Case Report of Atypical Infiltrative Lipomatosis of the Equine Mesojejunum W

EQUINE VETERINARY EDUCATION / AE / MAY 2013 237

Case Report Case report of atypical infiltrative lipomatosis of the equine mesojejunum W. Linnenkohl*,T.Mair† and D. Fews‡ Department of Clinical Sciences, College of Veterinary Medicine, Cornell University, Ithaca, New York, USA; †Bell Equine Veterinary Clinic, Mereworth, Kent; and ‡University of Bristol, School of Veterinary Sciences, Department of Pathology, Langford, Bristol, UK. *Corresponding author email: [email protected]

Keywords: horse; lipomatosis; mesojejunum; colic; lipoma

Summary represent a post differentiation genetic aberration of the A 25-year-old mare was examined for persistent colic. Over normal adipocyte karyotype (Erkert et al. 2007). the previous month, the mare had experienced several This report describes the clinical and post mortem findings episodes of mild recurrent colic. The episode of colic in a mature horse subjected to euthanasia due to persistent persisted for 18 h and the pain became unresponsive to signs of colic. Post mortem examination revealed diffuse analgesic therapy. Euthanasia was undertaken. Post mortem infiltrative lipomatosis of the mesojejunum, which to the examination revealed an atypical infiltrative lipoma of the authors’ knowledge has not been previously reported in the mesojejunum, confirmed histopathologically. To the authors’ horse. knowledge, this is the first case report of a diffuse infiltrative lipoma of the equine mesojejunum. Case history A 25-year-old mare presented with a history of acute colic of Introduction approximately 10 hours’ duration. Over the previous month, the Obesity is well recognised as having a significant effect on mare had experienced several episodes of mild recurrent colic. human morbidity and mortality and there is increasing Prior to admission, the mare had been managed medically on awareness of the potential pathological effects of adipose the yard for a suspected large colon impaction with enteral tissue in horses, including horses affected by colic (Packer fluids and electrolytes (250 g magnesium sulphate in 10 litres of et al. 2011). Benign lipomas of the mesentery are common water administered by nasogastric tube) and ketoprofen findings in the adult horse, with prevalence increasing with (Ketofen 10%)1 (2.2 mg/kg bwt i.v). The mare initially responded age (Blikslager et al. 1992; Edwards and Proudman 1994; well to medical management, but 3–4 h later displayed signs of Freeman and Schaeffer 2001). Strangulation of the small colic once again, which were refractory to administration of intestine or small colon is possible when the stalk of a further analgesics (phenylbutazone (Equipalazone Solution for pedunculated lipoma becomes twisted around a section of Injection)2, 4.4 mg/kg bwt i.v). Therefore, she was referred to the the viscus or its mesentery (Garcia-Seco et al. 2005; Prange hospital for further assessment and monitoring. et al. 2010). Alternatively, in cases where the mass arises close On presentation, the mare was quiet and depressed, but to the intestine, the pedicle may act as an extraluminal not showing any signs of pain. Her heart rate was 44 beats/min obstruction, compressing the intestine and causing a simple, (reference range [rr] 36–44 beats/min) and respiratory rate 10 often intermittent colic (Mair and Edwards 2003). In contrast to breaths/min (rr 8–16 Vf). Rectal temperature was 37.5°C (rr well circumscribed pedunculated lipomas, lipomatosis is a 37–38°C). Mucous membrane colour and capillary refill time nonencapsulated infiltrative adipose tissue lesion not limited were both within normal limits. Gastrointestinal sounds were by tissue planes resulting in massive tissue infiltration and decreased on both sides of the abdomen. Initial rectal expansion (Archer 2007). It is a rare condition that has been examination on admission revealed moderate gas distention of reported infrequently in the veterinary literature affecting the large colon. Abdominocentesis yielded normal peritoneal various species including dogs, llamas, geese, equids and man fluid (total nucleated cell count 0.5 ¥ 109 cells/l and total (Siskind et al. 1984; Dsoust et al. 1991; Bindseil and Madsen protein 26 g/l; rr <2.0 ¥ 109 cells/l and 10–28 g/l, respectively). 1997; Klopfleisch et al. 2009). Transabdominal ultrasonography was unremarkable. Initial Lipomatosis has been reported in various anatomical haematological evaluation revealed a mild leucocytosis (total locations in numerous species, including subcutaneous/ white blood cell count 10.3 ¥ 109/l, rr 5–10 ¥ 109/l) characterised musculoskeletal tissues (Bristol and Fubini 1984; Dsoust et al. by a granulocytosis (granulocytes 9.1 ¥ 109/l, rr 3–8 ¥ 109/l) and 1991; Hammer et al. 2002; Erkert et al. 2007), the epidural space a mild lymphopenia (lymphocytes 1.1 ¥ 109/l, rr 1.5–4 ¥ 109/l). A (Chan et al. 2009; Flisberg et al. 2009), salivary gland (Bindseil biochemistry profile revealed hyperfibrinogenaemia (plasma and Madsen 1997), liver (Klopfleisch et al. 2009), mesentery/ fibrinogen 5.6 g/l, rr 1–4 g/l) and increased serum activity of gastrointestinal tract (Henry and Yamini 1995; Riley et al. 2007) creatinine kinase (CK 7218 iu/l, rr 150–385 iu/l), lactate and the myocardium/mediastinum (Erkert et al. 2007). The dehydrogenase (LDH 2054 iu/l, rr 192–482 iu/l) and aspartate pathogenesis of this uncapsulated, infiltrative lesion is unknown. transaminase (AST 580 iu/l, rr 258–554 iu/l). It has been hypothesised to perhaps result from congenital The mare was admitted to the clinic and placed in a stable abnormalities in younger animals, while in older animals it may for observation. Over the next 4 h, the mare continued to show

