oncotomskСибирский онкологический журналSiberian journal of oncology1814-48612312-3168Tomsk National Research Medical Сепtеr of the Russian Academy of Sciences10.21294/1814-4861-2025-24-5-198-205oncotomsk-3871Research ArticleСЛУЧАЙ ИЗ КЛИНИЧЕСКОЙ ПРАКТИКИCASE REPORTSСиндром Реклингхаузена–Энгеля. Скелетное проявление гиперпаратиреоидизма: обзор литературы и клиническое наблюдениеRecklinghausen–Engel syndrome and tumor-like skeletal lesions in hyperparathyroidism: literature review and clinical casehttps://orcid.org/0000-0002-4066-2688ВасильевН. В.VasilievN. V.

Васильев Николай Вольтович - доктор медицинских наук, старший научный сотрудник отделения общей и молекулярной патологии, Научно-исследовательский институт онкологии

SPIN-код: 1627-5226

Researcher ID (WOS): D-1144-2012

Author ID (Sсоpus): 57206755190

634009, Томск, пер. Кооперативный, 5

Nikolay V. Vasiliev - MD, DSc, Senior Researcher, Department of General and Molecular Pathology, Cancer Research Institute

Researcher ID (WOS): D-1144-2012

Author ID (Sсоpus): 57206755190

5, Kooperativny St., Tomsk, 634009

https://orcid.org/0000-0002-1195-4008ВторушинС. В.VtorushinS. V.

Вторушин Сергей Владимирович - доктор медицинских наук, профессор, заместитель директора по научной работе и трансляционной медицине, руководитель отделения общей и молекулярной патологии, Научно-исследовательский институт онкологии, Томский НМЦ РАН; профессор кафедры патологической анатомии, ФГБОУ ВО «Сибирский государственный медицинский университет» Минздрава России (г. Томск, Россия)

SPIN-код: 2442-4720

Researcher ID (WOS): S-3789-2016

Author ID (Scopus): 26654562300

634009, Томск, пер. Кооперативный, 5; 634050, Томск, Московский тракт, 2

Sergey V. Vtorushin - MD, DSc, Professor, Deputy Director for Research and Translational Medicine, Head of Department of General and Molecular Pathology, Cancer Research Institute, Tomsk NRMC, Russian Academy of Sciences; Professor, Pathology Department, Siberian SMU, MHR

Researcher ID (WOS): S-3789-2016

Author ID (Scopus): 26654562300

5, Kooperativny St., Tomsk, 634009; 2, Moskovsky trakt, Tomsk, 634050

https://orcid.org/0000-0002-8676-2932ХакимовХ. И.KhakimovKh. I.

Хакимов Хуршед Илхомжонович - кандидат медицинских наук, младший научный сотрудник отделения общей онкологии

SPIN-код: 4799-2673

Researcher ID (WOS): AGB-1459-2022

Author ID (Scopus): 57996937000

634009, Томск, пер. Кооперативный, 5; 634050, Томск, Московский тракт, 2

Khurshed I. Khakimov - MD, PhD, Junior Researcher, Department of General Oncology, Cancer Research Institute, Tomsk National Research Medical Center, Russian Academy of Sciences; Assistant Professor, Department of Oncology

Researcher ID (WOS): AGB-1459-2022

Author ID (Scopus): 57996937000

5, Kooperativny St., Tomsk, 634009; 2, Moskovsky trakt, Tomsk, 634050

khurshed.1319@gmail.comhttps://orcid.org/0000-0003-3882-4665АнисеняИ. И.AnisenyaI. I.

Анисеня Илья Иванович - кандидат медицинских наук, старший научный сотрудник отделения общей онкологии

SPIN-код: 3003-8744

Researcher ID (WOS): D-8434-2012

Author ID (Scopus): 6507640381

634009, Томск, пер. Кооперативный, 5

Ilya I. Anisenya - MD, PhD, Senior Researcher, Department of General Oncology, Cancer Research Institute

Researcher ID (WOS): D-8434-2012

Author ID (Scopus): 6507640381

5, Kooperativny St., Tomsk, 634009

https://orcid.org/0000-0002-4098-6076ЛоосД. М.LoosD. M.

Лоос Дмитрий Максимович - патологоанатом отделения общей и молекулярной патологии

SPIN-код: 9355-4470

Author ID (Scopus): 57224221490

634009, Томск, пер. Кооперативный, 5

Dmitriy M. Loos - MD, Pathologist, Department of General and Molecular Pathology, Cancer Research Institute

Author ID (Scopus): 57224221490

5, Kooperativny St., Tomsk, 634009

https://orcid.org/0000-0003-2179-5685БоковаУ. А.BokovaU. A.

