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Case Report

A Tropical Fever - Mimicking a Surgical Emergency

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13 October 2023

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17 October 2023

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Abstract
Introduction: Dengue is a mosquito transmitted arboviral infection. In dengue fever spontaneous bleeding in different parts of body occurs; but spontaneous bleeding into rectus muscle leading to haematoma formation is rare. Case Presentation: A 72-year-old hypertensive female presented with high grade intermittent fever with chills and rigors for last four days. She was diagnosed to have dengue fever (NS1Ag -Reactive) on the day before admission. At admission, on examination she was found to be have dehydration. Immediately she was put on IV fluids, antiemetics and other supportive therapy. At admission she had low platelets, low haemoglobin, total leucocyte counts, with raised liver enzymes. On day 3 of illness, she had significant drop of haematocrit with low blood pressure and subsequently she managed with packed red blood cell (PRBC) transfusion. But on 9th day of illness, she has severe right iliac fossa pain mimicking acute appendicitis. CECT of abdomen showed soft tissue lesion in right lower abdomen (Inflammatory/ hematoma along rectus sheath) which was managed conservatively as per surgical opinion. 10 days past discharge she had no pain in right iliac fossa and size of hematoma was significantly reduced. Discussion: In dengue fever, hematoma can be formed any vulnerable part in the but there are only a few cases reported to be having rectus sheath hematoma. Rectus sheath hematoma (RSH) has been mistaken for many acute abdominal diseases like- acute appendicitis. Our case mimicked acute appendicitis and managed conservatively with IV fluids, analgesics, Blood transfusion as it fits into RSH type II. Conclusion: It is important to be vigilant in the patients who presents with abdominal pain in severe dengue cases. Prompt imaging in relevant areas could make the diagnosis and father treatment possible.
Keywords: 
Subject: Medicine and Pharmacology  -   Tropical Medicine

Background

Dengue is a mosquito transmitted arboviral infection, which has become a disease of public health concern globally. It usually presents as fever with myalgia, body ache may often be complicated by bleeding, shock, organ involvement. Majority of patients present with fever, flu like symptoms, constitutional symptoms and some forms of plasma leakage. However, reports of dengue cases with organ involvement in the form of hepatitis, kidney injury, myocarditis, encephalitis, pancreatitis, acute respiratory distress syndrome (ARDS) are on the rise and this is called expanded dengue syndrome (EDS).(1) There has been reports of spontaneous bleeding in different parts of body but spontaneous bleeding into rectus muscle leading to haematoma formation is rare.(2) We report an interesting case of spontaneous rectus sheath haematoma leading to severe right iliac fossa pain mimicking acute appendicitis which was formed during the recovery phase of dengue haemorrhagic fever (DHF).

