Hindawi Publishing Corporation Case Reports in Endocrinology Volume 2011, Article ID 624020, 4 pages doi:10.1155/2011/624020

Case Report An Unusual Case of Myonecrosis

P. Mu khop a d hy ay , 1, 2 R. Barai,1 C. A. Philips,1 J. Ghosh,1 and S. Saha1

1 Department of General Medicine, Nilratan Sircar Medical College and Hospital, Kolkata 700014, India 2 Phani Kutir, Udaypur (South), 82 Olay Chandi Road, Kolkata 700049, India

Correspondence should be addressed to P. Mukhopadhyay, [email protected]

Received 25 May 2011; Accepted 27 June 2011

Academic Editors: I. Broom, K. Iida, and T. Konrad

Copyright © 2011 P. Mukhopadhyay et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Diabetic Myonecrosis is a rare complication of long-standing Mellitus Type 1 and 2. The most likely affected areas are of proximal lower limbs, mostly the quadriceps muscle. The presenting features are myriad and a diagnostic conundrum for the physician. There has been previously mentioned, through few case reports, the classical presentation of diabetes-related muscle infarction. Here we present a patient of diabetic myonecrosis, in whom the initial presentation of diabetes mellitus was that of bilateral symmetric proximal upper limb predominant muscle infarction, which has never been reported before.

1. Introduction to have episodes of polyuria and polydipsia for a period of 2 years but considered these trivial. His mother and an elder Diabetic Myonecrosis is an enigmatic, rare complication of sibling suffered from Diabetes Mellitus type 2 with good long-standing diabetes mellitus type 1 and 2. Most patients glycemic control on oral hypoglycemic agents. have associated retinopathy, nephropathy, or neuropathy. It On examination there were local warmth, tenderness, ff commonly a ects the lower limb muscles, predominantly and swelling around the shoulder joints and upper part of the quadriceps. The pathogenesis of this condition is less both arms associated with mild erythema, no induration, understood for which more physicians are identifying this and no palpable crepitus (Figure 1). The patient was afebrile uncommon entity, since its first description in 1965 by and normotensive with a Body Mass Index (the weight in Angervall and Stener [1]. The treatment for this particular kilograms divided by the square of height in meters) of 29.4. manifestation of diabetes is watchful conservative manage- All the peripheral pulses were palpable. Deep tendon reflexes ment and strict glycemic control and in most cases produces were present and normal in all four limbs. A fundoscopy a good outcome. revealed small microaneurysms of the retinal vessels with small blot hemorrhages. Other systems examination was 2. Case Report noncontributory. The hemogram revealed hemoglobin levels to be 13 g/dl, a slightly raised total leucocyte count of A 58-year-old male patient, presented to our Emergency 12.9 × 109 (normal—3.8 to 9.8 × 109/L) with a neutrophilic Department with features of bilateral proximal upper limb predominance (94%) in the differential and normal liver predominant muscle pain which started off as dull ache in function and renal function tests. An initial random blood character and over the course of 8 days, became excruci- glucose on admission was 440 mg/dl and a subsequent ating in nature associated with swelling and restriction of fasting blood glucose and 2-hour postprandial blood glu- movements at the shoulder joints. He was neither a known cose done the next day revealed values of 280 mg/dl and hypertensive nor a diabetic. There was no history of direct or 308 mg/dl, respectively. The Hba1c was 10.1% (normal— indirect trauma; skin changes with any “over-the-counter” less than 5.5%). The serum (CK) level was or herbal drug intake, apart from occasional acetaminophen 80 U/L (normal—24 to 195 U/L) while serum lactic acid (500 mg) tablets for pain. On further question, he admitted dehydrogenase (LD) was 168 U/L (normal—45 to 90 U/L). 2 Case Reports in Endocrinology

(a) (b)

Figure 1: The muscles around the right and left shoulder joints showing features of swelling and mild erythema.

