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    A and B, Ulceration of the tips of the second and third fingers (white arrows) due to median neuropathy. C, Atrophy of flexor carpi ulnaris (black arrows) and brachioradialis muscles (arrowheads).

  • 1

    Leach RE, Hammond G, Stryker WS, 1967. Anterior tibial compartment syndrome. Acute and chronic. J Bone Joint Surg Am 49 :451–462.

  • 2

    Power RA, Greengross P, 1991. Acute lower leg compartment syndrome. Br J Sports Med 25 :218–220.

  • 3

    Imbriglia J, Boland D, 1984. An exercise induced compartment syndrome of the forearm: a case report. J Hand Surg 9A :142–143.

  • 4

    Klodell C Jr, Pokorny R, Carrillo EH, Heniford BT, 1996. Exercise induced compartment syndrome: a case report. Am Surg 62 :469–471.

  • 5

    Beall S, Garner J, Oxley D, 1983. Anterolateral compartment syndrome related to drug-induced bleeding. A case report. Am J Sports Med 11 :454–455.

    • Search Google Scholar
    • Export Citation
  • 6

    Matsen FA 3rd, Winquist RA, Krugmire RB Jr, 1980. Diagnosis and management of compartmental syndromes. J Bone Joint Surg Am 62 :286–291.

    • Search Google Scholar
    • Export Citation
  • 7

    Perry MO, 1988. Compartment syndromes and reperfusion injury. Surg Clin North Am 68 :853–864.

  • 8

    Lee BY, Brancato RF, Park IH, Shaw WW, 1984. Management of compartmental syndrome. Diagnostic and surgical considerations. Am J Surg 148 :383–388.

    • Search Google Scholar
    • Export Citation
  • 9

    Fryberg ER, 1995. Compartment syndrome. Cameron JL, ed. Current Surgical Therapy. Fifth edition. St. Louis: Mosby, 850–855.

  • 10

    Rorabeck CH, Bourne RB, Fowler PJ, Finlay JB, Nott L, 1988. The role of tissue pressure measurement in diagnosing chronic anterior compartment syndrome. Am J Sports Med 16 :143–146.

    • Search Google Scholar
    • Export Citation
  • 11

    Whitesides TE, Haney TC, Morimoto K, Harada H, 1975. Tissue pressure measurements as a determinant for the need of fasciotomy. Clin Orthop Rel Res :43–51.

    • Search Google Scholar
    • Export Citation

 

 

 

 

COMPARTMENT SYNDROME: AN UNUSUAL COURSE FOR A RARE DISEASE

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  • 1 Neurology Department, Department of Infectious Diseases,and Department of Operative Orthopedics, Zahedan University of Medical Sciences, Zahedan, Iran

We report a case of compartment syndrome of the left upper limb following hemorrhage due to Crimean-Congo hemorrhagic fever in a 45-year-old man. As far as we know, there is not such a report in the literature. We discuss clinical manifestations, electrophysiologic findings, differential diagnosis, and management of the patient. A high degree of awareness for an early diagnosis may participate to improve the poor prognosis.

CASE REPORT

A 45-year-old shepherd was admitted to our hospital because of fever and hemorrhagic rashes. He was well until three days before admission when fever, headache, myalgia, and hemorrhagic rashes suddenly occurred. On physical examination, he appeared ill. His temperature was 39.0°C, and he was tachycardic and tachypneic. His neck was supple but the conjunctivae were injected. Pharyngeal mucosa was erythematous. On abdominal examination, he had epigastric tenderness without organomegaly. A diffuse maculopapular rash and petechiae were seen over the entire body, especially the chest and abdomen. On the fifth day of admission, oral and nasal bleeding occurred. Large ecchymoses were seen at venipuncture sites on the volar portion of the left forearm. The results of laboratory tests are shown in Table 1.