Case presentation

A 72-year-old hypertensive female resident of Kolkata, West Bengal, India, presented with high grade intermittent fever with chills and rigors for last four days. She also had severe muscle pain, body ache, nausea and vomiting associated with fever. She was unable to eat or drink due to the illness. She did not have any cough, shortness of breath, urinary symptoms, altered bowel habits. She was on telmisartan 40 mg and hydrochlorothiazide 12.5 mg for control of hypertension. She was diagnosed to have dengue fever (NS1Ag -Reactive) on the day before admission. At admission, on examination she was found to be have dehydration, mild pallor, BP- 110/70 mm of Hg, Pulse- 122/min (regular), temperature- 102.6⁰F, SpO2- 98% @ room air, alert, co-operative, chest- bilateral vesicular breath sounds, CVS- S1, S2 audible, Tender 2 cm hepatomegaly below right costal margin, no free fluid. Immediately she was put on IV fluids (volume calculated according to Holliday-Segar formula), antiemetics and other supportive therapy. At admission she had low platelets, low haemoglobin, total leucocyte counts, with raised liver enzymes. Serial blood parameters are mentioned in Table 1.
On day 3 of illness, she had significant drop of haematocrit with low blood pressure (90/60 mm of Hg). So, she was immediately transfused packed red blood cell (PRBC). Her Blood culture, urine cultures were showing no growth. Malaria was not found in peripheral blood smear and on Day 7 of illness her Dengue IgM was reactive. After the initial hypotension and drop in haematocrit she continued to have low haemoglobin (7~8 gm/dl) with normal blood pressure (without antihypertensives). Initially she complained of severe nausea and vomiting due to raised liver enzymes. But on 9th day of illness, she has severe right iliac fossa pain mimicking acute appendicitis with continued nausea. There was no history of trauma during this period also. At that time her stool occult blood test was strongly positive. Immediately surgical opinion was sought for this new onset abdominal pian and as acute appendicitis was the primary differential diagnosis, she was again started on IV fluids, Ceftriaxone, IV metronidazole and other supportive therapy. CECT of abdomen showed soft tissue lesion in right lower abdomen (? Inflammatory/? hematoma along rectus sheath). [Picture 1].
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After that antibiotics were discontinued in view of normalised total leucocyte and absence of fever. After 2~3 days of conservative management she was stable with mild tender right iliac fossa mass. On surgical opinion she was discharged with a device to do a follow up ultrasonography. 10 days past discharge she had no pain in right iliac fossa and size of hematoma was significantly reduced (from 11 cm X 3 cm to 4 cm X 2 cm) on sonography. On Follow up after 15 days she had no right iliac fossa pain or mass felt. On ultrasonography also there was very minimal hematoma.

Discussion

Rectus sheath hematoma (RSH) usually presents as bleeding within the rectus abdominis muscle sheath. This is commonly be caused by ruptured epigastric vessels or a tear of the rectus muscle.(3) Usual known risk factors of spontaneous RSH are female gender, pregnancy, obesity, anticoagulant drugs, abdominal straining, severe vomiting, and intractable cough. (4) Complications in patients with DHF in the form of bleeding usually a result from the combination of thrombocytopenia, increased vascular fragility, increased fibrinolysis, pro-coagulation and anticoagulation factors imbalance.(5) In critical stage of DHF there is defervescence of fever and higher degree of plasma leakage and low platelet count, which in some cases may lead to circulatory failure or bleeding complications.(6) We did an extensive literature review of spontaneous hematoma in dengue fever which is shown in Table 2.
We can see from this literature review that maximum cases were reported from South East Asia as dengue fever mostly endemic here. There were slightly more males than females who had this spontaneous hematoma in different parts of the body in dengue fever. Age group was more in younger population. Majority had no co-morbidities. Hematoma can be formed any vulnerable part in the but there are quite a few cases reported to be having rectus sheath hematoma. Also, RSH did not need any surgical intervention; though splenic hematoma mostly managed with splenectomy. There were few mortalities when patient presented with cerebral or splenic hematomas.
RSH can be divided by the severity of haemorrhage as seen on CT scan. Type-I RSH are one sided hematoma contained only within the muscle. Type-II RSH is bilateral hematomas/ hematomas not contained within the muscle sheath. Type-III RSH is called when blood enters the pre-vesicular space or peritoneum. Usually type I & II RSH can be manged conservatively but type III usually needs surgery. Treatment of spontaneous RSH is generally conservative including resuscitation, correction of coagulopathy, analgesia and treatment of the underlying condition.(30) Interventional radiological intervention and embolization is preferred in cases of continued bleeding with surgery being considered as the last option.(31) RSH has been mistaken for many acute abdominal diseases like- acute appendicitis, dissecting abdominal aneurysm, cholecystitis and biliary colic, cholelithiasis, diverticular disease, peptic ulcer disease, acute gastritis, obstructed intestinal hernias. Our case mimicked acute appendicitis and managed conservatively with IV fluids, analgesics, Blood transfusion as it fits into RSH type II.
In conclusion, it is important to be vigilant in the patients who presents with abdominal pain or any neuro-deficit during convalescent phase of dengue fever. Because it could be a presenting symptom of spontaneous hematoma in different parts of the body. Prompt imaging in relevant areas could make the diagnosis and father treatment possible.