Fig 1: A section of mid-jejunum showing diffuse infiltrative Fig 2: The coalescing multi-nodular fat tissue extended to the lipomatous tissue throughout the mesojejunum. Note the multifocal mesenteric border of jejunum. areas of haemorrhage. escalating signs of discomfort and her heart rate was increased above the normal range (66 beats/min). A nasogastric tube was passed and 15 litres of gastric reflux was obtained. Due to the horse’s history of colic and her failure to respond to medical management, an exploratory laparotomy was recommended. The owner declined surgical intervention due to financial constraints and the mare was humanely subjected to euthanasia.

Post mortem findings A necropsy was performed immediately after euthanasia. The pelvic flexure of the large colon was found to be displaced into the right cranial abdomen. A large multinodular infiltrative fatty mass was present in a 2 m segment of mid-jejunal mesojejunum (Fig 1); the small intestine proximal to this lesion was moderately distended with fluid. There was a sharp line of 200 μm demarcation at the oral and aboral borders of the lesion Fig 3: Mesenteric adipose tissue shows a disrupted adipocyte with between normal mesentery and the lipomatosis-affected associated infiltration by macrophages, including mutinuclated section of bowel. In the affected section of bowel, the giant cells (H&E stained and viewed at 100¥ objective abnormal tissue was present from the root of the mesentery to magnification). the mesenteric border of the intestine (Fig 2). Diffuse multifocal haemorrhage was noted within the fatty mass (Fig 1). No (Fig 3). Histopathological examination of the haemorrhagic fat physical obstruction or strangulation was apparent in the small revealed relatively extensive areas of recent haemorrhage intestine at the time of necropsy; however, there was (Fig 4). Otherwise the features of the adipose tissue were moderate fluid distention of the small intestine. Two sections of similar to the sections associated with the jejunum. the jejunum with associated mesentery and one area of mesentery with visible haemorrhage were submitted for histopathological evaluation. Discussion Tissue samples were fixed in 10% neutral buffered formalin, To the authors’ knowledge, this case report represents the embedded in paraffin wax and sectioned (4 mm). For first reported occurrence of lipomatosis of the equine histopathological examination, sections were stained with mesojejunum. Lipomatosis is defined as excessive haematoxylin and eosin (H&E). In the sections of jejunum there accumulation of fat in the body or organ that is not was mild infiltration of the lamina propria by eosinophils and a encapsulated or limited by tissue planes and pathologically normal to mildly increased population of lymphocytes and differs from the behaviour of benign lipomas. It occurs plasma cells. The serosa was slightly oedematous. The uncommonly and has several different presentations (Nielson associated adipose tissue of the mesentery was composed et al. 2001). predominantly of adipocytes of normal appearance; There are currently 2 case reports of lipomatosis of the however, areas of necrosis were also present. Occasionally, equine gastrointestinal tract, both of which were associated adipocytes appeared disrupted by infiltration of with the ascending or descending colon (Henry and Yamini macrophages and small vessels were frequently congested 1995; Riley et al. 2007). In the case report by Riley et al. (2007),

large cutaneous nonencapsulated masses of fatty tissue were identified infiltrating the subcutaneous and underlying muscular tissue. In all horses, no evidence of neoplastic processes was identified histopathologically, which is similar to the benign lipomas seen associated with the gastrointestinal tract. It was hypothesised in these reports that the cutaneous presentation of lipomatosis represents a different pathological process to that of neoplastic infiltrative lesions such as liposarcomas (Bristol and Fubini 1984; Erkert et al. 2007). Based on the post mortem findings, it is impossible to know if the source of gastrointestinal pain was directly associated with the lipomatous tissue of the mesojejunum. The presence of reflux prior to euthanasia and the absence of evidence of ischaemic bowel make a nonstrangulating extraluminal obstruction the likely cause of the colic signs, which could have been caused by the weight of the mesojejunum

