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  IN THIS Article
 ::  Abstract
  ::  Introduction
  ::  Case History
  ::  Discussion
 ::  References
 ::  Article Figures

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CASE REPORT
Year : 2023  |  Volume : 69  |  Issue : 1  |  Page : 50-52

Progressive nodule-like lesions on bilateral lower limbs


China Medical University Hospital, China Medical University, Taichung, Taiwan

Date of Submission06-Feb-2022
Date of Decision06-Mar-2022
Date of Acceptance04-Apr-2022
Date of Web Publication19-Dec-2022

Correspondence Address:
Dr. J W Chou
China Medical University Hospital, China Medical University, Taichung
Taiwan
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jpgm.jpgm_136_22

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 :: Abstract 


Pancreatic panniculitis is a rare disease characterized by subcutaneous fat necrosis. It could be the result of an associated pancreatic tumor. Herein, we reported a 63-year-old man who presented with progressive bilateral lower limb edema accompanied with nodule-like lesions for 1 month. His serum lipase was 3,927 U/L (normal, 0–160 U/L). Histopathology of the skin specimen revealed lobular panniculitis, favoring a diagnosis of pancreatic panniculitis. Abdominal computed tomography (CT) scan with contrast showed a huge mass in his left upper quadrant. Endoscopic ultrasound showed a mixed echoic tumor, measuring 11.9 × 7.8 cm in dimensions, originating from the pancreatic tail. Biopsy performed via an endoscopic ultrasound showed a poorly differentiated acinar cell carcinoma. Because of the unresectable status of the tumor, the patient underwent chemotherapy with paclitaxel and gemcitabine. After chemotherapy, his skin lesions improved progressively. It is important to treat pancreatic panniculitis with its underlying pancreatic disease.


Keywords: Acinar cell carcinoma, chemotherapy, pancreatic panniculitis


How to cite this article:
Yang F A, Cheng K S, Chou J W. Progressive nodule-like lesions on bilateral lower limbs. J Postgrad Med 2023;69:50-2

How to cite this URL:
Yang F A, Cheng K S, Chou J W. Progressive nodule-like lesions on bilateral lower limbs. J Postgrad Med [serial online] 2023 [cited 2023 May 30];69:50-2. Available from: https://www.jpgmonline.com/text.asp?2023/69/1/50/364508





 :: Introduction Top


Pancreatic panniculitis is a rare disease characterized by subcutaneous fat necrosis. It presents with tender, ill-defined, red-brown nodules, especially in lower extremities clinical manifestation include tender, sometimes fluctuate subcutaneous nodules that commonly involve the legs, but sometimes other sites of the body in some case reports.[1],[2] It may cause ulceration and drain an oily substance.[1] The release of pancreatic enzymes (lipase) into the bloodstream, causing hyperlipidemia in the setting of pancreatic injury may be the leading cause of it, resulting in fat necrosis in subcutaneous tissue.[2] Pancreatic panniculitis has been reported in acute and chronic pancreatitis and pancreatic neoplasms (acinar cell carcinoma in 80% of cases).[3] We herein report a case of pancreatic panniculitis that was associated with pancreatic acinar cell carcinoma.


 :: Case History Top


A 63-year-old male presented to our hospital because of progressive bilateral lower limbs edema accompanied with nodule-like lesions for 1 month [Figure 1]. He was diagnosed with pancreatic tumor at another hospital 3 months ago. One month ago, progressive bilateral lower limb edema accompanied with multiple nodule-like skin lesions were noted. Initially, the lesions were erythematous, painful, and firm on palpation. As time went by, pus and some oily substance started to drain from the lesions. Ulcers formed after these substances were drained out. He denied having either fever/trauma history, or any specific gastrointestinal symptoms except for weight loss of 10 kg.
Figure 1: Physical examination of the patient showed several painful, erythematous nodules with ulcerations and crusts in the left leg

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On presentation, his vital signs were stable without fever. Auscultation revealed left lower lung crackles accompanied with decreased breath sounds. His abdomen was soft on palpation without focal tenderness. There were several nodular, erythematous, and painful lesions over his lower limbs. Some ulcerations that drained pus and oily substance were seen from these lesions. Chest x-ray showed a left sided massive pleural effusion. Thus, thoracentesis was performed and some pleural fluids were aspirated. Pleural fluid examination revealed that it was exudate with an elevated white count with neutrophil predominant. Laboratory tests revealed a white blood cell count of 13.5 × 109/L (normal range, 3.5–11 × 109/L) with a neutrophil predominance and an elevated C-reactive protein level of 17.99 mg/dL (normal range, <1 mg/dL). His serum lipase was 3,927 U/L (normal range, 0–160 U/L). His serum tumor markers were within the normal limits except for the mild elevation of serum AFP (AFP: 16.39 ng/mL). He was admitted to our gastrointestinal ward for further evaluation and treatment.

