A rare case of a splenic hamartoma in a patient with a huge palpable abdominal mass: a case report

Vlachou, Paraskevi A.; Fagkrezos, Dimitris; Tzivelopoulou, Anastasia; Kyriakopoulou, Georgia; Maniatis, Petros; Triantopoulou, Charikleia; Papailiou, John

doi:10.1186/1752-1947-9-4

Cited by 7 publications

(10 citation statements)

References 11 publications

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“…We didn't order an MRI because of the lack of this study in our public hospital. Additionally, this study may distinguish fibrous from non-fibrous hamartomas [ 15 ]. Although SH has some clinical and image features that may suggest the type of lesion, the definitive diagnosis depends on histopathological findings [ 14 , 15 ].…”

Section: Discussionmentioning

confidence: 99%

“…Our patient presented with only one giant lesion of 14 cm on his greater dimension, consequently compressing the spleen. This tumor may vary in size with a median size of 5 cm and a maximum reported size of 23cm [ 15 ].…”

Section: Discussionmentioning

confidence: 99%

“…Littoral cell angioma is characterized by expression of both endothelial and histiocytic markers, it is associated with splenomegaly, and appears as a low attenuating lesion in contrast-enhanced CT. Lymphangioma manifests as a subcapsular nodule or as diffuse lymphangiomatosis in young patients. Hemangioendothelioma, has an intermediate histology between that of a hemangioma and an angiosarcoma, with lining cells showing an intermediate degree of atypia and the latter characterized for being a primary malignant tumor of non-lymphoid origin, plus for being highly aggressive with a poor prognosis [ 15 ]. At last, a sclerosing angiomatoid nodular transformation of the spleen, also identified as multinodular hemangioma, is altered red pulp entrapped by nonneoplastic stromal proliferation [ 4 ].…”

Section: Discussionmentioning

confidence: 99%

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A giant splenic hamartoma associated with hematologic disorders: A case report

González-Urquijo

Rodarte-Shade

Rangel-Rangel

et al. 2018

Annals of Medicine and Surgery

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IntroductionSplenic hamartoma is a primary benign tumor of the spleen, with approximately 150 cases documented in the literature to date, with only a few cases associated with symptoms and hematologic disorders.Presentation of caseA 49-year-old female with no past medical history, presented to the emergency department complaining of a three-month history of intermittent abdominal pain and 12 kg of weight loss. Physical examination revealed abdominal distension and a big palpable and painless mass on the left side of her abdomen measuring 14 cm. Laboratory tests were significant for anemia and thrombocytopenia, with levels of 9.7 g/dL and 47 × 109/L respectively. Ultrasonography showed splenomegaly with a hypoechoic splenic mass and the computed tomography showed a 14 cm splenic mass with heterogeneous enhancement during the arterial phase. A laparotomy with splenectomy was unremarkably accomplished. Histological examination revealed abnormal red pulp proliferation and showed unorganized sinusoid-like vascular channels, compatible with splenic hamartoma. The patient was discharged on postoperative day 3 without complications. She was seen at the ambulatory clinic 6-months after the surgical procedure with a normal blood count.DiscussionAlthough splenic hamartoma is very rare, it must be included in the differential diagnosis of splenic mass-forming lesions. This type of tumor has some specific radiological features. However, the diagnosis of this disease must be based on clinical features and confirmed by pathology.ConclusionIn patients with splenic tumors, splenectomy is indicated in cases where malignancy cannot be excluded, when symptoms occur, or in the rare cases of consequent hematologic disorders.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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A giant splenic hamartoma associated with hematologic disorders: A case report

González-Urquijo

Rodarte-Shade

Rangel-Rangel

et al. 2018

Annals of Medicine and Surgery

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“…Symptomatic cases are invariably of the red pulp type due to its sinusoidal nature, which can sequester cells and result in hypersplenism and expansion [ 13 ]. Typical microscopic features are of disorganized narrow vascular channels lined by CD8-positive, rounded endothelial cells without atypia, showing a low mitotic rate with a low Ki-67 index, in a background of a red pulp stroma, with or without white pulp [ 17 – 19 ]. However, lymphoid follicles are characteristically not present [ 20 ].…”