Treatment was begun with oral ribavirin and intravenous ceftriaxone, along with platelet transfusions and supportive therapy. The result of an IgM enzyme-linked immunosorbent assay (ELISA) for Crimean-Congo hemorrhagic fever virus was positive on the fifth day of admission, and the results of both IgM and IgG ELISAs were positive on the 10th day of admission. During the first three days of treatment, massive bleeding occurred in the left forearm. The limb became tense and nonpiting edema occurred. Only medical supportive therapies were continued. No surgical procedure was done. Two weeks later, the patient was improved.

Three months later, the next physical examination showed atrophy of left forearm and hand. Trophic changes of the thumb were prominent (Figure 1). On neurologic examination, pain and pinprick sensation were decreased and motor power was graded 3/5 distally. Brachioradialis muscle stretch reflex was decreased in comparison to the other side. Nerve conduction studies showed decreased amplitude of compound motor action potentials, decreased conduction velocities, and increased distal latencies of both median and ulnar nerves. Electromyography (EMG) showed long duration polyphasic motor unit potentials (MUPs), with single MUP recruitment pattern in the ulnar and median innervated muscles of the forearm and hand. Severe axonal damage of the median nerve after innervation of the pronator teres and axonal injury of the ulnar nerve before innervation of the flexor carpi ulnaris without presence of reinnervation MUPs were the other major EMG findings.

DISCUSSION

Acute compartment syndrome (ACS) with its risk of irreversible muscle and nerve necrosis can be both life- and limb-threatening. It has been described in association with trauma, crush injury,1 ischemia, and reperfusion episodes.2 Non-traumatic ACS occurs most commonly in the lower leg, but has also been reported in the forearm following muscle overuse,3 undue exertion,4 and bleeding diathesis.5 It requires urgent diagnosis and treatment. Crimean-Congo hemorrhagic fever is a viral disease characterized by fever, thrombocytopenia, myalgia, and subcutaneous bleedings. Disseminated intravascular coagulation is the major cause of death.

Compartment syndromes are characterized by increased tissue pressure within the confined space of the fascial sheaths. The lower extremities are affected more commonly than the upper extremities. Loss of integrity of the microcirculation with fluid exudation into the interstitial space results in edema formation, muscle swelling, and raised intracompartmental pressure, eventually leading to compression of blood vessels and nerves.6 Varieties of non-traumatic conditions have been described as etiologic factors in the development of compartment syndrome.7

Acute compartment syndrome is of particular concern because the diagnosis must be made essentially on clinical grounds and must be acted upon promptly if serious and potentially irreversible injury to the relevant compartment is to be avoided. It is progressive and irreversible unless surgical decompression is performed.8 Continuous tissue pressure greater than 30–40 mm of Hg lead to irreversible nerve and muscle damage after 6–12 hours.6 Fryberg has reported that a tissue pressure 30 mm of Hg less than patient’s diastolic pressure is diagnostic.9

Increased intracompartmental pressure may be common due to hemorrhage between fascial sheaths but may evade recognition as a compartment syndrome because it reflects only a transient response to altered intracompartmental pressure that resolves spontaneously, especially with successful treatment of the basic condition. Physical examination, including passive stretching of muscle groups, should be conducted frequently to detect early signs of compartment syndrome, especially in the limbs with widespread ecchymoses.

Direct percutaneous monitoring of patients with intracompartmental pressure has been proposed although criteria have varied regarding the accepted useful diagnostic readings.10,11 Surgical decompression is impossible or very difficult in these patients because of thrombocytopenia, disordered coagulation, and presence of a highly transmissible infection. Blood sampling is an important cause of interfascial hemorrhage. Venipuncture sites should be controlled and compressed carefully to prevent continuous bleeding. Reducing blood sampling and controlling venipuncture sites are useful measures for deceasing interfascial hemorrhage in viral hemorrhagic fevers. Progressive increase in muscle compartment pressure may be prevented by early recognition, conservative therapy, and elevation of the affected compartment. Measuring of blood pressure using the involved limb especially with diffuse echymoses could be an inducing factor, especially when venous pressure is less than tissue pressure.

