Tropical diabetic hand syndrome in a Solomons Islands adult: A case report of a rare complication

Abstract

Background

Tropical diabetic hand syndrome (TDHS) is a rare and often unrecognized complication that can lead to lifelong disability or even death among diabetic patients living in the tropics.

Presentation of case

This study reports the case of a 47-year-old male patient in the Solomon Islands who developed TDHS caused by Klebsiella pneumonia. The patient presented with symptoms of localized cellulitis of the fourth digit of the left hand after being discharged 10.5 weeks prior for an infection on the second digit of the left hand. Subsequent physical exams, surgical debridement, and patient monitoring indicated that the cellulitis spread and developed into necrotizing fasciitis. Despite serial surgical debridement and a fasciotomy, as well as administration of antidiabetic agents and antibiotics, the patient developed sepsis and died forty-five days post-admission.

Discussion

Medication shortages, late presentation, and failure to pursue aggressive surgery increases risk of TDHS patient morbidity and mortality.

Conclusion

TDHS requires early detection and presentation, aggressive surgical management, and efficient administration of antidiabetic agents and intravenous antibiotics.

Highlights

• •Tropical diabetic hand syndrome (TDHS) is a rare acute condition affecting diabetic patients living in the tropics.
• •Early identification, aggressive surgical intervention, and early administration of broad spectrum IV antibiotics are key.
• •Limited resource settings pose various challenges to the effective management of infection and TDHS.
• •Access to medications, diagnostic equipment, and knowledge about identifying and treating TDHS may help reduce mortality.

1. Introduction

Tropical diabetic hand syndrome (TDHS) is a term used to describe a condition occurring in tropical regions in which acute infection of the hand leads to localized swelling or cellulitis and subsequently to sepsis in diabetic patients [1]^, [2]. This is a commonly unrecognized and underreported complication in the Pacific Region with most cases being reported in Africa or India [3]^, [4]. Prior research has indicated that poorly controlled diabetes, neuropathy, insulin treatment, and malnutrition are risk factors for TDHS [5]. Early presentation, aggressive surgical intervention, rapid treatment with intravenous and broad spectrum antibiotics, as well as effective glycemic control are important to preventing major limb amputation and mortality in cases of TDHS [5]^, [6]. Managing diabetic wounds, infection, and sepsis in resource-limited settings can be challenging, particularly if medical infrastructure and patient awareness is limited [7]^, [8]. To the authors' knowledge, this is the first case report of TDHS in the South Pacific, a region combatting limited healthcare resources and the highest rate of diabetes globally [9]^, [10].

This work has been reported in line with the SCARE guidelines [11].

2. Case presentation

A 47-year-old male Melanesian patient with previously diagnosed type 2 diabetes mellitus (T2DM) and hypertension (HTN) ambulated to the emergency room of the National Referral Hospital of the Solomon Islands complaining of four days left-hand pain, swelling, fever, headache, and generalized weakness. The patient reported that three weeks prior to admission, he noticed a blister on the anterior fourth digit of his left hand that did not heal and began oozing pus four days prior to admission. Ten and a half weeks earlier, the patient had sought care for an infected wound on the second digit of the same hand. At that time, the medical team found that the patient's diabetes was uncontrolled. The patient was managed in the medical ward for seven days with five days intravenous (IV) cloxacillin (1 g 4×/day), as well as anti-diabetic and anti-hypertensive agents. The patient responded well to treatment and was counseled on lifestyle modification and instructed to return for follow up care. At the time of discharge metformin was added to his T2DM regimen and the patient was managed as an outpatient with Glipizide.

Upon readmission to the medical ward, the patient had uncontrolled blood sugar (above quantifiable limit) despite reporting full medication compliance. Physical examination revealed generalized body weakness, pale conjunctiva, dehydration, reduced range of movement of the left wrist, loss of sensation in the left hand, blood pressure of 95/60 mm Hg, pulse rate of (110×/min), respiratory rate of 22×/min, blood oxygen saturation of 98 %, and temperature of 38 °C. Bloodwork results are shown in Table 1.

Table 1.

This table lists the patientsblood results during hospital stay.