After admission, we consulted a dermatologist for the evaluation of his skin lesions. A skin biopsy and culture were both done. The culture of skin lesions was negative for any atypical infection. In due course, the skin lesions extended to his right buttock, abdomen, and upper extremities. Abdominal computed tomography (CT) scan with contrast showed a heterogeneous hypodense lesion, measuring 11 × 10 cm in size, with some gas bubbles inside in left upper abdomen [[Figure 2]a, arrows]. Endoscopic ultrasound was performed to get samples from the mass. The mass was a mixed echoic tumor, measuring 11.9 × 7.8 cm in dimensions, originating from the pancreatic tail [[Figure 2]b, arrow]. Histopathological report of the skin biopsy specimen showed infiltrations of histiocytes, lymphocytes, neutrophils, and lobular focal fat necrosis with saponification and “ghost cells” [[Figure 3], arrows, hematoxylin and eosin staining; original magnification, ×40]. Histopathological examination of biopsy specimen from the pancreatic tumor showed poorly differentiated acinar cell carcinoma. Based on histopathological findings, a final diagnosis of poorly differentiated acinar cell carcinoma of the pancreas with secondary pleural effusion and pancreatic panniculitis was made. Due to the unresectable condition of the cancer, he was transferred to the oncological department. He accepted chemotherapy with paclitaxel and gemcitabine and was later discharged under stable condition. At 3-months follow-up after discharge, his skin lesions had resolved and his serum lipase level had decreased to 2,542 U/L. Follow-up abdominal CT scan with contrast revealed no progression of the pancreatic cancer. The patient will keep current treatment in our hospital in the future.
Figure 2: (a) Axial view of abdominal CT showed a huge mass in the left upper quadrant (arrows). (b) Endoscopic ultrasound showed a mass lesion, 11.9 × 7.8 cm in size, in the pancreatic tail (arrow)

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Figure 3: (a) Histopathology of biopsy specimen from the skin showed infiltrations of histiocytes, lymphocytes, neutrophils, and lobular focal fat necrosis with saponification and “ghost” cells, in favor of lobular panniculitis (arrows, H & E staining; ×40); (b) Histopathology of biopsy specimen from the pancreatic tumor showed poorly differentiated acinar cell carcinoma (H & E staining; x400)

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 :: Discussion Top


Pancreatic panniculitis is a rare extra-pancreatic manifestation of acinar cell carcinoma of the pancreas.[1] Pancreatic panniculitis is mostly associated with pancreatic diseases.[1],[2] Classical findings of pancreatic panniculitis include tender, sometimes fluctuating subcutaneous nodules that commonly involve the legs, but sometimes other sites of the body in some case reports.[2] The nodules can ulcerate, releasing oily, thick, yellowish-brown material indicative of necrotic adipose tissue.[2],[3],[4] The eruption is owing to the massive release of lipolytic enzymes (lipase) into the bloodstream and causes hyperlipidemia.[2]

The diagnosis of pancreatic panniculitis is by histopathology. Pancreatic panniculitis shows the characteristic features of lobular panniculitis with intense necrosis of the adipocytes.[4] Fully matured lesions of pancreatic panniculitis show a characteristic coagulative necrosis of the adipocytes, which leads to “ghost adipocytes.”[5] Ghost adipocytes are those that have lost their nuclei and show a finely granular and basophilic material within their cytoplasm because of calcium deposition, and the process is known as saponification.[1],[4] Ghost adipocytes appear in groups as small clusters at the center of the fat lobule, whereas an inflammatory infiltrate of neutrophils is present at the periphery. In older lesions, fat necrosis and calcified ghost adipocytes are less evident and the fat lobules are replaced by a granulomatous inflammatory infiltrate composed of foamy histiocytes and multinucleated giant cells.[4] The treatment of pancreatic panniculitis is to treat underlying pancreatic disease.[5] In patients with an unresectable malignancy, it is recommended they be treated with chemotherapy, which can result in symptom reduction.[6],[7],[8]

In conclusion, although pancreatic panniculitis is a rare event in clinical practice, clinicians should pay an attention in patients with unexplained skin lesions with an elevated serum lipase level. Moreover, it is crucial to treat pancreatic panniculitis with its underlying pancreatic disease. We hope that the present case will assist in shared decision making between patients, caregivers, and their clinicians.

Declaration of patient consent

The authors certify that appropriate patient consent was obtained.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 :: References Top

1.
Yoshihiro T, Nio K, Tsuchihashi K, Ariyama H, Kohashi K, Tsuruta N, et al. Pancreatic acinar cell carcinoma presenting with panniculitis, successfully treated with FOLFIRINOX: A case report. Mol Clin Oncol 2017;6:866-70.  Back to cited text no. 1
    
2.
Rongioletti F, Caputo V. Pancreatic panniculitis. G Ital Dermatol Venereol 2013;148:419-25.  Back to cited text no. 2
    
3.
Zhang G, Cao Z, Yang G, Wu W, Zhang T, Zhao Y. Pancreatic panniculitis associated with pancreatic carcinoma: A case report. Medicine (Baltimore) 2016;95:e4374.  Back to cited text no. 3
    
4.
Requena L, Sánchez Yus E. Panniculitis. Part II. Mostly lobular panniculitis. J Am Acad Dermatol 2001;45:325-61.  Back to cited text no. 4
    
5.
Miksch RC, Schiergens TS, Weniger M, Ilmer M, Kazmierczak PM, Guba MO, et al. Pancreatic panniculitis and elevated serum lipase in metastasized acinar cell carcinoma of the pancreas: A case report and review of literature. World J Clin Cases 2020;8:5304-12.  Back to cited text no. 5
    
6.
Singh S, Gorouhi F, Konia T, Burrall B. Pancreatic acinar cell carcinoma-induced panniculitis. JAAD Case Rep 2018;4:719-21.  Back to cited text no. 6
    
7.
Yoo C, Kim BJ, Kim KP, Lee JL, Kim TW, Ryoo BY, et al. Efficacy of chemotherapy in patients with unresectable or metastatic pancreatic acinar cell carcinoma: Potentially improved efficacy with oxaliplatin-containing regimen. Cancer Res Treat 2017;49:759-65.  Back to cited text no. 7
    
8.
Zheng ZJ, Gong J, Xiang GM, Mai G, Liu XB. Pancreatic panniculitis associated with acinar cell carcinoma of the pancreas: A case report. Ann Dermatol 2011;23:225-8.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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