A spot urine for albumin to creatine ratio was 110 microgram Usual initial manifestations are acute or subacute onset albumin per milligram creatine (normal in male—30 to 300 of intense pain of involved muscles. There is a clear microgram per milligram). His thyroid profile tests, serum predilection for thigh muscles and the disease rarely presents uric acid, and electrolytes were well within normal limits. as bilateral disease and further rarely involving the upper A Roentgenogram of the shoulder joints did not reveal limb muscles. The most commonly affected muscles are any bony abnormalities, but there was evidence of mild quadriceps, hip adductors, and ham strings [4]. CK levels soft tissue swelling. Ultrasonography around both shoulder are generally normal, with a normal or slightly elevated joints and upper arms showed diffuse swelling of soft tissue LD levels. There may be associated leucocytosis. Upper of muscle compartments without any evidence of local limb involvement in diabetes mellitus is extremely rare. or muscle tumor and a subsequent Doppler study Very few reports have shown unique involvement of the revealed absence of . unilateral arm muscles [5, 6]. Initial presentation of dia- A Magnetic Resonance Imaging of the right and left betes mellitus in a patient as diabetic myonecrosis has upper limbs showed features of myositis, myoedema, and been described only once before by the classical lower focal necrotic regions (greater on the left) and subcutaneous limb predominant presentation [7]. Bilateral upper limb edema with increased signal intensities on T2-weighted proximal muscle involvement has never been reported images, involving deltoid, supraspinatus, and pectoralis mus- before. cles. There were also mild increased signal changes seen from Muscle is the gold standard for diagnosis. But subscapularisaswellasteresmuscle(Figures2(a) and 2(b)). current studies and experience teach us that a muscle Considering the clinical and radiological findings, a biopsy or surgical excision of the affected muscles will either diagnosis of Diabetic myonecrosis was made and the patient prolong the disease in the individual or acutely exacerbate was started on , aggressive therapy, and this condition. Usually MRI and clinical examination will absolute bed rest. Within 4 weeks of admission, he showed suffice, in coming to a diagnosis of diabetic muscle infarction signs and symptoms of improvement with decrement in pain [8]. The differential diagnosis includes infections (myositis, with near normalization of his glycemic status. A repeat , abscess, necrotizing fasciitis, and ), level was 6.1 after a month and a half. trauma (hematoma, muscle rupture, myositis ossificans), vascular (deep vein thrombosis, compartment syndrome), 3. Discussion tumors, inflammatory muscle diseases, and drug-related myositis (statin group) [9]. The short-term prognosis is Diabetic myonecrosis or Diabetic muscle infarction as it is generally good, but the long-term prognosis is complicated called is an uncommon complication of Diabetes Mellitus. by recurrences in a previously affected muscle or muscles Most patients have long-standing diabetes with or without of the opposite limb. Nearly half of these recurrences occur extensive end organ damage, as a result of microvascular within 2 months of initial presentation. Treatment includes disease [2]. Severe diabetic microangiopathy has been pro- adequate rest, analgesics, and good glycemic control. Some posed as the underlying mechanism that leads to sponta- authors propose the use of antiplatelet therapy to treat neous nongangrenous and focalized muscle infarction. Other the underlying micro-vasculopathy, but this is not a strict proposed theories include -fibrinolysis derange- recommendation [10]. ment, hypoxia-reperfusion abnormalities, and recently, the There has been only one case report from India on this role of anti-phospholipid antibodies [3]. condition previously [11]. Case Reports in Endocrinology 3

· Signa 1.5T SYS GEMSOW S 146 EKO CT and MRI SCAN Centre MOH · S 146 EKO CT and MRI SCAN Centre MOH Ex: 5327 Pramatha N. Biswas 65/M Signa 1.5T SYS GEMSOW Pramatha N. Biswas 65/M Se: 9 17423 Ex: 5327 17423 Im: 11 Se: 9 OSag R180.9 Im: 12 May0211 OSag R174.4 May0211 04:03:37 PM 04:03:38 PM Mag = 1.7 Mag = 1.7 FL: FL: ROT: ROT: ET: 16 ET: 16

A A 1 P 1 P 0 9 0 9 4 6 4 6

FSE-XL/90 FSE-XL/90 TR: 4440 TR: 4440 TE: 96.6/Ef TE: 96.6/Ef EC: 1/1 20.9 kHz EC: 1/1 20.9 kHz

TORSOPA TORSOPA FOV: 34 × 34 FOV: 34 × 34 5.0 thk/1.5 sp 5.0 thk/1.5 sp 23/03:42 23/03:42 320X192/2 NEX RT. side v> 320X192/2 NEX RT. side v> St:SI/NP/WB I 54 W = 515 L = 207 St:SI/NP/WB I 54 W = 515 L = 207