Test type	On admission	33 days post-admission (prior to fasciotomy)	39 days post admission
Hemoglobin (g/L)	106 (110–180)	123 (110–180)	67 (110–180)
Erythrocyte (×1012/L)	3.75 (3.9–5.1)	4.54 (3.9–5.1)	2.43 (3.9–5.1)
MCV (fL)	78.9 (84–94)	76.0 (84–94)	77.0 (84–94)
MCH (pg)	28.3 (27–34)	27.1 (27–34)	27.6 (27–34)
MCHC (g/L)	358 (300–350)	357 (300–350)	358 (300–350)
White blood cell count (×109/L)	17.21 (4.0–11.0)	20.21 (4.0–11.0)	34.27 (4.0–11.0)
Neutrophil (%)	81 (40–75)	87.4 (40–75)	90.9 (40–75)
Lymphocyte (%)	8.9 (20–45)	5.5 (20–45)	4.3 (20–45)
Monocyte (%)	1.35 (2−10)	5.7 (2–10)	4.5 (2–10)
Platelets (×109/L)	405 (150–400)	468 (150–400)	369 (150–400)
ESR (mm/h)	>100 (0–7)	N/A	N/A
CRP mg/L	354.5 (0–5)	N/A	N/A
Sodium (mmol/L)	124 (135–146)	133.2 (135–147)	N/A
Potassium (mmol/L)	5.7 (3.5–5.1)	3.92 (3.4–5.3)	N/A

The patient was diagnosed with clinical sepsis secondary to a diabetic wound infection. Radiography of the left hand showed periosteal reaction, a known phenomenon in diabetic infection, generalized soft tissue swelling, and evidence of decreased bone density on the fourth digit (Fig. 1) [12]. The surgical department was consulted, and the patient was scheduled for surgical debridement.

Fig. 1.

Radiograph of the left hand on admission showing marked periosteal reaction on the second, third, and fourth digits as well as generalized soft tissue swelling.

2.1. Initial infection control

The patient was initially managed in the medical ward for three days and started on Cloxicillin (500 mg 6×/day), Flagyl (400 mg 3×/day), Mixtard¹ (20 units morning, 15 units evening), Paradol (1 g 4×/day), and Metformin (1 g 2×/day) the day after admission. A surgical debridement of the left palm and fourth digit was performed. Intraoperative culture results identified amoxicillin and ampicillin-resistant Klebsiella pneumonia. On debridement, the surgical team noted that the infection had spread to the distal 4th digit and proximally along the entire palmar surface of the hand. Infection was limited to soft tissues and the tendon of the 4th digit. Following debridement, the surgical team elevated the patient's hand, performed routine wound care, and ordered IV antibiotics. Due to a shortage of intravenous medications, oral Cloxacillin and Flagyl were continued, and oral Ciprofloxacin (500 mg 2×/day) was added on receipt of microbiology results. The patient was cared for on the surgical ward as no high acuity care unit exists at the hospital.

2.2. Surgical control

Despite the initial debridement, wound care, and antibiotic treatment, the patient's infection progressed to the anterior and posterior surfaces of his left hand. Skin sloughing was noted. A second surgical debridement was performed seventeen days post-admission. Necrotic tissue was excised from the palm and posterior aspect of the fourth digit of the left hand. Repeat wound cultures were obtained (Fig. 2, Fig. 3). Microbiology results were consistent with prior cultures. Antibiotics, wound care, and diabetes management were continued. Blood sugar levels proved difficult to control, with fasting blood glucose ranging from 5.2 mmol/L to 25.7 mmol/L during the patient's stay in hospital.

Fig. 2.

Posterior hand post-debridement. Prominent edema and skin sloughing can be visualized.

Fig. 3.

Anterior hand post-debridement. The tendon has been exposed and necrotic tissue has been excised from the palm.

Twenty-eight days post-admission, the patient complained of pain spreading proximally to the upper arm. Pre-operative bloodwork showed anemia (Hgb 88 g/L) delaying immediate surgical intervention in order to transfuse PRBCs. Surgical debridement of the hand, forearm, and distal arm was performed. During surgery infectious tenosynovitis involving the fourth digit, as well as malodorous necrotic tissue, and pus were found to extend up to the left axilla. A fasciotomy of the hand, wrist, forearm, and brachium was performed at which time necrosis extended proximally into the axilla (Table 1) (Fig. 4). Post-operatively, the patient was managed with regular wound checks, dressing changes and intravenous Ceftriaxone (1 g 2×/day). 1 unit of packed red blood cells was transfused.

Fig. 4.

Left arm edema and necrosis.

Thirty-eight days post-admission, surgical ward nurses noted swelling in the patient's left axilla and paralysis of the left arm. A radical debridement of the arm from the left 4th digit to the left proximal arm and axilla was performed the same day. Following surgery, the patient had significant bleeding, >500 cm³, and a pressure dressing was applied. The patient developed hemorrhagic and septic shock within 3 h post-debridement. Initial vitals were blood pressure 90/50 mm Hg, pulse oximetry 82 %, a weak and thready pulse of 90 x/min, temperature of 38 °C, and cool and clammy skin. Resuscitation with saline IV fluids and oxygen via nasal cannula (4 L/min) was performed. An additional two units of blood were ordered but only one unit of blood was available. The patient subsequently regained consciousness and vitals stabilized to blood pressure 90/50 mm Hg, pulse oximetry 99 %, and heart rate 90 x/min. The patient continued oxygen via nasal cannula as needed.