Funding

This case report did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Conflicts of Interest

The authors declare no conflict of interest.

References

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Table 1. Treands of blood parameters day-wise with associated clinical events.
Table 1. Treands of blood parameters day-wise with associated clinical events.
Day of illness Sample Hb (gm/dl) Hct TLC (/cmm) Platelet count (/cmm) SGPT (IU/l) SGOT
(IU/l)		 Phase /clinical events
Day 4 Admission 8.6 21.5 2,300 80,000 61 158 Febrile Phase
Evening 8.7 22.8 2,200 70,000 - -
Day 5 Morning 9.1 26.9 1,400 60,000 98 232 Critical Phase
Evening 9.1 25.0 1,200 69,000 - -
Day 6 Morning 9.4 28.4 1,700 30,000 176 548 Critical Phase; Severe Nausea and Vomiting; 1 unit of PRBC given
Evening 7.0 21.4 1,600 22,000 - -
Day 7 Morning 8.2 23.5 2,500 30,000 190 534 Critical Phase; Nausea, vomiting present
Evening 7.8 22.4 2,700 32,000 - -
Day 8 Morning 6.8 19.2 2,800 33,000 186 504 Critical Phase ended; Nausea, vomiting present;
1 unit of PRBC given; Stool for OBT- strongly positive
Evening 6.7 18.7 3,300 35,000 - -
Day 9 Morning 7.8 22.7 4,300 40,000 178 456 Nausea, vomiting present; Severe RIF pain; 1 unit of PRBC given
Evening 7.6 23.9 4,200 45,000 - -
Day 10 Morning 8.2 24.6 4,300 55,000 170 345 Nausea, vomiting subsided, RIF pain continued
Evening 8.0 25.8 4,700 76,000 - -
Day 11 Morning 7.6 23.7 5,800 90,000 134 235 RIF pain continued, RIF tender mass felt; antibiotics and IV fluids started; 1 unit of PRBC given
Evening 7.2 2.6 5,700 1,12,000 - -
Day 12 Morning 8.6 26.7 6,300 1,25,000 87 176 RIF tender mass felt; 1 unit of PRBC given
Evening 8.9 27.9 7,600 1,56,000 - -
Day 13 Morning 8.5 27.0 8,900 2,12,000 67 112 Imaging showed right lower abdominal rectus muscle hematoma, 1 unit of PRBC given; antibiotics discontinued
Day 14 Morning 9.2 28.3 8,700 2,54,000 56 78 Mild RIF tenderness; discharged
Table 2. Literature review of Spontaneous Hematoma in Dengue fever.
Table 2. Literature review of Spontaneous Hematoma in Dengue fever.
Location Sex Age Co-morbidities/ drugs Presentation Diagnosis Treatment Outcome
Our case
2023		 India F 72 HTN RIF pain; Recurrent Vomiting (dengue hepatitis) RSH (CT scan) Conservative Survival
Pahari et al. 2023(7) Nepal M 54 Nil Left upper abdominal pain Splenic sub-capsular hematoma (CT scan) Conservative Death
Kaushik et al. 2022(8) India F 48 Nil Acute onset paraplegia with bladder and bowel dysfunction and anaesthesia below the umbilicus Compression of spinal cord due to intradural hematoma at the D7–D8 level (MRI) Emergency D7–D8 laminectomy with excision of the clot and Dural
repair		 Survival with residual neuro-deficit
Corré et al. 2022(9) France M 33 Nil Chest pain Coronary hematoma (angiography): Acute myocarditis (CMRI) Conservative with dual antiplatelets Survival
Siahann et al. 2022(10) Indonesia M 65 Nil Altered consciousness and focal neuro- deficits Subdural hematoma (CT scan) Craniotomy Survival
Chang et al. 2021(11) Malaysia F 59 DM Hypotensive shock Retroperitoneal hematoma (Ultrasonography)
Associated with HLH and AIHA		 Conservative Death
Ungthammakhun et al. 2021(12) Thailand M 22 Nil Left upper abdominal pain Splenomegaly with active contrast extravasation at spleen surrounded with
