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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 35  |  Issue : 2  |  Page : 99-102

A rare and interesting case of wandering unilateral edema like a wave


1 Department of General Medicine, Geetanjali Medical College and Hospital, Udaipur, Rajasthan, India
2 Department of Community Medicine, Geetanjali Medical College and Hospital, Udaipur, Rajasthan, India

Date of Submission08-Jan-2021
Date of Acceptance18-Jan-2022
Date of Web Publication08-Jul-2022

Correspondence Address:
Dr. Navgeet Mathur
Department of General Medicine, Geetanjali Medical College and Hospital, Udaipur - 313 001, Rajasthan
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijaai.ijaai_2_21

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  Abstract 


This case report is about a 55-year-old female who presented with wandering unilateral edema in the sequence of the left breast (for 7 days) followed by left upper limb (for 1 month), followed by the left nape of the neck (for 1 day), followed by left scalp (for 1 day), followed by left-sided face (for 3 days), followed by right-sided face (for 2 days). Edema progressed from one site to another site in the above sequence with the overlapping period of only 12–24 h and later subsided completely from its previous site. In the patient's language edema was moving from site to site like a slow wave moving forward. The patient took multiple consultations but could not get any relief. The patient was evaluated according. Serum total eosinophil count found 3100/cumm (30.1% of total leucocyte count) and diagnosed with angioedema. The patient was completely recovered by steroid and antihistaminic. To the best of our knowledge, this is the first case reporting wandering unilateral edema like a wave.

Keywords: Allergy, edema, steroid


How to cite this article:
Mathur N, Mathur M. A rare and interesting case of wandering unilateral edema like a wave. Indian J Allergy Asthma Immunol 2021;35:99-102

How to cite this URL:
Mathur N, Mathur M. A rare and interesting case of wandering unilateral edema like a wave. Indian J Allergy Asthma Immunol [serial online] 2021 [cited 2022 Aug 8];35:99-102. Available from: https://www.ijaai.in/text.asp?2021/35/2/99/350076




  Introduction Top


Angioedema was first described by Milton in 1876. Mental stress was recognized as one of the exacerbation factors and it was termed as angioneurotic edema by Quinke in 1982. Etiology may be heredity or acquired. Pathogenesis is usually allergy due to different substances. Common areas involved in angioedema were the face, lips, tongue, larynx, pharynx, hands, feets, and genitalia. It may also involve mucous membranes.[1],[2],[3],[4] Laryngeal involvement may be dreadful. There is multiple differential diagnosis of edema. Early diagnosis and treatment may resolve angioedema completely without any dreadful situation. Unusual presentation of even a common entity is tending to misdiagnose. This case report is about a rare presentation of angioedema as migrating unilateral edema. This case report also highlighted the diagnostic approach and management of this unusual presentation.


  Case Report Top


This case report is about a 55-year-old, Hindu, housewife female resident of a village of Dungarpur district, Rajasthan. She had a history of insidious onset, painless, wandering unilateral edema following sequence of the left breast (for 7 days) followed by left upper limb (for 1 month), followed by the left nape of the neck (for 1 day), followed by left scalp (for 1 day), followed by left-sided face (for 3 days), followed by right-sided face (for 2 days) [Figure 1] and [Figure 2]. Edema progressed from one site to another site in the above sequence with overlapping periods of only 12–24 h and later subsided completely from its previous site. In the patient's language edema was moving from site to site like a slow wave moving forward. Shifting of edema was not in the jumping pattern (regress fully at one site and afterward reappear at another side). There was no history of fever, cough, burning micturition, decreased or increased urine output, yellowish discoloration of body, bleeding, abdominal pain, diarrhea, malformed stool, chest pain or discomfort, pedal edema, joint pain, rashes, any mass over the body, headache, facial pain, running nose, sneezing, nasal blockage, any apparent dilated vessel, weight gain or loss, toothache, earache, worm in stool and trauma.
Figure 1: Edema at left-sided face

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Figure 2: Edema predominantly at right-sided face

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The patient took multiple consultations for the last one and a half months but could not get any relief. All the previous records of the patient were reviewed. Previous investigations had no significant abnormality. Most of the time she was treated with diuretics with nonspecific medications like multivitamin, proton-pump inhibitors. One of the prescriptions also had diclofenac indicating inflammatory etiology was thought. The patient presented to us at the stage of right-sided unilateral facial swelling in December 2020. There was no history of diabetes, hypertension, tuberculosis, allergic rhinitis, asthma, food, and drug allergy, or any other significant illness. The patient was vegetarian by diet, nonsmoker, nonalcoholic, bowel, bladder habits and sleep was adequate. The patient was not on any kind of medication before the illness. No significant family history was there. The patient had menopause 5 years back. The patient was having two sons born by uncomplicated vaginal delivery at home.

On examination blood pressure was 124/82 mm of Hg, pulse was 88/minute regular, respiratory rate was 14/min. She was afebrile. There was unilateral facial swelling over the right-sided face which was nonpitting, nontender, moreover upper part than the lower part of right-sided face. There was no lymphadenopathy, pallor, icterus, cyanosis, clubbing, pedal edema, or raised jugular venous pressure. On systemic examination, no significant finding was found.

As the patient was presented with unilateral facial edema, underlying sinusitis, tooth or ear infection, superior veina cava syndrome, angioneurotic edema, minor trauma were kept as primary possible etiologies. Other etiologies of edema such as renal failure, cardiac failure, hepatic failure, anemia, hypothyroidism, protein-losing enteropathy were also evaluated as contributing factors, although these entities usually not present as unilateral edema. As discussed above there was no history of any kind of allergy, trauma, toothache, earache before, and no underlying facial tenderness or lymphadenopathy was found. Gynecologist also evaluated the patient by breast palpation and mammography of both breasts to look out any lump or palpable lymph node, but it was not present.

