in a special group of patients like pregnancy and puerperium. Multidisciplinary approach in management and prevention is essential principally during antenatal care of the patient.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Hasan W, Leonard D, Russell J. Ludwig's angina-A controversial surgical emergency: How we do it. Int J Otolaryngol 2011;2011:231816.  Back to cited text no. 1
[PUBMED]    
2.
Kathleen M, Maryam G. Case Report Pregnancy Complicated by Ludwig's Angina Requiring Delivery. Infectious Diseases in Obstetrics and Gynaecology Volume 2010, doi: 10.1155/2010/158264.  Back to cited text no. 2
    
3.
Osunde O, Bassey G, Ver-Or N. Management of Ludwig's angina in pregnancy: A review of 10 cases. Ann Med Health Sci Res 2014;4:361-4.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
WHO. WHO recommendations on postnatal care of the mother and newborn. Geneva: WHO library Cataloguing-in-Publication Data; 2013. p. 62.  Back to cited text no. 4
    
5.
Patil AB, Anuranjani L. A rare case of pregnancy with Ludwig's angina. Int J Health Sci Res 2014;4:248-53.  Back to cited text no. 5
    
6.
Braimah RO, Taiwo OA, Ibikunle AA. Ludwig's angina: Analysis of 28 cases seen and managed in Sokoto Northwest Nigeria. Saudi Surg J 2016;4:77-83.  Back to cited text no. 6
  [Full text]  
7.
Taiwo OA, Sulaiman OA, Shoremi OO, Danlami J, Adeniji UO, Olawole OW. Pattern and indications for adult permanent teeth extraction in Zamfara State, Noertwest Nigeria. J Stomatol 2015;68:183-90.  Back to cited text no. 7
    
8.
Ling KU, Hasan MS, Ha KO, Wang CY. Superficial cervical plexus block combined with auriculotemporal nerve block for drainage of dental abscess in adults with difficult airways. Anaesth Intensive Care 2009;37:124-6.  Back to cited text no. 8
[PUBMED]    
9.
Robert NS. An impact of socio-cultural practices on maternal mortality in Masasi District, Tanzania. Malays J Med Biol Res 2015;2:211-20.  Back to cited text no. 9
    
10.
Hughes P, Bradrick JP, Yowler CJ. Nutrition for the oral and maxillofacial surgery patient. In: Fonseca RJ, Barber HD, Walker RV, Powers MP, Frost DE, editors. Oral and Maxillofacial Trauma. 4th ed. St. Louis, Missouri: Elsevier Saunders; 2013. p. 30-47.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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