Bisceglia Michele, Chiaramonte Antonio, Panniello Gaetano, Tucci Antonio, Orcioni Giulio Fraternali, Colby Thomas V
*Pathologist, School of Biomedical Sciences, Etromapmacs Pole, Lesina (FG) †Division of Thoracic Surgery, IRCCS Casa Sollievo della Sofferenza Hospital, San Giovanni Rotondo ‡Division of Clinical Dermatology, Ospedali Riuniti di Foggia, Foggia §Director, School of Biomedical Sciences, Etromapmacs Pole, Lesina (FG) ∥Division of Anatomic Pathology, "San Martino-IST" Polyclinic, Genova, Italy ¶Department of Pathology, Mayo Clinic Scottsdale, Scottsdale, AZ.
Adv Anat Pathol. 2015 Jan;22(1):59-68. doi: 10.1097/PAP.0000000000000051.
Two cases of diffuse dendriform pulmonary ossification (DPO) are presented, one of the secondary type and the other of the idiopathic type. Case 1 was an adult female patient who underwent thoracic surgery to remove a posterior mediastinal bronchogenic cyst, which was discovered on a computed tomography scan performed after an episode of pneumonia when traction bronchiectasis with interstitial lung disease/fibrosis was also suspected in the lungs. Histologic examination performed on the resected lung tissue revealed numerous scattered small osseous spicules on a background of intense interstitial inflammation and fibrosis, leading to further clinical and laboratory investigations and the final diagnosis of DPO secondary to lung involvement by scleroderma. Case 2 was an adult male patient who underwent thoracoscopic exploration after a computed tomography scan, which revealed traction bronchiectasis with linear thickening of the interstitial lung tissue. Histologic examination of the lung tissue specimen revealed numerous osseous spicules in the absence of interstitial septal inflammation. Noteworthy in this case were also some nodules of collagenized tissue similar to those seen in the lungs of patients affected by Ehlers-Danlos syndrome. The absence of any clinical sign or symptoms related to Ehlers-Danlos syndrome attest to the nonspecificity of these pulmonary fibrous nodules. No case of DPO secondary to scleroderma has been reported in the literature so far, although around half of the patients with scleroderma manifest pulmonary diseases. Idiopathic DPO is even rarer, usually discovered postmortem, with only 20 cases diagnosed in life with lung biopsies taken by open surgery or through a thoracoscopic approach. DPO is often misdiagnosed radiologically as bronchiectasis and/or interstitial lung disease/fibrosis.
本文报告了两例弥漫性树状肺骨化(DPO)病例,一例为继发性,另一例为特发性。病例1是一名成年女性患者,因肺炎发作后进行计算机断层扫描发现后纵隔支气管源性囊肿,遂接受胸外科手术切除,当时肺部还怀疑有牵引性支气管扩张伴间质性肺疾病/纤维化。对切除的肺组织进行组织学检查,发现在强烈的间质性炎症和纤维化背景上有许多散在的小骨针,这促使进一步进行临床和实验室检查,最终诊断为硬皮病累及肺部继发DPO。病例2是一名成年男性患者,计算机断层扫描显示有牵引性支气管扩张伴间质性肺组织线性增厚,随后接受了胸腔镜探查。肺组织标本的组织学检查显示有许多骨针,但无间质间隔炎症。该病例中值得注意的还有一些胶原化组织结节,类似于埃勒斯-当洛综合征患者肺部所见。与埃勒斯-当洛综合征相关的任何临床体征或症状均不存在,证明这些肺部纤维结节具有非特异性。迄今为止,文献中尚未报道过硬皮病继发DPO的病例,尽管约一半的硬皮病患者有肺部疾病表现。特发性DPO更为罕见,通常在尸检时发现,仅有20例通过开放手术或胸腔镜途径进行肺活检在生前确诊。DPO在影像学上常被误诊为支气管扩张和/或间质性肺疾病/纤维化。