Signa 1.5T SYS·GEMSOW S 146 EKO CT and MRI SCAN Centre MOH Signa 1.5T SYS·GEMSOW S 146 EKO CT and MRI SCAN Centre MOH Ex: 5327 Pramatha N. Biswas 65/M Ex: 5327 Pramatha N. Biswas 65/M Se: 9 17423 Se: 9 17423 Im: 15 Im: 16 OSag R154.9 May 02 11 OSag R148.4 May0211 04:03:38 PM 04:03:38 PM Mag = 1.7 Mag = 1.7 ET: 16 FL: ET: 16 FL: ROT: ROT:

A A 1 P 1 P 0 9 0 9 4 6 4 6

FSE-XL/90 FSE-XL/90 TR: 4440 TR: 4440 TE: 96.6/Ef TE: 96.6/Ef EC: 1/1 20.9 kHz EC: 1/1 20.9 kHz TORSOPA TORSOPA FOV: 34 × 34 FOV: 34 × 34 5.0 thk/1.5 sp 5.0 thk/1.5 sp 23/03:42 23/03:42 320X192/2 NEX 320X192/2 NEX RT. side v> St:SI/NP/WB RT. side v> St:SI/NP/WB I 54 W = 516 L = 210 I 54 W = 516 L = 210

(a)

Signa 1.5T SYS·GEMSOW AI EKO CT and MRI SCAN Centre MOH Signa 1.5T SYS·GEMSOW AI EKO CT and MRI SCAN Centre MOH Ex: 5327 Pramatha N. Biswas 65/M Ex: 5327 Pramatha N. Biswas 65/M Se: 12 17423 Se: 12 17423 Im: 4 Im: 5 OAx S113.7 OAx S102.7 May0211 May 02 11 04:15:53 PM 04:15:53 PM Mag = 1.8 Mag = 1.8 ET: 17 FL: ET: 17 FL: ROT: ROT: FLT: e1 FLT: e1

L R L R I S I S P A P A

FSE-XL/90 FSE-XL/90 TR: 3060 TR: 4440 TE: 86.6/Ef TE: 96.6EF EC: 1/1 20.8 kHz EC: 1/1 20.9 Hz

Body Body FOV: 34 × 26 FOV: 34 × 26 5.0 thk/6.0 sp 5.0 thk/6.0 sp 23/02:15 23/02:15 320X192/2 NEX LT. side v> 320X192/2 NEX LT. side v> VB PS W = 575 L = 203 VB PS W = 575 L = 203

Signa 1.5T SYS·GEMSOW AI EKO CT and MRI SCAN Centre MOH Signa 1.5T SYS·GEMSOW AI EKO CT and MRI SCAN Centre MOH Ex: 5327 Pramatha N. Biswas 65/M Ex: 5327 Pramatha N. Biswas 65/M Se: 12 17423 Se: 12 17423 Im: 8 Im: 9 OAx S69.7 OAx S58.7 May 02 11 May0211 04:15:53 PM 04:15:53 PM Mag = 1.8 Mag = 1.8 FL: FL: ET: 17 ROT: ET: 17 ROT: FLT: e1 FLT: e1

R L R L S I S I A P A P

FSE-XL/90 FSE-XL/90 TR: 4440 TR: 4440 TE: 96.6EF TE: 96.6EF EC: 1/1 20.9 Hz EC: 1/1 20.9 Hz

Body Body FOV: 34 × 26 FOV: 34 × 26 5.0 thk/6.0 sp 5.0 thk/6.0 sp 23/02:15 23/02:15 320X192/2 NEX LT. side v> 320X192/2 NEX LT. side v> VB PS W = 575 L = 203 VB PS W = 575 L = 203

(b)

Figure 2: (a) Magnetic Resonance Imaging (T2W) of muscles around the right shoulder joint and right anterior chest region showing areas of increased signal intensities with areas of myositis, muscle swelling (red arrows), and subcutaneous edema. (b) Magnetic Resonance Imaging (T2W) of the muscles around the left shoulder joint revealing areas of hyperintensities suggestive of (yellow arrows) and inflammation of muscles with subcutaneous edema. 4 Case Reports in Endocrinology

Our case is unique in the sense that the initial presenta- tion of Diabetes Mellitus was that of myonecrosis involving bilateral proximal muscles of the upper limb, mainly the deltoids and pectoralis muscles.

References

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