200 μm compressing the intestine and restricting the passage of ingesta along the small intestine; however, we cannot rule out Fig 4: Mesenteric adipose tissue shows multifocal acute obstruction of the duodenum by the displaced colon being haemorrhage (H&E stained and viewed at 40¥ objective the source of the gastrointestinal reflux. A report by Ozel et al. magnification). (2004) reports a giant nonstrangulating mesenteric lipoma in a 7-year-old girl as a cause of ileus which caused clinical signs of vomiting and abdominal pain since the age of 3 years. a 2-year-old Tennessee Walking Horse developed a refractory Additionally, the visceral lipomatosis seen in the 2 previously small colon impaction secondary to infiltration of fatty tissue discussed case reports in equids had similar signs of pain into the mesenteric and anti-mesenteric surfaces of the small associated with impactions as a sequel to reduced peristaltic colon. Henry and Yamini (1995) reported similar findings in a function of the small colon (Erkert et al. 2007). It is possible that 7-year-old Quarter Horse that had multi-focal to coalescing the section of bowel affected by lipomatosis experienced fatty masses associated with the anti-mesenteric serosal some degree of ileus, which could have been the cause for surface of the transverse and small colon. Large lipomatous the gastrointestinal reflux. On the other hand, the absence of diverticula were associated with the colonic wall in both any previous history of colic in this horse suggests that the cases. Histopathologically, both cases had evidence of lipomatosis probably existed as a sub-clinical condition prior to infiltration of adipocytes into the muscularis layer of the colon the development of the persistent colic episode. with associated fibrosis, presumed to be responsible for the In conclusion, lipomatosis is a rare and multifaceted clinical signs seen in both patients. In our case, there was no condition that has numerous clinical presentations. This case identified infiltration of adipocytes into the small intestinal wall, report highlights the clinical features of lipomatosis associated although histopathological examination was limited to 2 small with the small intestinal mesentery. Although histologically the sections from a total of 2 m of affected jejunum so it is possible lipomatous mass appeared to be benign, the locally invasive that affected sites were not sampled. and infiltrative nature of the lesion likely contributed to the Lipomatosis in people has been associated with cutaneous, signs of colic. This case report will serve to add more clinical visceral and epidural pathology, each of which have differing and pathological information regarding lipomatosis as it is the clinical signs (Siskind et al. 1984; Nielson et al. 2001; Cha et al. first reported instance of infiltrative lipomatosis in the 2009; Chan et al. 2009; Flisberg et al. 2009). Siskind et al. (1984) mesojejunum of an equid. published a case report linking prolonged steroid use to mesenteric lipomatosis. It is well documented in many species that corticosteroids can induce alterations of fat distribution, Authors’ declaration of interests particularly intra-abdominal fat in man (Wajchenberg et al. No conflicts of interest have been declared. 1995) and subcutaneous and omental fat in horses (Johnson 2002), as seen in horses affected by Cushing’s disease or equine metabolic syndrome. Our patient had no previous Manufacturers’ addresses documented history of endocrinopathy, but it is known that a 1Merial Animal Health Ltd, Harlow, UK. horse’s risk for pituitary-pars intermedia dysfunction (PPID) is 2Dechra Veterinary Products, Shrewsbury, UK. greatly increased with age. It is possible that this patient had an underlying undiagnosed endocrinopathy that could have contributed to the development of this condition; however, References many aged horses have clinical evidence of PPID but the Archer, D. (2007) Lipomatosis: just another fatty lump? Equine Vet. Educ. condition of lipomatosis of the gastrointestinal tract appears to 19, 489-490. be extremely rare. Bindseil, E. and Madsen, J. (1997) Lipomatosis causing tumour-like Another clinical presentation of lipomatosis reported in swelling of a mandibular salivary gland in a dog. Vet. Rec. 140, 583-584. several species, including horses, is cutaneous lipomatosis Blikslager, A.T., Bowman, K.F., Haven, M.L., Lloyd, P.T. and Bristol, D.G. (Bristol and Fubini 1984; Erkert et al. 2007), which has also been (1992) Pedunculated lipomas as a cause of intestinal obstruction in referred to as infiltrative lipomas. This condition was reported in horses: 17 cases (1983-1990). J. Am. Vet. Med. Ass. 201, 249- several young horses, all less than 2 years of age, in which 1252.

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