Section: Discussionmentioning

confidence: 99%

Splenic Hamartoma: A Case Report and Literature Review

et al. 2022

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Patient: Male, 19-year-old Final Diagnosis: Splenic hamartoma Symptoms: Abdominal pain Medication:— Clinical Procedure: Laparoscopic splenectomy Specialty: Surgery Objective: Rare disease Background: Splenic hamartoma (SH) is a benign vascular lesion, usually found incidentally on abdominal images or at autopsy. Only around 200 cases have been reported since 1861, when SH was first described by Rokitansky. Although it is very rare, it is important to be familiar with it, as it may be a diagnostic challenge to distinguish SH from other mass lesions of the spleen based solely on preoperative investigations. Case Report: We describe a case of symptomatic, isolated, single splenic hamartoma in a 19-year-old, otherwise healthy young man who presented with upper abdominal pain, nausea, and vomiting for a few months. The examination was unremarkable. The patient has been previously evaluated with abdominal ultrasonography, which found a suspicious splenic hyperechoic lesion. Computed tomography revealed a heterogeneous 5×7 cm enhancing lesion in the spleen, concerning for splenic hamartoma. The patient underwent laparoscopic splenectomy and recovered well. The histopathology examination confirmed the diagnosis of splenic hamartoma. Conclusions: Splenic hamartoma is a rare benign vascular lesion of debated etiology. Most cases are asymptomatic and are found incidentally on images, in splenectomies performed for other reasons, or at autopsy. Radiologic findings may suggest the diagnosis and new modalities have shown accuracy in distinguishing splenic hamartomas. However, resection with formal or partial splenectomy is usually still needed since the differential diagnosis is wide, from benign to aggressive lesions, and histopathology remains the criterion standard for diagnosis. Given its benign nature, we found no cases of recurrence or metastasis in the literature.

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“…Among the autoimmune pathologies possibly triggered by the anti-TNFα agents used for ulcerative colitis treatment have been reported cases of autoimmune hepatitis, Lewis–Sumner syndrome, Takayasu arteritis, Guillian–Barre syndrome. [ 2 , 3 ]…”

Section: Discussionmentioning

confidence: 99%

Sarcoidosis associated with infliximab therapy in ulcerative colitis

et al. 2017

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Rationale:Although immunomodulatory therapy has been clearly stated as an important landmark in treatment of ulcerative colitis, significantly improving the quality of life for patients with inflammatory bowel disease, there are several aspects to be considered regarding the possible side-effects of anti-TNF alpha agents. In spite of a good safety profile, biologic TNF antagonists may induce paradoxical inflammation, which can manifest as sarcoid-like granulomatosis, consisting of noncaseating granulomas in the affected organs.Patient concerns:We report the case of a 30-year-old male patient, with no personal or familial history of lung disease, with a personal history of ulcerative colitis (UC), under clinical remission following infliximab therapy in maintenance dose, who was admitted for treatment administration, but also for dyspnea, nocturnal sweating, and nonproductive cough.Diagnoses:Based on clinical manifestations, biological landmarks excluding various infections, CT scan, fibrobronchoscopy with bronchoalveolar lavage for culture and immunohistochemical examination, followed by mediastinoscopy with sampling of paratracheal lymph node, which underwent histopathological examination, the patient was diagnosed with drug- induced stage II pulmonary sarcoidosis.Interventions:Since the patient had developed severe allergic reaction after being administered Infliximab at admission, the biological treatment was immediately discontinued. Following the diagnosis of pulmonary sarcoidosis, corticotherapy was initiated.Patient outcomes:After corticotherapy was initiated, the patient had a favorable outcome at 3 months reevaluation, both regarding the course of ulcerative colitis and sarcoidosis.Lessons:Patients under biological therapy using anti-TNF alpha agents must be carefully monitored, in order to early identify potential paradoxical inflammation (such as sarcoidosis) as a side-effect. The drug-related pulmonary disease tends to improve upon withdrawal of the drug, with occasional requirement of steroid treatment. However, a thorough strategy should be assembled in the case of UC relapse in this patient category, with switching to adalimumab or surgical approach as main possibilities.

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A rare case of a splenic hamartoma in a patient with a huge palpable abdominal mass: a case report

Cited by 7 publications

References 11 publications

A giant splenic hamartoma associated with hematologic disorders: A case report

A giant splenic hamartoma associated with hematologic disorders: A case report

Splenic Hamartoma: A Case Report and Literature Review

Sarcoidosis associated with infliximab therapy in ulcerative colitis

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