At forty-three days post-admission the infection had tracked from the left axilla distally along the left lateral chest and abdominal wall down to the left anterior superior iliac spine. The patient was given intravenous morphine (5 mg 4×/day). The surgical team prepared the patient for an extensive debridement of the axilla, chest, and abdomen wall. Fearing pain from further surgical intervention, the patient and his family refused further debridement. Comfort measure treatments were continued including wound care, pain management, and antibiotics. Forty-four days post-admission, the patient experienced respiratory distress, intermittent apnea, and lapsed into unconsciousness. Forty-five days post-admission the patient became completely apneic and was pronounced dead.

All surgical interventions were performed by a senior surgical registrar. A consultant physician advised on patient management.

3. Discussion

Early presentation, diagnosis and treatment are key to treating TDHS in order to minimize fulminant sepsis, limb loss due to gangrene, and death. We present a case of TDHS occurring in Solomon Islands that had several challenges in providing treatment. First the patient, despite being treated for type 2 diabetes, delayed seeking treatment for a digit infection. Medical literacy can be a challenge in any environment and likely contributed to the outcome in this case. A delay in diagnosis also contributed to the case outcome.

Although TDHS has been seen in Papua New Guinea, this is the first case of TDHS reported in the Pacific Islands (David Watters, ChM, email communication, December 5, 2022). The prevalence of diabetes in Solomon Islands adults is 19.8 % [13]. With rates of diabetes in the Pacific Region estimated to increase over the next decade it is important that healthcare providers be made aware of this devastating complication of diabetes.

Initially the extent of the patient's infection was underrecognized which resulted in a delay in consulting surgery. Initial infection control via antibiotic treatment and debridement was based on physical exam and x-ray. Use of CT imaging of the hand and arm along with scheduled returns to the operating theater for wound debridement may have identified the proximal progression of the infection earlier in this case [14]. Additionally, the development of a locally tailored protocol may assist providers in managing TDHS cases as has been successful in other regions [15]. Prior research has identified factors such as time since diabetes diagnosis, gram-negative infection, time between symptom onset and presentation, polymicrobial infection, presence of end stage renal disease, peripheral neuropathy, and HbA1c >10 % as contributing to poor clinical outcomes in patients with diabetic hand infections [16]^, [17]^, [18]. These factors could aid in the development of a treatment strategy for TDHS.

Once cultures were obtained, antibiotics were selected based upon antibiotic sensitivity and availability in a resource-limited environment. Shortages of intravenous antibiotics posed a challenge to early and aggressive antibiotic treatment. Significantly, prior research has suggested the importance of effective glycemic control in managing diabetic infections [19]. In this case, despite insulin therapy and treatment with metformin, the patient's blood glucose was poorly controlled, possibly contributing to the poor outcome. Despite medical and surgical management, the infection progressed proximally resulting in necrotizing fasciitis and ultimately fulminant sepsis. We hope this report on what may be the first recognized case of TDHS in the South Pacific will alert healthcare providers to this problem in order to prevent delays in treatment.

4. Conclusion

In conclusion, we presented a rare case of TDHS in which the patient died from sepsis despite efforts to control the infection via surgical intervention and antibiotic treatment. Given its rarity, TDHS is challenging to treat, especially in resource-limited contexts. Early identification and aggressive treatment of TDHS can reduce mortality in these cases. Improved awareness and education may help improve TDHS management.

Consent

Written informed consent was obtained from the patient's family for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Provenance and peer review

Not commissioned, externally peer-reviewed.

Ethical approval

Ethical approval was provided by the authors' institution.

Funding

No funding received.

Guarantor

Dylan Bush.

Research registration number

N/A.

CRediT authorship contribution statement

• 1.Dylan Bush – Primary author of case report (conceptualization, data procurement, drafting of manuscript)
• 2.Dr. Eileen Natuzzi – Primary editor of case report (case analysis, revision of prior drafts)
• 3.Dr. Gordon Koburu - Lead surgeon on debridement & fasciotomy, editor (case note interpretation, photographing of limb)
• 4.Dr. Maggie Bana - Assisting surgeon on debridement & fasciotomy, editor (case note interpretation, data procurement)
• 5.Dr. Flins Taki - Assisting surgeon on debridement & fasciotomy, editor (case note interpretation, photographing of limb)
• 6.Dr. Augustin Melly– Consultant surgeon on debridement & fasciotomy, editor (case analysis, revision of prior drafts, final approval of manuscript).

Declaration of competing interest

The authors report no declaration of competing interest.