hematoma, and generalized hemoperitoneum (CT scan)		 Splenectomy Survival
Mushtaque et al. 2020(13) Pakistan M 32 Nil Decreased power in bilateral lower
limbs and pain in right leg		 Bilateral iliopsoas hematoma (CT scan) Conservative Survival
Matthias etal. 2019(14) Sri Lanka M 28 Nil Left groin and inguinal region Left psoas
haematoma (Ultrasonography)		 Conservative Survival
Baruah et al. 2018(15) India F Not Mentioned HTN, Bronchial Asthma, OSA Acute quadriparesis with urinary incontinence Cervicodorsal anterior epidural hematoma (MRI) Surgical evacuation (durotomy) Death
Ghosh et al. 2018(16) India F 74 HTN Recurrent Vomiting RSH (CT scan) Surgical evacuation for Abdominal Compartment Syndrome Survival
Tamilasran et al. 2018(17) India M 36 Nil Sore throat Hematoma involving
both the vocal cords and immediate sub-glottis (Video Laryngoscopy)		 Conservative Survival
Anam et al. 2017(18) Bangladesh M 45 Nil Left calf swelling Calf muscle hematoma (Ultrasonography) Surgical evacuation via fasciotomy Survival
Nelwan et al. 2017(19) Indonesia F 58 Nil lower abdominal pain RSH (Cullen’s sign + CT scan) Conservative Survival
Jayasinghe et al. 2016(20) Sri Lanka F 24 Nil Headache, loss of consciousness, seizure Intracranial haemorrhages and
sub arachnoid haemorrhages (CT scan)		 Conservative as platelet count was 40,000/cmm Death
Singh et al. 2016(21) India M 30 Nil RIF pain Intra-parenchymal haematoma in the liver with extension to
right peritoneum and right psoas muscle haematoma		 Conservative Survival
Sharma et al. 2014(22) India M 40 Nil Severe pain in
paraumbilical region, extending to right hypochondrium and
lumbar regions		 RSH (CT scan) Conservative Survival
Waseem et al. 2014(23) Pakistan M 55 HTN, DM, Aspirin Pain in right upper quadrant of abdomen RSH (CT scan) Surgical Evacuation Survival
Mehtani et al. 2013 (24) India M 36 Nil Right forearm pain with tingling and numbness along the distribution of ulnar nerve Hematoma compressing ulnar neurovascular bundle (MRI) Surgical evacuation Survival with residual neuro-deficit
Sarkar et al. 2011(25) India M Young Nil Inability to protrude his tongue and was unable to speak Tongue- enlarged and swollen (Clinical)
 Conservative Survival
Mittal et al. 2011(26) India F 27 Nil Recurrent seizure Bilateral subdural haematoma (MRI) Conservative Survival with residual neuro-deficit
Tong et al. 2010(27) Republic of Singapore F 37 Adenomyosis, OCP RIF pain RSH (CT scan), DVT (US doppler) Conservative; Anti-coagulation after stabilisation Survival
Seravali et al. 2008(28) Brazil M 27 Nil Shock Spleen congested with rupture
of middle third and peri-splenic hematoma (Intra-operative)		 Splenectomy Survival
Seravali et al. 2008(28) Brazil M 20 Nil Left upper abdomen pain Splenic hematoma (Ultrasonography) Splenectomy Survival
Miranda et al. 2003(29) Brazil M 27 Nil Diffuse abdominal pain Splenic hematoma (CT scan) Splenectomy Survival
HTN- Hypertension; RIF- Right iliac fossa; RSH- Rectus sheath hematoma; CT scan- Computed tomography scan; MRI- Magnetic resonance imaging; CMRI- Cardiac Magnetic resonance imaging; HLH- Hemophagocytic Lymph histiocytosis; AIHA- Auto-immune haemolytic anaemia: DVT- Deep vein thrombosis.
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