Patients that are at the risk of developing compartment syndromes especially in the forearm. We emphasize the importance of clinical signs in the early diagnosis of this potentially serious complication of viral hemorrhagic fevers. With greater awareness among physicians, patients with bleeding diatheses, especially secondary to transmissible viral infections, are more likely to have a mild disease course.

Table 1

Laboratory data for the patient on admission and five days later*

TestFirst dayFifth day
* WBCs = white blood cells; Hb = hemoglobin; AST = aspartate aminotransferase; ALT = alanine aminotransferase; ALP = alkaline phosphatase; PT prothrombin time.
WBCs/L2.9 × 1092.2 × 109
Hb (g/dL)13.510.3
Platelets (L)72 × 10915 × 109
AST (U/L)4853
ALT (U/L)75128
ALP (U/L)272285
PT (seconds)1517
Figure 1.
Figure 1.

A and B, Ulceration of the tips of the second and third fingers (white arrows) due to median neuropathy. C, Atrophy of flexor carpi ulnaris (black arrows) and brachioradialis muscles (arrowheads).

Citation: The American Journal of Tropical Medicine and Hygiene Am J Trop Med Hyg 73, 2; 10.4269/ajtmh.2005.73.450

*

Address correspondence to Dr. Ali Moghtaderi, Neurology Department, Zahedan University of Medical Sciences, Zahedan, Iran. E-mail: moghtaderi@zdmu.ac.ir

Authors’ addresses: Ali Moghtaderi, Neurology Department, Zahedan University of Medical Sciences, Zahedan, Iran, E-mail: moghtaderi@zdmu.ac.ir. Roya Alavi-Naini, Department of Infectious Diseases, Zahedan University of Medical Sciences, Zahedan, Iran, E-mail: ranaini@zdmu.ac.ir. Hadi Azimi, Department of Operative Orthopedics, Zahedan University of Medical Sciences, Zahedan, Iran.

REFERENCES

  • 1

    Leach RE, Hammond G, Stryker WS, 1967. Anterior tibial compartment syndrome. Acute and chronic. J Bone Joint Surg Am 49 :451–462.

  • 2

    Power RA, Greengross P, 1991. Acute lower leg compartment syndrome. Br J Sports Med 25 :218–220.

  • 3

    Imbriglia J, Boland D, 1984. An exercise induced compartment syndrome of the forearm: a case report. J Hand Surg 9A :142–143.

  • 4

    Klodell C Jr, Pokorny R, Carrillo EH, Heniford BT, 1996. Exercise induced compartment syndrome: a case report. Am Surg 62 :469–471.

  • 5

    Beall S, Garner J, Oxley D, 1983. Anterolateral compartment syndrome related to drug-induced bleeding. A case report. Am J Sports Med 11 :454–455.

    • Search Google Scholar
    • Export Citation
  • 6

    Matsen FA 3rd, Winquist RA, Krugmire RB Jr, 1980. Diagnosis and management of compartmental syndromes. J Bone Joint Surg Am 62 :286–291.

    • Search Google Scholar
    • Export Citation
  • 7

    Perry MO, 1988. Compartment syndromes and reperfusion injury. Surg Clin North Am 68 :853–864.

  • 8

    Lee BY, Brancato RF, Park IH, Shaw WW, 1984. Management of compartmental syndrome. Diagnostic and surgical considerations. Am J Surg 148 :383–388.

    • Search Google Scholar
    • Export Citation
  • 9

    Fryberg ER, 1995. Compartment syndrome. Cameron JL, ed. Current Surgical Therapy. Fifth edition. St. Louis: Mosby, 850–855.

  • 10

    Rorabeck CH, Bourne RB, Fowler PJ, Finlay JB, Nott L, 1988. The role of tissue pressure measurement in diagnosing chronic anterior compartment syndrome. Am J Sports Med 16 :143–146.

    • Search Google Scholar
    • Export Citation
  • 11

    Whitesides TE, Haney TC, Morimoto K, Harada H, 1975. Tissue pressure measurements as a determinant for the need of fasciotomy. Clin Orthop Rel Res :43–51.

    • Search Google Scholar
    • Export Citation
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