Бокова Устинья Анатольевна - кандидат биологических наук, научный сотрудник лаборатории биологии опухолевой прогрессии, Научно-исследовательский институт онкологии

SPIN-код: 3546-0527

Researcher ID (WOS): AAX-9705-2021

Author ID (Scopus): 57226147765

634009, Томск, пер. Кооперативный, 5

Ustinia A. Bokova - PhD, Researcher, Laboratory of Tumor Progression Biology, Cancer Research Institute

Researcher ID (WOS): AAX-9705-2021

Author ID (Scopus): 57226147765

5, Kooperativny St., Tomsk, 634009

Научно-исследовательский институт онкологии, Томский национальный исследовательский медицинский центр Российской академии наукРоссияCancer Research Institute, Tomsk National Research Medical Center, Russian Academy of Sciences; Siberian State Medical University, Ministry of Health of RussiaRussian FederationНаучно-исследовательский институт онкологии, Томский национальный исследовательский медицинский центр Российской академии наук; ФГБОУ ВО «Сибирский государственный медицинский университет» Минздрава РоссииРоссияCancer Research Institute, Tomsk National Research Medical Center, Russian Academy of Sciences; Siberian State Medical University, Ministry of Health of RussiaRussian FederationНаучно-исследовательский институт онкологии, Томский национальный исследовательский медицинский центр Российской академии наукРоссияCancer Research Institute, Tomsk National Research Medical Center, Russian Academy of SciencesRussian Federation202520112025245198205Copyright © Васильев Н.В., Вторушин С.В., Хакимов Х.И., Анисеня И.И., Лоос Д.М., Бокова У.А., 20252025Васильев Н.В., Вторушин С.В., Хакимов Х.И., Анисеня И.И., Лоос Д.М., Бокова У.А.Vasiliev N.V., Vtorushin S.V., Khakimov K.I., Anisenya I.I., Loos D.M., Bokova U.A.Данная работа распространяется под лицензией Creative Commons Attribution 4.0.This work is licensed under a Creative Commons Attribution 4.0 License.https://www.siboncoj.ru/jour/article/view/3871

Цель исследования – представить обзор с современными сведениями о номенклатуре и терминологических особенностях синдрома Реклингхаузена–Энгеля (паратиреоидная остеодистрофия), его патогенезе, типичных морфологических и лучевых проявлениях. Уделить особое внимание вопросам дифференциальной диагностики бурой опухоли и гигантоклеточной опухоли скелета. Привеcти клинико-морфологические критерии разграничения этих состояний, систематизированные в виде сравнительной таблицы.

Материал и методы

Материал и методы. Поиск соответствующих источников производился в системах Medline, PubMed, Elibrary и др. Из 100 найденных исследований 30 были использованы для написания систематического обзора. Приведен клинический случай резецированной бурой опухоли бедренной кости у 58-летнего мужчины с ранее не распознанным гиперпаратиреозом, продемонстрированы особенности морфологического диагноза и динамика заболевания.

Результаты

Результаты. Синдром Реклингхаузена–Энгеля (паратиреоидная остеодистрофия) представляет собой редкое, но клинически важное метаболическое поражение скелета, развивающееся на фоне гиперпаратиреоидизма. Он характеризуется выраженной остеокластической активностью, формированием кистозных очагов, субпериостальной резорбцией и так называемых бурых опухолей. Несмотря на доброкачественную природу, данный синдром часто имитирует агрессивные гигантоклеточные новообразования кости, что создает сложности в интерпретации клинико-рентгенологических и морфологических данных и повышает риск диагностических ошибок.

Заключение

Заключение. Обсуждаются трудности диагностики при отсутствии выраженной клинической симптоматики гиперпаратиреоза и подчеркивается ключевая роль патоморфолога в составе междисциплинарной команды. Своевременное выявление гиперсекреции паратгормона и радикальное удаление гиперплазированной или опухолевой паращитовидной железы обеспечивают регресс изменений костной ткани и благоприятный прогноз.

Aim of the study

Aim of the study: to provide a contemporary overview of the nomenclature and terminology of Recklinghausen– Engel syndrome (parathyroid osteodystrophy), its pathogenesis, and characteristic morphological and radiological manifestations. Special attention is paid to the differential diagnosis between brown tumor and skeletal giant cell tumor. The study aims to present the clinical and morphological criteria for distinguishing these conditions, systematized in a comparative table.

Material and Methods

Material and Methods. A literature search was conducted in the Medline, PubMed, Elibrary, and other databases. Of the 100 identified publications, 30 were included in this systematic review. We report a case of a resected brown tumor of the femur in a 58-year-old man with previously undiagnosed hyperparathyroidism, detailing the morphological diagnostic features and the disease course.

Results

Results. Recklinghausen-Engel syndrome (parathyroid osteodystrophy) is a rare but clinically significant metabolic skeletal disorder that develops in the setting of hyperparathyroidism. It is characterized by marked osteoclastic activity, the formation of cystic lesions, subperiosteal resorption, and so-called brown tumors. Despite its benign nature, this syndrome often mimics aggressive giant cell bone tumors, which complicates the interpretation of clinical, radiological, and morphological data and increases the risk of diagnostic errors.

Conclusion

Conclusion. This article discusses the diagnostic challenges in the absence of overt clinical symptoms of hyperparathyroidism and emphasizes the key role of the pathomorphologist within a multidisciplinary team. Timely detection of parathyroid hormone hypersecretion and radical removal of the hyperplastic or neoplastic parathyroid gland lead to the regression of bone lesions and a favorable prognosis.

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The authors declare that there are no conflicts of interest present.