The patient was investigated further accordingly. Her hemoglobin was 12.7 g/dl, total leukocyte was 10,300/cumm, platelet count was 179,000/cumm, ESR was 06. Random blood sugar was 122 mg/dl, urea was 22.3 mg/dl, creatnine was 0.7 mg/dl, total bilirubin was 0.9 mg/dl total protein was 8.0 g/dl, TSH was 1.21 uIU/ml. Urine examination shows no protein urea, hematuria or pus cells. Electrocardiography, chest X-ray, ultrasound abdomen, the 2D echo of the heart showed no significant abnormality. Antinuclear antibody titer was also in the normal range. Total eosinophil count was 3100/cumm and differential leucocyte counts were neutrophil 50.3%, lymphocyte 17.3%, monocyte 2.2%, eosinophil 30.1%, basophil 0.1%.

Due to nonpitting edema and raised eosinophil count, angioneurotic edema was kept as the diagnosis. The patient was not able to recognize any substance or factor which can be kept as responsible allergen. The patient was treated with steroids in form of methylprednisolone 16 mg once a day and a combination of montelukast 10 mg, levocetirizine 5 mg once a day. She was advised to follow up after 5 days. After 5 days facial edema was completely subsided [Figure 3]. Complete recovery in response to steroid and antihistaminic also favored allergic etiology. She was further treated with tapering dose of steroid for the next 10 days and combination of montelukast, levocetirizine for a month. The further plan was kept as allergy test for common allergens after 3 days of completion of current treatment.
Figure 3: Completely recovered edema after treatment

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  Discussion Top


The case of unilateral wandering edema is unique in its presentation as no other case report was found involving the current pattern of edema after an extensive search. To the best of our knowledge, this is the first case reporting wandering unilateral edema like a slow wave in the current pattern. In previous case reports, various etiologies were identified of facial swelling like hereditary angioedema,[1],[2] drug-induced angioedema,[3],[4] superior venacaval obstruction,[5] Ramsay Hunt syndrome,[6] arteriovenous malformation,[7] Erysipelas,[8] pericoronitis of tooth,[9] multiple myeloma,[10] underlying mass or hematoma,[11] etc., Unilateral upper limb edema was reported secondary to etiologies like venous and lymphatic obstruction due to mass,[12],[13] lymphedema,[14] etc.

Angioedema can be differentiated from other types of edema by features like self-limiting, asymmetrical distribution, onset in minutes to hours, spontaneous resolution in hours to several days, nongravitational, non position-dependent, involvement of areas with loose connective tissue (lips, eyelids, mouth, throat, uvula, larynx, extremities, genitalia, bowel wall).[15] Several disorders may be mistaken for angioedema like contact dermatitis, cellulitis, erysipelas, rosacea, autoimmune disorders, superior vena cava syndrome, parasitic infections, hypothyroidism, or cheilitis granulomatosa.[15]

This case report highlighted the diagnostic approach and management of rare presentation of wandering unilateral edema. It can also help to know the spectrum of presentation of angioedema. Early and correct diagnosis with appropriate management may do complete recovery like in current case report. Clinical recovery in response to steroids and antihistaminic is quick. Misdiagnosis of rare presentation even of common entity is common. Awareness about the spectrum of presentation and differential diagnosis of angioedema is needed among clinicians.


  Conclusion Top


This case report favors keeping angioedema as the differential diagnosis of wandering or unilateral edema. The patient in the current case report took multiple consultations before but could not be diagnosed indicates awareness about the entity is needed among clinicians.

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.
Behera S, Zafar L, Hasan A. Hereditary angioedema: A case report and literature review. Indian J Allergy Asthma Immunol 2015;29:92-5.  Back to cited text no. 1
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Jan AM, McGuire TP, Clokie CM, Sándor GK. Unilateral facial swelling caused by Ramsay Hunt syndrome resembles odontogenic infection. J Can Dent Assoc 2006;72:829-32.  Back to cited text no. 6
    
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Morgan JA. Unilateral facial swelling and fever. Clin Pearls 1995;2:320-7.  Back to cited text no. 8
    
9.
Costa F, Cian R, Robiony M, Zerman N, Politi M. Unilateral swelling of the cheek. J Oral Maxillofac Surg 2008;66:342-8.  Back to cited text no. 9
    
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Thomas AE, Kurup S, Jose R, Soman C. Facial swelling as a primary manifestation of multiple myeloma. Case Rep Dent 2015;2015:319231.  Back to cited text no. 10
    
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Agrawal A, Singh V, Kumar P, Bhagol A, Narwal A, Singh I. Unilateral swelling of cheek. Natl J Maxillofac Surg 2017;8:157-61.  Back to cited text no. 11
[PUBMED]  [Full text]  
12.
Freischlag J, Machleder HI. Unilateral upper extremity edema. A case report. J Cardiovasc Surg (Torino) 1993;34:251-4.  Back to cited text no. 12
    
13.
Martins F, Pereira E. Episodic facial edema as a presentation of pleomorphic adenoma – Case report. Galicia Clin 2016;77:137-8.  Back to cited text no. 13
    
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Idowu A, Lee K, Gujral S, Manushakian J. Unilateral swollen hand: A rare case of primary lymphedema tarda. Eplasty 2015;15:ic53.  Back to cited text no. 14
    
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Nedelea I, Deleanu D. Isolated angioedema: An overview of clinical features and etiology. Exp Ther Med 2019;17:1068-72.  Back to cited text no. 15
    


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